Irreversible Leukoencephalopathy After Reduced-intensity Stem Cell Transplantation in a Dyskeratosis Congenita Patient With TINF2 Mutation

被引:8
作者
Isoda, Takeshi [1 ]
Mitsuiki, Noriko [1 ]
Ohkawa, Teppei [1 ]
Kaneko, Setsuko [1 ]
Endo, Akifumi [1 ]
Ono, Toshiaki [1 ]
Aoki, Yuki [1 ]
Tomizawa, Daisuke [1 ]
Kajiwara, Michiko [2 ]
Araki, Satoshi [1 ]
Nagasawa, Masayuki [1 ]
Morio, Tomohiro [1 ]
Takagi, Masatoshi [1 ]
Mizutani, Shuki [1 ]
机构
[1] Tokyo Med & Dent Univ, Dept Pediat & Dev Biol, Yushima, Tokyo 1138519, Japan
[2] Tokyo Med & Dent Univ, Hosp Blood Transfus Ctr, Yushima, Tokyo 1138519, Japan
关键词
dyskeratosis congenita; TINF2; hematopoietic stem cell transplantation; complication; sequelae; encephalopathy; REVERSIBLE ENCEPHALOPATHY SYNDROME; POSTERIOR; CHILDREN; LENGTH;
D O I
10.1097/MPH.0b013e318279e5ca
中图分类号
R73 [肿瘤学];
学科分类号
100214 ;
摘要
Hematopoietic stem cell transplantation (HSCT) for dyskeratosis congenita (DC) is challenging due to severe treatment-related adverse effects. Development of pulmonary fibrosis or veno-occlusive disease is well described in DC. However, neurological complication after HSCT has not been reported. A 9-year-old Japanese male with DC harboring the TINF2 mutation received reduced-intensity HSCT. Unfortunately, patient developed posterior reversible encephalopathy syndrome-like symptoms plausibly result by combination of thrombotic microangiopathy, graft-versus-host disease, and persistent hypertension and has been persisted mental retardation. Therefore, to decrease risk in DC cases after HSCT, strict control of hypertension, graft-versus-host disease, and thrombotic microangiopathy is required.
引用
收藏
页码:E178 / E182
页数:5
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