scholarly journals A117 A CASE OF ADULT AUTOIMMUNE ENTEROPATHY AND PRIMARY SCLEROSING CHOLANGITIS

2020 ◽  
Vol 3 (Supplement_1) ◽  
pp. 135-137
Author(s):  
Y Lei ◽  
H Azhari ◽  
M Raman
Keyword(s):  
Primary Sclerosing Cholangitis ◽  
Sclerosing Cholangitis ◽  
Chronic Diarrhea ◽  
Villous Atrophy ◽  
Case Reports ◽  
Rare Condition ◽  
Mucosal Damage ◽  
Severe Diarrhea ◽  
Autoimmune Enteropathy ◽  
Repeat Endoscopy

Abstract Background Autoimmune enteropathy (AIE) is a rare, immune-mediated condition causing severe diarrhea and mucosal damage. The diagnostic criteria are chronic diarrhea with malabsorption, blunting of the small bowel villi, specific histologic changes, and the exclusion of other causes of villous atrophy. Anti-enterocyte and anti-goblet cell antibodies are supportive of the diagnosis, but not specific or sensitive enough to make the diagnosis. It has several clinical and histologic phenotypes and affects children more than adults. To our knowledge, there are only 2 published cases of AIE with concurrent primary sclerosing cholangitis (PSC). Aims To review the literature for adult AIE, any previous cases of concurrent PSC, and present a case of both conditions. Methods We searched PubMed for “autoimmune enteropathy psc”, “autoimmune enteropathy”, “autoimmune enteropathy case report”, “autoimmune enteropathy adult” and collected all the relevant articles. Relevant clinical features of demographics, IBD status, liver diseases, immune conditions, antibody status, and treatment agents were summarized. Results A 31-year-old man presented with a 20-year history of diarrhea and 11kg weight loss over 3 months. He had gout, no regular medications, and relevant no family history. All initial investigations were normal. Repeat endoscopy showed increased intra-epithelial lymphocytes (IEL) in the colon consistent with lymphocytic colitis. He was given budesonide with moderate improvement. He then developed biliary stasis, and was found to have early PSC. Repeat colonoscopy for ongoing diarrhea showed increased colonic IEL. Ulcerative colitis (UC) was suspected, and he was given oral 5-ASA and prednisone, with subsequent switch to vedolizumab. He then relapsed, requiring IV fluids, total parenteral nutrition (TPN), and IV steroids. Repeat endoscopy showed persistent colonic IEL, and villous blunting in the terminal ileum and duodenum. Celiac disease, IBD, combined variable immunodeficiency (CVID), and enteric infections were excluded. In this context, AIE was considered. He improved with IV steroids, and was switched to infliximab. Despite initial improvement, he had worsening diarrhea and malnutrition. After dose escalation of infliximab, and further IV steroids and TPN, he was started on a trial of ustekinumab. We found 35 articles that reported on 100 adult cases of AIE. There were two previous adult cases of concurrent PSC. We found 7 cases where TNF-alpha inhibitors were used, one case where vedolizumab was used, and no cases using ustekinumab. Details are in Table 1. Conclusions AIE is a rare condition causing chronic diarrhea and gut mucosal damage reported only in case reports and series. There have only been 2 other reported cases of concurrent PSC with AIE in adults. This is the first reported case of ustekinumab being tried to control symptoms. Funding Agencies None

Comparison of pharmacodynamics and celiac effects of olmesartan medoxomil formulations by using olmesartan-induced celiac-rat-model

2021 ◽  
Vol 18 ◽  
Author(s):  
Yelda Komesli ◽  
Bekir Ugur Ergur ◽  
Ercument Karasulu
Keyword(s):  
Weight Loss ◽  
Side Effects ◽  
Rat Model ◽  
Villous Atrophy ◽  
Case Reports ◽  
Olmesartan Medoxomil ◽  
Albino Rats ◽  
Pressure System ◽  
Severe Diarrhea ◽  
Clinical Changes

: Olmesartan Medoxomil (OM) is an angiotensin receptor blocker and has the adverse effect of celiac like enteropathy which was accepted by the FDA in 2013. This disease is characterized by severe diarrhea, weight loss and enteropathy. Although there are many case reports associated with olmesartan-related enteropathy in humans, it has not been described in a long-term animal model study so far. We developed a self-microemulsifying drug delivery system (OM-SMEDDS) in our previous study to reduce this side effect of drug and to enhance bioavailability. In this study, an artificial hypertension model was established with dose of 185 µmol /kg L-NAME (N ω-nitro-L-arginine methyl ester) twice in a day intraperitoneally in Wistar albino rats. To determine and compare side effects, the OM-Suspension and OM-SMEDDS were administered at 1.3 mg/kg therapeutic dose during one-month period to the rats. Tension of rats was recorded by measuring from their tails with non invasive blood pressure system. We observed celiac like enteropathy findings like villous atrophy and intraepithelial lymphocytosis and clinical changes like weight loss and severe diarrhea after the treatment with OM-Suspension during one-month experiment. It was also observed that the antihypertensive efficacy of the OM-SMEDDS formulation was higher than the suspension during the experiment and did not cause enteropathy, diarrhea and weight loss by reducing intestinal exposure. Hereby we evaluated the side effects of two different pharmaceutical forms by designing a sustainable and reproducible celiac rat model that can be induced with olmesartan medoxomil.

scholarly journals Spruelike Enteropathy Associated with Olmesartan: An Unusual Case of Severe Diarrhea

2013 ◽  
Vol 2013 ◽  
pp. 1-3 ◽  
Author(s):  
Stephanie E. Dreifuss ◽  
Yutaka Tomizawa ◽  
Nicholas J. Farber ◽  
Jon M. Davison ◽  
Adam E. Sohnen
Keyword(s):  
Clinical Response ◽  
Unusual Case ◽  
Chronic Diarrhea ◽  
Villous Atrophy ◽  
Case Series ◽  
Screening Colonoscopy ◽  
Drug Induced ◽  
Severe Diarrhea ◽  
History Of ◽  
High Index Of Suspicion

A 64-year-old male with a history of hypertension presented with worsening diarrhea and 25-pound weight loss over the preceding three months. Prior screening colonoscopy was unremarkable, and the patient failed conservative management. On presentation, the patient had orthostatic hypotension associated with prerenal azotemia for which olmesartan (40 mg/day) was held. Initial workup for chronic diarrhea was essentially unremarkable. Then, EGD was performed with small bowel biopsy, which showed a moderate villous blunting and an intraepithelial lymphocyte infiltration. Celiac disease was excluded by negative conventional serology tests and the absence of clinical response to a gluten-free diet. In the interim, diarrhea became resolving without any other interventions, and clinical response was achieved even with gluten-containing diet. Two months later, he achieved a complete resolution of diarrhea and regained 20-pound weight. Spruelike enteropathy is a clinical entity manifested by chronic diarrhea and intestinal villous atrophy. Spruelike enteropathy associated with olmesartan as a cause of drug-induced diarrhea is rare, and it has been reported only in a case series to date. This case highlighted the importance for clinicians to maintain a high index of suspicion for olmesartan as a precipitant of spruelike enteropathy.

scholarly journals Biliary Involvement in Type 2 Autoimmune Pancreatitis

2019 ◽  
Vol 13 (1) ◽  
pp. 200-206 ◽  
Author(s):  
Diana Ollo ◽  
Sylvain Terraz ◽  
Gregoire Arnoux ◽  
Giacomo Puppa ◽  
Jean-Louis Frossard ◽  
...  
Keyword(s):  
Primary Sclerosing Cholangitis ◽  
Autoimmune Pancreatitis ◽  
Sclerosing Cholangitis ◽  
Hepatic Duct ◽  
Intrahepatic Bile Duct ◽  
Rare Condition ◽  
Left Hepatic Duct ◽  
Mr Cholangiography ◽  
Igg4 Related Disease

Autoimmune pancreatitis (AIP) is a rare condition classified in 2 subtypes. Their distinction relies on a combination of clinical, serological, morphological and histological features. Type 1 is a pancreatic manifestation of IgG4-related disease characterized by multiorgan infiltration by IgG4 plasmocytes. In this condition, hepatobiliary infiltration is frequent and often mimics cholangiocarcinoma or primary sclerosing cholangitis. On the other hand, type 2 is commonly limited to the pancreas. Herein, we describe the case of a patient who presented a type 2 AIP associated with cholangiopathy, a condition not described in the established criteria. He first developed a pancreatitis identified as type 2 by the typical histopathological features and lack of IgG4 in the serum and tissue. Despite a good clinical response to steroids, cholestasis persisted, identified by MR cholangiography as a stricture of the left hepatic duct with dilatation of the intrahepatic bile duct in segments 2 and 3. Biliary cytology was negative. Evolution was favorable but after steroid tapering a few months later, the patient suffered from recurrence of the pancreatitis as well as progression of biliary attempt, suspicious for cholangiocarcinoma. As the investigations again ruled out neoplastic infiltration or primary sclerosing cholangitis, azathioprine was initiated with resolution of both pancreatic and biliary attempts.

New Therapeutic Option With N-acetylcysteine for Primary Sclerosing Cholangitis: Two Case Reports

2011 ◽  
Vol 18 (3) ◽  
pp. e71-e74 ◽  
Author(s):  
Burhan Ozdil ◽  
Arif Cosar ◽  
Hikmet Akkiz ◽  
Macit Sandikci ◽  
Can Kece
Keyword(s):  
Primary Sclerosing Cholangitis ◽  
Sclerosing Cholangitis ◽  
Case Reports ◽  
Therapeutic Option

scholarly journals Is Bartonella a Cause of Primary Sclerosing Cholangitis? A Case Study

2020 ◽  
Vol 2 (1) ◽  
pp. 48-57 ◽  
Author(s):  
Daniel A. Kinderlehrer
Keyword(s):  
Primary Sclerosing Cholangitis ◽  
Sclerosing Cholangitis ◽  
Case Reports ◽  
Liver Function Tests ◽  
Causal Link ◽  
Oral Vancomycin ◽  
Autoimmune Reaction ◽  
Small Series ◽  
Magnetic Resonance Imaging Mri

The etiology of primary sclerosing cholangitis (PSC) is unknown. I present a case which may be indicative of a causal link between Bartonella infection and PSC. The patient presented with complaints of abdominal pain and bloody diarrhea. A colonoscopy demonstrated chronic inflammation and changes consistent with ulcerative colitis. Routine laboratory studies revealed elevated liver function tests (LFTs); ultrasound and magnetic resonance imaging (MRI) confirmed the diagnosis of PSC. Bartonella serology was positive. It is established that Bartonella infection is associated with both gastrointestinal inflammation and autoimmunity; indeed, there is an animal model for Bartonella-induced PSC. Bartonella is susceptible to treatment with vancomycin and there are case reports and small series that demonstrate that PSC responds to treatment with oral vancomycin. Because of this, it is postulated that at least some cases of PSC may be associated with Bartonella infection. The patient in this report was treated with oral vancomycin and, since then, has been in remission for both colitis and PSC. Since vancomycin is not systemically absorbed, the premise is that he suffered from Bartonella colitis and an autoimmune reaction to Bartonella causing PSC. This premise warrants further study.

Beneficial effect of bezafibrate on primary sclerosing cholangitis (three case reports)

2002 ◽  
Vol 97 (7) ◽  
pp. 1849-1851 ◽  
Author(s):  
Ryuichi Kita ◽  
Yuri Kita-Sasai ◽  
Ikuko Hanaoka ◽  
Toru Kimura ◽  
Hiroyuki Kokuryu ◽  
...  
Keyword(s):  
Beneficial Effect ◽  
Primary Sclerosing Cholangitis ◽  
Sclerosing Cholangitis ◽  
Case Reports
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