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Long-term outcome after surgery in a patient with intestinal Behcet’s disease complicated by myelodysplastic syndrome and trisomy 8

Intestinal Research 2020년 18권 4호 p.469 ~ 475
Mori Yuki, Iwamoto Fumihiko, Ishida Yasuaki, Kuno Toru, Kobayashi Shoji, Yoshida Takashi, Yamaguchi Tatsuya, Sato Tadashi, Sudo Makoto, Ichikawa Daisuke, Enomoto Nobuyuki,
소속 상세정보
 ( Mori Yuki ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Iwamoto Fumihiko ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Ishida Yasuaki ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Kuno Toru ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Kobayashi Shoji ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Yoshida Takashi ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Yamaguchi Tatsuya ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Sato Tadashi ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine
 ( Sudo Makoto ) - University of Yamanashi Faculty of Medicine Department of Surgery
 ( Ichikawa Daisuke ) - University of Yamanashi Faculty of Medicine Department of Surgery
 ( Enomoto Nobuyuki ) - University of Yamanashi Faculty of Medicine Department of Internal Medicine

Abstract


Behcet’s disease (BD) is a multisystem inflammatory disease of unknown origin. Rarely, BD occurs together with myelodysplastic syndrome (MDS). Interestingly, it is speculated that these are not simple coexistence but that the etiology of intestinal BD is at least partly derived from MDS itself. Furthermore, there is a relationship between MDS in patients with intestinal BD and trisomy 8. Immunosuppressive agents alone are insufficient to control MDS-associated BD, and many of these patients die of infection or hemorrhage. Surgery is considered for intestinal BD patients who are unresponsive to medical treatment or those with bowel complications such as perforation or persistent bleeding. We report a case of intestinal BD associated with MDS and trisomy 8. The patient was unresponsive to oral steroids and immunosuppressive treatment; the patient improved by surgical repair of a bowel perforation. Five years after the surgery, the patient is free of recurrence and not on medication. Our experience suggests that surgery may provide an effective therapeutic option for the treatment of MDS-related BD.

키워드

Behcet syndrome; Myelodysplastic syndrome; Trisomy 8

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