Nishio Nobuhiro, Takahashi Yoshiyuki, Ohashi Haruhiko, Doisaki Sayoko, Muramatsu Hideki, Hama Asahito, Shimada Akira, Yagasaki Hiroshi, Kojima Seiji
Department of Pediatrics, Nagoya University Graduate School of Medicine, Clinical Research Center for Blood Diseases, National Hospital Organization Nagoya Medical Center, Nagoya, Japan.
Pediatr Transplant. 2011 Mar;15(2):161-6. doi: 10.1111/j.1399-3046.2010.01431.x. Epub 2010 Dec 22.
DC is an inherited bone marrow failure syndrome mainly characterized by nail dystrophy, abnormal skin pigmentation, and oral leukoplakia. Bone marrow failure is the most common cause of death in patients with DC. Because previous results of HSCT with a myeloablative regimen were disappointing, we used a reduced-intensity conditioning regimen for two patients with classic DC, and one patient with cryptic DC who harbored the TERT mutation. Graft sources included two mismatched-related bone marrow (BM) donors and one unrelated BM donor. Successful engraftment was achieved with few regimen-related toxicities in all patients. They were alive 10, 66, and 72 months after transplantation, respectively. Long-term follow-up is crucial to determine the late effects of our conditioning regimen.
先天性角化不良(DC)是一种遗传性骨髓衰竭综合征,主要特征为指甲营养不良、皮肤色素沉着异常和口腔黏膜白斑。骨髓衰竭是DC患者最常见的死亡原因。由于先前采用清髓性方案进行造血干细胞移植(HSCT)的结果令人失望,我们对两名典型DC患者以及一名携带端粒酶逆转录酶(TERT)突变的隐匿性DC患者采用了减低强度预处理方案。移植物来源包括两名HLA配型不合的亲属骨髓供者和一名非亲属骨髓供者。所有患者均成功植入,且与方案相关的毒性反应较少。移植后他们分别存活了10个月、66个月和72个月。长期随访对于确定我们预处理方案的远期效应至关重要。