Successful cord blood transplantation for myelodysplastic syndrome with Behçet disease-like refractory stomatitis and multiple ileocecal ulcerations

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  • 臍帯血移植が有効であったBehçet病類似の難治性口内炎と多発回盲部潰瘍を伴う骨髄異形成症候群
  • 症例報告 臍帯血移植が有効であったBehcet病類似の難治性口内炎と多発回盲部潰瘍を伴う骨髄異形成症候群
  • ショウレイ ホウコク サイタイケツ イショク ガ ユウコウ デ アッタ Behcetビョウ ルイジ ノ ナンチセイ コウナイエン ト タハツカイ モウブ カイヨウ オ トモナウ コツズイイケイセイ ショウコウグン

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Description

<p>A 12-year-old boy was diagnosed with aplastic anemia. He was followed as an outpatient without medication, and his cytopenia improved after several years. When he was 26 years old, an annual medical checkup revealed leukocytopenia, and at the age of 31 years, he was diagnosed with myelodysplastic syndrome (MDS), refractory cytopenia with multilineage dysplasia. Chromosomal analysis of his bone marrow cells revealed trisomy 8. Ten months after being diagnosed with MDS, he developed refractory stomatitis. Two months later, he experienced abdominal pain and bloody stool, and simple punched-out ulcers similar to intestinal Behçet's disease (BD) were noted in the terminal ileum on colonoscopy. Steroids, mesalazine, and adalimumab were ineffective. Nineteen months after the MDS diagnosis, he underwent cord blood transplantation from an HLA 1-locus mismatched unrelated donor in accordance with a non-myeloablative pretransplant conditioning regimen. The patient's stomatitis and ileocecal ulcers improved following the transplantation. Currently, both MDS and BD-like symptoms are in complete remission at 36 months post transplantation, and the patient continues to take low-dose oral tacrolimus for chronic skin GVHD. Allogeneic hematopoietic stem cell transplantation could become a therapeutic choice for MDS associated with BD, even if refractory intestinal BD symptoms are present.</p>

Journal

  • Rinsho Ketsueki

    Rinsho Ketsueki 60 (10), 1436-1442, 2019

    The Japanese Society of Hematology

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