Trisomy 8-Associated Intestinal Behcet’s Disease That Achieved Complete Remission With Mesalazine

Satoshi Tanida, Keiji Ozeki, Tsutomu Mizoshita, Mamoru Tanika, Hirotada Nishie, Yasuyuki Okamoto, Takaya Shimura, Eiji Kubota, Hiromi Kataoka

Abstract


A 39-year-old man was referred to our hospital because of ileocecal ulcer and pancytopenia. Past medical history included psoriatic arthritis and acne vulgaris. Physical examination findings showed recurrent oral ulcerations and acneiform eruptions on his body. Colonoscopy confirmed the presence of an ulcer at the ileocecal valve. Human leukocyte antigen was positive for B51. Based on the bone marrow findings of hypercellularity accompanied by trilineage dysplastic changes and cytogenetic findings of 47,XY,dup(1)(q21q32), idem+8, a definite diagnosis of intestinal Behcet’s disease (BD), incomplete type, associated with myelodysplastic syndrome (MDS) with positivity for trisomy 8 was finally confirmed. Treatment of intestinal BD with mesalazine at 3,000 mg daily induced complete ulcer healing after 4 months. Thereafter, two cycles of azacytidine was administered, followed by bone marrow transplantation. The patient has done well with continuous healing of the ulcer for a year. Differentiating episodic MDS with positivity for trisomy 8 from drug-induced MDS by examination of the bone marrow and the cytogenetic changes is indispensable. Intestinal BD patients who develop pancytopenia would probably need sufficient doses of mesalazine for treatment.




J Med Cases. 2018;9(8):255-259
doi: https://doi.org/10.14740/jmc3082w

Keywords


Pancytopenia; Myelodysplastic syndrome; HLAB51; Ulcer healing

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