Severe stomatitis and ileocecal perforation developed after all-<i>trans</i> retinoic acid monotherapy in an HLA-B51-positive patient with acute promyelocytic leukemia

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  • KIMURA Kenji
    Department of Hematology, Chiba University Hospital
  • TAKEUCHI Masahiro
    Department of Hematology, Chiba University Hospital
  • HASEGAWA Nagisa
    Department of Hematology, Chiba University Hospital
  • TOGASAKI Emi
    Department of Hematology, Chiba University Hospital
  • SHIMIZU Ryoh
    Department of Hematology, Chiba University Hospital
  • KAWAJIRI Chika
    Department of Hematology, Chiba University Hospital
  • MUTO Tomoya
    Department of Hematology, Chiba University Hospital
  • TSUKAMOTO Shokichi
    Department of Hematology, Chiba University Hospital
  • TAKEDA Yusuke
    Department of Hematology, Chiba University Hospital
  • OHWADA Chikako
    Department of Hematology, Chiba University Hospital
  • SAKAIDA Emiko
    Department of Hematology, Chiba University Hospital
  • SAKAI Shio
    Department of Hematology, Chiba University Hospital Department of Transfusion Medicine and Cell Therapy, Chiba University Hospital
  • MIMURA Naoya
    Department of Hematology, Chiba University Hospital Department of Transfusion Medicine and Cell Therapy, Chiba University Hospital
  • OTA Satoshi
    Department of Pathology, Chiba University Hospital
  • ISEKI Tohru
    Department of Hematology, Chiba University Hospital Department of Transfusion Medicine and Cell Therapy, Chiba University Hospital
  • NAKASEKO Chiaki
    Department of Hematology, Chiba University Hospital

Bibliographic Information

Other Title
  • All-<i>trans</i> retinoic acid単独投与後に重度の口内炎と回盲部腸管穿孔を発症したHLA-B51陽性急性前骨髄球性白血病
  • 症例報告 第169回日本血液学会例会 : 臼杵憲祐例会長 推薦演題 All-trans retinoic acid単独投与後に重度の口内炎と回盲部腸管穿孔を発症したHLA-B51陽性急性前骨髄球性白血病
  • ショウレイ ホウコク ダイ169カイ ニホン ケツエキ ガッカイ レイカイ : ウスキケンユウレイカイチョウ スイセン エンダイ All-trans retinoic acid タンドク トウヨ ゴ ニ ジュウド ノ コウナイエン ト カイ モウブ チョウカン センコウ オ ハッショウ シタ HLA-B51 ヨウセイ キュウセイ ゼン コツズイキュウセイ ハッケツビョウ

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Abstract

A 34-year-old man who had been referred to our hospital was diagnosed with acute promyelocytic leukemia (APL). All-trans retinoic acid (ATRA), oral administration, was initiated. On day 25, he developed fever and respiratory distress with bilateral pulmonary infiltrates, suggesting differentiation syndrome (DS) caused by ATRA. These symptoms showed amelioration after discontinuing ATRA and initiating methylprednisolone. ATRA was re-started on day 29 at half the original dose because of residual APL blasts. The patient subsequently developed fever, severe stomatitis, and oropharyngeal ulcers, which persisted even after discontinuing ATRA. On day 48, he suddenly developed severe abdominal pain with free air, observable on an abdominal X-ray, and underwent emergency ileocecal resection. Pathological examination of the resected ileocecal intestines revealed multiple ulcers and perforations. No leukemic cell infiltration was observed. In this case, only ATRA was administered for APL treatment. These findings suggest that ileocecal ulcerations and perforations, as well as oropharyngeal ulcers, might have been caused by DS or ATRA. Furthermore, DNA typing of the HLA-B locus revealed that the patient had HLA-B51 associated with Behçet's disease. Therefore, hypercytokinemia with DS might have induced Behçet's disease-like symptoms, including stomatitis and ileocecal perforation, complications that are particularly observed in patients with HLA-B51.

Journal

  • Rinsho Ketsueki

    Rinsho Ketsueki 57 (6), 765-770, 2016

    The Japanese Society of Hematology

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