Behcet disease and celiac disease: to screen or not?

Abstract

Behcet’s disease (BD) is a chronic, relapsing, inflammatory disease characterized by recurrent oral aphthae and any of the several systemic manifestations including genital aphthae, ocular disease, skin lesions, gastrointestinal involvement, neurologic disease, vascular disease, or arthritis. BD is an autoimmune disorder and may be associated with other autoimmune diseases, but the underlying pathogenetic mechanism of the disease is not well understood [1]. Up to now, only a few cases have been described with BD and celiac disease (CD). Herein, we report a case of BD overlap with CD. A 17-year-old young was admitted to our clinic because of diarrhea, bloating, and abdominal distension. The patient’s medical history included BD. In addition, he had been taking colchicine for 2 years for BD. Microscopic examination of stool was normal. Serum biochemical parameters were within the normal range. Complete blood count revealed mild anemia with hb: 13.2 g/l and iron: 87(70–180) and total iron binding capacity: 417(227–428) and ferritine: 12 ng/ml (15–140). Because of iron deficiency, diarrhea and abdominal symptoms serum tissue transglutaminase (tTG) and gliadin antibodies were checked for celiac disease. Immunoglobulin A class tTG and IgG gliadin antibodies were found positive with normal serum immunoglobulin levels. In order to confirm the diagnosis of CD, an upper gastrointestinal system endoscopy with duodenal biopsy was performed. It showed scalloping of folds, and histopathological examination showed subtotal villous atrophy (Marsh 3a). Gluten-free diet was instituted, and all the symptoms significantly decreased in follow-up. Celiac disease can be defined as a small-bowel disorder characterized by mucosal inflammation, villous atrophy, and crypt hyperplasia, which occur upon exposure to dietary gluten and which demonstrate improvement after withdrawal of gluten from the diet [2]. Patients affected by CD have greater risk of developing autoimmune diseases, among them rheumatic diseases. BD and CD share many clinical presentations, such as recurrent aphthous stomatitis, as well as gastrointestinal, musculoskeletal, and neurologic involvements. There are only three studies concerning the association of BD and CD in the literature [3–5]. In these reports, patients with BD were screened for CD and only two of them were diagnosed with histologically proven CD. In addition, two other cases with CD and BD have been reported in the literature. Most of these cases showed alarm symptoms for early further investigations. In our case, patient referred to our clinic because of diarrhea and bloating. Colchicine is commonly used in the treatment of BD and has many gastrointestinal side effects including abdominal pain, cramping, nausea, vomiting, and diarrhea. Differential diagnosis of CD and colchicine side effects may be difficult because of similar symptoms and may result interruption of treatment. In conclusion, despite the rarity of this combination, both disease have immunogenic mechanism and share many clinical manifestations. It is still unclear whether all patients with BD should be screened for CD or not, and in addition, colchicine treatment may cause differential diagnosis more difficult. If diarrhea and iron deficiency are seen in a patient with BD, the serologic tests with smallbowel biopsy should be performed to exclude CD. B. Ergul E. Kocak (&) S. Koklu Department of Gastroenterology, Ankara Education and Research Hospital, Keklikpinari mahallesi, 240 sokak, Duru apartmani, 8/6, Dikmen, Cankaya, Ankara, Turkey e-mail: kocak67@hotmail.com