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International Journal of Hematology

, Volume 84, Issue 2, pp 166–169 | Cite as

Recurrent Intramural Hematoma of the Small Intestine in a Severe Hemophilia A Patient with a High Titer of Factor VIII Inhibitor: A Case Report and Review of the Literature

  • Akira KatsumiEmail author
  • Tadashi Matsushita
  • Kanji Hirashima
  • Toshihiro Iwasaki
  • Tatsuya Adachi
  • Koji Yamamoto
  • Tetsuhito Kojima
  • Junki Takamatsu
  • Hidehiko Saito
  • Tomoki Naoe
Case Report

Abstract

A 17-year-old man with severe hemophilia A (factor VIII <1%) developed intermittent left upper quadrant pain. He had a high titer of factor VIII inhibitor (1024 Bethesda units/mL) and was diagnosed with intramural hematoma of the jejunum. He was managed conservatively with activated prothrombin complex concentrate (APCC), resulting in the resolution of symptoms. He developed recurrent intramural hematoma of the small intestine over the next 54 months, and was successfully treated with APCC. This case highlights a rare clinical manifestation in hemophilia patients, and also indicates the effectiveness of APCC instead of exploratory surgery for intramural hematoma. Cases of intramural hematoma of the gastrointestinal tract among hemophilia patients are also reviewed.

Key words

Hemophilia A Inhibitor Intramural hematoma Small intestine Activated prothrombin complex concentrate 

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Copyright information

© The Japanese Society of Hematology 2006

Authors and Affiliations

  • Akira Katsumi
    • 1
    Email author
  • Tadashi Matsushita
    • 1
  • Kanji Hirashima
    • 1
  • Toshihiro Iwasaki
    • 1
  • Tatsuya Adachi
    • 1
  • Koji Yamamoto
    • 2
  • Tetsuhito Kojima
    • 3
  • Junki Takamatsu
    • 2
  • Hidehiko Saito
    • 4
  • Tomoki Naoe
    • 1
  1. 1.Department of Hematology and OncologyNagoya University Graduate School of MedicineNagoyaJapan
  2. 2.Department of Blood Transfusion ServiceNagoya University HospitalNagoyaJapan
  3. 3.Department of Medical TechnologyNagoya University School of Health SciencesNagoyaJapan
  4. 4.Nagoya Medical CenterNagoyaJapan

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