Journal of St. Marianna University
Online ISSN : 2189-0277
Print ISSN : 2185-1336
ISSN-L : 2185-1336
Case Report
Successful Switch to Adalimumab after Long-Term Thalidomide-Based Maintenance Therapy for Juvenile Onset Intestinal Behçet's Disease: A Case Report
Masaki YamashitaHiroshi Yasuda Masaki KatoHirofumi KiyokawaYoshinori SatoTadateru MaehataSatoko KimuraKeisuke Tateishi
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2022 Volume 13 Issue 2 Pages 149-154

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Abstract

The patient was a 32-year-old woman who had a 20-year history of intestinal Behçet's disease without ocular involvement. At the age of 12, she developed oral aphthosis, genital ulcers, erythema nodosum on the lower limbs, and soon developed lower abdominal pain. Colonoscopic examination revealed a round punched-out ulceration in the ileocecal region. Human leukocyte antigen (HLA)-B51 (B5) antigen was negative. Drug therapies with salazosufapyridine and prednisolone was started. When prednisolone was reduced to 15 mg/d, the ileocecal lesion recurred and in addition, diabetes mellitus developed. The patient had been suffered corticosteroid dependence with severe side effects. Since the induction of thalidomide at the age of 14, partial remission that allowed for outpatient visits was maintained for more than 10 years, but negative C-reactive protein (CRP) and endoscopically mucosal healing of the ileocecal ulcer on colonoscopy were not achieved.

Switch from thalidomide to adalimumab (ADA) was introduced at the age of 26 because of reproductive concern. Since introduction of ADA, no recurrence of genital ulcer or skin lesions had been observed. Negative inflammatory reaction was maintained. Colonoscopy performed after the switch to ADA revealed sustained endoscopic mucosal healing in ileocecal ulcer. ADA may have a more specific anti-inflammatory effect on intestinal BD than thalidomide.

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© 2022 St. Marianna University Society of Medical Science
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