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Yu P, Zhou X, Yue L, Zhang L, Zhou Y, Jiang F. Comparative diagnostic performance of imaging modalities in chronic pancreatitis: a systematic review and Bayesian network meta-analysis. BMC Med Imaging 2025; 25:1. [PMID: 39748339 PMCID: PMC11697682 DOI: 10.1186/s12880-024-01541-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/17/2024] [Accepted: 12/19/2024] [Indexed: 01/04/2025] Open
Abstract
PURPOSE We aimed to perform a Bayesian network meta-analysis to assess the comparative diagnostic performance of different imaging modalities in chronic pancreatitis(CP). METHODS The PubMed, Embase and Cochrane Library databases were searched for relevant publications until March 2024. All studies evaluating the head-to-head diagnostic performance of imaging modalities in CP were included. Bayesian network meta-analysis was performed to compare the sensitivity and specificity between the imaging modalities. The Quality Assessment of Diagnostic Performance Studies (QUADAS-2) tool was used to evaluate the quality of studies. RESULTS This meta-analysis incorporated 17 studies. Network meta-analytic results indicated that endoscopic ultrasonography (EUS) achieved the highest surface under the cumulative ranking (SUCRA) value at 0.86 for sensitivity. Conversely, magnetic resonance imaging (MRI) demonstrated best specificity, recording the highest SUCRA value at 0.99. Ultrasonography (US) displayed comparatively lower sensitivity than endoscopic retrograde cholangiopancreatography (ERCP) (relative risk[RR]: 0.83, 95% Confidence Interval[CI]: 0.69-0.99) and EUS (RR: 0.73, 95% CI: 0.57-0.91). MRI outperformed all other imaging modalities in terms of specificity. CONCLUSIONS It appears that EUS demonstrates higher sensitivity, while MRI exhibits higher specificity in patients with chronic pancreatitis. However, it is crucial to note that our analysis was limited to the diagnostic performance and did not evaluate the cost-effectiveness of these various imaging modalities. Consequently, further extensive studies are needed to assess the benefit-to-risk ratios comprehensively.
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Affiliation(s)
- Ping Yu
- Department of Pharmacy, Taihe Hospital, Hubei Provincial Clinical Research Center for Umbilical Cord Blood Hematopoietic Stem Cells, Hubei University of Medicine, Shiyan, Hubei, 442000, China
| | - Xujia Zhou
- Department of Pharmacy, Taihe Hospital, Hubei Provincial Clinical Research Center for Umbilical Cord Blood Hematopoietic Stem Cells, Hubei University of Medicine, Shiyan, Hubei, 442000, China
- Department of Radiology, Taihe Hospital, Hubei University of Medicine, Shiyan, Hubei, 442000, China
| | - Li Yue
- Office of Administration and Management, Taihe Hospital, Hubei University of Medicine, Shiyan, Hubei, 442000, China
| | - Ling Zhang
- Department of Pharmacy, Taihe Hospital, Hubei Provincial Clinical Research Center for Umbilical Cord Blood Hematopoietic Stem Cells, Hubei University of Medicine, Shiyan, Hubei, 442000, China
| | - Yuan Zhou
- Department of Pharmacy, Taihe Hospital, Hubei Provincial Clinical Research Center for Umbilical Cord Blood Hematopoietic Stem Cells, Hubei University of Medicine, Shiyan, Hubei, 442000, China.
- Department of Respiratory, Taihe Hospital, Hubei University of Medicine, Shiyan, Hubei, 442000, China.
| | - Fei Jiang
- Department of Pharmacy, Taihe Hospital, Hubei Provincial Clinical Research Center for Umbilical Cord Blood Hematopoietic Stem Cells, Hubei University of Medicine, Shiyan, Hubei, 442000, China.
- Department of Respiratory, Taihe Hospital, Hubei University of Medicine, Shiyan, Hubei, 442000, China.
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Shimada R, Yamada Y, Okamoto K, Murakami K, Motomura M, Takaki H, Fukuzawa K, Asayama Y. Pancreatic volume change using three dimensional-computed tomography volumetry and its relationships with diabetes on long-term follow-up in autoimmune pancreatitis. World J Radiol 2024; 16:644-656. [PMID: 39635311 PMCID: PMC11612800 DOI: 10.4329/wjr.v16.i11.644] [Citation(s) in RCA: 2] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/25/2024] [Revised: 09/07/2024] [Accepted: 09/23/2024] [Indexed: 11/27/2024] Open
Abstract
BACKGROUND Several studies found that early pancreatic atrophy detected by computed tomography (CT) within 6 months was associated with a high incidence of diabetes in patients with type-1 autoimmune pancreatitis (AIP) receiving steroid therapy; however, no long-term follow-up studies have been performed. AIM To investigate pancreatic volume (PV) changes using three dimensional (3D)-CT volumetry and their relationship with IgG4 and diabetes in patients with AIP. METHODS This retrospective study included 33 patients with type-1 AIP receiving steroid therapy. Patients were divided into diffuse (D-type) and mass-forming type (M-type) AIP. PV was determined by semi-automated 3D-CT volumetry, and changes between initial and follow-up values were calculated. The relationship between PV and serum IgG4 levels was analyzed by Spearman's rank correlation. The PV atrophy ratio compared with the presumed normal PV at the time of last follow-up CT and its relationship with diabetes were investigated. RESULTS There were 16 D-type and 17 M-type patients with long-term follow-up (mean, 95.8 months). The regression curve of mean relative PV change reduced exponentially and rapidly during the first 25 months and then more slowly in both groups. The overall cumulative pancreas re-enlargement rates at 1, 3, 5, 7 and 10 years were 6.1%, 12.2%, 29.2%, 47.5% and 55.0%, respectively. There was a moderate-to-very strong positive correlation (ρ ≥ 0.4) between PV and serum IgG4 levels in nine (9/13, 69.2%) patients. All 33 patients showed pancreatic atrophy (mean 59.3%) after long-term follow-up. Patients with D-type AIP had a significantly higher atrophy rate and higher incidence of diabetes than M-type patients (P < 0.05). CONCLUSION PV change initially reduced exponentially and then more slowly and is considered an important factor associated with diabetes. Serum IgG4 levels were positively correlated with PV during follow-up.
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Affiliation(s)
- Ryuichi Shimada
- Department of Radiology, Oita University Faculty of Medicine, Yufu 879-5593, Oita, Japan
| | - Yasunari Yamada
- Department of Radiology, Oita Red Cross Hospital, Oita 870-0033, Oita, Japan
| | - Kazuhisa Okamoto
- Department of Gastroenterology, Oita University Faculty of Medicine, Yufu 879-5593, Oita, Japan
| | - Kazunari Murakami
- Department of Gastroenterology, Oita University Faculty of Medicine, Yufu 879-5593, Oita, Japan
| | - Mitsuteru Motomura
- Department of Hepato-Biliary-Pancreatic Internal Medicine, Oita Red Cross Hospital, Oita 870-0033, Oita, Japan
| | - Hajime Takaki
- Department of Radiology, Oita Red Cross Hospital, Oita 870-0033, Oita, Japan
| | - Kengo Fukuzawa
- Department of Surgery, Oita Red Cross Hospital, Oita 870-0033, Oita, Japan
| | - Yoshiki Asayama
- Department of Radiology, Oita University Faculty of Medicine, Yufu 879-5593, Oita, Japan
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Sharma RK, Vitali F, Chhabra P. Editorial: Diagnosis and management of acute, chronic, and autoimmune pancreatitis. Front Med (Lausanne) 2024; 11:1517007. [PMID: 39624037 PMCID: PMC11609844 DOI: 10.3389/fmed.2024.1517007] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/25/2024] [Accepted: 11/04/2024] [Indexed: 01/18/2025] Open
Affiliation(s)
- Ravi Kumar Sharma
- Department of University Institute of Biotechnology, Chandigarh University, Mohali, Punjab, India
| | - Francesco Vitali
- Department of Internal Medicine, A University Hospital of Greifswald, Greifswald, Germany
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Li Y, Zheng C, Zhang Y, He T, Chen W, Zheng K. Enhancing preoperative diagnosis of pancreatic ductal adenocarcinoma and mass-forming chronic pancreatitis: a study on normalized conventional MR imaging parameters. Abdom Radiol (NY) 2024:10.1007/s00261-024-04652-7. [PMID: 39488674 DOI: 10.1007/s00261-024-04652-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2024] [Revised: 10/10/2024] [Accepted: 10/17/2024] [Indexed: 11/04/2024]
Abstract
PURPOSE To assess the utility of signal intensity ratio (SIR) in distinguishing between mass-forming chronic pancreatitis (MFCP) and pancreatic ductal adenocarcinoma (PDAC), thereby reducing unnecessary pancreatectomies or delayed diagnosis brought by misdiagnosis. MATERIALS AND METHODS This retrospective study included 170 participants (34 with MFCP and 136 with PDAC) who underwent radical pancreatic surgery and were diagnosed via specimen pathology. The study group was carefully selected with a 1:4 ratio matching for sex, age, and operation time between two entities. T1 SIR, T2 SIR, arterial phase (AP) SIR, portal venous phase (VP) SIR, delay phase (DP) SIR, DWI0-50 SIR, and DWI500-1000 SIR, were calculated by dividing the signal intensity of lesions by that of the paraspinal muscle, serving as a reference organ. Intraclass Correlation Coefficient (ICC) was estimated to evaluate the intraobserver and interobserver reliability. Wilcoxon tests were employed for univariate analysis, and receiver operating characteristic (ROC) curves were generated to determine optimal cutoff points and AUC values for selected predictors. A tenfold cross-validation method was applied to validate the robustness of the results. RESULTS The ICC demonstrated excellent correlation for both intraobserver and interobserver(ICCs > 0.8). T1 SIR, AP SIR, VP SIR, and DP SIR were significantly lower in the PDAC group compared to the MFCP group, and exhibited good independent predictive properties with the sensitivities of 61.8, 61.8, 70.6, and 73.5%, specificities of 66.2, 68.4, 59.6, and 55.9%, and AUCs of 0.620, 0.659, 0.670, and 0.668, respectively, hovering around 0.7. The tenfold cross-validation confirmed the reliability and robustness of our findings, with consistent AUC, sensitivity, specificity, and 95% confidence intervals over 1000 iterations. CONCLUSION T1 SIR, AP SIR, VP SIR, and DP SIR show promise as potential imaging biomarkers for distinguishing between MFCP and PDAC.
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Affiliation(s)
- Yuxiao Li
- Department of Radiology, Changhai Hospital Affiliated to Navy Medical University, 168 Changhai Road, Shanghai, People's Republic of China
| | - Chenxi Zheng
- Department of Hepatobiliary Pancreatic Surgery, Changhai Hospital Affiliated to Navy Medical University, 168 Changhai Road, Shanghai, People's Republic of China
| | - Yang Zhang
- Department of Oncology Radiation, Shuguang Hospital affiliated to Shanghai University of Traditional Chinese Medicine, 528 Zhangheng Road, Shanghai, People's Republic of China
| | - Tianlin He
- Department of Hepatobiliary Pancreatic Surgery, Changhai Hospital Affiliated to Navy Medical University, 168 Changhai Road, Shanghai, People's Republic of China
| | - Wei Chen
- Department of Radiology, Changhai Hospital Affiliated to Navy Medical University, 168 Changhai Road, Shanghai, People's Republic of China
| | - Kailian Zheng
- Department of Hepatobiliary Pancreatic Surgery, Changhai Hospital Affiliated to Navy Medical University, 168 Changhai Road, Shanghai, People's Republic of China.
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Luo P, Guo Y, He Y, Wang C. Clinical characteristics, treatment and outcome of pembrolizumab-induced acute pancreatitis. Invest New Drugs 2024; 42:369-375. [PMID: 38829427 DOI: 10.1007/s10637-024-01452-0] [Citation(s) in RCA: 8] [Impact Index Per Article: 8.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/08/2024] [Accepted: 05/30/2024] [Indexed: 06/05/2024]
Abstract
Acute pancreatitis (AP) is a rare adverse event of pembrolizumab with unclear clinical features. This study investigated the clinical features of pembrolizumab-induced AP to provide a reference for prevention and treatment. Case reports, case series and clinical studies of pembrolizumab-induced AP were collected by searching Chinese and English databases up to January 31, 2024. Thirty-one patients were included, with a median age of 59 years (range 39, 82). The median time from administration to onset of AP was 5.05 months (range 0.5, 16) and the median cycle was 7 cycles (range 1, 35). Twenty-two (71.0%) patients had elevated pancreatic amylase with a median value of 860 IU/L (range 105-12562), and 16 (51.6%) patients had elevated lipase with a median value of 282 IU/L (range 153-1034). Pancreatic biopsy showed neutrophil infiltration (9.7%) and lymphocyte infiltration (6.5%). Immunohistochemical staining showed CD8 dominated inflammatory infiltration (6.5%). The computed tomography showed diffuse enlargement (51.6%) and focal enlargement (51.6%) of the pancreas. Endoscopic ultrasound showed enlarged hypoechoic pancreas(16.1%). PET/CT showed increased FDG uptake (16.1%). The magnetic resonance cholangial pancreatography showed narrowing of main pancreatic duct (12.9%). AP symptoms and pancreatic enzymes improved after discontinuation of pembrolizumab and administration of steroids and infliximab. Clinicians should be aware that AP is a rare adverse reaction to pembrolizumab. Pembrolizumab induced AP can be initiated with steroids for control, and infliximab can be initiated with steroid-refractory AP.
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Affiliation(s)
- Panpan Luo
- Department of Spine Surgery, The First Hospital of Hunan University of Chinese Medicine, Changsha, 410007, Hunan, China
- College of pharmacy, Changsha Medical University, Changsha, 410219, Hunan, China
| | - Yuge Guo
- Department of Spine Surgery, The First Hospital of Hunan University of Chinese Medicine, Changsha, 410007, Hunan, China
| | - Yang He
- Department of pharmacy, The First Hospital of Hunan University of Chinese Medicine, No.95 Shaoshan Middle Road, Changsha, 410007, Hunan, China.
| | - Chunjiang Wang
- Department of pharmacy, The Third Xiangya Hospital, Central South University, No. 138 Tongzipo Road, YueLu District, Changsha, 410013, Hunan, China.
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de Pretis N, Martinelli L, Palmeri E, Caldart F, Crucillà S, Zorzi A, Brillo A, Crinò SF, Conti Bellocchi MC, Bernardoni L, De Marchi G, Amodio A, Campagnola P, Ciccocioppo R, Gabbrielli A, Marcon A, Frulloni L. The effect of steroid therapy on pancreatic exocrine function in autoimmune pancreatitis. Pancreatology 2024; 24:538-544. [PMID: 38693038 DOI: 10.1016/j.pan.2024.04.006] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/21/2023] [Revised: 04/19/2024] [Accepted: 04/23/2024] [Indexed: 05/03/2024]
Abstract
BACKGROUND/OBJECTIVES Autoimmune pancreatitis (AIP) is a steroid-responsive inflammatory disease of the pancreas. Few studies investigated pancreatic exocrine function (PEF) in patients suffering from AIP and no definitive data are available on the effect of steroids in PEF recovery. Aim of the study is the evaluation of severe pancreatic insufficiency (sPEI) prevalence in AIP at clinical onset and after steroid treatment. METHODS 312 Patients with diagnosis of AIP between January 1st, 2010 and December 31st, 2020 were identified in our prospectively maintained register. Patients with a pre-steroid treatment dosage of fecal elastase-1 (FE-1) were included. Changes in PEF were evaluated in patients with available pre- and post-treatment FE (between 3 and 12 months after steroid). RESULTS One-hundred-twenty-four patients were included, with a median FE-1 of 122 (Q1-Q3: 15-379) μg/g at baseline. Fifty-nine (47.6 %) had sPEI (FE-1<100 μg/g). Univariable analysis identified type 1 AIP, radiological involvement of the head of the pancreas (diffuse involvement of the pancreas or focal involvement of the head), weight loss, age and diabetes as associated with a greater risk of sPEI. However, at multivariable analysis, only the involvement of the head of the pancreas was identified as independent risk factor for sPEI. After steroids, mean FE-1 changed from 64 (15-340) to 202 (40-387) μg/g (P = 0.058) and head involvement was the only predictor of improvement of sPEI. CONCLUSION The inflammatory involvement of the head of the pancreas is associated with PEF severity, as well as PEF improvement after treatment with steroids in patients with AIP.
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Affiliation(s)
- Nicolò de Pretis
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy.
| | - Luigi Martinelli
- Department of Diagnostics and Public Health, University of Verona, Verona, Italy
| | - Enrico Palmeri
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Federico Caldart
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Salvatore Crucillà
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Alberto Zorzi
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Alessandro Brillo
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | | | | | - Laura Bernardoni
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Giulia De Marchi
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Antonio Amodio
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Pietro Campagnola
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Rachele Ciccocioppo
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Armando Gabbrielli
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
| | - Alessandro Marcon
- Department of Diagnostics and Public Health, University of Verona, Verona, Italy
| | - Luca Frulloni
- Gastroenterology Unit, Department of Medicine, University of Verona, Verona, Italy
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7
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Overbeek KA, Poulsen JL, Lanzillotta M, Vinge-Holmquist O, Macinga P, Demirci AF, Sindhunata DP, Backhus J, Algül H, Buijs J, Levy P, Kiriukova M, Goni E, Hollenbach M, Miksch RC, Kunovsky L, Vujasinovic M, Nikolic S, Dickerson L, Hirth M, Neurath MF, Zumblick M, Vila J, Jalal M, Beyer G, Frost F, Carrara S, Kala Z, Jabandziev P, Sisman G, Akyuz F, Capurso G, Falconi M, Arlt A, Vleggaar FP, Barresi L, Greenhalf B, Czakó L, Hegyi P, Hopper A, Nayar MK, Gress TM, Vitali F, Schneider A, Halloran CM, Trna J, Okhlobystin AV, Dagna L, Cahen DL, Bordin D, Rebours V, Mayerle J, Kahraman A, Rasch S, Culver E, Kleger A, Martínez-Moneo E, Røkke O, Hucl T, Olesen SS, Bruno MJ, Della-Torre E, Beuers U, Löhr JM, Rosendahl J. Type 1 Autoimmune Pancreatitis in Europe: Clinical Profile and Response to Treatment. Clin Gastroenterol Hepatol 2024; 22:994-1004.e10. [PMID: 38184096 DOI: 10.1016/j.cgh.2023.12.010] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/29/2023] [Revised: 12/04/2023] [Accepted: 12/05/2023] [Indexed: 01/08/2024]
Abstract
BACKGROUND & AIMS Autoimmune pancreatitis (AIP) is an immune-mediated disease of the pancreas with distinct pathophysiology and manifestations. Our aims were to characterize type 1 AIP in a large pan-European cohort and study the effectiveness of current treatment regimens. METHODS We retrospectively analyzed adults diagnosed since 2005 with type 1 or not-otherwise-specified AIP in 42 European university hospitals. Type 1 AIP was uniformly diagnosed using specific diagnostic criteria. Patients with type 2 AIP and those who had undergone pancreatic surgery were excluded. The primary end point was complete remission, defined as the absence of clinical symptoms and resolution of the index radiologic pancreatic abnormalities attributed to AIP. RESULTS We included 735 individuals with AIP (69% male; median age, 57 years; 85% White). Steroid treatment was started in 634 patients, of whom 9 (1%) were lost to follow-up. The remaining 625 had a 79% (496/625) complete, 18% (111/625) partial, and 97% (607/625) cumulative remission rate, whereas 3% (18/625) did not achieve remission. No treatment was given in 95 patients, who had a 61% complete (58/95), 19% partial (18/95), and 80% cumulative (76/95) spontaneous remission rate. Higher (≥0.4 mg/kg/day) corticosteroid doses were no more effective than lower (<0.4 mg/kg/day) doses (odds ratio, 0.428; 95% confidence interval, 0.054-3.387) and neither was a starting dose duration >2 weeks (odds ratio, 0.908; 95% confidence interval, 0.818-1.009). Elevated IgG4 levels were independently associated with a decreased chance of complete remission (odds ratio, 0.639; 95% confidence interval, 0.427-0.955). Relapse occurred in 30% of patients. Relapses within 6 months of remission induction were independent of the steroid-tapering duration, induction treatment duration, and total cumulative dose. CONCLUSIONS Patients with type 1 AIP and elevated IgG4 level may need closer monitoring. For remission induction, a starting dose of 0.4 mg/kg/day for 2 weeks followed by a short taper period seems effective. This study provides no evidence to support more aggressive regimens.
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Affiliation(s)
- Kasper A Overbeek
- Department of Gastroenterology & Hepatology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
| | - Jakob L Poulsen
- Centre for Pancreatic Diseases, Department of Gastroenterology & Hepatology, Aalborg University Hospital, Aalborg, Denmark
| | - Marco Lanzillotta
- Unit of Immunology, Rheumatology, Allergy and Rare Diseases (UnIRAR), San Raffaele Scientific Institute, Milan, Italy
| | - Olof Vinge-Holmquist
- Department of Digestive Surgery, Akershus University Hospital, Loerenskog, Norway; Department of Digestive Surgery, St. Olavs Hospital, Trondheim University Hospital, Trondheim, Norway
| | - Peter Macinga
- Department of Gastroenterology and Hepatology, Institute for Clinical and Experimental Medicine, Prague, Czech Republic
| | - A Fatih Demirci
- Department of Internal Medicine, Marmara University Research and Education Hospital, Istanbul, Turkey
| | - Daniko P Sindhunata
- Department of Gastroenterology & Hepatology, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands
| | - Johanna Backhus
- Department of Internal Medicine I, University Hospital Ulm, Ulm, Germany
| | - Hana Algül
- Department of Medicine II, Technische Universität München, München, Germany
| | - Jorie Buijs
- Department of Gastroenterology & Hepatology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Philippe Levy
- Pancreatology Unit, APHP Beaujon Hospital, Clichy, France
| | - Mariia Kiriukova
- Department of Upper Gastrointestinal, Pancreatic, and Biliary Diseases, A.S. Loginov Moscow Clinical Research Center, Moscow, Russia
| | - Elisabetta Goni
- Department of Medicine II, University Hospital, Ludwig Maximilian University Munich, Munich, Germany
| | - Marcus Hollenbach
- Division of Gastroenterology, Medical Department II - Oncology, Gastroenterology, Hepatology, Pulmonology, Infectious Diseases, University of Leipzig Medical Center, Leipzig, Germany
| | - Rainer C Miksch
- Department of General, Visceral, and Transplantation Surgery, University Hospital, Ludwig Maximilian University Munich, Munich, Germany
| | - Lumir Kunovsky
- 2nd Department of Internal Medicine, Gastroenterology and Geriatrics, University Hospital Olomouc, Faculty of Medicine and Dentistry, Palacky University, Olomouc, Czech Republic; Department of Surgery, University Hospital Brno, Faculty of Medicine, Masaryk University, Brno, Czech Republic; Department of Gastroenterology and Digestive Endoscopy, Masaryk Memorial Cancer Institute, Brno, Czech Republic
| | - Miroslav Vujasinovic
- Department of Upper Abdominal Diseases, Karolinska University Hospital, Stockholm, Sweden
| | - Sara Nikolic
- Department of Upper Abdominal Diseases, Karolinska University Hospital, Stockholm, Sweden
| | - Luke Dickerson
- Department of Molecular and Clinical Cancer Medicine, Institute of Translational Medicine, University of Liverpool, Liverpool, United Kingdom
| | - Michael Hirth
- Department of Medicine II, University Medical Center Mannheim, Medical Faculty at Mannheim, University of Heidelberg, Mannheim, Germany
| | - Markus F Neurath
- Department of Medicine I, Deutsches Zentrum Immuntherapie (DZI), Kussmaul Campus for Medical Research, University Erlangen-Nürnberg, Erlangen, Germany
| | - Malte Zumblick
- Department of Gastroenterology and Endocrinology, Philipps-University Marburg, Marburg, Germany
| | - Josephine Vila
- HPB Unit, Freeman Hospital, Newcastle Upon Tyne, United Kingdom
| | - Mustafa Jalal
- Department of Infection, Immunity and Cardiovascular Disease, University of Sheffield, Sheffield, United Kingdom
| | - Georg Beyer
- Department of Medicine II, University Hospital, Ludwig Maximilian University Munich, Munich, Germany
| | - Fabian Frost
- Department of Medicine A, University Medicine Greifswald, Greifswald, Germany
| | - Silvia Carrara
- Gastrointestinal Endoscopy Unit, Humanitas Mater Domini, Castellanza, Italy
| | - Zdenek Kala
- Department of Surgery, University Hospital Brno, Faculty of Medicine, Masaryk University, Brno, Czech Republic
| | - Petr Jabandziev
- Department of Pediatrics, University Hospital Brno, Faculty of Medicine, Masaryk University, Brno, Czech Republic; Central European Institute of Technology, Masaryk University, Brno, Czech Republic
| | - Gurhan Sisman
- Acibadem Mehmet Ali Aydinlar University School of Medicine, Istanbul, Turkey
| | - Filiz Akyuz
- Department of Gastroenterology, Istanbul University, Istanbul Faculty of Medicine, Istanbul, Turkey
| | - Gabriele Capurso
- Pancreato-Biliary Endoscopy & Endosonography Division, Pancreas Translational & Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milan, Italy
| | - Massimo Falconi
- Division of Pancreatic Surgery, Pancreas Translational & Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milan, Italy
| | - Alexander Arlt
- Department of Internal Medicine I, University Hospital Schleswig-Holstein, Kiel, Germany; Department for Internal Medicine and Gastroenterology, University Hospital, Klinikum Oldenburg AöR, Oldenburg, Germany
| | - Frank P Vleggaar
- Department of Gastroenterology and Hepatology, University Medical Center Utrecht, Utrecht, The Netherlands
| | - Luca Barresi
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, Mediterranean Institute for Transplantation and Advanced Specialized Therapies (IRCSS-ISMETT), Palermo, Italy
| | - Bill Greenhalf
- Department of Molecular and Clinical Cancer Medicine, Institute of Translational Medicine, University of Liverpool, Liverpool, United Kingdom
| | - László Czakó
- Institute for Translational Medicine, Szentágothai Research Centre, Medical School, University of Pécs, Pécs, Hungary
| | - Peter Hegyi
- Institute for Translational Medicine, Szentágothai Research Centre, Medical School, University of Pécs, Pécs, Hungary; Division of Pancreatic Diseases, Heart and Vascular Centre, Semmelweis University, Budapest, Hungary
| | - Andrew Hopper
- Department of Infection, Immunity and Cardiovascular Disease, University of Sheffield, Sheffield, United Kingdom
| | - Manu K Nayar
- HPB Unit, Freeman Hospital, Newcastle Upon Tyne, United Kingdom
| | - Thomas M Gress
- Department of Gastroenterology and Endocrinology, Philipps-University Marburg, Marburg, Germany
| | - Francesco Vitali
- Department of Medicine I, Deutsches Zentrum Immuntherapie (DZI), Kussmaul Campus for Medical Research, University Erlangen-Nürnberg, Erlangen, Germany
| | - Alexander Schneider
- Department of Medicine II, University Medical Center Mannheim, Medical Faculty at Mannheim, University of Heidelberg, Mannheim, Germany
| | - Chris M Halloran
- Department of Molecular and Clinical Cancer Medicine, Institute of Translational Medicine, University of Liverpool, Liverpool, United Kingdom
| | - Jan Trna
- Department of Gastroenterology and Digestive Endoscopy, Masaryk Memorial Cancer Center Institute, Faculty of Medicine, Masaryk University, Brno, Czech Republic
| | | | - Lorenzo Dagna
- Unit of Immunology, Rheumatology, Allergy and Rare Diseases (UnIRAR), San Raffaele Scientific Institute, Milan, Italy
| | - Djuna L Cahen
- Department of Gastroenterology & Hepatology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Dmitry Bordin
- Department of Upper Gastrointestinal, Pancreatic, and Biliary Diseases, A.S. Loginov Moscow Clinical Research Center, Moscow, Russia; Department of Outpatient Therapy and Family Medicine, Tver State Medical University, Tver, Russia
| | | | - Julia Mayerle
- Department of Medicine II, University Hospital, Ludwig Maximilian University Munich, Munich, Germany
| | - Alisan Kahraman
- Division of Gastroenterology and Hepatology, Department of Internal Medicine, Essen University Hospital, University of Duisberg-Essen, Essen, Germany
| | - Sebastian Rasch
- Department of Medicine II, Technische Universität München, München, Germany
| | - Emma Culver
- Translational Gastroenterology Unit, John Radcliffe Hospital and Nuffield Department of Medicine, University of Oxford, Oxford, United Kingdom
| | - Alexander Kleger
- Department of Internal Medicine I, University Hospital Ulm, Ulm, Germany
| | - Emma Martínez-Moneo
- Biocruces, Grupo Transplante Hepático, Osakidetza, Hospital Universitario Cruces, Servicio Aparato Digestivo, Barakaldo, Spain
| | - Ola Røkke
- Department of Digestive Surgery, Akershus University Hospital, Loerenskog, Norway; Faculty of Medicine, Campus Ahus, University of Oslo, Oslo, Norway
| | - Tomas Hucl
- Department of Gastroenterology and Hepatology, Institute for Clinical and Experimental Medicine, Prague, Czech Republic
| | - Søren S Olesen
- Centre for Pancreatic Diseases, Department of Gastroenterology & Hepatology, Aalborg University Hospital, Aalborg, Denmark
| | - Marco J Bruno
- Department of Gastroenterology & Hepatology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Emanuel Della-Torre
- Unit of Immunology, Rheumatology, Allergy and Rare Diseases (UnIRAR), San Raffaele Scientific Institute, Milan, Italy
| | - Ulrich Beuers
- Department of Gastroenterology & Hepatology, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands
| | - J-Matthias Löhr
- 2nd Department of Internal Medicine, Gastroenterology and Geriatrics, University Hospital Olomouc, Faculty of Medicine and Dentistry, Palacky University, Olomouc, Czech Republic; Department of Surgery, University Hospital Brno, Faculty of Medicine, Masaryk University, Brno, Czech Republic; Department of Gastroenterology and Digestive Endoscopy, Masaryk Memorial Cancer Institute, Brno, Czech Republic
| | - Jonas Rosendahl
- Department of Internal Medicine I, Martin Luther University, Halle (Saale), Germany.
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8
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Thomas AS, Abreo M, Ahmed AS, Rao Manikonda SP, Eyada M, Issac A, Abraham F, Jacob JS, Wang Y, Yedururi S, Chari ST. Immune Checkpoint Inhibitor-Induced Pancreatic Injury: Clinical and Radiological Profile and Response to Steroids. GASTRO HEP ADVANCES 2023; 3:361-367. [PMID: 39131139 PMCID: PMC11308033 DOI: 10.1016/j.gastha.2023.11.020] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 08/23/2023] [Accepted: 11/30/2023] [Indexed: 08/13/2024]
Abstract
Background and Aims Immune checkpoint inhibitor therapy causes numerous immune-related adverse events, including autoimmune pancreatic injury (AIPI), which results in rapid organ atrophy. We profiled the clinico-radiological features, short-term natural history, and response to steroids of AIPI. Methods We retrospectively reviewed medical records of 229/11,165 (2.1%) adult patients with AIPI. One hundred and ten out of 229 (48%) had abdominal computerized tomography (CT) scan at lipase elevation; data of 110 without pancreatic metastases were analyzed. We analyzed serial CT-based pancreas volumetry data in 48 patients with AIPI (32 with normal CT and 16 with pancreatitis on CT at lipase elevation). We examined impact of steroids on pain and disease course. Results In AIPI (n = 229), median lipase elevation was 4x upper limit of normal (range: 3-40x). The injury was more often asymptomatic than painful (143/229 (62%) vs 86/229 (38%), P < .000). Majority (83/110 (75%) had normal CT, often in painless vs painful disease: 51/57 (90%) vs 32/53 (60%), P < .001) 25% had interstitial pancreatitis. On serial pancreas volumetry, marked volume (cc) loss occurred 1 year after vs 3 months before lipase elevation in both normal CT (median 81.6 vs 61.3, P = .00) and pancreatitis on CT groups (91.8 vs 60.5, P = .00), ≥20% volume loss occurred in 47% vs 73%, respectively (P = .08). Steroids, when used did not mitigate pain, biochemical relapse, pancreas volume loss or 1-year diabetes incidence (7.2%). Conclusion Autoimmune pancreatic injury (AIPI) is uniquely characterized by painless lipase elevation, normal pancreas on CT and rapid pancreatic volume loss on follow-up. Steroids do not appear to have a role in management.
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Affiliation(s)
- Anusha Shirwaikar Thomas
- Department of Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas
| | - Michael Abreo
- Department of Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas
| | - Ahmed Sayed Ahmed
- Department of Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas
| | | | - Mostafa Eyada
- Division of Internal Medicine, University of Texas Medical Branch at Galveston, Galveston, Texas
| | - Aaron Issac
- Division of Internal Medicine, Emory University School of Medicine, Atlanta, Georgia
| | - Fiyinfoluwa Abraham
- Division of Internal Medicine, Emory University School of Medicine, Atlanta, Georgia
| | - Jake Sheraj Jacob
- Department of Internal Medicine, Baylor College of Medicine, Houston, Texas
| | - Yinghong Wang
- Department of Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas
| | - Sireesha Yedururi
- Department of Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas
| | - Suresh T. Chari
- Department of Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas
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9
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Li Y, Song H, Meng X, Li R, Leung PSC, Gershwin ME, Zhang S, Sun S, Song J. Autoimmune pancreatitis type 2 (idiopathic duct-centric pancreatitis): A comprehensive review. J Autoimmun 2023; 140:103121. [PMID: 37826920 DOI: 10.1016/j.jaut.2023.103121] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/26/2023] [Revised: 09/20/2023] [Accepted: 10/02/2023] [Indexed: 10/14/2023]
Abstract
Autoimmune pancreatitis (AIP) is an uncommon fibro-inflammatory disorder precipitated by autoimmune/inflammatory reactions. Currently, there are two clinical subtypes of AIP (type 1 [AIP-1] and type 2 [AIP-2]) that correspond to two histologic descriptors (lymphoplasmacytic sclerosing pancreatitis and idiopathic duct-centric pancreatitis, respectively). While our understanding of AIP-1 has evolved considerably over the years, little is known about AIP-2 due to its rarity, often leading to misdiagnosis, delayed treatment, and even unnecessary surgical resection. Compared to AIP-1, AIP-2 exhibits distinct clinical and histologic features. Because AIP-2 is a pancreas-restricted disease without a specific serum marker, the evaluation of histologic features (e.g., granulocytic epithelial lesions) is essential for an accurate diagnosis. Patients with AIP-2 respond well to glucocorticoids, with anti-tumor necrosis factor-alpha antibodies as a promising alternative therapy. The prognosis of AIP-2 is generally favorable and relapse is uncommon. Here, we provide an overview of our current knowledge on the clinical features, diagnosis, therapeutic regimens, prognosis, and putative mechanisms underlying AIP-2. Notably, the diagnostic differentiation between AIP-2, especially the mass-forming/focal type, and pancreatic cancer is important, but challenging. In this regard, endoscopic ultrasound-guided core biopsy has a key role, but novel diagnostic markers and modalities are clearly needed.
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Affiliation(s)
- Yang Li
- Department of Critical Care Medicine, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China
| | - Hanyi Song
- Department of Gastroenterology, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China
| | - Xiangzhen Meng
- Department of General Surgery, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China
| | - Runzhuo Li
- Department of Gastroenterology, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China
| | - Patrick S C Leung
- Division of Rheumatology/Allergy and Clinical Immunology, School of Medicine, University of California, Davis, CA 95616 USA
| | - M Eric Gershwin
- Division of Rheumatology/Allergy and Clinical Immunology, School of Medicine, University of California, Davis, CA 95616 USA
| | - Shucheng Zhang
- Department of Pediatrics, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China.
| | - Siyu Sun
- Department of Gastroenterology, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China.
| | - Junmin Song
- Department of Gastroenterology, Shengjing Hospital of China Medical University, Shenyang 110004, Liaoning Province, PR China.
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10
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Vitali F, Zundler S, Jesper D, Strobel D, Wildner D, de Pretis N, Frulloni L, Crinó SF, Neurath MF. Endoscopic Ultrasound in Pancreatology: Focus on Inflammatory Diseases and Interventions. Visc Med 2023; 39:131-139. [PMID: 37899796 PMCID: PMC10601533 DOI: 10.1159/000533433] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/05/2023] [Accepted: 08/03/2023] [Indexed: 10/31/2023] Open
Abstract
Background Endoscopic ultrasound (EUS) is a main tool in pancreatology for both diagnosis and therapy. It allows minimally invasive differentiation of various diseases, with a minimal degree of inflammation or anatomic variations. EUS also enables interventional direct access to the pancreatic parenchyma and the retroperitoneal space, the pancreatic duct, the pancreatic masses, cysts, vascular structures for diagnostic and therapeutic purposes. Summary This review aimed to summarize the new developments of EUS in the field of pancreatology, with special interest on inflammation and interventions. EUS enables way to perform pseudocyst drainage, necrosectomy, transenteral drainage and transenteric access of the main pancreatic duct, or the direct visualization or therapy of vascular structures adjacent to the pancreas. Key Messages EUS has a deep impact on pancreatology, and the development of new diagnostic and interventional approaches to the retroperitoneal space and the pancreas has increased in the last years exponentially, allowing minimal invasive diagnostics and therapy and avoiding surgery and percutaneous therapy.
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Affiliation(s)
- Francesco Vitali
- Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nuremberg, Erlangen, Germany
| | - Sebastian Zundler
- Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nuremberg, Erlangen, Germany
| | - Daniel Jesper
- Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nuremberg, Erlangen, Germany
| | - Deike Strobel
- Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nuremberg, Erlangen, Germany
| | - Dane Wildner
- Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nuremberg, Erlangen, Germany
| | - Nicoló de Pretis
- Gastroenterology and Digestive Endoscopy Unit, The Pancreas Institute, G.B. Rossi University Hospital, Verona, Italy
| | - Luca Frulloni
- Gastroenterology and Digestive Endoscopy Unit, The Pancreas Institute, G.B. Rossi University Hospital, Verona, Italy
| | - Stefano Francesco Crinó
- Gastroenterology and Digestive Endoscopy Unit, The Pancreas Institute, G.B. Rossi University Hospital, Verona, Italy
| | - Markus F. Neurath
- Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nuremberg, Erlangen, Germany
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11
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Suenaga A, Sawa N, Ikuma D, Oba Y, Sekine A, Yamanouchi M, Hasegawa E, Mizuno H, Suwabe T, Kono K, Shintani-Domoto Y, Kinowaki K, Ohashi K, Suzuki Y, Miyazono M, Takemura T, Yamaguchi Y, Ubara Y. Immunoglobulin G4-related Tubulointerstitial Nephritis with Simultaneous Resolution of Plasma Cell Infiltration and Fibrosis after Steroid Treatment. Intern Med 2023; 62:2699-2706. [PMID: 36725037 PMCID: PMC10569928 DOI: 10.2169/internalmedicine.1121-22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/13/2022] [Accepted: 12/11/2022] [Indexed: 02/03/2023] Open
Abstract
We performed 3 kidney biopsies in a 71-year-old man. At the first biopsy, we made the diagnosis of immunoglobulin G4 (IgG4)-related interstitial nephritis characterized by the simultaneous presence of IgG4-positive plasma cells and characteristic fibrosis with a bird's-eye pattern. At the second biopsy, rather than finding fibrosis as a post-inflammatory scar, we noted that steroid treatment had caused the simultaneous disappearance of IgG4-positive plasma cells and fibrosis and had restored the normal tubular structure. The third biopsy showed the recurrence of the disease with inflammatory cells accompanied by fibrosis. These findings suggest that IgG4-positive plasma cells and fibrosis occur simultaneously.
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Affiliation(s)
- Atsuhiko Suenaga
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
- Department of Nephrology, Saga University School of Medicine, Japan
| | - Naoki Sawa
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
- Okinaka Memorial Institute for Medical Research, Toranomon Hospital, Japan
| | - Daisuke Ikuma
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
| | - Yuki Oba
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
| | - Akinari Sekine
- Department of Nephrology and Rheumatology, Toranomon Hospital, Japan
| | - Masayuki Yamanouchi
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
| | - Eiko Hasegawa
- Department of Nephrology, Saga University School of Medicine, Japan
- Okinaka Memorial Institute for Medical Research, Toranomon Hospital, Japan
| | - Hiroki Mizuno
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
| | - Tatsuya Suwabe
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
| | - Kei Kono
- Department of Pathology, Toranomon Hospital, Japan
| | | | | | - Kenichi Ohashi
- Department of Pathology, Toranomon Hospital, Japan
- Department of Human Pathology, Tokyo Medical and Dental University, Japan
| | | | - Motoaki Miyazono
- Department of Nephrology, Saga University School of Medicine, Japan
| | - Tamiko Takemura
- Department of Pathology, Kanagawa Cardiovascular and Respiratory Center, Japan
| | | | - Yoshifumi Ubara
- Department of Nephrology and Rheumatology, Toranomon Hospital Kajigaya, Japan
- Okinaka Memorial Institute for Medical Research, Toranomon Hospital, Japan
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12
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Hogeboom A, Sánchez ER, Ibarrola C, Pérez-Campos A. Pancreatic head mass: To Whipple or not to Whipple. Cytojournal 2023; 20:28. [PMID: 37681077 PMCID: PMC10481877 DOI: 10.25259/cytojournal_12_2023] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/06/2023] [Accepted: 04/29/2023] [Indexed: 09/09/2023] Open
Affiliation(s)
- Adriana Hogeboom
- Department of Pathology, 12 de Octubre University Hospital, Madrid, Spain
| | | | - Carolina Ibarrola
- Department of Pathology, 12 de Octubre University Hospital, Madrid, Spain
| | - Ana Pérez-Campos
- Department of Pathology, 12 de Octubre University Hospital, Madrid, Spain
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13
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Caba O, Diéguez-Castillo C, Martínez-Galán J, González-Cebrián I, Jiménez-Luna C. Serum biomarkers for the differentiation of autoimmune pancreatitis from pancreatic ductal adenocarcinoma. World J Gastrointest Oncol 2023; 15:268-275. [PMID: 36908319 PMCID: PMC9994052 DOI: 10.4251/wjgo.v15.i2.268] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/20/2022] [Revised: 11/29/2022] [Accepted: 01/17/2023] [Indexed: 02/14/2023] Open
Abstract
Autoimmune pancreatitis (AIP), a chronic inflammation caused by the immune system attacking the pancreas, usually presents imaging and clinical features that overlap with those of pancreatic ductal adenocarcinoma (PDAC). Serum biomarkers, substances that quantitatively change in sera during disease development, are a promising non-invasive tool with high utility for differentiating between these diseases. In this way, the presence of AIP is currently suspected when serum concentrations of immunoglobulin G4 (IgG4) antibody are elevated. However, this approach has some drawbacks. Notably, IgG4 antibody concentrations are also elevated in sera from some patients with PDAC. This review focuses on the most recent and relevant serum biomarkers proposed to differentiate between AIP and PDAC, evaluating the usefulness of immunoglobulins, autoantibodies, chemokines, and cytokines. The proposed serum biomarkers have proven useful, although most studies had a small sample size, did not examine their presence in patients with PDAC, or did not test them in humans. In addition, current evidence suggests that a single serum biomarker is unlikely to accurately differentiate these diseases and that a set of biomarkers will be needed to achieve adequate specificity and sensitivity, either alone or in combination with clinical data and/or radiological images.
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Affiliation(s)
- Octavio Caba
- Institute of Biopathology and Regenerative Medicine (IBIMER), University of Granada, Granada 18016, Spain
- Biosanitary Institute of Granada (ibs.GRANADA), 18014 Granada, Spain
| | | | - Joaquina Martínez-Galán
- Department of Medical Oncology, Virgen de las Nieves University Hospital, Granada 18014, Spain. Biosanitary Institute of Granada (ibs.GRANADA), 18014 Granada, Spain
| | | | - Cristina Jiménez-Luna
- Institute of Biopathology and Regenerative Medicine (IBIMER), University of Granada, Granada 18016, Spain
- Biosanitary Institute of Granada (ibs.GRANADA), 18014 Granada, Spain
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14
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de Pretis N, Amodio A, De Marchi G, Marconato E, Ciccocioppo R, Frulloni L. The role of serological biomarkers in the diagnosis and management of autoimmune pancreatitis. Expert Rev Clin Immunol 2022; 18:1119-1124. [PMID: 36125384 DOI: 10.1080/1744666x.2022.2125379] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022]
Abstract
INTRODUCTION Autoimmune pancreatitis (AIP) is a fibroinflammatory disease of the pancreas. Type 1 AIP is the pancreatic manifestation of a systemic IgG4-related disease and is associated with serum elevation of IgG4, tissue infiltration of IgG4-positive plasma cells, and multiorgan involvement. Although serum IgG4 elevation is considered a useful diagnostic tool, the concomitant presence of more diagnostic criteria is needed to achieve diagnosis. No other biomarkers have been approved in clinical practice in type 1 AIP. Type 2 AIP is a pancreatic-specific disease associated with inflammatory bowel disease. No specific biomarkers for type 2 AIP have been identified. AREAS COVERED The role of serum IgG4 in the diagnosis, management and follow-up of patients with type 1 AIP. Moreover, data on other emerging biomarkers for type 1 and 2 AIP have been reported. EXPERT OPINION The diagnosis of AIP is challenging in clinical practice, especially for focal forms without multiorgan involvement, where distinction from pancreatic cancer can be difficult. Despite the strong association with type 1 AIP, serum IgG4 should only be measured when the suspicion for the disease is high, considering its limited sensitivity. New biomarkers with high diagnostic yield for both type 1 and type 2 AIP are needed.
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Affiliation(s)
- Nicolò de Pretis
- Department of Medicine, Pancreas Center, University of Verona, Verona, Italy
| | - Antonio Amodio
- Department of Medicine, Pancreas Center, University of Verona, Verona, Italy
| | - Giulia De Marchi
- Department of Medicine, Pancreas Center, University of Verona, Verona, Italy
| | - Eugenio Marconato
- Department of Medicine, Pancreas Center, University of Verona, Verona, Italy
| | - Rachele Ciccocioppo
- Department of Medicine, Pancreas Center, University of Verona, Verona, Italy
| | - Luca Frulloni
- Department of Medicine, Pancreas Center, University of Verona, Verona, Italy
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15
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Chen MX, Su HH, Shiao CY, Chang YT, Chang MC, Kao CC, Wang SY, Shih HC, Tsai IL. Affinity Purification Coupled to Stable Isotope Dilution LC-MS/MS Analysis to Discover IgG4 Glycosylation Profiles for Autoimmune Pancreatitis. Int J Mol Sci 2021; 22:ijms222111527. [PMID: 34768956 PMCID: PMC8584136 DOI: 10.3390/ijms222111527] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/28/2021] [Revised: 10/20/2021] [Accepted: 10/22/2021] [Indexed: 01/13/2023] Open
Abstract
Type 1 autoimmune pancreatitis (AIP) is categorized as an IgG4-related disease (IgG4-RD), where a high concentration of plasma IgG4 is one of the common biomarkers among patients. IgG Fc-glycosylation has been reported to be potential biosignatures for diseases. However, human IgG3 and IgG4 Fc-glycopeptides from populations in Asia were found to be isobaric ions when using LC-MS/MS as an analytical tool. In this study, an analytical workflow that coupled affinity purification and stable isotope dilution LC-MS/MS was developed to dissect IgG4 glycosylation profiles for autoimmune pancreatitis. Comparing the IgG4 and glycosylation profiles among healthy controls, patients with pancreatic ductal adenocarcinoma (PDAC), and AIP, the IgG4 glycosylations from the AIP group were found to have more digalactosylation (compared to PDAC) and less monogalactosylation (compared to HC). In addition, higher fucosylation and sialylation profiles were also discovered for the AIP group. The workflow is efficient and selective for IgG4 glycopeptides, and can be used for clinical biosignature discovery.
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Affiliation(s)
- Michael X. Chen
- Department of Pathology and Laboratory Medicine, The University of British Columbia, Victoria, BC V8Z6R5, Canada;
- Division of Medical Sciences, University of Victoria, Victoria, BC V8W2Y2, Canada
| | - Ho-Hsuan Su
- Department of Biochemistry and Molecular Cell Biology, School of Medicine, College of Medicine, Taipei Medical University, Taipei 11031, Taiwan; (H.-H.S.); (C.-Y.S.)
| | - Ching-Ya Shiao
- Department of Biochemistry and Molecular Cell Biology, School of Medicine, College of Medicine, Taipei Medical University, Taipei 11031, Taiwan; (H.-H.S.); (C.-Y.S.)
| | - Yu-Ting Chang
- Department of Internal Medicine, National Taiwan University Hospital, Taipei 100, Taiwan; (Y.-T.C.); (M.-C.C.)
- Department of Internal Medicine, College of Medicine, National Taiwan University, Taipei 100, Taiwan
| | - Ming-Chu Chang
- Department of Internal Medicine, National Taiwan University Hospital, Taipei 100, Taiwan; (Y.-T.C.); (M.-C.C.)
- Department of Internal Medicine, College of Medicine, National Taiwan University, Taipei 100, Taiwan
| | - Chih-Chin Kao
- Division of Nephrology, Department of Internal Medicine, Taipei Medical University Hospital, Taipei 11031, Taiwan;
- Division of Nephrology, Department of Internal Medicine, School of Medicine, College of Medicine, Taipei Medical University, Taipei 11031, Taiwan
- Taipei Medical University-Research Center of Urology and Kidney (TMU-RCUK), Taipei Medical University, Taipei 11031, Taiwan
| | - San-Yuan Wang
- School of Pharmacy, College of Pharmacy, Taipei Medical University, Taipei 11031, Taiwan;
- Master Program in Clinical Genomics and Proteomics, College of Pharmacy, Taipei Medical University, Taipei 11031, Taiwan
| | - Hsi-Chang Shih
- Genomics Research Center, Academia Sinica, Taipei 115, Taiwan;
| | - I-Lin Tsai
- Department of Biochemistry and Molecular Cell Biology, School of Medicine, College of Medicine, Taipei Medical University, Taipei 11031, Taiwan; (H.-H.S.); (C.-Y.S.)
- School of Pharmacy, College of Pharmacy, Taipei Medical University, Taipei 11031, Taiwan;
- Master Program in Clinical Genomics and Proteomics, College of Pharmacy, Taipei Medical University, Taipei 11031, Taiwan
- Graduate Institute of Medical Sciences, College of Medicine, Taipei Medical University, Taipei 11031, Taiwan
- International Ph.D. Program for Cell Therapy and Regeneration Medicine, College of Medicine, Taipei Medical University, Taipei 11031, Taiwan
- Pulmonary Research Center, Wan Fang Hospital, Taipei Medical University, Taipei 116, Taiwan
- Correspondence:
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16
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Goodchild G, Peters RJ, Cargill TN, Martin H, Fadipe A, Leandro M, Bailey A, Collier J, Firmin L, Chouhan M, Rodriguez-Justo M, Sadler R, Chapman RW, Bungay H, Fryer E, David J, Luqmani R, Barnes E, Webster GJ, Culver EL. Experience from the first UK inter-regional specialist multidisciplinary meeting in the diagnosis and management of IgG4-related disease. Clin Med (Lond) 2021; 20:e32-e39. [PMID: 32414739 DOI: 10.7861/clinmed.2019-0457] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
Immunoglobulin G4-related disease (IgG4-RD) is a complex multisystem fibro-inflammatory disorder, requiring diagnostic differentiation from malignancy and other immune-mediated conditions, and careful management to minimise glucocorticoid-induced toxicity and prevent progressive organ dysfunction. We describe the experience of the first inter-regional specialist IgG4-RD multidisciplinary team meeting (MDM) incorporating a broad range of generalists and specialists, held 6-weekly via web-link between Oxford University Hospitals NHS Foundation Trust and University College London Hospitals NHS Foundation Trust. Over 3 years, there were 206 discussions on 156 patients. Of these, 97 (62%) were considered to have definite or possible IgG4-RD; 67% had multi-organ involvement and 23% had a normal serum IgG4. The average number of specialist opinions sought prior to MDM was four per patient. Management was changed in the majority of patients (74%) with the treatment escalation recommended in 61 cases, including 19 for rituximab. Challenges arose from delays and misdiagnosis, cross-specialty presentation and the management of sub-clinical disease. Our cross-discipline IgG4-RD MDM enabled important diagnostic and management decisions in this complex multisystem disorder, and can be used as a model for other centres in the UK.
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Affiliation(s)
- George Goodchild
- University College London Hospitals NHS Foundation Trust, London, UK
| | - Rory Jr Peters
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK and University of Oxford, Oxford, UK
| | - Tamsin N Cargill
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK and University of Oxford, Oxford, UK
| | - Harry Martin
- University College London Hospitals NHS Foundation Trust, London, UK
| | | | - Maria Leandro
- University College London Hospitals NHS Foundation Trust, London, UK
| | - Adam Bailey
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Jane Collier
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Louisa Firmin
- University College London Hospitals NHS Foundation Trust, London, UK
| | - Manil Chouhan
- University College London Hospitals NHS Foundation Trust, London, UK
| | | | - Ross Sadler
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Roger W Chapman
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK and University of Oxford, Oxford, UK
| | - Helen Bungay
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Eve Fryer
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Joel David
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Raashid Luqmani
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK
| | - Eleanor Barnes
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK and University of Oxford, Oxford, UK
| | - George J Webster
- University College London Hospitals NHS Foundation Trust, London, UK
| | - Emma L Culver
- Oxford University Hospitals NHS Foundation Trust, Oxford, UK and University of Oxford, Oxford, UK
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Review of Diagnostic Biomarkers in Autoimmune Pancreatitis: Where Are We Now? Diagnostics (Basel) 2021; 11:diagnostics11050770. [PMID: 33923064 PMCID: PMC8146865 DOI: 10.3390/diagnostics11050770] [Citation(s) in RCA: 9] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2021] [Revised: 04/22/2021] [Accepted: 04/23/2021] [Indexed: 12/16/2022] Open
Abstract
Autoimmune pancreatitis (AIP) is a pancreatic manifestation of an IgG4-related disease (IgG4-RD). AIP lacks disease-specific biomarkers, and therefore, it is difficult to distinguish AIP from malignancies, especially pancreatic cancer. In this review, we have summarized the latest findings on potential diagnostic biomarkers for AIP. Many investigations have been conducted, but no specific biomarkers for AIP are identified. Therefore, further studies are required to identify accurate diagnostic biomarkers for AIP.
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Shah DS, Prajapati B, Sanghavi K, Kanhere S, Kothari J, Dubal J. Mass Mimicking Autoimmune Pancreatitis—A Report of Two Cases and Review of Literature. JOURNAL OF GASTROINTESTINAL AND ABDOMINAL RADIOLOGY 2021. [DOI: 10.1055/s-0041-1722813] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/08/2022] Open
Abstract
AbstractAutoimmune pancreatitis (AIP) consists of two clinically histologically distinct forms (type I and II) of chronic pancreatitis that are histologically different. These forms of AIP classically respond to oral steroids. The focal form of AIP resembles pancreatic carcinoma both clinically and radiologically and it is of utmost importance to make an early correct diagnosis between these two diseases in order to identify the optimal therapeutic strategy and to avoid unnecessary laparotomy or pancreatic resection in AIP patients. Here we report focal forms of type I and II AIP with clinical and imaging features closely mimicking pancreatic carcinoma.
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Affiliation(s)
- Diva S. Shah
- Department of Radiology, HCG Cancer Centre, Ahmedabad, Gujarat, India
| | - Bharat Prajapati
- GI Onco-Surgery Department, HCG Cancer Centre, Ahmedabad, Gujarat, India
| | - Kintan Sanghavi
- Department of Histopathology and Cytology, Strand Life Science, Ahmedabad, Gujarat, India
| | - Shubhda Kanhere
- Department of Histopathology and Cytology, Strand Life Science, Ahmedabad, Gujarat, India
| | - Jagdish Kothari
- GI Onco-Surgery Department, HCG Cancer Centre, Ahmedabad, Gujarat, India
| | - Jignesh Dubal
- Department of Radiology, HCG Cancer Centre, Ahmedabad, Gujarat, India
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Beyer G, Habtezion A, Werner J, Lerch MM, Mayerle J. Chronic pancreatitis. Lancet 2020; 396:499-512. [PMID: 32798493 DOI: 10.1016/s0140-6736(20)31318-0] [Citation(s) in RCA: 302] [Impact Index Per Article: 60.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/07/2020] [Revised: 05/02/2020] [Accepted: 05/07/2020] [Indexed: 12/15/2022]
Abstract
Chronic pancreatitis is a multifactorial, fibroinflammatory syndrome in which repetitive episodes of pancreatic inflammation lead to extensive fibrotic tissue replacement, resulting in chronic pain, exocrine and endocrine pancreatic insufficiency, reduced quality of life, and a shorter life expectancy. The incidence and prevalence of chronic pancreatitis is rising and no curative treatment is available. Using novel diagnostic algorithms, definitive chronic pancreatitis can be diagnosed by imaging criteria alone, whereas probable chronic pancreatitis requires clinical features and imaging criteria. Criteria for the diagnosis of early chronic pancreatitis are still under discussion and need prospective validation in clinical trials. Cross-sectional imaging should be used first; endoscopic ultrasound is needed only when CT or MRI are inconclusive or to plan therapeutic interventions. Management of chronic pancreatitis requires an interdisciplinary approach including primary care practitioners, gastroenterologists, surgeons, radiologists, pain specialists, and nutritional therapists. Patients with chronic pancreatitis should be seen at least once a year and re-evaluated for causal risk factors, symptom control, and complications such as malnutrition, pancreatic exocrine insufficiency, and diabetes; refer to a specialised centre if symptoms are poorly controlled or there is risk of deterioration. Scoring systems to monitor disease progression have been developed and validated internationally. Interventional treatments for pain or cholestasis should be done by specialists only, and early discussion of treatment approaches should include all medical disciplines involved in care. Throughout this Seminar, we address research needs such as staging of pancreatitis, aspects of malnutrition and pain, and cancer surveillance, to help improve the care of patients.
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Affiliation(s)
- Georg Beyer
- Medical Department II, University Hospital, LMU Munich, Munich, Germany
| | - Aida Habtezion
- Division of Gastroenterology and Hepatology, Stanford Medicine, Stanford University, Stanford, CA, USA
| | - Jens Werner
- Department of General, Visceral and Transplant Surgery, University Hospital, LMU Munich, Munich, Germany
| | - Markus M Lerch
- Department of Medicine A, University Medicine, University of Greifswald, Greifswald, Germany
| | - Julia Mayerle
- Medical Department II, University Hospital, LMU Munich, Munich, Germany.
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Chhoda A, Rustagi T. EUS-guided needle biopsy for autoimmune pancreatitis. Clin J Gastroenterol 2020; 13:669-677. [DOI: 10.1007/s12328-020-01153-0] [Citation(s) in RCA: 15] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/10/2019] [Accepted: 06/02/2020] [Indexed: 12/11/2022]
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Nakamaru K, Tomiyama T, Kobayashi S, Ikemune M, Tsukuda S, Ito T, Tanaka T, Yamaguchi T, Ando Y, Ikeura T, Fukui T, Nishio A, Takaoka M, Uchida K, Leung PSC, Gershwin ME, Okazaki K. Extracellular vesicles microRNA analysis in type 1 autoimmune pancreatitis: Increased expression of microRNA-21. Pancreatology 2020; 20:318-324. [PMID: 32147308 DOI: 10.1016/j.pan.2020.02.012] [Citation(s) in RCA: 13] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/16/2019] [Revised: 01/24/2020] [Accepted: 02/18/2020] [Indexed: 12/11/2022]
Abstract
BACKGROUND The molecular basis of type 1 autoimmune pancreatitis (AIP) remains unclear. Recent attention on the role of extracellular vesicles microRNA (EV miRNA) in immune homeostasis has prompted us to perform an extensive miRNA screening of serum-derived EV in AIP. METHODS EV miRNA expression was analyzed using microarrays in AIP, chronic pancreatitis (CP), and healthy adult (HC) samples (n = 10 from each group). Differences in signals, > 3 or <1/3 times, represented significant differences in expression. Another cohort of AIP (n = 14), CP (n = 10), and HC (n = 10) samples of EV miRNA was analyzed using reverse-transcription polymerase chain reaction (RT-PCR). miRNA expression in pancreatic tissues was evaluated using in situ hybridization (ISH) in three additional subjects from each group. RESULTS Signals of eight miRNAs (miR-659-3p, -27a-3p, -99a-5p, -21-5p, -205-5p, -100-5p, -29c-3p, and -125b-1-3p) were significantly higher, while those of two miRNAs (miR-4252 and -5004-5p) were significantly lower in AIP than in HC. EV miR-21-5p was significantly up-regulated in AIP than in HC (P = 0.035) and CP (P = 0.048). The number of miR-21-5p positive inflammatory cells was significantly elevated in AIP than in CP (P = 0.014). CONCLUSIONS Circulating EVs exhibited altered miRNA expression patterns with elevated miR-21-5p in AIP when compared with those in HC and CP. miR-21-5p was highly expressed in pancreatic inflammatory cells in AIP. Our data suggests that miR-21-5p may be involved in the regulation of effector pathways in the pathophysiology of AIP, thus differentiating AIP from CP.
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Affiliation(s)
- Koh Nakamaru
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Takashi Tomiyama
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan.
| | - Sanshiro Kobayashi
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Manami Ikemune
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Satoshi Tsukuda
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Takashi Ito
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Toshihiro Tanaka
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Takashi Yamaguchi
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Yugo Ando
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Tsukasa Ikeura
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Toshiro Fukui
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Akiyoshi Nishio
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Makoto Takaoka
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
| | - Kazushige Uchida
- Division of Gastroenterology and Hepatology, Kochi Medical School, Kochi University, Kohasu Oko-cho, Nankoku, Kochi, 783-8505, Japan
| | - Patrick S C Leung
- Division of Rheumatology, Allergy and Clinical Immunology, University of California at Davis, 451 Health Sciences Drive Suite 6510, Davis, CA, 95616, USA
| | - M E Gershwin
- Division of Rheumatology, Allergy and Clinical Immunology, University of California at Davis, 451 Health Sciences Drive Suite 6510, Davis, CA, 95616, USA
| | - Kazuichi Okazaki
- Third Department of Internal Medicine, Division of Gastroenterology and Hepatology, Kansai Medical University, 2-5-1 Shinmachi, Hirakata, Osaka, 573-1010, Japan
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Rebours V, Lévy P. Pancreatic and biliary tract involvement in IgG4-related disease. Presse Med 2020; 49:104015. [PMID: 32234378 DOI: 10.1016/j.lpm.2020.104015] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/27/2018] [Revised: 04/25/2019] [Accepted: 07/04/2019] [Indexed: 02/07/2023] Open
Abstract
The diagnosis and treatment of pancreatic and biliary tract involvement in IgG4 disease can be challenging for physicians. A French series shows that the pancreas is the most frequently involved organ in systemic IgG4 disease. Pancreatitis may be found in more than 50% of patients with IgG4 disease and the biliary tract is involved in one third. Pancreatic or biliary involvement may be isolated, metachronous or synchronous of other IgG4-related organ injuries. Pancreatitis related to IgG4 disease is called autoimmune pancreatitis (AIP) type 1. The diagnosis is mainly suspected in the presence of symptoms and morphological features. Changes observed on conventional imaging are not typical and are usually similar to lesions observed in autoimmune pancreatitis type 2. AIP type 1 can also sometimes have a clinical or morphological presentation that mimics pancreatic cancer, especially pseudo-tumoral forms, associated with obstructive jaundice, weight loss and fatigue. Thus, the first challenge is to confirm the diagnosis of autoimmune pancreatitis and to exclude cancer. The AIP type must then be determined to decide on the most appropriate treatment.
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Affiliation(s)
- Vinciane Rebours
- Pancreatology unit, Inserm, UMR 1149, pancreatic rare diseases (PaRaDis), centre de reference de maladies rares, Beaujon hospital, 100, boulevard Gal-Leclerc, 92110 Clichy, France.
| | - Philippe Lévy
- Pancreatology unit, Inserm, UMR 1149, pancreatic rare diseases (PaRaDis), centre de reference de maladies rares, Beaujon hospital, 100, boulevard Gal-Leclerc, 92110 Clichy, France
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Glinka J, Calderón F, de Santibañes M, Hyon SH, Gadano A, Mullen E, Pol M, Spina J, de Santibañes E. Early pancreatic cancer in IgG4-related pancreatic mass: A case report. World J Gastrointest Surg 2019; 11:443-448. [PMID: 31879536 PMCID: PMC6912071 DOI: 10.4240/wjgs.v11.i12.443] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/23/2019] [Revised: 09/18/2019] [Accepted: 11/07/2019] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND IgG4-related disease can manifest diversely, including autoimmune pancreatitis and IgG4-related cholangiopathy. We are reporting a very unusual cause of pancreatic cancer triggered in a previously unknown IgG4-related disease.
CASE SUMMARY A 75-year-old man was diagnosed with a 43 mm × 33 mm pancreatic head tumor after consulting for abdominal pain and jaundice. A pancreaticoduodenectomy was carried out uneventfully, and the histopathology report showed an early stage of acinar-cell pancreatic cancer. The patient reconsulted on the 30th postoperative day with fever, jaundice and asthenia. Magnetic resonance cholangiopancreatography evidenced an extense bile duct stricture. A percutaneous biliary drainage proved to be ineffective, even after exchanging it with larger bore drainage. Reviewing the surgical specimen, features compatible with IgG4-related disease were observed. Consequently, empiric treatment with steroids was initiated achieving excellent results.
CONCLUSION IgG4-related disease may cause chronic inflammation of the pancreas and can condition pancreatic malignancies.
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Affiliation(s)
- Juan Glinka
- Department of General Surgery, Hepato-bilio-pancreatic Unit, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Francisco Calderón
- Department of General Surgery, Hepato-bilio-pancreatic Unit, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Martín de Santibañes
- Department of General Surgery, Hepato-bilio-pancreatic Unit, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Sung Ho Hyon
- Department of General Surgery, Image Guided Minimally Invasive Surgery Unit, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Adrián Gadano
- Hepatology Section, Hospital Italiano Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Eduardo Mullen
- Department of Surgical Pathology, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Melina Pol
- Department of Surgical Pathology, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Juan Spina
- Department of Radiology, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
| | - Eduardo de Santibañes
- Department of General Surgery, Hepato-bilio-pancreatic Unit, Hospital Italiano de Buenos Aires, Buenos Aires C1181ACH, Argentina
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Sánchez Rodríguez E, García García de Paredes A, Albillos A. Current management of acute idiopathic pancreatitis and acute recurrent pancreatitis. Rev Clin Esp 2019. [DOI: 10.1016/j.rceng.2019.03.014] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
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Sánchez Rodríguez E, García García de Paredes A, Albillos A. Manejo actual de la pancreatitis aguda idiopática y la pancreatitis aguda recurrente. Rev Clin Esp 2019; 219:266-274. [DOI: 10.1016/j.rce.2018.12.011] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/11/2018] [Accepted: 12/28/2018] [Indexed: 02/07/2023]
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Dickerson LD, Farooq A, Bano F, Kleeff J, Baron R, Raraty M, Ghaneh P, Sutton R, Whelan P, Campbell F, Healey P, Neoptolemos JP, Yip VS. Differentiation of Autoimmune Pancreatitis from Pancreatic Cancer Remains Challenging. World J Surg 2019; 43:1604-1611. [PMID: 30815742 DOI: 10.1007/s00268-019-04928-w] [Citation(s) in RCA: 25] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
BACKGROUND Autoimmune pancreatitis (AIP) is an uncommon form of chronic pancreatitis. Whilst being corticosteroid responsive, AIP often masquerades radiologically as pancreatic neoplasia. Our aim is to appraise demographic, radiological and histological features in our cohort in order to differentiate AIP from pancreatic malignancy. METHODS Clinical, biochemical, histological and radiological details of all AIP patients 1997-2016 were analysed. The initial imaging was re-reviewed according to international guidelines by three blinded independent radiologists to evaluate features associated with autoimmune pancreatitis and pancreatic cancer. RESULTS There were a total of 45 patients: 25 in type 1 (55.5%), 14 type 2 (31.1%) and 6 AIP otherwise not specified (13.3%). The median (IQR) age was 57 (51-70) years. Thirty patients (66.6%) were male. Twenty-six patients (57.8%) had resection for suspected malignancy and one for symptomatic chronic pancreatitis. Three had histologically proven malignancy with concurrent AIP. Two patients died from recurrent pancreatic cancer following resection. Multidisciplinary team review based on radiology and clinical history dictated management. Resected patients (vs. non-resected group) were older (64 vs. 53, p = 0.003) and more frequently had co-existing autoimmune pathologies (22.2 vs. 55.6%, p = 0.022). Resected patients also presented with less classical radiological features of AIP, which are halo sign (0/25 vs. 3/17, p = 0.029) and loss of pancreatic clefts (18/25 vs. 17/17, p = 0.017). There were no differences in demographic features other than age. CONCLUSION Despite international guidelines for diagnosing AIP, differentiation from pancreatic cancer remains challenging. Resection remains an important treatment option in suspected cancer or where conservative treatment fails.
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Affiliation(s)
- L D Dickerson
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - A Farooq
- Department of Radiology, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - F Bano
- Department of Radiology, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - J Kleeff
- Department of Visceral, Vascular and Endocrine Surgery, Martin-Luther-University Halle-Wittenberg, 06120, Halle (Saale), Germany
| | - R Baron
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - M Raraty
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - P Ghaneh
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
- Department of Molecular and Clinical Cancer Medicine, Institute of Translational Medicine, University of Liverpool, Liverpool, L69 3GA, UK
| | - R Sutton
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
- Department of Molecular and Clinical Cancer Medicine, Institute of Translational Medicine, University of Liverpool, Liverpool, L69 3GA, UK
| | - P Whelan
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - F Campbell
- Department of Pathology, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - P Healey
- Department of Radiology, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK
| | - J P Neoptolemos
- Department of Molecular and Clinical Cancer Medicine, Institute of Translational Medicine, University of Liverpool, Liverpool, L69 3GA, UK
- Department of General, Visceral and Transplantation Surgery, University of Heidelberg, Im Neuenheimer Feld 110, 69120, Heidelberg, Germany
| | - V S Yip
- Pancreas Unit, Department of General Surgery, Royal Liverpool University Hospital, Prescot Street, Liverpool, L7 8XP, UK.
- Department of Hepatobiliary and Pancreas Surgery, 13C Royal London Hospital, Whitechapel Road, London, E1 1BB, UK.
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Shi Y, Cang L, Zhang X, Cai X, Wang X, Ji R, Wang M, Hong Y. The use of magnetic resonance elastography in differentiating autoimmune pancreatitis from pancreatic ductal adenocarcinoma: A preliminary study. Eur J Radiol 2018; 108:13-20. [PMID: 30396645 DOI: 10.1016/j.ejrad.2018.09.001] [Citation(s) in RCA: 24] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/26/2018] [Revised: 08/30/2018] [Accepted: 09/03/2018] [Indexed: 12/12/2022]
Abstract
PURPOSE To assess the value of magnetic resonance elastography (MRE) in patients with autoimmune pancreatitis (AIP) and in the differentiation of AIP from pancreatic ductal adenocarcinoma (PDAC). METHOD AND MATERIALS This prospective study included 14 AIP patients, 26 PDAC patients, and 14 healthy volunteers. All participants underwent pancreatic MRE (40-Hz; 3 T scanner) at enrollment, and 7 AIP patients underwent a second MRE after initiation of steroid therapy. Pancreatic stiffness values were obtained by MRE and a new logistic regression model (the calculated Rad score) was used to combine pancreatic stiffness and the distribution and shape of high-stiffness areas for differentiation of AIP and PDAC. The area under the curve (AUC) was calculated for all parameters using receiver operating characteristic (ROC) analysis. RESULTS Pancreatic stiffness was significantly higher (2.67 kPa [interquartile range, 2.24-3.56 kPa]) in AIP than in healthy pancreas (1.24 kPa [1.18-1.24 kPa]) and significantly lower in AIP than in PDAC (3.78 kPa [3.22-5.11 kPa]; both P < 0.05). Diffuse (n = 4 vs 1; P = 0.043) and multiple (n = 3 vs 0; P = 0.037) lesions were more common in AIP, while solitary (n = 25 vs 7; P = 0.001) and nodular lesions (n = 18 vs 2; P = 0.002) were more frequent in PDAC. Rad scores outperformed individual imaging parameters in distinguishing AIP from PDAC (AUC, 0.948 vs 0.607 to 0.782; all P < 0.05), with 84.6% specificity and 92.9% sensitivity. Pancreatic stiffness in AIP decreased significantly, from 2.66 kPa [2.29 to 3.05 kPa] to 1.55 kPa [1.43 to 1.67 kPa] (P = 0.016), during treatment. CONCLUSIONS MRE shows promise as a quantitative imaging method for differentiating AIP from PDAC and for monitoring the treatment response in AIP.
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Affiliation(s)
- Yu Shi
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Lizhuo Cang
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Xianyi Zhang
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Xiaoli Cai
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | | | - Ruoyun Ji
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Min Wang
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Yang Hong
- Department of Neurosurgery, Shengjing Hospital, China Medical University, Shenyang, PR China.
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Epstein R, Shaniuk PM, Sabe R. Fever, Abdominal Pain, and Jaundice in a 7-Year-Old Male. Clin Pediatr (Phila) 2018; 57:865-867. [PMID: 28990431 DOI: 10.1177/0009922817734365] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Affiliation(s)
- Rebecca Epstein
- 1 Rainbow Babies and Children's Hospital, Cleveland, OH, USA
| | - Paul M Shaniuk
- 1 Rainbow Babies and Children's Hospital, Cleveland, OH, USA.,2 Case Western Reserve University, Cleveland, OH, USA.,3 University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| | - Ramy Sabe
- 1 Rainbow Babies and Children's Hospital, Cleveland, OH, USA.,2 Case Western Reserve University, Cleveland, OH, USA
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Muñoz LE, Leppkes M, Fuchs TA, Hoffmann M, Herrmann M. Missing in action-The meaning of cell death in tissue damage and inflammation. Immunol Rev 2018; 280:26-40. [PMID: 29027227 DOI: 10.1111/imr.12569] [Citation(s) in RCA: 22] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
Billions of cells die every day in higher organisms as part of the normal process of tissue homeostasis. During special conditions like in development, acute infections, mechanical injuries, and immunity, cell death is a common denominator and it exerts profound effects in the outcome of these scenarios. To prevent the accumulation of aged, superfluous, infected, damaged and dead cells, professional phagocytes act in a rapid and efficient manner to clear the battle field and avoid spread of the destruction. Neutrophils are the most abundant effector immune cells that extravasate into tissues and can turn injured tissues into gory battle fields. In peace times, neutrophils tend to patrol tissues without provoking inflammatory reactions. We discuss in this review actual and forgotten knowledge about the meaning of cell death during homeostatic processes and drive the attention to the importance of the action of neutrophils during patrolling and for the maintenance or recovery of the homeostatic state once the organism gets attacked or injured, respectively. In this fashion, we disclose several disease conditions that arise as collateral damage of physiological responses to death.
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Affiliation(s)
- Luis E Muñoz
- Department of Internal Medicine 3 - Rheumatology and Immunology, Friedrich-Alexander-University Erlangen-Nürnberg (FAU), Universitätsklinikum Erlangen, Erlangen, Germany
| | - Moritz Leppkes
- Department of Internal Medicine 1 - Gastroenterology, Pulmonology and Endocrinology, Friedrich-Alexander-University Erlangen-Nürnberg (FAU), Universitätsklinikum Erlangen, Erlangen, Germany
| | - Tobias A Fuchs
- Institute of Clinical Chemistry and Laboratory Medicine, University Medical Center Hamburg-Eppendorf, Hamburg, Germany
| | - Markus Hoffmann
- Department of Internal Medicine 3 - Rheumatology and Immunology, Friedrich-Alexander-University Erlangen-Nürnberg (FAU), Universitätsklinikum Erlangen, Erlangen, Germany
| | - Martin Herrmann
- Department of Internal Medicine 3 - Rheumatology and Immunology, Friedrich-Alexander-University Erlangen-Nürnberg (FAU), Universitätsklinikum Erlangen, Erlangen, Germany
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Schneider A, Michaely H, Weiss C, Hirth M, Rückert F, Wilhelm TJ, Schönberg S, Marx A, Singer MV, Löhr JM, Ebert MP, Pfützer RH. Prevalence and Incidence of Autoimmune Pancreatitis in the Population Living in the Southwest of Germany. Digestion 2017; 96:187-198. [PMID: 28957814 DOI: 10.1159/000479316] [Citation(s) in RCA: 23] [Impact Index Per Article: 2.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/27/2017] [Accepted: 07/07/2017] [Indexed: 02/04/2023]
Abstract
BACKGROUND/AIMS The prevalence and incidence of autoimmune pancreatitis (AiP) in those living in western countries are largely unknown. We aimed to determine the prevalence of AiP among patients with pancreatitis presenting to our tertiary referral center in Mannheim, Germany; and to estimate the incidence of AiP in the Southwest of Germany. METHODS We performed a retrospective cross-sectional analysis and determined the prevalence of AiP in patients with acute pancreatitis (AP) or chronic pancreatitis (CP). Patients (n = 704; alcoholic pancreatitis n = 373, nonalcoholic pancreatitis n = 331) were stratified into the Retrospective-Pancreas-Cohort (RPC, period 1998-2008, n = 534) and the Pancreas-Clinic-Cohort (PCC, periods 2008-2010 and 2013-2014, n = 170, with detailed investigation for features of AiP). Diagnosis of AiP was established by International-Consensus-Diagnostic-Criteria and Unifying-Autoimmune-Pancreatitis-Criteria. RESULTS In the RPC, the prevalence of AiP was 5.9% (n = 13/221) among individuals with nonalcoholic pancreatitis (n = 1/61 with AP, 1.6%; n = 12/160 with CP, 7.5%). In the PCC, the prevalence of AiP was 9.1% (n = 10/110) among patients with nonalcoholic pancreatitis (n = 2/24 with AP, 8.3%; n = 8/86 with CP, 9.3%), and 1.7% (n = 1/60) among subjects with alcoholic pancreatitis. We estimated the incidence of AiP with 0.29 per 100,000 population each year. CONCLUSION The prevalence rate of AiP may account for 9% of patients with nonalcoholic pancreatitis but is almost never observed in patients with alcoholic pancreatitis. The incidence of AiP in Germany appears lower than 1 per 100,000 population.
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Affiliation(s)
- Alexander Schneider
- Department of Medicine II, University Medical Centre Mannheim, Medical Faculty of the University of Heidelberg, Mannheim, Germany
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Kolasinski NT, Johannsen MT, Hollon JR. Fifteen-Year-Old Male with Type 2 Autoimmune Pancreatitis: An Argument for Endoscopy. Case Rep Gastroenterol 2017. [PMID: 28626379 PMCID: PMC5471753 DOI: 10.1159/000453659] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/11/2023] Open
Abstract
Type 2 autoimmune pancreatitis, an increasingly recognized etiology of pancreatitis in patients less than 20 years old, has characteristically been diagnosed with the histological finding of duct-centric pancreatitis in a patient who lacks elevated serum immunoglobulin G4. We present the case of a nonobese 15-year-old male, without any chronic medical conditions, who presented with the chief complaint of abdominal pain. The laboratory study results were remarkable for a lipase level of 5,419 U/L and a γ-glutamyl transferase level of 373 U/L. Magnetic resonance cholangiopancreatography revealed delayed contrast enhancement of the pancreas, diffuse parenchymal enlargement, and lack of normal lobulation. The patient's serum immunoglobulin G4 level was found to be 66 mg/dL, which was within normal limits and supportive of a diagnosis of type 2 autoimmune pancreatitis. Despite the absence of intestinal complaints, the patient underwent subsequent endoscopy due to the correlation of type 2 autoimmune pancreatitis with inflammatory bowel disease that has been described in recent literature. Pan-colonic mild colitis was visualized, and the patient began treatment with steroids, to which he quickly responded. Performing endoscopy on this patient allowed for confident initiation of early therapy for both autoimmune pancreatitis and inflammatory bowel disease, and may have limited further surgical intervention and disease progression. For these reasons, this case highlights the utility of endoscopy in pediatric patients with suspected type 2 autoimmune pancreatitis, even in the absence of intestinal symptoms.
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Affiliation(s)
- Nathan T Kolasinski
- Department of Pediatrics, Naval Medical Center Portsmouth, Portsmouth, Virginia, USA
| | - Marc T Johannsen
- Department of Radiology, Naval Medical Center Portsmouth, Portsmouth, Virginia, USA
| | - Justin R Hollon
- Department of Pediatrics, Naval Medical Center Portsmouth, Portsmouth, Virginia, USA
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Abstract
Autoimmune pancreatitis (AIP) occurring in association with inflammatory bowel disease (IBD) is rather rare and carries a worse prognosis and greater disease severity compared with IBD alone. Although it is an infrequently documented association, progress over the last 20 years has led to better understanding of the association between AIP and IBD. IBD has a stronger association with type 2 than with type 1 AIP. Clinical and histologic features of AIP-IBD more often reveal features of type 2 AIP. Imaging is not helpful in facilitating the diagnosis of AIP and IBD. Similarly, attempts to identify a serological marker have not yielded better result. A proposed lymphocyte homing mechanism provides some insight into the mechanism of pathogenesis. This review represents an update of our current knowledge of the association between AIP and IBD.
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Shi ZD. Recent advances in research of immunoglobulin G4-related autoimmune pancreatits. Shijie Huaren Xiaohua Zazhi 2016; 24:3946-3952. [DOI: 10.11569/wcjd.v24.i28.3946] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Autoimmune pancreatitis (AIP) is a rare type of chronic pancreatitis that belongs to the spectrum of immunoglobulin G4 (IgG4) related diseases. AIP is characterized by lymphoplasmacytic infiltration, storiform fibrosis, obliterative phlebitis, and increased IgG4+ plasma cells. Patients with AIP present clinically with recurrent pancreatitis and obstructive jaundice, symptoms involving bile ducts and salivary glands and so on. Serum IgG4 level is often elevated. The main imaging manifestation of AIP is a "sausage-shaped pancreas" and multiple strictures of the main pancreatic duct. AIP responds sensitively to glucocorticosteroids but relapses easily. In relapsed cases, re-administration of glucocorticosteroids alone or in combination with immune modulators is effective.
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Kim M, Jang KM, Kim JH, Jeong WK, Kim SH, Kang TW, Kim YK, Cha DI, Kim K. Differentiation of mass-forming focal pancreatitis from pancreatic ductal adenocarcinoma: value of characterizing dynamic enhancement patterns on contrast-enhanced MR images by adding signal intensity color mapping. Eur Radiol 2016; 27:1722-1732. [PMID: 27510628 DOI: 10.1007/s00330-016-4522-0] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/03/2016] [Revised: 07/18/2016] [Accepted: 07/20/2016] [Indexed: 12/17/2022]
Abstract
OBJECTIVES To evaluate the value of dynamic enhancement patterns on contrast-enhanced MR images by adding signal intensity colour mapping (SICM) to differentiate mass-forming focal pancreatitis (MFFP) from pancreatic ductal adenocarcinoma (PDAC). METHODS Forty-one clinicopathologically proven MFFPs and 144 surgically confirmed PDACs were enrolled. Laboratory and MR imaging parameters were used to differentiate MFFP from PDAC. In particular, enhancement patterns on MR images adding SICM were evaluated. By using classification tree analysis (CTA), we determined the predictors for the differentiation of MFFP from PDAC. RESULTS In the CTA, with all parameters except enhancement pattern on SICM images, ductal obstruction grade and T1 hypointensity grade of the pancreatic lesion were the first and second splitting predictor for differentiation of MFFP from PDAC, in order. By adding an enhancement pattern on the SICM images to CTA, the enhancement pattern was the only splitting predictor to differentiate MFFP from PDAC. The CTA model including enhancement pattern on SICM images has sensitivity of 78.0 %, specificity of 99.3 %, and accuracy of 94.6 % for differentiating MFFP from PDAC. CONCLUSION The characterization of enhancement pattern for pancreatic lesions on contrast-enhanced MR images adding SICM would be helpful to differentiate MFFP from PDAC. KEY POINTS • SICM was useful to characterize enhancement pattern. • Enhancement pattern on SICM was the only splitting predictor on CTA. • This model may be useful for differentiating MFFP from PDAC.
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Affiliation(s)
- Mimi Kim
- Department of Radiology, Hanyang Medical Center, Hanyang University College of Medicine, Seoul, Republic of Korea
| | - Kyung Mi Jang
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea. .,Department of Radiology and Center for Imaging Science, Samsung Medical Center, Sungkyunkwan University School of Medicine, 50 Irwon-Dong, Gangnam-gu, Seoul, 135-710, Korea.
| | - Jae-Hun Kim
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
| | - Woo Kyoung Jeong
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
| | - Seong Hyun Kim
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
| | - Tae Wook Kang
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
| | - Young Kon Kim
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
| | - Dong Ik Cha
- Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
| | - Kyunga Kim
- Biostatics and Clinical Epidemiology Center, Research Institute for Future Medicine, Samsung Medical Center, Seoul, Republic of Korea
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López-Serrano A, Crespo J, Pascual I, Salord S, Bolado F, Del-Pozo-García AJ, Ilzarbe L, de-Madaria E, Moreno-Osset E. Diagnosis, treatment and long-term outcomes of autoimmune pancreatitis in Spain based on the International Consensus Diagnostic Criteria: A multi-centre study. Pancreatology 2016; 16:382-90. [PMID: 26944001 DOI: 10.1016/j.pan.2016.02.006] [Citation(s) in RCA: 25] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/22/2015] [Revised: 12/18/2015] [Accepted: 02/12/2016] [Indexed: 12/11/2022]
Abstract
OBJECTIVES Autoimmune pancreatitis (AIP) is a form of chronic pancreatitis that has been reported worldwide for the last two decades. The aim of this study is to analyse the clinical profile of patients from Spain with AIP, as well as treatments, relapses and long-term outcomes. METHODS Data from 59 patients with suspected AIP that had been diagnosed in 15 institutions are retrospectively analysed. Subjects are classified according to the International Consensus Diagnostic Criteria (ICDC). Patients with type 1 AIP (AIP1) and type 2 AIP (AIP2) are compared. Kaplan-Meier methodology is used to estimate the overall survival without relapses. RESULTS Fifty-two patients met ICDC, 45 patients were AIP1 (86.5%). Common manifestations included abdominal pain (65.4%) and obstructive jaundice (51.9%). Diffuse enlargement of pancreas was present in 51.0%; other organ involvement was present in 61.5%. Serum IgG4 increased in 76.7% of AIP1 patients vs. 20.0% in AIP2 (p = 0.028). Tissue specimens were obtained in 76.9%. Initial successful treatment with steroids or surgery was achieved in 79.8% and 17.3%, respectively. Maintenance treatment was given in 59.6%. Relapses were present in 40.4% of AIP1, with a median of 483 days. Successful long-term remission was achieved in 86.4%. CONCLUSIONS AIP1 is the most frequent form of AIP in Spain in our dataset. Regularly, ICDC allows AIP diagnosis without the need for surgery. Steroid and chirurgic treatments were effective and safe in most patients with AIP, although maintenance was required many times because of their tendency to relapse. Long-term serious consequences were uncommon.
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Affiliation(s)
- Antonio López-Serrano
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain.
| | - Javier Crespo
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Isabel Pascual
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Silvia Salord
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Federico Bolado
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Andrés J Del-Pozo-García
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Lucas Ilzarbe
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Enrique de-Madaria
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
| | - Eduardo Moreno-Osset
- Department of Gastroenterology, Hospital Universitari Dr. Peset, University of Valencia, Valencia, Spain
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Rasch S, Phillip V, Schmid RM, Algül H. Epidemiology, clinical presentation, diagnosis and treatment of autoimmune pancreatitis: A retrospective analysis of 53 patients. Pancreatology 2015; 16:73-7. [PMID: 26694138 DOI: 10.1016/j.pan.2015.11.006] [Citation(s) in RCA: 23] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/10/2015] [Revised: 09/03/2015] [Accepted: 11/17/2015] [Indexed: 12/11/2022]
Abstract
BACKGROUND Most of the data about epidemiology, clinical presentation and treatment of autoimmune pancreatitis (AIP) is based on case series or small study groups. We therefore analyzed all cases of AIP treated at our clinic retrospectively. METHODS We searched our clinical database for the diagnosis pancreatitis between January 2007 and June 2014, selected patients with AIP and entered all relevant information in a database for statistical analysis. RESULTS In total 53 patients with AIP were treated at our institution, 62% with type 1 and 23% with type 2 AIP. Gender distribution was male/female 3.1:1 for type 1 and 1:1.2 for type 2 AIP. The median age was 63.0 and 32.5 years for type 1 and type 2 AIP, respectively. The most common symptom is abdominal pain particular in patients with type 2 AIP whereas jaundice was only apparent in patients with type 1 AIP. The international diagnostic criteria seem to facilitate diagnosis of AIP as unnecessary pancreatic surgery in patients with AIP decreases. In 62.6% of the patients therapy was indicated and 84.8% showed a response to initial therapy with steroids. Recurring disease occurred in 28.3% of the cases but only 3.8% suffered a second relapse. Permanent maintenance therapy with steroids or additional therapy with immunomodulatory drugs is successful in recurring disease. CONCLUSION Our data further corroborate previous findings on epidemiology, clinical presentation and treatment of AIP. AIP is a well manageable autoimmune disease in most patients. Better biopsy techniques and simplified diagnostic criteria might further alleviate diagnosis of AIP.
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Affiliation(s)
- Sebastian Rasch
- II. Medizinische Klinik und Poliklinik, Klinikum rechts der Isar, Technische Universität München, Ismaninger Straße 22, 81675 München, Germany.
| | - Veit Phillip
- II. Medizinische Klinik und Poliklinik, Klinikum rechts der Isar, Technische Universität München, Ismaninger Straße 22, 81675 München, Germany
| | - Roland M Schmid
- II. Medizinische Klinik und Poliklinik, Klinikum rechts der Isar, Technische Universität München, Ismaninger Straße 22, 81675 München, Germany
| | - Hana Algül
- II. Medizinische Klinik und Poliklinik, Klinikum rechts der Isar, Technische Universität München, Ismaninger Straße 22, 81675 München, Germany
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Nakano E, Kanno A, Masamune A, Yoshida N, Hongo S, Miura S, Takikawa T, Hamada S, Kume K, Kikuta K, Hirota M, Nakayama K, Fujishima F, Shimosegawa T. IgG4-unrelated type 1 autoimmune pancreatitis. World J Gastroenterol 2015; 21:9808-9816. [PMID: 26361429 PMCID: PMC4562966 DOI: 10.3748/wjg.v21.i33.9808] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/14/2015] [Revised: 04/14/2015] [Accepted: 06/26/2015] [Indexed: 02/06/2023] Open
Abstract
A 50-year-old male was referred to our hospital for the evaluation of hyperproteinemia. Fluorodeoxyglucose positron emission tomography revealed high fluorodeoxyglucose uptake in the pancreas, bilateral lacrimal glands, submandibular glands, parotid glands, bilateral pulmonary hilar lymph nodes, and kidneys. Laboratory data showed an elevation of hepatobiliary enzymes, renal dysfunction, and remarkably high immunoglobulin (Ig) G levels, without elevated serum IgG4. Abdominal computed tomography revealed swelling of the pancreatic head and bilateral kidneys. Endoscopic retrograde cholangiopancreatography showed an irregular narrowing of the main pancreatic duct in the pancreatic head and stricture of the lower common bile duct. Histological examination by endoscopic ultrasonography-guided fine-needle aspiration revealed findings of lymphoplasmacytic sclerosing pancreatitis without IgG4-positive plasma cells. Abnormal laboratory values and the swelling of several organs were improved by the treatment with steroids. The patient was diagnosed as having type 1 autoimmune pancreatitis (AIP) based on the International Consensus Diagnostic Criteria. Therefore, we encountered a case of compatible type 1 AIP without elevated levels of serum IgG4 or IgG4-positive plasma cells. This case suggests that AIP phenotypes are not always associated with IgG4.
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Fernández-Codina A, Martínez-Valle F, Pinilla B, López C, DeTorres I, Solans-Laqué R, Fraile-Rodríguez G, Casanovas-Martínez A, López-Dupla M, Robles-Marhuenda Á, Barragán-González MJ, Cid MC, Prieto-González S, Brito-Zerón P, Cruces-Moreno MT, Fonseca-Aizpuru E, López-Torres M, Gil J, Núñez-Fernández MJ, Pardos-Gea J, Salvador-Cervelló G. IgG4-Related Disease: Results From a Multicenter Spanish Registry. Medicine (Baltimore) 2015; 94:e1275. [PMID: 26266361 PMCID: PMC4616706 DOI: 10.1097/md.0000000000001275] [Citation(s) in RCA: 87] [Impact Index Per Article: 8.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/24/2022] Open
Abstract
IgG4-related disease (IgG4-RD) is a rare entity consisting of inflammation and fibrosis that has been described in multiple organs. Concrete diagnostic criteria have been established recently and there is a lack of large series of patients.To describe the clinical presentation, histopathological characteristics, treatment and evolution of a series of IgG4-RD Spanish patients.A retrospective multicenter study was performed. Twelve hospitals across Spain included patients meeting the current 2012 consensus criteria on IgG4-RD diagnosis.Fifty-five patients were included in the study, 38 of whom (69.1%) were male. Median age at diagnosis was 53 years. Thirty (54.5%) patients were included in the Histologically Highly Suggestive IgG4-RD group and 25 (45.5%) in the probable IgG4-RD group. Twenty-six (47.3%) patients had more than 1 organ affected at presentation. The most frequently affected organs were: retroperitoneum, orbital pseudotumor, pancreas, salivary and lachrymal glands, and maxillary sinuses.Corticosteroids were the mainstay of treatment (46 patients, 83.6%). Eighteen patients (32.7%) required additional immunosuppressive agents. Twenty-four (43.6%) patients achieved a complete response and 26 (43.7%) presented a partial response (<50% of regression) after 22 months of follow-up. No deaths were attributed directly to IgG4-RD and malignancy was infrequent.This is the largest IgG4-RD series reported in Europe. Patients were middle-aged males, with histologically probable IgG4-RD. The systemic form of the disease was frequent, involving mainly sites of the head and abdomen. Corticosteroids were an effective first line treatment, sometimes combined with immunosuppressive agents. Neither fatalities nor malignancies were attributed to IgG4-RD.
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Affiliation(s)
- Andreu Fernández-Codina
- From the Internal Medicine Department, Hospital Universitari Vall d'Hebron, Barcelona, Spain (AF-C, FM-V, RS-L, JP-G); Hospital General Universitario Gregorio Marañón, Madrid, Spain (BP, CL); Pathology Department, Hospital Universitari Vall d'Hebron, Barcelona, Spain (IDT); Hospital Ramón y Caja, Madrid, Spain (GF-R); Consorci Sanitari Parc Taulí, Spain (AC-M); Hospital Universitari Joan XXIII, Tarragona, Spain (ML-D); Hospital La Paz, Madrid, Spain (AR-M); Hospital Valle del Nalón, Langreo, Spain (MJB-G); Hospital Clínic i Provincial, Barcelona, Spain (MCC, SP-G, PB-Z); Hospital Universitario San Cecilio, Granada, Spain (MTC-M); Hospital de Cabueñes, Gijón, Spain (EF-A); Hospital Universitario La Princesa, Madrid, Spain (ML-T); Hospital Universitario Río Hortega, Valladolid, Spain (JG); Complejo Hospitalario de Pontevedra, Pontevedra, Spain (MJN-F); and Hospital de Manises, Manises, Spain (GS-C)
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Kawa S, Okazaki K, Notohara K, Watanabe M, Shimosegawa T. Autoimmune pancreatitis complicated with inflammatory bowel disease and comparative study of type 1 and type 2 autoimmune pancreatitis. J Gastroenterol 2015; 50:805-815. [PMID: 25399203 DOI: 10.1007/s00535-014-1012-5] [Citation(s) in RCA: 28] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/01/2014] [Accepted: 10/27/2014] [Indexed: 02/04/2023]
Abstract
BACKGROUND Two types of autoimmune pancreatitis (AIP) have been reported, lymphoplasmacytic sclerosing pancreatitis and idiopathic duct-centric chronic pancreatitis (IDCP), which are now recognized as type 1 and type 2 AIP, respectively. Since the clinical features of type 2 AIP have not been fully elucidated and this condition is frequently accompanied by inflammatory bowel disease (IBD), we performed a nationwide survey of patients with AIP complicated with IBD to precisely characterize this disease entity. METHODS We collected 138 cases of pancreatitis with complicating IBD from affiliated institutes specializing in AIP or IBD, and comparative study between the IDCP groups and type 1 AIP was performed. RESULTS Histological examination revealed 15 AIP cases to be IDCP of institutional diagnosis, among which 11 cases were upgraded to IDCP of central diagnosis by an expert pathologist. The IDCP group exhibited younger onset age, no gender bias, frequent abdominal pain, and normal IgG4 value, similar to those of type 2 AIP reported previously. We also witnessed a lower prevalence of jaundice in type 2 AIP than in type 1 AIP that corresponded to imaging findings of less frequent pancreatic head swelling and scarce bile duct stenosis. CONCLUSIONS A characteristic feature of type 2 AIP compared with type 1 AIP is a low frequency of obstructive jaundice that is related to rare lower bile duct stricture due to lower prevalence of pancreatic head swelling. Contrary to type 1 AIP, lower bile duct stricture in this condition has no apparent relation to sclerosing cholangitis.
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Affiliation(s)
- Shigeyuki Kawa
- Center for Health, Safety, and Environmental Management, Shinshu University, 3-1-1 Asahi, Matsumoto, 390-8621, Japan,
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Cystic fibrosis transmembrane conductance regulator gene variants are associated with autoimmune pancreatitis and slow response to steroid treatment. J Cyst Fibros 2015; 14:661-7. [PMID: 25869325 DOI: 10.1016/j.jcf.2015.03.009] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/05/2014] [Revised: 03/22/2015] [Accepted: 03/22/2015] [Indexed: 12/24/2022]
Abstract
BACKGROUND Autoimmune pancreatitis (AIP) is a distinct type of chronic pancreatitis. To date, the association of CFTR gene variants with AIP has not been studied. METHODS The entire coding and intronic regions of the CFTR gene were examined using next-generation sequencing in 89 AIP patients. Clinical features, including imaging, histology, serology, steroid treatment response and extra-pancreatic involvement, were compared between AIP patients with and without CFTR gene variants. RESULTS A total of 28.1% (25/89) of the AIP patients carried 26 CFTR variants, including nine with I556V, seven with 5T, four with S42F, two with I125T, and one each with R31C, R553X, S895N, and G1069R. The presence of CFTR variants and age was independent predictors of the response to steroid treatment, as shown by multivariate analysis. CONCLUSIONS CFTR variants are associated with AIP. Because AIP patients with CFTR variants show slower and reduced steroid treatment responses, different treatments should be considered in AIP patients with CFTR variants.
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Senosiain Lalastra C, Foruny Olcina JR. [Autoimmune pancreatitis]. GASTROENTEROLOGIA Y HEPATOLOGIA 2015; 38:549-55. [PMID: 25799073 DOI: 10.1016/j.gastrohep.2015.01.006] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 12/16/2014] [Revised: 01/20/2015] [Accepted: 01/23/2015] [Indexed: 01/06/2023]
Abstract
Autoimmune pancreatitis is a benign fibroinflammatory disease of the pancreas of probable autoimmune origin, which includes 2 different phenotypes: type 1 (lymphoplasmacytic sclerosing pancreatitis) and type 2 (idiopathic duct-centric pancreatitis). Its clinical presentation as obstructive jaundice in patients with a pancreatic mass is common and therefore it must be included in the differential diagnosis of pancreatic neoplasia. Many diagnostic criteria have been described throughout history. The most famous are the HISORT criteria of the Mayo Clinic and the international consensus criteria of 2011. One of the main features of autoimmune pancreatitis is its dramatic response to steroid therapy, without the need for surgical treatment. Knowledge of this disease can dramatically change the management of patients with obstructive jaundice, who would otherwise be subjected to a pancreaticoduodenectomy.
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Affiliation(s)
- Carla Senosiain Lalastra
- Servicio de Aparato Digestivo, Hospital Universitario Príncipe de Asturias, Alcalá de Henares, Madrid, España.
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Autoimmune pancreatitis: a case of difficult diagnosis. GASTROENTEROLOGY REVIEW 2015; 10:51-3. [PMID: 25960816 PMCID: PMC4411406 DOI: 10.5114/pg.2014.47500] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 10/27/2012] [Revised: 01/07/2013] [Accepted: 03/22/2013] [Indexed: 12/16/2022]
Abstract
Autoimmune pancreatitis (AIP) is an insidious disease of non-specific symptomatology. To make correct diagnosis three different findings must correlate: radiological imaging, serological markers, and histology. This is not easy, and furthermore an incorrect diagnosis can lead to incorrect management and even patient death. We present our experience with a case of AIP in a young woman (34 years old) affected by different autoimmune pathologies with a history of abdominal pain. The diagnosis was made correlating histological findings and anamnestic data, although there were no radiological or serological findings. However, the management of this case was complicated by acute pancreatitis. In our case, we had only a histological sample and anamnestic data. So in these cases of positive history for autoimmune disorders and unclear clinical signs, AIP should be considered in differential diagnosis.
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Pancreatic duct "Icicle sign" on MRI for distinguishing autoimmune pancreatitis from pancreatic ductal adenocarcinoma in the proximal pancreas. Eur Radiol 2014; 25:1551-60. [PMID: 25501271 DOI: 10.1007/s00330-014-3548-4] [Citation(s) in RCA: 47] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/10/2014] [Revised: 11/20/2014] [Accepted: 12/01/2014] [Indexed: 12/24/2022]
Abstract
OBJECTIVE To determine reliable MRI features of autoimmune pancreatitis (AIP) in the proximal pancreas that could allow its differentiation from pancreatic ductal adenocarcinoma (PDAC). METHODS Twenty-three patients with AIP and 61 patients with PDAC in the proximal pancreas underwent MRI. Two observers analyzed MRI for lesion morphology, hypointensity degree on T1-weighted images, enhancement pattern during dynamic phases, capsule-like rim, presence of cysts and duct penetrating sign, morphology of bile duct, and icicle appearance and tortuosity of the upstream pancreatic duct. Sensitivity and specificity for the diagnosis of AIP were calculated for each category or combined. RESULTS When isointensity on the portal and late phase of MRI and/or the icicle sign of pancreatic duct are applied, 100 % sensitivity for the diagnosis of AIP in the proximal pancreas was achieved. Applying both mild T1 hypointensity similar to the spleen and the icicle sign enabled 100 % specificity for the diagnosis of AIP by differentiating it from PDAC. CONCLUSION The combination of the icicle sign in the upstream pancreatic duct and mild T1 hypointensity or isointensity on portal and late phase of dynamic MRI could be reliable MR features for the diagnosis of AIP in the proximal pancreas by allowing its differentiation from PDAC. KEY POINTS • The icicle sign of the pancreatic duct is useful for diagnosing AIP. • Mild T1 hypointensity similar to the spleen is useful for diagnosing AIP. • Isointensity on portal and late phases MRI is useful for diagnosing AIP.
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Franchello A, Gonella F, Campra D, Limerutti G, Bruno M, De Angelis C, Cassine D, Fronda G, Silvestri S. A rare case of IgG4-related systemic disease manifesting with pancreatic head mass mimicking borderline resectable cancer. Int J Surg Case Rep 2014; 5:1095-7. [PMID: 25460484 PMCID: PMC4275972 DOI: 10.1016/j.ijscr.2014.11.014] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/08/2014] [Accepted: 11/06/2014] [Indexed: 01/02/2023] Open
Abstract
Diagnostic algorithm for pancreatic head mass. Differential diagnosis between pancreatic cancer and mass forming autoimmune pancreatitis. Diagnosis and treatment of IgG4-related systemic disease. INTRODUCTION Autoimmune pancreatitis (AIP) is a rare pancreatic disorder among chronic pancreatitis that can mimick pancreatic cancer (PC). Patients with type 1 AIP usually present obstructive jaundice associated with high level of IgG4 in serum and a pancreatic mass at radiological imaging; these disorders may be associated with other organs lesions presenting the same histopathological features, and in these cases AIP should be considered a pancreatic localization of an IgG4-related systemic disease. PRESENTATION OF CASE We report the case of a young man with initial suspect of PC to be treated with surgery, and final diagnosis of AIP in the context of an IgG4-related systemic disease. DISCUSSION Because of its similar features, several algorithms have been proposed for AIP diagnosis, based on combination of clinical/serological and radiological criteria. However, histology represents the only way to obtain definitive diagnosis, even if sometimes it is difficult to obtain biological samples. CONCLUSION IgG4-related systemic disease must be taken into account among differential diagnosis during the workup for PC, in order to avoid unnecessary surgery.
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Affiliation(s)
| | | | - Donata Campra
- Department of Surgery, Molinette Hospital, Turin, Italy
| | | | - Mauro Bruno
- Department of Gastroenterology, Molinette Hospital, Turin, Italy
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Beyer G, Schwaiger T, Lerch MM, Mayerle J. IgG4-related disease: a new kid on the block or an old aquaintance? United European Gastroenterol J 2014; 2:165-72. [PMID: 25360299 DOI: 10.1177/2050640614532457] [Citation(s) in RCA: 24] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/13/2014] [Accepted: 03/13/2014] [Indexed: 12/24/2022] Open
Abstract
IgG4-related systemic disease is a recently recognized systemic condition characterized by unique pathological features that can affect a variety of organs. It includes a growing number of medical conditions which have the following features in common: diffuse organ swelling or focal mass formation, sclerosing storiforme (whirl-shaped) fibrosis with a lymphoplasmacytic infiltrate rich in IgG4-bearing plasma cells, as well as elevated levels of serum IgG4. It invariably responds to steroid treatment and is mostly diagnosed in elderly men. Well-known syndromes like Mikulicz's disease of the salivary or lacrimal gland, Küttner's tumour of the submandibular gland, Riedel's thyroiditis, or retroperitoneal fibrosis, as well as novel entities such as autoimmune pancreatitis type 1, are now regarded to be manifestations of this systemic disease. This article provides an overview of the epidemiology, concepts of pathogenesis, clinical presentation, proposed diagnostic approaches, treatment options, and differential diagnosis of IgG4-related disease.
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Affiliation(s)
- Georg Beyer
- Department of Medicine A, University Medicine Greifswald, Ernst-Moritz-Arndt-University Greifswald, Germany
| | - Theresa Schwaiger
- Department of Medicine A, University Medicine Greifswald, Ernst-Moritz-Arndt-University Greifswald, Germany
| | - Markus M Lerch
- Department of Medicine A, University Medicine Greifswald, Ernst-Moritz-Arndt-University Greifswald, Germany
| | - Julia Mayerle
- Department of Medicine A, University Medicine Greifswald, Ernst-Moritz-Arndt-University Greifswald, Germany
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Fritz S, Bergmann F, Grenacher L, Sgroi M, Hinz U, Hackert T, Büchler MW, Werner J. Diagnosis and treatment of autoimmune pancreatitis types 1 and 2. Br J Surg 2014; 101:1257-65. [DOI: 10.1002/bjs.9574] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/23/2013] [Revised: 01/31/2014] [Accepted: 04/24/2014] [Indexed: 01/05/2023]
Abstract
Abstract
Background
Autoimmune pancreatitis (AIP) is characterized by diffuse or focal swelling of the pancreas. AIP has been divided into types 1 and 2. The aim of the study was to evaluate and compare the clinicopathological characteristics, therapy and outcome of patients with AIP.
Methods
The medical records of patients diagnosed with AIP between January 2003 and July 2011 were reviewed. Characteristics of patients with AIP types 1 and 2 were compared with those of patients with pancreatic ductal adenocarcinoma (PDAC).
Results
AIP was classified as type 1 in 40 patients and type 2 in 32 according to the HISORt (Histology, Imaging, Serology, Other organ involvement, Response to therapy) criteria. Patients with histologically confirmed AIP type 2 were younger than those with type 1 (P = 0·005). Some 30 of 32 patients with AIP type 2 were found to have a localized tumour-like pancreatic mass and underwent pancreatectomy, compared with only 16 of 40 with type 1 (P < 0·001). Three of 25 patients with AIP type 2 presented with raised serum levels of IgG4 compared with 21 of 38 with type 1 (P < 0·001). There was no difference in symptoms and involvement of other organs between AIP types 1 and 2. Presentation with weight loss was more common among patients with PDAC than those with AIP, but there was no difference in pain or jaundice between the groups. Raised serum carbohydrate antigen 19-9 levels were more prevalent in patients with PDAC.
Conclusion
Patients with AIP type 2 frequently present with abdominal pain and a tumour-like mass. Differentiating AIP from PDAC is difficult, so making the clinical decision regarding operative versus conservative management is challenging.
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Affiliation(s)
- S Fritz
- Department of General, Visceral and Transplantation Surgery, University of Heidelberg, Heidelberg, Germany
| | - F Bergmann
- Department of Pathology, University of Heidelberg, Heidelberg, Germany
| | - L Grenacher
- Diagnostic and Interventional Radiology, University of Heidelberg, Heidelberg, Germany
| | - M Sgroi
- Department of General, Visceral and Transplantation Surgery, University of Heidelberg, Heidelberg, Germany
| | - U Hinz
- Department of General, Visceral and Transplantation Surgery, University of Heidelberg, Heidelberg, Germany
| | - T Hackert
- Department of General, Visceral and Transplantation Surgery, University of Heidelberg, Heidelberg, Germany
| | - M W Büchler
- Department of General, Visceral and Transplantation Surgery, University of Heidelberg, Heidelberg, Germany
| | - J Werner
- Department of General, Visceral and Transplantation Surgery, Ludwig-Maximilians University of Munich, Munich, Germany
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Immunoglobulin G4-related pancreatic and biliary diseases. CANADIAN JOURNAL OF GASTROENTEROLOGY = JOURNAL CANADIEN DE GASTROENTEROLOGIE 2014; 27:523-30. [PMID: 24078937 DOI: 10.1155/2013/180461] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
BACKGROUND Autoimmune pancreatitis and autoimmune cholangitis are new clinical entities that are now recognized as the pancreatico-biliary manifestations of immunoglobulin (Ig) G4-related disease. OBJECTIVE To summarize important clinical aspects of IgG4-related pancreatic and biliary diseases, and to review the role of IgG4 in the diagnosis of autoimmune pancreatitis (AIP) and autoimmune cholangitis (AIC). METHODS A narrative review was performed using the PubMed database and the following keywords: "IgG4", "IgG4 related disease", "autoimmune pancreatitis", "sclerosing cholangitis" and "autoimmune cholangitis". A total of 955 articles were retrieved; of these, 381 contained relevant data regarding the IgG4 molecule, pathogenesis of IgG-related diseases, and diagnosis, management and long-term follow-up for patients with AIP and AIC. Of these 381 articles, 66 of the most pertinent were selected. RESULTS The selected studies demonstrated the increasing clinical importance of both AIP and AIC, which can mimic pancreatic cancer and cholangiocarcinoma, respectively. IgG4 titration in tissue or blood cannot be used alone to diagnose all IgG4-related diseases; however, it is often a useful adjunct to clinical, radiological and histological features. AIP and AIC respond to steroids; however, relapse is common and long-term maintenance treatment often required. CONCLUSIONS A review of the diagnosis and management of both AIC and AIP is timely and pertinent to clinical practice because the amount of information regarding these conditions has increased substantially in the past few years, resulting in significant impact on the clinical management of affected patients.
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Rebours V. [Acute pancreatitis: an overview of the management]. Rev Med Interne 2014; 35:649-55. [PMID: 24837648 DOI: 10.1016/j.revmed.2014.04.009] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2014] [Accepted: 04/15/2014] [Indexed: 12/18/2022]
Abstract
Over the past decades, the incidence and the number of hospital admissions for acute pancreatitis have increased in the Western countries. The two most common etiological factors of acute pancreatitis are gallstones (including small gallstones or microlithiasis) and alcohol abuse. Acute pancreatitis is associated with a significant mortality (4-10%) and 25% in case of pancreatic necrosis, especially. Edematous pancreatitis is benign and oral feeding can be restarted once abdominal pain is decreasing and inflammatory markers are improving. Enteral tube feeding should be the primary therapy in patients with predicted severe acute pancreatitis who require nutritional support. Enteral nutrition in acute pancreatitis can be administered via either the nasojejunal or nasogastric route. In case of necrosis, preventive antibiotics are not recommended. The single indication is infected necrosis confirmed by fine needle aspiration. The incidence trends of acute pancreatitis possibly reflect a change in the prevalence of main etiological factors (e.g. gallstones and alcohol consumption) and cofactors such as tobacco, obesity and genetic susceptibility. Priority is to search for associated causes, especially in cases with atypical symptoms. In case of first acute pancreatitis in patients older than 50 years, the presence of a tumor (benign or malignant) has to be specifically ruled out, using CT-scan, MRI and endoscopic ultrasound.
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Affiliation(s)
- V Rebours
- Service de pancréatologie, gastro-entérologie, hôpital Beaujon, 100, boulevard du Général-Leclerc, 92110 Clichy, France.
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Vaquero EC, Salcedo MT, Cuatrecasas M, De León H, Merino X, Navarro S, Ginès A, Abu-Suboh M, Balsells J, Fernández-Cruz L, Molero X. Autoimmune pancreatitis type-1 associated with intraduct papillary mucinous neoplasm: report of two cases. Pancreatology 2014; 14:316-8. [PMID: 25062884 DOI: 10.1016/j.pan.2014.04.032] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/26/2014] [Revised: 03/31/2014] [Accepted: 04/28/2014] [Indexed: 12/11/2022]
Abstract
Chronic pancreatitis lesions usually embrace both intraduct papillary mucinous neoplasm (IPMN) and pancreatic ductal adenocarcinoma (PDAC). Patients at genetically-determined high risk of PDAC often harbor IPMN and/or chronic pancreatitis, suggesting IPMN, chronic pancreatitis and PDAC may share pathogenetic mechanisms. Chronic autoimmune pancreatitis (AIP) may also herald PDAC. Concurrent IPMN and AIP have been reported in few patients. Here we describe two patients with IPMN who developed type-1 AIP fulfilling the Honolulu and Boston diagnostic criteria. AIP diffusively affected the whole pancreas, as well as peripancreatic lymph nodes and the gallbladder. Previous pancreatic resection of focal IPMN did not show features of AIP. One of the patients carried a CFTR class-I mutation. Of notice, serum IgG4 levels gradually decreased to normal values after IPMN excision. Common risk factors to IPMN and AIP may facilitate its coincidental generation.
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Affiliation(s)
- Eva C Vaquero
- Department of Gastroenterology, Institut de Malalties Digestives i Metabòliques, Hospital Clínic, CIBEREHD, IDIBAPS, Barcelona, Spain
| | - Maria T Salcedo
- Department of Pathology, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
| | - Míriam Cuatrecasas
- Department of Pathology, Centre de Diagnòstic Biomèdic (CDB), Hospital Clínic, University of Barcelona and Banc de Tumors-Biobanc Clinic-IDIBAPS-XBTC, Barcelona, Spain
| | - Hannah De León
- Exocrine Pancreatic Diseases Research Group, Hospital Universitari Vall d'Hebron, Institut de Recerca (VHIR), Universitat Autònoma de Barcelona, CIBEREHD, Barcelona, Spain
| | - Xavier Merino
- Department of Radiology, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
| | - Salvador Navarro
- Department of Gastroenterology, Institut de Malalties Digestives i Metabòliques, Hospital Clínic, CIBEREHD, IDIBAPS, Barcelona, Spain
| | - Angels Ginès
- Department of Gastroenterology, Institut de Malalties Digestives i Metabòliques, Hospital Clínic, CIBEREHD, IDIBAPS, Barcelona, Spain
| | - Monder Abu-Suboh
- Department of Endoscopy, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
| | - Joaquim Balsells
- Department of Surgery, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
| | - Laureano Fernández-Cruz
- Department of Surgery, Institut de Malalties Digestives i Metabòliques, Hospital Clínic, CIBEREHD, IDIBAPS, Barcelona, Spain
| | - Xavier Molero
- Exocrine Pancreatic Diseases Research Group, Hospital Universitari Vall d'Hebron, Institut de Recerca (VHIR), Universitat Autònoma de Barcelona, CIBEREHD, Barcelona, Spain.
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