IgA vasculitis in a patient with ulcerative colitis under infliximab: drug-induced or genetic?
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CASE REPORT
IgA vasculitis in a patient with ulcerative colitis under infliximab: drug‑induced or genetic? Jun Urushikubo1 · Shunichi Yanai1 · Shotaro Nakamura1 · Yosuke Toya1 · Kensuke Asakura1 · Takahiro Gonai1 · Makoto Eizuka2 · Noriyuki Uesugi2 · Tamotsu Sugai2 · Takayuki Matsumoto1 Received: 11 August 2020 / Accepted: 19 October 2020 © Japanese Society of Gastroenterology 2020
Abstract We present the case of a 17-year-old male patient with ulcerative colitis (UC) under infliximab therapy and a family history of IgA vasculitis (IgAV). During a maintenance infliximab session, he developed palpable purpura, abdominal pain, and hematochezia. Computed tomography and endoscopy revealed findings compatible with gastrointestinal manifestations of IgAV. He was successfully treated by intravenous prednisolone and did not develop recurrence of IgAV during subsequent infliximab infusions. Keywords Ulcerative colitis · Inflammatory bowel disease · IgA vasculitis · Henoch–Schönlein purpura · Tumor necrosis factor
Introduction
Case report
Ulcerative colitis (UC) is an idiopathic inflammatory bowel disease (IBD) affecting the colon and rectum, and is characterized by a chronic and relapsing clinical course [1, 2]. Tumor necrosis factor (TNF) inhibitors are effective for the management of IBD, as well as other autoimmune diseases. However, increasing use of these agents in clinical practice may paradoxically induce secondary autoimmune conditions [3, 4]. IgA vasculitis (IgAV), previously referred to as Henoch–Schönlein purpura, is an acute vasculitis, characterized by cutaneous purpura, arthritis, and gastrointestinal and renal impairment. Several cases of IgAV associated with anti-TNF treatment have been reported [5, 6]. We herein present a case of an adolescent male with UC, which was complicated by IgAV during treatment with infliximab.
A 17-year-old male patient with a prior diagnosis of UC was admitted to our institution for severe abdominal pain and hematochezia. The patient was diagnosed with pan-colitis UC at the age of 14, and had been treated with 5-aminosalicylic acid (5-ASA), azathioprine, and intravenous infliximab every 8 weeks, which had allowed for clinical remission for the last 2 years. Two weeks prior to admission, he presented to our institution for evaluation of palpable purpura on bilateral lower extremities (Fig. 1a, b) and abdominal pain of 3 day duration. He underwent skin biopsy of purpura on the foot and histological examination revealed leukocytoclastic vasculitis (Fig. 1c), prompting a diagnosis of IgAV. He was subsequently started on daily oral prednisolone (PSL, 30 mg/day). Although his symptoms initially improved, he returned 2 weeks later for severe abdominal pain and hematochezia. Upon further investigation, we discovered that his father had a history IgAV, diagnosed at the age of 10 years. Laboratory data on admission revealed hemoglobin 16.0 g/dL, white blood cell (WBC) count 12,620/μL, serum C-reactive protein (CRP) 0.3 mg/dL, albumin 4.3 g/dL, IgG 1400 mg/ dL, IgA 400 mg/dL, IgM 9
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