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George NA, Surendran S, Paulose RR, Pradeep M. Hyperacute reactivation of cytomegalovirus-induced gastroduodenitis during remission induction in a young male patient with granulomatosis with polyangiitis: a case report and review of literature. J Med Case Rep 2025; 19:68. [PMID: 39994816 PMCID: PMC11849236 DOI: 10.1186/s13256-025-05103-7] [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: 09/21/2024] [Accepted: 01/31/2025] [Indexed: 02/26/2025] Open
Abstract
BACKGROUND Cytomegalovirus is a pathogen known to aggravate the inflammatory response in autoimmune diseases via molecular mimicry. Although it is recognized that cytomegalovirus activation can happen during extended but variable periods of immunosuppression (14-90 days), it is rarely reported in conjunction with an acute flare-up of an autoimmune disease. Currently, there is no consensus on cytomegalovirus prophylaxis for patients initiating remission induction. CASE PRESENTATION Here, we present the case of a 31-year-old male patient of South Indian ethnicity, presenting with a 2-month history of fever, conductive hearing loss, and ear discharge. This was associated with symmetrical inflammatory polyarthritis for 1 month, unilateral painful conjunctivitis, and skin erythema for 5 days. Blood analyses showed elevated inflammatory markers; strongly positive anti-proteinase 3 and cytoplasmic antineutrophil cytoplasmic antibody levels; normal procalcitonin and complement levels; and negative anti-myeloperoxidase and perinuclear antineutrophil cytoplasmic antibody levels. A nasal endoscopy revealed a midline granuloma with vasculitis features on biopsy. Imaging revealed pulmonary nodules and otomastoiditis. Now diagnosed with granulomatosis with polyangiitis, the patient developed signs of gastroduodenitis within a day of initiation of immunosuppression with high-dose "pulse" intravenous methylprednisolone. We evaluated him for mesenteric ischemia/gastrointestinal vasculitis. However, the duodenal biopsies from the bleeding ulcers revealed a probable cytomegalovirus infection, confirmed with high serum viral loads. We treated him with a ganciclovir regimen and transitioned him to steroid-sparing immunosuppressant therapy with mycophenolate mofetil, which was selected over cyclophosphamide for its noninferior effectiveness and better safety profile in non-life-threatening granulomatosis with polyangiitis disease. The patient recovered uneventfully and is currently in remission. CONCLUSION Cytomegalovirus reactivation is possible during short-term steroid pulse therapy. Further research is needed to evaluate whether routine cytomegalovirus screening is warranted before starting immunosuppressive treatment with high-dose steroids in autoimmune conditions.
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Affiliation(s)
- Nisha Annie George
- Department of Infectious Diseases and HIV Medicine, University Hospitals of Leicester NHS Trust, Leicester, UK
| | - Sandeep Surendran
- Department of Rheumatology and Clinical Immunology, Amrita Institute of Medical Sciences, Amrita Vishwa Vidyapeetham, Kochi, Kerala, India.
| | - Roopa Rachel Paulose
- Department of Pathology, Amrita Institute of Medical Sciences, Amrita Vishwa Vidyapeetham, Kochi, Kerala, India
| | - Manu Pradeep
- Department of Rheumatology and Clinical Immunology, Amrita Institute of Medical Sciences, Amrita Vishwa Vidyapeetham, Kochi, Kerala, India
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Miller CQ, Saeed OAM, Collins K. Gastrointestinal histoplasmosis complicating pediatric Crohn disease: A case report and review of literature. World J Gastrointest Endosc 2022; 14:648-656. [PMID: 36303809 PMCID: PMC9593511 DOI: 10.4253/wjge.v14.i10.648] [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: 07/17/2022] [Revised: 09/05/2022] [Accepted: 09/21/2022] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Infection with Histoplasma capsulatum (H. capsulatum) can lead to disseminated disease involving the gastrointestinal tract presenting as diffuse abdominal pain and diarrhea which may mimic inflammatory bowel disease (IBD).
CASE SUMMARY We report a case of 12-year-old boy with presumptive diagnosis of Crohn disease (CD) that presented with several months of abdominal pain, weight loss and bloody diarrhea. Colonoscopy showed patchy moderate inflammation characterized by erythema and numerous pseudopolyps involving the terminal ileum, cecum, and ascending colon. Histologic sections from the colon biopsy revealed diffuse cellular infiltrate within the lamina propria with scattered histiocytic aggregates, and occasional non-necrotizing granulomas. Grocott-Gomori’s Methenamine Silver staining confirmed the presence of numerous yeast forms suggestive of Histoplasma spp., further confirmed with positive urine Histoplasma antigen (6.58 ng/mL, range 0.2-20 ng/mL) and serum immunoglobulin G antibodies to Histoplasma (35.9 EU, range 10.0-80.0 EU). Intravenous amphotericin was administered then transitioned to oral itraconazole. Follow-up computed tomography imaging showed a left lower lung nodule and mesenteric lymphadenopathy consistent with disseminated histoplasmosis infection.
CONCLUSION Gastrointestinal involvement with H. capsulatum with no accompanying respiratory symptoms is exceedingly rare and recognition is often delayed due to the overlapping clinical manifestations of IBD. This case illustrates the importance of excluding infectious etiologies in patients with “biopsy-proven” CD prior to initiating immunosuppressive therapies. Communication between clinicians and pathologists is crucial as blood cultures and antigen testing are key studies that should be performed in all suspected cases of histoplasmosis to avoid misdiagnosis and inappropriate treatment.
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Affiliation(s)
- C Quinn Miller
- Department of Pathology, Indiana University School of Medicine, Indianapolis, IN 46202, United States
| | - Omer A M Saeed
- Department of Pathology, Indiana University School of Medicine, Indianapolis, IN 46202, United States
| | - Katrina Collins
- Department of Pathology, Indiana University School of Medicine, Indianapolis, IN 46202, United States
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Shojaei E, Walsh JC, Sangle N, Yan B, Silverman MS, Hosseini-Moghaddam SM. Gastrointestinal Histoplasmosis Mimicking Crohn's Disease. Open Forum Infect Dis 2021; 8:ofab249. [PMID: 34262987 PMCID: PMC8274358 DOI: 10.1093/ofid/ofab249] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/10/2021] [Accepted: 05/13/2021] [Indexed: 11/13/2022] Open
Abstract
Disseminated histoplasmosis is a life-threatening disease usually seen in immunocompromised patients living in endemic areas. We present an apparently immunocompetent patient with gastrointestinal histoplasmosis who was initially diagnosed with biopsy-proven Crohn's disease. Following discontinuation of anti-inflammatory drugs and institution of antifungal therapy, his gastrointestinal illness completely improved. Specific fungal staining should be routinely included in histopathologic assessment of tissue specimens diagnosed as Crohn's disease.
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Affiliation(s)
- Esfandiar Shojaei
- Division of Infectious Diseases, Department of Medicine, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada
| | - Joanna C Walsh
- Department of Pathology and Laboratory Medicine, London Health Sciences Centre, Western University, London, Ontario, Canada
| | - Nikhil Sangle
- Department of Pathology and Laboratory Medicine, London Health Sciences Centre, Western University, London, Ontario, Canada
| | - Brian Yan
- Division of Gastroenterology, Department of Medicine, Schulich School of Medicine & Dentistry, London Health Sciences Centre, Western University, London, Ontario, Canada
| | - Michael S Silverman
- Division of Infectious Diseases, Department of Medicine, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada
- Department of Epidemiology and Biostatistics, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada
| | - Seyed M Hosseini-Moghaddam
- Division of Infectious Diseases, Department of Medicine, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada
- Division of Infectious Diseases, Department of Medicine, University Health Network, University of Toronto, Toronto, Ontario, Canada
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Bhaskaran H, Balan S. Unusual cause for intestinal perforation in juvenile dermatomyositis. BMJ Case Rep 2019; 12:12/8/e229395. [PMID: 31444261 DOI: 10.1136/bcr-2019-229395] [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] [Indexed: 11/04/2022] Open
Abstract
We report a case of juvenile dermatomyositis (JDM) with cytomegalovirus (CMV) colitis which was further complicated with perforation. The patient, a 6-year-old girl, was diagnosed with JDM 1 month prior to the current presentation. After 2 weeks of optimising her treatment with steroid, intravenous Ig and methotrexate, she was readmitted with diffuse abdominal pain. Erect abdominal X-ray revealed gas under diaphragm. An exploratory laparotomy showed perforation of the large intestine. A biopsy showed inclusion bodies of CMV with immunohistochemistry for CMV positive. Strong positive CMV DNA PCR from tissue specimen, positive IgG CMV and negative IgM CMV in blood suggested a reactivation of CMV. The treatment followed included surgery and strategic use of antiviral agents as well as immunomodulators. CMV enteritis with complications should also be suspected in optimally treated autoimmune disease patients, including JDM, when they present with abdominal symptoms.
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Affiliation(s)
- Harikrishnan Bhaskaran
- Department of Clinical Immunology and Rheumatology, Amrita Institute of Medical Sciences and Research Centre, Ernakulam, Kerala, India
| | - Suma Balan
- Department of Clinical Immunology and Rheumatology, Amrita Institute of Medical Sciences and Research Centre, Ernakulam, Kerala, India
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Ikeda K, Nakajima S, Tanji K, Hirai T, Uomori K, Morimoto S, Tomita S, Fukunaga M, Tamura N, Sekigawa I. Intestinal perforation due to hemorrhagic Cytomegalovirus enteritis in a patient with severe uncontrolled lupus nephritis: a case and review of the literature. Rheumatol Int 2017; 37:1395-1399. [PMID: 28283734 DOI: 10.1007/s00296-017-3693-6] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/21/2016] [Accepted: 02/28/2017] [Indexed: 10/20/2022]
Abstract
A 31-year-old woman with systemic lupus erythematosus and lupus nephritis was treated with prednisone and immunosuppressants. After her lupus nephritis symptoms worsened, both high-dose steroid and cyclophosphamide pulse therapy were administered. The patient developed an intestinal perforation, and laparoscopic Hartmann's surgery was performed on the sigmoid colon. Serum Cytomegalovirus (CMV) antigen C7HRP was detected, and the patient was diagnosed with CMV colitis and underwent a colon resection. Severe hematochezia continued despite ganciclovir administration, and the patient underwent laparoscopic total colectomy and partial ileostomy. CMV enteritis should be considered in patients treated with prednisone and immunosuppressants and those who have abdominal pain and hematochezia. Immunocompromised patients with intestinal perforation due to CMV enteritis have a poor prognosis. We report a case with along with the results of a literature review.
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Affiliation(s)
- Keigo Ikeda
- Department of Internal Medicine and Rheumatology, Juntendo University Urayasu Hospital, 2-1-1 Tomioka Urayasu-shi, Chiba, 279-0021, Japan.
| | - Shihoko Nakajima
- Department of Internal Medicine and Rheumatology, Juntendo University School of Medicine, Tokyo, Japan
| | - Kana Tanji
- Department of Internal Medicine and Rheumatology, Juntendo University School of Medicine, Tokyo, Japan
| | - Takuya Hirai
- Department of Internal Medicine and Rheumatology, Juntendo University Urayasu Hospital, 2-1-1 Tomioka Urayasu-shi, Chiba, 279-0021, Japan
| | - Kaori Uomori
- Department of Internal Medicine and Rheumatology, Juntendo University School of Medicine, Tokyo, Japan
| | - Shinji Morimoto
- Department of Internal Medicine and Rheumatology, Juntendo University Urayasu Hospital, 2-1-1 Tomioka Urayasu-shi, Chiba, 279-0021, Japan
| | - Shigeki Tomita
- Department of Pathology, Juntendo University Urayasu Hospital, 2-1-1 Tomioka Urayasu-shi, Chiba, 279-0021, Japan
| | - Masaki Fukunaga
- Department of Surgery, Juntendo University Urayasu Hospital, 2-1-1 Tomioka Urayasu-shi, Chiba, 279-0021, Japan
| | - Naoto Tamura
- Department of Internal Medicine and Rheumatology, Juntendo University School of Medicine, Tokyo, Japan
| | - Iwao Sekigawa
- Department of Internal Medicine and Rheumatology, Juntendo University Urayasu Hospital, 2-1-1 Tomioka Urayasu-shi, Chiba, 279-0021, Japan
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Baniak N, Kanthan R. Cytomegalovirus Colitis: An Uncommon Mimicker of Common Colitides. Arch Pathol Lab Med 2017; 140:854-8. [PMID: 27472242 DOI: 10.5858/arpa.2015-0176-rs] [Citation(s) in RCA: 16] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Cytomegalovirus latency, though ubiquitous in the human population, is known to cause colitis in both immunocompromised and immunocompetent hosts. Furthermore, the clinical, endoscopic, and histologic appearance of cytomegalovirus colitis can mimic that of inflammatory bowel disease, an extremely well-documented disease. In this context, though many reports have looked at inflammatory bowel disease with superimposed cytomegalovirus infection, less attention has been paid to cytomegalovirus as a primary cause of isolated colitis. Owing to the rarity of this phenomenon, it is important to consider this diagnosis and implement proper testing to avoid misdiagnosis and mismanagement.
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Affiliation(s)
| | - Rani Kanthan
- From the Department of Pathology and Laboratory Medicine, College of Medicine, University of Saskatchewan, Saskatoon, Canada
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Abstract
Systemic lupus erythematosus (SLE) is an autoimmune disease characterized by multisystem organ involvement, heterogeneity of clinical features, and variety in degree of severity. The differential diagnosis is a crucial aspect in SLE as many other autoimmune diseases portray clinical similarities and autoantibody positivity. Lupus mimickers refer to a group of conditions that exhibit both clinical features and laboratory characteristics, including autoantibody profiles that resemble those present in patients with SLE, and prompt a diagnostic challenge in everyday clinical practice. Thus, lupus mimickers may present as a lupus-like condition (i.e., 2 or 3 criteria) or as one meeting the classification criteria for SLE. Herein we review and classify the current literature on lupus mimickers based on diverse etiologies which include infections, malign and benign neoplasms, medications, and vaccine-related reactions.
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Affiliation(s)
- Omar-Javier Calixto
- Center for Autoimmune Diseases Research (CREA), School of Medicine and Health Sciences, Universidad del Rosario, Bogotá, Colombia; Mederi, Hospital Universitario Mayor, Bogotá, Colombia
| | - Juan-Sebastian Franco
- Center for Autoimmune Diseases Research (CREA), School of Medicine and Health Sciences, Universidad del Rosario, Bogotá, Colombia; Mederi, Hospital Universitario Mayor, Bogotá, Colombia
| | - Juan-Manuel Anaya
- Center for Autoimmune Diseases Research (CREA), School of Medicine and Health Sciences, Universidad del Rosario, Bogotá, Colombia; Mederi, Hospital Universitario Mayor, Bogotá, Colombia.
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[Pharmacological therapy of urogenital cancer: rational routine diagnostic imaging]. Urologe A 2013; 52:1564-73. [PMID: 24197084 DOI: 10.1007/s00120-013-3253-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/26/2022]
Abstract
BACKGROUND Imaging studies are an integral and important diagnostic modality to stage, monitor, and follow-up patients with metastatic urogenital cancer. The currently available guidelines on diagnosis and treatment of urogenital cancer do not provide the clinician with evidence-based recommendations for daily routine. It is the aim of the current manuscript to develop scientifically valid recommendations with regard to the most appropriate imaging technique and the most useful time interval in metastatic urogenital cancer patients undergoing systemic therapy. RESULTS Therapeutic response of soft tissue metastases is evaluated with the use of the RECIST criteria. In skeletal metastases, bone scans with validated algorithms must be performed to assess response. In patients with testicular germ cell tumors, computed tomography (CT) of the chest, the retroperitoneum, and the abdomen represents the standard imaging technique of choice usually performed prior to and at the end of systemic chemotherapy. Only in seminomas with residual tumors > 3 cm in diameter should FDG-PET/CT be performed about 6 weeks after chemotherapy. Metastatic renal cell carcinomas treated with molecular targeted therapies are routinely evaluated by CT scans at 3 month intervals. In specific cases, FDG-PET/CT is able to predict responses as early as 8 weeks after initiation of treatment. In patients with metastatic urothelial carcinomas, imaging studies should be performed after every second cycle of cytotoxic therapy. In patients with metastatic prostate cancer, the modality and the frequency of imaging studies depends on the type of the treatment. In men undergoing androgen deprivation therapy, no routine imaging studies are recommended except for patients with new onset symptoms or significant PSA progression prior to change of treatment. In men with metastatic castration-resistant PCA who are treated with cytotoxic regimes, routine imaging studies in the presence of decreasing or stable PSA serum concentrations are not indicated. In men treated with lyase inhibitor or inhibitors of the androgen receptor signaling cascade, imaging studies should be performed at 3 month intervals due to the low correlation of PSA serum concentrations with clinical response. CONCLUSIONS Imaging studies to assess therapeutic response to systemic treatment in metastatic cancers of the urogenital tract must be chosen depending on the treatment regime, primary organ, and potential consequences of the findings. Routine imaging studies without specific clinical or therapeutic relevance are not justified.
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Coexistence of Crohn's disease in a patient with systemic lupus erythematosus. Rheumatol Int 2012; 33:2145-8. [PMID: 22249306 DOI: 10.1007/s00296-011-2357-1] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/09/2011] [Accepted: 12/22/2011] [Indexed: 12/15/2022]
Abstract
The concurrence of inflammatory bowel disease with systemic lupus erythematosus (SLE) is rare. The concomitant diagnosis of Crohn's disease and SLE is even more rare. The patient, a 40-year-old woman, was admitted to our hospital because of relapsing episodes of abdominal pain, diarrheas upper and lower extremities arthralgias, Raynaud's phenomenon with positive antinuclear antibodies, and fever for the last 2 years. The patient was diagnosed elsewhere with SLE and treated with hydroxychloroquine. Her medical history also included tonsillectomy and total hip replacement after a car accident. Family history was unremarkable. Physical examination was unremarkable except of very mild pain at lower left abdominal quadrant. Laboratory tests showed erythrocyte sedimentation rate at 32 mm/h, C-reactive protein at 36 mg/dl, positive rheumatoid factor, and increased C3, C4, positive antinuclear antibodies with the presence of anti-Sm and anti-RNP antibodies. Ileocolonoscopy revealed colonic inflammation with ulcers and pseudopolyps. Subsequent biopsies were diagnostic of Crohn's disease. Patient was diagnosed with Crohn's colitis concomitant to systemic lupus erythematosus and was started on therapy with azathioprine 2 mg/Kg, methylprednisolone 16 mg/d with slow tapering, mesalazine 1.5 g/day, and hydroxychloroquine. Patient is in excellent health status on the six-month follow-up.
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Navaneethan U, Venkatesh PGK, Wang J. Cytomegalovirus ileitis in a patient after liver transplantation-differentiating from de novo IBD. J Crohns Colitis 2011; 5:354-9. [PMID: 21683307 DOI: 10.1016/j.crohns.2011.01.010] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/01/2011] [Revised: 01/31/2011] [Accepted: 01/31/2011] [Indexed: 02/08/2023]
Abstract
Cytomegalovirus (CMV) infection of the gastrointestinal (GI) tract has been reported in immunocompromised patients and is seen following liver transplantation. Although CMV infection can affect any part of the GI tract, involvement of the terminal ileum is rarely encountered after liver transplantation. We report a case of a 32-year-old male who developed CMV infection of the terminal ileum while receiving immunosuppression for liver transplantation. Initial ganciclovir treatment did not improve the patient's symptoms and therapy was then switched to foscarnet which ultimately resulted in resolution of infection. However the patient continued to have symptoms because of intermittent small bowel obstruction because of ulcerations and fibrosis ultimately requiring surgical resection. CMV DNA polymerase chain reaction (PCR) was negative throughout the course of infection. Surgical resected specimen revealed no evidence of inflammatory bowel disease (IBD). Follow up colonoscopy up to a year after infection also did not reveal any evidence of IBD. Compartmentalization in the clinical presentation of CMV involving GI tract can be seen with a negative blood DNA PCR. Histological diagnosis thus forms an important part in the clinical follow-up of liver transplant patients undergoing intense immunosuppression and should be aggressively pursued in patients with GI symptoms. De novo IBD should be considered in the differential diagnosis in these patients who do not improve with anti-viral treatment.
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Histologic features of cytomegalovirus enteritis in small bowel allografts. Transplant Proc 2011; 42:2671-5. [PMID: 20832567 DOI: 10.1016/j.transproceed.2010.04.059] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/13/2010] [Accepted: 04/21/2010] [Indexed: 01/09/2023]
Abstract
BACKGROUND Cytomegalovirus (CMV) is one of the most common viral infections to affect solid organ transplant patients, most frequently owing to reactivation of a latent infection as a result of immunosuppression. CMV enteritis (CE) may enter into the differential diagnosis of acute rejection in biopsies of small bowel (SB) allografts, where differentiation is important due to disparate therapies. OBJECTIVE The aim of this study was to identify histologic features in SB allografts that may suggest CE. METHODS The case files for a single institution were queried for all cases of SB mucosal biopsies with cells positive by CMV immunoperoxidase staining. Morphologic and clinical information was reviewed. RESULTS Six biopsies demonstrating immunoperoxidase-confirmed CE were identified in a retrospective review of the records of a single institution. A common predisposing factor was the administration of high-dose steroids within a month before CE diagnosis. Most cases (66%) displayed a demarcated area of villous/crypt loss with an abundance of plasma cells and lymphocytes and a paucity of eosinophils. One case showed an acute enteritis-like pattern of injury, corresponding with a higher number of CMV-positive cells. CMV inclusions were visible on hematoxylin-eosin stains in all but 1 case. In no case were histologic criteria for acute cellular rejection met. CONCLUSIONS The presence of circumscribed area of mucosal injury with few eosinophils or an acute enteritis pattern should prompt the identification of viral inclusions or the acquisition of a CMV immunostain.
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