1
|
Wen H, Niu X, Yu R, Zhao R, Wang Q, Sun N, Ma L, Li Y. Association of Serum AGR With All-Cause and Cause-Specific Mortality Among Individuals With Diabetes. J Clin Endocrinol Metab 2025; 110:e266-e275. [PMID: 38571296 DOI: 10.1210/clinem/dgae215] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/26/2023] [Revised: 03/14/2024] [Accepted: 04/02/2024] [Indexed: 04/05/2024]
Abstract
CONTEXT There are insufficient data to support a link between serum albumin-to-globulin ratio (AGR) and mortality in individuals with diabetes. OBJECTIVE This prospective study sought to investigate the relationship between serum AGR and all-cause and cause-specific mortality in adult diabetics. METHODS This study included 8508 adults with diabetes from the National Health and Nutrition Examination Survey (NHANES) from 1999 to 2018. Death outcomes were ascertained by linkage to National Death Index records through December 31, 2019. Hazard ratios (HR) and 95% CIs for mortality from all causes, cardiovascular disease (CVD), and cancer were estimated using weighted Cox proportional-hazards models. RESULTS A total of 2415 all-cause deaths, including 688 CV deaths and 413 cancer deaths, were recorded over an average of 9.61 years of follow-up. After multivariate adjustment, there was a significant and linear relationship between higher serum AGR levels and reduced all-cause and cause-specific mortality in a dose-response manner. The multivariate-adjusted HR and 95% CI for all-cause mortality (Ptrend < .0001), cardiovascular mortality (Ptrend < .001), and cancer mortality (Ptrend < .01) were 0.51 (0.42-0.60), 0.62 (0.46-0.83), and 0.57 (0.39-0.85), respectively, for individuals in the highest AGR quartile. There was a 73% decreased risk of all-cause death per 1-unit rise in natural log-transformed serum AGR, as well as a 60% and 63% decreased risk of mortality from CVD and cancer, respectively (all P < .001). Both the stratified analysis and the sensitivity analyses revealed the same relationships. CONCLUSION AGR is a promising biomarker in risk predictions for long-term mortality in diabetic individuals, particularly in those younger than 60 years and heavy drinkers.
Collapse
Affiliation(s)
- He Wen
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
- School of Public Health, Xi'an Jiaotong University Health Science Center, Xi'an, Shaanxi Province, 710061 China
| | - Xiaona Niu
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
| | - Rui Yu
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
| | - Ran Zhao
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
| | - Qiuhe Wang
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
| | - Nan Sun
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
| | - Le Ma
- School of Public Health, Xi'an Jiaotong University Health Science Center, Xi'an, Shaanxi Province, 710061 China
- Key Laboratory of Environment and Genes Related to Diseases (Xi'an Jiaotong University), Ministry of Education of China, Xi'an, Shaanxi Province, 710061 China
| | - Yan Li
- Department of Cardiology, Tangdu Hospital, Second Affiliated Hospital of Air Force Military Medical University, Xi'an City, Shaanxi Province, 710038 China
| |
Collapse
|
2
|
Thapaliya I, Yadav J. Hypocalcaemic tetany linked to vitamin D deficiency and hypomagnesemia in primary intestinal lymphangiectasia: a literature review. Ann Med Surg (Lond) 2024; 86:2049-2057. [PMID: 38576918 PMCID: PMC10990415 DOI: 10.1097/ms9.0000000000001850] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/16/2023] [Accepted: 02/11/2024] [Indexed: 04/06/2024] Open
Abstract
Primary intestinal lymphangiectasia (PIL) is a rare disorder in children causing protein-losing enteropathy. Vitamin D deficiency and hypomagnesemia contributed to the tetany. The literature review reflects the importance of screening for these deficiencies and regular serum magnesium monitoring in PIL cases with neuromuscular or ionic abnormalities.
Collapse
|
3
|
Terbah R, Testro A, Gow P, Majumdar A, Sinclair M. Portal Hypertension in Malnutrition and Sarcopenia in Decompensated Cirrhosis-Pathogenesis, Implications and Therapeutic Opportunities. Nutrients 2023; 16:35. [PMID: 38201864 PMCID: PMC10780673 DOI: 10.3390/nu16010035] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/03/2023] [Revised: 12/18/2023] [Accepted: 12/19/2023] [Indexed: 01/12/2024] Open
Abstract
Malnutrition and sarcopenia are highly prevalent in patients with decompensated cirrhosis and are associated with poorer clinical outcomes. Their pathophysiology is complex and multifactorial, with protein-calorie malnutrition, systemic inflammation, reduced glycogen stores and hormonal imbalances all well reported. The direct contribution of portal hypertension to these driving factors is however not widely documented in the literature. This review details the specific mechanisms by which portal hypertension directly contributes to the development of malnutrition and sarcopenia in cirrhosis. We summarise the existing literature describing treatment strategies that specifically aim to reduce portal pressures and their impact on nutritional and muscle outcomes, which is particularly relevant to those with end-stage disease awaiting liver transplantation.
Collapse
Affiliation(s)
- Ryma Terbah
- Liver Transplant Unit, Austin Health, 145 Studley Road, Heidelberg, VIC 3084, Australia; (R.T.); (A.T.); (P.G.); (A.M.)
- Department of Medicine, The University of Melbourne, Parkville, VIC 3050, Australia
| | - Adam Testro
- Liver Transplant Unit, Austin Health, 145 Studley Road, Heidelberg, VIC 3084, Australia; (R.T.); (A.T.); (P.G.); (A.M.)
- Department of Medicine, The University of Melbourne, Parkville, VIC 3050, Australia
| | - Paul Gow
- Liver Transplant Unit, Austin Health, 145 Studley Road, Heidelberg, VIC 3084, Australia; (R.T.); (A.T.); (P.G.); (A.M.)
- Department of Medicine, The University of Melbourne, Parkville, VIC 3050, Australia
| | - Avik Majumdar
- Liver Transplant Unit, Austin Health, 145 Studley Road, Heidelberg, VIC 3084, Australia; (R.T.); (A.T.); (P.G.); (A.M.)
- Department of Medicine, The University of Melbourne, Parkville, VIC 3050, Australia
| | - Marie Sinclair
- Liver Transplant Unit, Austin Health, 145 Studley Road, Heidelberg, VIC 3084, Australia; (R.T.); (A.T.); (P.G.); (A.M.)
- Department of Medicine, The University of Melbourne, Parkville, VIC 3050, Australia
| |
Collapse
|
4
|
Wang J, Chen L, Huang Z, Lu J, Yang Y, Zhao X, Tu J, Pan Y, Bao K, Chen W, Xiu J, Liu Y, Chen L, Chen S, Chen K. A Synergistic Association Between Inflammation, Malnutrition, and Mortality in Patients With Diabetics. Front Nutr 2022; 9:872512. [PMID: 35719150 PMCID: PMC9201752 DOI: 10.3389/fnut.2022.872512] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/09/2022] [Accepted: 04/04/2022] [Indexed: 11/20/2022] Open
Abstract
Background Although inflammation is a known predictor for poor prognosis in patients with diabetics, few data report the synergistic association between inflammation, malnutrition, and mortality in patients with diabetics. We aim to explore whether malnutrition modifies the predictor of inflammation on prognosis. Methods Nutritional status and inflammation were measured in 6,682 patients with diabetics undergoing coronary angiography or percutaneous coronary intervention between January 2007 to December 2018 from Cardiorenal Improvement Registry. Malnutrition was defined as Controlling Nutritional Status (CONUT) score, which was more than 1. High-sensitivity C-reactive protein (hs-CRP) exceeding the median was assessed as a high-risk inflammation. Cox regression models were used to estimate hazard ratios (HR) for mortality across combined hs-CRP and CONUT score categories. Results During a median follow-up of 5.0 years (interquartile range: 3.0–7.6 years), 759 (11.36%) patients died. The mortality of the four groups (normal nutrition and low hs-CRP level; normal nutrition and high hs-CRP level; malnutrition and low hs-CRP level; and malnutrition and high hs-CRP level) were 7.29, 7.12, 10.71, and 17.31%, respectively. Compared with normal nutrition and low hs-CRP level, an isolated condition of either malnutrition or high hs-CRP level was not associated with any significant risk for all-cause mortality. However, concomitant presence of both high hs-CRP level and malnutrition condition was associated with a significantly increased risk of all-cause mortality (HR: 1.51; 95% CI: 1.20–1.89; p < 0.001). The p-value for interaction between nutritional status and hs-CRP level on all-cause mortality was 0.03. Conclusion The interplay of inflammation and malnutrition in patients with diabetics significantly amplifies the deleterious effects of each as distinct disease entities. A prospective randomized clinical trial is needed in the future to verify the results.
Collapse
Affiliation(s)
- Junjie Wang
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Liling Chen
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Zhidong Huang
- Department of Cardiology, Guangdong Provincial Key Laboratory of Coronary Heart Disease Prevention, Guangdong Cardiovascular Institute, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangzhou, China
| | - Jin Lu
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Yanfang Yang
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Xiaoli Zhao
- Department of Cardiology, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, China
| | - Jiabin Tu
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Yuxiong Pan
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Kunming Bao
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Weihua Chen
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Jiaming Xiu
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
| | - Yong Liu
- Department of Cardiology, Guangdong Provincial Key Laboratory of Coronary Heart Disease Prevention, Guangdong Cardiovascular Institute, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangzhou, China
| | - Longtian Chen
- Department of Hematology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
- *Correspondence: Longtian Chen
| | - Shiqun Chen
- Department of Cardiology, Guangdong Provincial Key Laboratory of Coronary Heart Disease Prevention, Guangdong Cardiovascular Institute, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangzhou, China
- Shiqun Chen
| | - Kaihong Chen
- Department of Cardiology, Longyan First Affiliated Hospital of Fujian Medical University, Longyan, China
- Kaihong Chen
| |
Collapse
|
5
|
Lopez RN, Day AS. Primary intestinal lymphangiectasia in children: A review. J Paediatr Child Health 2020; 56:1719-1723. [PMID: 32463559 DOI: 10.1111/jpc.14837] [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: 02/06/2020] [Revised: 02/15/2020] [Accepted: 02/17/2020] [Indexed: 02/06/2023]
Abstract
Primary intestinal lymphangiectasia is an uncommon condition that usually presents early in childhood. This incurable condition is consequent to underlying lymphatic abnormalities that lead to loss of lymphatic contents into the intestinal lumen. This article outlines an approach to the assessment of children presenting with characteristic features and consideration of other conditions that could lead to enteric protein loss. An overview of the management of primary intestinal lymphangiectasia is outlined.
Collapse
Affiliation(s)
- Robert N Lopez
- Department of Gastroenterology, Hepatology and Liver Transplantation, Queensland Children's Hospital, Brisbane, Queensland, Australia
| | - Andrew S Day
- Department of Paediatrics, University of Otago, Christchurch, New Zealand.,Department of Paediatrics, Christchurch Hospital, Christchurch, New Zealand
| |
Collapse
|
6
|
Abstract
PURPOSE OF REVIEW The present review offers its readers a practical overview of protein-losing enteropathy, particularly with regard to diagnostic and therapeutic approaches. The aim is to support clinicians in their daily practice with a practical tool to deal with protein-losing enteropathy. RECENT FINDINGS The literature covering protein-losing enteropathy does not appear to be quite recent and also guidelines are scanty. The main innovations during the last decade probably regard the introduction of enteroscopic techniques in the diagnostic flowchart. The use of video-capsule and device-assisted enteroscopy has enabled the direct exploration of the small bowel and the identification of the damage causing the loss of proteins from the gastrointestinal tract. Other innovations are to do with the therapies of the disorder underlying protein-losing enteropathy, although the support with nutritional supplementation are the direct remedies to tackle the protein loss. SUMMARY Protein-losing enteropathy represents an important clinical aspect of different gastrointestinal and extra-intestinal diseases. An established flowchart is still unavailable, but the use of enteroscopy has deeply changed the modern diagnostic approach. Nutritional support and therapy of the underlying disease are pivotal to patients' management.
Collapse
|
7
|
Fonollosa A, Valcarcel M, Salado C, Pereiro X, Vecino E. Effect of somatostatin on human retinal pigment epithelial cells permeability. Exp Eye Res 2019; 184:15-23. [PMID: 30978347 DOI: 10.1016/j.exer.2019.04.007] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/01/2018] [Revised: 03/05/2019] [Accepted: 04/05/2019] [Indexed: 11/28/2022]
Abstract
PURPOSE To assess the effect of somatostatin (SST) on the permeability of human retinal pigment epithelial cells. METHODS We conducted two experiments, exposing cells from human-fetal retinal pigment epithelium (hfRPE) cultures to vascular endothelial growth factor (VEGF), with or without SST pretreatment, in one, and to hypoxic conditions, again with or without SST pretreatment, in the other. The paracellular permeability of hfRPE was assessed by measuring transepithelial electrical resistance (TER) and fluorescein isothiocyanate-sodium (FITC-sodium) flux. Immunochemistry analysis was used to assess the expression of occludin and Zonula occludens-1(ZO-1). RESULTS Both VEGF and hypoxia increased permeability of the hfRPE, as measured by TER and tracer flux, and decreased occludin and ZO-1staining, as measured by immunochemistry. Pretreatment of cultures with SST partially counteracted these effects. CONCLUSIONS Somatostatin may play a role in the regulation of permeability across retinal pigment epithelium. It may act as an anti-permeability factor in the retina through the enhancement of tight junction function.
Collapse
Affiliation(s)
- Alex Fonollosa
- Begiker-Ophthalmology Research Group, Department of Ophthalmology, Faculty of Medicine and Nursing, BioCruces Health Research Institute, Cruces Hospital, University of the Basque Country UPV/EHU, Bilbao, Spain; Experimental Ophthalmo-Biology Group, Department Cell Biology and Histology, Faculty of Medicine and Nursing, University of the Basque Country UPV/EHU, Leioa, Spain.
| | - Maria Valcarcel
- Innoprot SL, Parque Científico y Tecnológico de Bizkaia, Derio, Spain
| | - Clarisa Salado
- Innoprot SL, Parque Científico y Tecnológico de Bizkaia, Derio, Spain
| | - Xandra Pereiro
- Experimental Ophthalmo-Biology Group, Department Cell Biology and Histology, Faculty of Medicine and Nursing, University of the Basque Country UPV/EHU, Leioa, Spain
| | - Elena Vecino
- Experimental Ophthalmo-Biology Group, Department Cell Biology and Histology, Faculty of Medicine and Nursing, University of the Basque Country UPV/EHU, Leioa, Spain
| |
Collapse
|
8
|
Abstract
Protein-losing enteropathy (PLE) is a rare complication of intestinal diseases. Its main manifestation is hypoproteinemic edema. The diagnosis of PLE is based on the verification of protein loss into the intestinal lumen, by determining fecal α1-antitrypsin concentration and clearance. The localization of the affected colonic segment is clarified using radiologic and endoscopic techniques. The mainstay of treatment for PLE is a fat-free diet enriched with medium-chain triglycerides. Surgical resection of the affected segment of the colon may be the treatment of choice for severe hypoproteinemia resistant to drug therapy.
Collapse
Affiliation(s)
- A I Parfenov
- Moscow Clinical Research and Practical Center, Moscow Healthcare Department, Moscow, Russia
| | - L M Krums
- Moscow Clinical Research and Practical Center, Moscow Healthcare Department, Moscow, Russia
| |
Collapse
|
9
|
Lee SJ, Song HJ, Boo SJ, Na SY, Kim HU, Hyun CL. Primary intestinal lymphangiectasia with generalized warts. World J Gastroenterol 2015; 21:8467-8472. [PMID: 26217101 PMCID: PMC4507119 DOI: 10.3748/wjg.v21.i27.8467] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/22/2015] [Revised: 02/25/2015] [Accepted: 04/17/2015] [Indexed: 02/06/2023] Open
Abstract
Primary intestinal lymphangiectasia (PIL) is a rare protein-losing enteropathy with lymphatic leakage into the small intestine. Dilated lymphatics in the small intestinal wall and mesentery are observed in this disease. Laboratory tests of PIL patients revealed hypoalbuminemia, lymphocytopenia, hypogammaglobulinemia and increased stool α-1 antitrypsin clearance. Cell-mediated immunodeficiency is also present in PIL patients because of loss of lymphocytes. As a result, the patients are vulnerable to chronic viral infection and lymphoma. However, cases of PIL with chronic viral infection, such as human papilloma virus-induced warts, are rarely reported. We report a rare case of PIL with generalized warts in a 36-year-old male patient. PIL was diagnosed by capsule endoscopy and colonoscopic biopsy with histological tissue confirmation. Generalized warts were observed on the head, chest, abdomen, back, anus, and upper and lower extremities, including the hands and feet of the patient.
Collapse
|
10
|
Troskot R, Jurčić D, Bilić A, Gomerčić Palčić M, Težak S, Brajković I. How to treat an extensive form of primary intestinal lymphangiectasia? World J Gastroenterol 2015; 21:7320-7325. [PMID: 26109821 PMCID: PMC4476896 DOI: 10.3748/wjg.v21.i23.7320] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/10/2015] [Revised: 02/27/2015] [Accepted: 03/31/2015] [Indexed: 02/07/2023] Open
Abstract
We report a case of a 42-year-old man with a rare disorder known as primary intestinal lymphangiectasia, which is characterized by dilated intestinal lymphatics that lead to the development of protein-losing enteropathy. The patient presented with a grand mal seizure caused by malabsorption-derived electrolytes and a protein disorder. Signs of the disease, including chronic diarrhea and peripheral edema, manifested 10 years ago, but a diagnosis was never made. The diagnosis was suspected because of the clinical manifestations, laboratory tests, imaging and endoscopic findings. Hyperemic and edematous mucosa of the small intestine corresponded to scattered white spots with dilated intestinal lymphatics and whitish villi in the histological specimen of the biopsied jejunal mucosa. Although numerous therapeutic strategies are available, only octreotide therapy proved to be an effective means of therapeutic resolution in this patient. Although the patient had a partial remission following the use of a slow release formula of octreotide, his prognosis, clinical course, and future treatment challenges are yet to be determined.
Collapse
|
11
|
Shin JK, Jung YH, Bae MN, Baek IW, Kim KJ, Cho CS. Successful treatment of protein-losing enteropathy due to AA amyloidosis with octreotide in a patient with rheumatoid arthritis. Mod Rheumatol 2014. [DOI: 10.3109/s10165-012-0675-0] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
|
12
|
Sinani SA, Rawahi YA, Abdoon H. Octreotide in Hennekam syndrome-associated intestinal lymphangiectasia. World J Gastroenterol 2012; 18:6333-6337. [PMID: 23180957 PMCID: PMC3501785 DOI: 10.3748/wjg.v18.i43.6333] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.5] [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
A number of disorders have been described to cause protein losing enteropathy (PLE) in children. Primary intestinal lymphangiectasia (PIL) is one mechanism leading to PLE. Few syndromes are associated with PIL; Hennekam syndrome (HS) is one of them. The principal treatment for PIL is a high protein, low fat diet with medium chain triglycerides supplementation. Supportive therapy includes albumin infusion. Few publications have supported the use of octreotide to diminish protein loss and minimize hypoalbuminemia seen in PIL. There are no publications on the treatment of PIL with octreotide in patients with HS. We report two children with HS and PLE in which we used octreotide to decrease intestinal protein loss. In one patient, octreotide increased serum albumin to an acceptable level without further need for albumin infusions. The other patient responded more dramatically with near normal serum albumin levels and cessation of albumin infusions. In achieving a good response to octreotide in both patients, we add to the publications supporting the use of octreotide in PIL and suggest that octreotide should be tried in patients with PIL secondary to HS. To the best of our knowledge, this is the first case report on the use of octreotide in HS-associated PIL.
Collapse
|
13
|
Jabeen SA, Murthy A, Kandadai RM, Meena AK, Borgohain R, Uppin MS. Cryptoccocal menigitis as a primary manifestation in a patient with intestinal lymphangictasia. Ann Indian Acad Neurol 2012; 15:218-20. [PMID: 22919199 PMCID: PMC3424804 DOI: 10.4103/0972-2327.99725] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/09/2011] [Revised: 08/25/2011] [Accepted: 11/11/2011] [Indexed: 12/19/2022] Open
Abstract
Opportunistic infections usually occur in patients with an immunocompromised state, and can be severe. Cryptoccocal meningitis is a fatal condition if left untreated, and is usually found in such patients. We report the case of an adult patient with cryptoccocal meningitis secondary to intestinal lymphangiectasia. A 30 year old female was admitted to our hospital for meningitis. Biochemical and radiological investigations were performed. A cerebrospinal fluid latex agglutination test showed positive cryptoccocal antigen. In addition, there were features of humoral and cell mediated immunity deficiency (lymphopenia, hypoalbuminemia, hypogammaglobulinemia), with a negative human immunodeficiency virus (HIV) test by enzyme linked immunosorbent assay and polymerase chain reaction. An upper gastroduodenoscopy was performed, which showed multiple lymphangiectasias, and a biopsy confirmed the diagnosis of primary intestinal lymphangiectasia (PIL). The patient was treated with intravenous amphotericin B and oral flucytosine, and the meningitis resolved. PIL should be suspected in patients with cryptoccocal meningitis, combined with humoral and cell mediated immunity with a negative HIV test. The management issues, in addition to antifungal therapy, include nutritional supplements for the protein losing enteropathy.
Collapse
Affiliation(s)
- Shaik Afshan Jabeen
- Department of Neurology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh, India
| | | | | | | | | | | |
Collapse
|
14
|
Dahlqvist GE, Jamar F, Zech F, Geubel AP. In-111 transferrin scintigraphy in cirrhosis with hypoalbuminemia: evidence for protein-losing enteropathy in a small group of selected cases. Scand J Gastroenterol 2012; 47:1247-52. [PMID: 22989334 DOI: 10.3109/00365521.2012.696682] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Abstract
BACKGROUND Hypoalbuminemia commonly observed in cirrhosis is considered to be mainly related to hepatocellular dysfunction. However, the correlation between the decrease in serum albumin and liver function is far from linear and arguments in favor of an additive role of protein-losing enteropathy have been brought by a few studies. AIM To assess the potential role of protein-losing enteropathy in a group of patients with cirrhosis, portal hypertension, and hypoalbuminemia. DESIGN AND METHODS Eleven patients with documented cirrhosis, portal hypertension, and a low serum albumin level compared to liver function underwent an (111)In-transferrin scintigraphy. RESULTS Using this sensitive method of investigation, nine exhibited features suggestive of exudative enteropathy. Serum albumin level and digestive protein loss were even correlated (Pearson's coefficient = -0.529, one-sided p = 0.047). Protein loss were however not correlated with the degree of portal hypertension or with the extent of liver dysfunction evaluated by the aminopyrine breath test. CONCLUSIONS Our preliminary data obtained in a small group of selected patients with cirrhosis, portal hypertension, and hypoalbuminemia indicate that protein-losing enteropathy may represent an appreciable and underestimated cause of hypoproteinemia.
Collapse
Affiliation(s)
- Géraldine E Dahlqvist
- Department of Hepato-gastroenterology, St Luc University Hospital, Université Catholique de Louvain, Brussels, Belgium
| | | | | | | |
Collapse
|
15
|
Shin JK, Jung YH, Bae MN, Baek IW, Kim KJ, Cho CS. Successful treatment of protein-losing enteropathy due to AA amyloidosis with octreotide in a patient with rheumatoid arthritis. Mod Rheumatol 2012; 23:406-11. [PMID: 22815005 DOI: 10.1007/s10165-012-0675-0] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2012] [Accepted: 05/08/2012] [Indexed: 12/20/2022]
Abstract
Protein-losing enteropathy (PLE) is a rare syndrome of gastrointestinal protein loss that may complicate a variety of diseases. This excessive protein loss across the gut epithelium can be explained by several mechanisms, such as augmentation of the intestinal mucosal capillary permeability, mucosal disruption, intestinal or mesenteric vasculitis, and lymphangiectasia. However, these pathophysiologic alterations of the gut are closely linked to the underlying cause, and primary treatment for PLE should be directed at the underlying condition. Here, we report a female patient with rheumatoid arthritis who developed severe PLE due to AA amyloidosis and was successfully treated with octreotide. She had been suffered from rheumatoid arthritis for 18 years, and her arthritic symptoms at the time of presentation were not definite but manifested as severe diarrhea and general edema with hypoalbuminemia. PLE due to gastrointestinal amyloidosis was confirmed by increased fecal α1-antitrypsin clearance and a colonoscopic biopsy that was positive for amyloid deposits. The diarrhea dissipated with conventional treatment, but the general edema resolved only after introducing a long-acting somatostatin analog (octreotide), along with a gradual recovery of the serum albumin level. This case teaches us that in the case of PLE due to AA amyloidosis that is refractory to conventional treatment, the administration of octreotide should be considered.
Collapse
Affiliation(s)
- Jin-Kyeong Shin
- Division of Rheumatology, Department of Internal Medicine, College of Medicine, Yeouido St. Mary's Hospital, The Catholic University of Korea, 62 Yeouido-dong, Yeongdeungpo-ku, Seoul, Korea, Republic of Korea
| | | | | | | | | | | |
Collapse
|
16
|
Octreotide management of intestinal lymphangiectasia in a teenage heart transplant patient. J Pediatr Gastroenterol Nutr 2012; 54:824-7. [PMID: 21768882 DOI: 10.1097/mpg.0b013e31822d2dd4] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/17/2022]
|
17
|
Abstract
Protein-losing enteropathy (PLE) is a debilitating potential complication of ulcerative colitis (UC). We report a case of PLE in a 26-year-old male patient with UC. The patient lost 50 pounds in the setting of a UC flare and was found to have an albumin level of 1.2 g/dl. Although the patient's UC was clinically controlled with steroids, the weight loss and hypoalbuminemia persisted with the patient's course complicated by development of deep vein thrombosis and pulmonary embolism. The diagnosis of PLE was confirmed with measurement of stool alpha-1-antitrypsin clearance. The patient's condition significantly improved following procto-colectomy.
Collapse
Affiliation(s)
- Ryan Ungaro
- Department of Internal Medicine, The Mount Sinai School of Medicine, New York, N.Y., USA
| | - Mark W. Babyatsky
- Department of Internal Medicine, The Mount Sinai School of Medicine, New York, N.Y., USA
| | - Hongfa Zhu
- Department of Pathology, The Mount Sinai School of Medicine, New York, N.Y., USA
| | - Jeffrey S. Freed
- Department of Surgery, The Mount Sinai School of Medicine, New York, N.Y., USA
| |
Collapse
|
18
|
Lee HJ, Rha MY, Cho YY, Kim ER, Chang DK. A Case of Protein Supplement Effect in Protein-Losing Enteropathy. Clin Nutr Res 2012; 1:94-8. [PMID: 23430393 PMCID: PMC3572805 DOI: 10.7762/cnr.2012.1.1.94] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/05/2012] [Revised: 07/12/2012] [Accepted: 07/13/2012] [Indexed: 12/14/2022] Open
Abstract
The objective of this article is to report improvement of nutritional status by protein supplements in the patient with protein-losing enteropathy. The patient was a female whose age was 25 and underwent medical treatment of Crohn's disease, an inflammatory bowl disease, after diagnosis of cryptogenic multifocal ulcerous enteritis. The weight was 33.3 kg (68% of IBW) in the severe underweight and suffered from ascites and subcutaneous edema with hypoalbuminemia (1.3 g/dL) at the time of hospitalization. The patient consumed food restrictively due to abdominal discomfort. Despite various attempts of oral feeding, the levels of calorie and protein intake fell into 40-50% of the required amount, which was 800-900 kcal/d (24-27 kcal/kg/d) for calorie and 34 g/d (1 g/kg/d) for protein. It was planned to supplement the patient with caloric supplementation (40-50 kcal/kg) and protein supplementation (2.5 g/kg) to increase body weight and improve hypoproteinemia. It was also planned to increase the level of protein intake slowly to target 55 g/d in about 2 weeks starting from 10 g/d and monitored kidney load with high protein supplementation. The weight loss was 1.0 kg when the patient was discharged from the hospital (hospitalization periods of 4 weeks), however, serum albumin was improved from 1.3 g/dL to 2.5 g/dL and there was no abdominal discomfort. She kept supplement of protein at 55 g/d for 5 months after the discharge from the hospital and kept it at 35 g/d for about 2 months and then 25 g/d. The body weight increased gradually from 32.3 kg (65% of IBW) to 44.0 kg (89% of IBW) by 36% for the period of F/u and serum albumin was kept above 2.8 g/dL without intravenous injection of albumin. The performance status was improved from 4 points of 'very tired' to 2 points of 'a little tired' out of 5-point scale measurement and the use of diuretic stopped from the time of 4th month after the discharge from the hospital owing to improvement in edema and ascites. During this period, the results of blood test such as BUN, Cr, and electrolytes were within the normal range. In conclusion, hypoproteinemia
and weight loss were improved by increasing protein intake through utilization of protein supplements in protein-losing enteropathy.
Collapse
Affiliation(s)
- Hyun Jeong Lee
- Department of Dietetics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul 135-710, Korea
| | - Mi Yong Rha
- Department of Dietetics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul 135-710, Korea
| | - Young Yun Cho
- Department of Dietetics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul 135-710, Korea
| | - Eun Ran Kim
- Division of Gastroenterology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul 135-710, Korea
| | - Dong Kyung Chang
- Division of Gastroenterology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul 135-710, Korea
| |
Collapse
|
19
|
Abstract
Protein-losing enteropathy (PLE) is a rare syndrome of gastrointestinal protein loss that may complicate a variety of diseases. The primary causes can be divided into erosive gastrointestinal disorders, nonerosive gastrointestinal disorders, and disorders involving increased central venous pressure or mesenteric lymphatic obstruction. The diagnosis of PLE should be considered in patients with hypoproteinemia after other causes, such as malnutrition, proteinuria, and impaired protein synthesis due to cirrhosis, have been excluded. The diagnosis of PLE is most commonly based on the determination of fecal alpha-1 antitrypsin clearance. Treatment of PLE targets the underlying disease but also includes dietary modification, supportive care, and maintenance of nutritional status. In this article, cases illustrating a variety of clinical presentations and etiologies of PLE are presented, and its diagnostic approach and treatment are reviewed.
Collapse
|
20
|
Abstract
Intestinal lymphangiectasia (IL) is a potentially fatal disorder of the lymphatic system if it is not recognized and proper treatment initiated. The disease is characterized by lymphocytopenia, peripheral edema, and hypoalbuminemia. Because IL is a rare disease, the symptoms, diagnostic workup, and treatment are unfamiliar to many clinicians. Current literature documents only a few reported cases of IL in a preterm infant. This case report of a preterm infant reviews history, symptomatology, and the diagnostic workup performed. The steps in making the diagnosis, the treatment, and the prognosis of this condition are also presented.
Collapse
MESH Headings
- Diagnosis, Differential
- Female
- Humans
- Infant, Newborn
- Infant, Premature
- Infant, Premature, Diseases/diagnosis
- Infant, Premature, Diseases/nursing
- Intensive Care Units, Neonatal
- Lymphangiectasis, Intestinal/diagnosis
- Lymphangiectasis, Intestinal/nursing
- Neonatal Nursing/methods
Collapse
|
21
|
First description of three patients with multifocal lymphangiomatosis and protein-losing enteropathy following palliation of complex congenital heart disease with total cavo-pulmonary connection. Pediatr Cardiol 2008; 29:771-4. [PMID: 18188635 DOI: 10.1007/s00246-007-9194-8] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/07/2007] [Revised: 12/09/2007] [Accepted: 12/10/2007] [Indexed: 01/19/2023]
Abstract
The objective was to identify abdominal lymphatic malformations in pediatric patients with protein-losing enteropathy after palliation of complex congenital heart disease with total cavo-pulmonary connection (TCPC). In 2006, we performed complete hemodynamic and laboratory workup and thoracic and abdominal MRT screens in three patients who newly presented with symptoms of protein-losing enteropathy. All three patients, aged 3, 5, and 7 years, showed excellent TCPC hemodynamics with central venous pressures of 10-13 mm Hg. None of the patients had right-to-left overflow. All three patients showed extensive thoracic and mesenterial lymphangiomatosis. One patient died after 18 months of therapy, which included long-term parenteral nutrition, somatostatin, subcutaneous heparin injections, and frequent albumin and immunoglobulin substitution. The other two patients are in stable condition. Lymphangiomatosis might play an unknown role in the pathogenesis of protein-losing enteropathy after TCPC. It remains unclear whether lymphangiomatosis is a primary congenital disease related to the cardiac disease or if it is triggered by repeated surgery or venous congestion. The presence of lymphangiomatosis should be given diagnostic and therapeutic consideration in TCPC patients in the future.
Collapse
|
22
|
Fang YH, Zhang BL, Wu JG, Chen CX. A primary intestinal lymphangiectasia patient diagnosed by capsule endoscopy and confirmed at surgery: A case report. World J Gastroenterol 2007; 13:2263-5. [PMID: 17465517 PMCID: PMC4146860 DOI: 10.3748/wjg.v13.i15.2263] [Citation(s) in RCA: 32] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Intestinal lymphangiectasia (IL) is a rare disease characterized by dilated lymphatic vessles in the intestinal wall and small bowel mesentery which induce loss of protein and lymphocytes into bowel lumen. Because it most often occurs in the intestine and cannot be detected by upper gastroendoscopy or colonoscopy, and the value of common image examinations such as X-ray and computerized tomography (CT) are limited, the diagnosis of IL is difficult, usually needing the help of surgery. Capsule endoscopy is useful in diagnosing intestinal diseases, such as IL. We here report a case of IL in a female patient who was admitted for the complaint of recurrent edema accompanied with diarrhea and abdominal pain over the last twenty years, and aggravated ten days ago. She was diagnosed by M2A capsule endoscopy as a primary IL and confirmed by surgical and pathological examination.
Collapse
Affiliation(s)
- You-Hong Fang
- Department of Gastroenterlogy, The First Affiliated Hospital of College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang Province, China
| | | | | | | |
Collapse
|
23
|
Simó R, Carrasco E, Fonollosa A, García-Arumí J, Casamitjana R, Hernández C. Deficit of somatostatin in the vitreous fluid of patients with diabetic macular edema. Diabetes Care 2007; 30:725-7. [PMID: 17327350 DOI: 10.2337/dc06-1345] [Citation(s) in RCA: 30] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/03/2023]
Affiliation(s)
- Rafael Simó
- Diabetes Research Unit, Endocrinology Division, Hospital Universitari Vall d'Hebron, Pg. Vall d'Hebron 119-129, 08035 Barcelona, Spain.
| | | | | | | | | | | |
Collapse
|
24
|
Robles-Medranda C, Lukashok HP, Novais P, Biccas B, Fogaça H. Chronic diarrhea as first manifestation of liver cirrhosis and hepatocarcinoma in a teenager: a case report and review of the literature. J Pediatr Gastroenterol Nutr 2006; 42:434-6. [PMID: 16641583 DOI: 10.1097/01.mpg.0000189342.38634.ac] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/21/2023]
Affiliation(s)
- Carlos Robles-Medranda
- Gastroenterology Division, University Hospital Clementino Fraga Filho, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.
| | | | | | | | | |
Collapse
|