A Case of Steroid-Refractory Nongranulomatous Chronic Idiopathic Enterocolitis Successfully Treated with Anti- TNF Agent

Case Report

J Gastroenterol Liver Dis. 2016; 1(1): 1006.

A Case of Steroid-Refractory Nongranulomatous Chronic Idiopathic Enterocolitis Successfully Treated with Anti- TNF Agent

Bhandari S¹*, Sanvanson P² and Perera LP³

¹Department of General Internal Medicine, Medical College of Wisconsin, USA

²Division of Gastroenterology and Hepatology, Medical College of Wisconsin, USA

³Gastroenterology and Hepatology, Aurora Health Care, USA

*Corresponding author: Sanjay Bhandari, Department of General Internal Medicine, Medical College of Wisconsin, 9200 W. Wisconsin Ave. Milwaukee, WI 53226, USA

Received: October 05, 2016; Accepted: December 15, 2016; Published: December 16, 2016

Abstract

Nongranulomatous Chronic Idiopathic Enterocolitis (NCIEC) has been reported in association with celiac sprue, lymphoma, and hypogammaglobinemia [1], but there have been very few cases reported in the literature where it exists as a primary entity. Nongranulomatous Chronic Idiopathic Enterocolitis (NCIEC) is generally corticosteroid-responsive [1,2]. We present a case of a 65 yearold female with chronic diarrhoea who was diagnosed with NCIEC. Our case is remarkable as it represents this rare disease. In addition, literature review reveals no previous reports of the use of anti-TNF agent Adalimumab (Humira®, Abbott Laboratories, Abbott Park, IL) as the treatment for steroid-refractory NCIEC, which resulted in successful clinical outcome in our case.

Keywords: Nongranulomatous chronic idiopathic enterocolitis; Corticosteroid; Adalimumab; Diarrhoea

Case Presentation

A 65 year-old Caucasian female who presented with diarrhoea for the past 3 weeks. Her past medical history was significant for hypertension and hyperlipidemia on lisinopril and atorvastatin, respectively. She had a previous laparoscopic hysterectomy. Family history was significant for cardiovascular disease and COPD. She was a former smoker-she smoked 1 pack a day for 15 years and quit 30 years ago. She was a retired clerk and was married with 2 children. She described diarrhoea as non-bloody, non-foul smelling and occurring with a frequency of 3-4 episodes per day. She reported decreased oral intake and weight loss of 25 lbs since the onset of symptoms. She denied any sick contacts or any recent travels out of the state. Diarrhoea occurred even at night. She reported she tried BRAT diet with no improvement of the symptoms. She was prescribed lomotil (Diphenoxylate and atropine) at an outpatient clinic about two weeks before the presentation, with no improvement of symptoms. 3 days after starting lomotil, she discontinued it. She was started on metronidazole about a week before presentation, which she could not tolerate because of nausea and vomiting. 3 days before the presentation to the hospital, she was started on Ciprofloxacin which she eventually stopped as it caused her abdominal pain, nausea and vomiting. On the presentation to the hospital, her physical examination was remarkable for dehydration. Abdomen was non-tender and revealed normal bowel sounds. Other aspects of the physical examination were unrevealing.

Laboratory work-up was significant for mild hypokalemia and hyponatremia from dehydration. The lab work-up on presentation is illustrated in Table 1. An extensive work-up was performed including stool ova and parasites, stool cultures, stool Clostridium difficile toxin, celiac disease antibody (tissue transglutaminase antibody), human immunodeficiency virus antibodies, quantitative immunoglobulins, hepatitis panel, anti-nuclear antibody screen, anti-enterocyte antibody (for autoimmune enteropathy) and thyroid stimulating hormone. All studies were negative. She underwent colonoscopy on day 2. It showed non-specific findings of diverticulosis of the sigmoid colon and small external hemorrhoids. Colonic biopsy revealed colonic mucosa with increased acute and chronic inflammation within the lamina propria and focal cryptitis. Terminal ileum biopsies revealed extensive infiltration of the lamina propria and epithelium with a dense lymphocytic infiltrate with foci of acute inflammation within the superficial lamina propria and epithelium. She underwent Esophagogastroduodenoscopy (EGD) on day 7, which revealed corrugation of the oesophagus, antral erythema, and nodular appearance of the duodenum. Biopsy of the specimen from duodenum and gastroesophageal junction showed markedly increased acute and chronic inflammatory changes in lamina propria, whereas gastric specimen showed only chronic inflammation. In addition, duodenal biopsy showed focal villous blunting. There was no evidence of any organisms (negative for CMV and H. pylori), dysplasia, or malignancy. The diagnosis was NCIEC.

Citation:Bhandari S, Sanvanson P and Perera LP. A Case of Steroid-Refractory Nongranulomatous Chronic Idiopathic Enterocolitis Successfully Treated with Anti-TNF Agent. J Gastroenterol Liver Dis. 2016; 1(1): 1006.