Successful allogeneic bone marrow transplantation after massive gastrointestinal bleeding in a patient with myelodysplastic syndrome associated with intestinal Behçet-like disease

Arata Ishii; Shokichi Tsukamoto; Tatsuzo Mishina; Shintaro Izumi; Yurie Nagai; Miki Yamazaki; Yutaro Hino; Kensuke Kayamori; Nagisa Oshima-Hasegawa; Tomoya Muto; Shio Mitsukawa; Yusuke Takeda; Naoya Mimura; Chikako Ohwada; Chiaki Nakaseko; Jun-Ichiro Ikeda; Emiko Sakaida

doi:10.1016/j.lrr.2021.100278

Successful allogeneic bone marrow transplantation after massive gastrointestinal bleeding in a patient with myelodysplastic syndrome associated with intestinal Behçet-like disease

Leuk Res Rep. 2021 Oct 29:16:100278. doi: 10.1016/j.lrr.2021.100278. eCollection 2021.

Authors

Arata Ishii^{1

2}, Shokichi Tsukamoto^{1

2}, Tatsuzo Mishina^{1

2}, Shintaro Izumi^{1

2}, Yurie Nagai^{1

2}, Miki Yamazaki^{1

2}, Yutaro Hino^{1

2}, Kensuke Kayamori^{1

2}, Nagisa Oshima-Hasegawa^{1

2}, Tomoya Muto^{1

2}, Shio Mitsukawa^{1

3}, Yusuke Takeda^{1

2}, Naoya Mimura^{1

3}, Chikako Ohwada^{1

2

4}, Chiaki Nakaseko^{1

2

4}, Jun-Ichiro Ikeda⁵, Emiko Sakaida^{1

2}

Affiliations

¹ Department of Hematology, Chiba University Hospital, Chiba, Japan.
² Blood and Marrow Transplant Center, Chiba University Hospital, Chiba, Japan.
³ Department of Transfusion Medicine and Cell Therapy, Chiba University Hospital, Chiba, Japan.
⁴ Department of Hematology, International University of Health and Welfare School of Medicine, Narita, Japan.
⁵ Department of Pathology, Chiba University Hospital, Chiba, Japan.

Abstract

A 45-year-old woman was diagnosed with myelodysplastic syndrome (MDS) with trisomy 8 and Behçet-like disease (BLD) with multiple colorectal ulcers. Nonspecific inflammatory cells were infiltrated in the intestinal mucosa, whereas fluorescence in situ hybridization (FISH) analysis revealed only sporadic trisomy 8-positive cells. She presented massive lower gastrointestinal bleeding early after bone marrow transplantation but achieved long-term remission of both MDS and BLD. This is the first report of massive gastrointestinal bleeding after transplantation for MDS with BLD. Based on FISH analysis, dysregulation of systemic inflammation may be involved in BLD rather than direct invasion by trisomy 8-positive MDS clones.

Keywords: Behçet-like disease; Bone marrow transplantation; Gastrointestinal bleeding; Myelodysplastic syndrome; Trisomy 8.

Publication types

Case Reports