遅発性増悪型急性リンパ芽球性白血病の小児におけるチオグアニン関連静脈閉塞性疾患に続発した眼球出血

Ocular hemorrhage secondary to thioguanine-associated veno-occlusive disease in a child with acute lymphoblastic leukemia in delayed intensification.

• Nabanita Bhunia
• Nicholas D Yeager
• Susan I Colace
• Dominic Buzzaco
• Rajinder P S Bajwa

抄録

肝VODは、幹細胞移植中に致命的な合併症となる可能性があり、非移植の場合にはほとんど見られない。遅発性増悪期のALL治療中に視覚障害を呈した5歳の男児の症例を報告する。化学療法による重篤な血小板減少を伴う両側網膜出血を認めた。VODはEBMTの基準に基づいて診断され、支持療法で管理された。VODは消失したが，視力は徐々に低下し失明に至った．この症例は、VOD発症のリスクがあるALL患者を遅発性増悪で注意深く観察することの重要性、診断上の特徴としての難治性血小板減少症の重要性、眼内出血を伴うVODの発現の可能性を浮き彫りにしている。

Hepatic VOD is a potentially fatal complication during stem cell transplantation and is rarely seen in the non-transplant setting. We report the case of a five-year-old boy who presented with visual complaints during delayed intensification phase of treatment for ALL. He was found to have bilateral retinal hemorrhages associated with profound thrombocytopenia due to chemotherapy. VOD was diagnosed based on EBMT criteria and was managed with supportive care. Despite resolution of VOD, his vision progressively deteriorated and resulted in blindness. This case highlights the significance of close monitoring of ALL patients in delayed intensification when they are at risk for developing VOD, the importance of refractory thrombocytopenia as a diagnostic feature and the potential for VOD to manifest with intraocular bleeding.

© 2019 Wiley Periodicals, Inc.