Division of Pediatric Critical Care Medicine, University of Utah, Salt Lake City, Utah.
Intermountain Healthcare Primary Children's Hospital, Salt Lake City, Utah.
Pediatr Transplant. 2020 Jun;24(4):e13669. doi: 10.1111/petr.13669. Epub 2020 Feb 18.
This case describes an uncommon acute complication of diffuse thrombotic angiopathy and associated aHUS/TTP in an 11-year-old girl with Danon disease who underwent orthotopic heart transplant. Shortly after transplant, despite an uncomplicated operative course, the patient developed severe kidney injury and progressive altered mental status, culminating in cerebral edema, brain herniation, and death. She had received a single dose of tacrolimus (FK506) and a single dose of antithymocyte globulin. Sources of progressive somnolence, including oversedation from impaired renal clearance of opiates, and severe myopathy as has been previously described in Danon disease, were ruled out, and the patient continued to decline. Initial brain CT scan early after transplant showed no signs of cerebral edema, but repeat CT indicated severe cerebral edema. Based on autopsy, diffuse thrombotic angiopathy, with signs of hemolytic anemia with schistocytes, was likely responsible for her deterioration in the broader condition of aHUS/TTP.
本病例描述了一例罕见的弥漫性血栓性血管病的急性并发症,并伴有 11 岁丹-诺病女孩心脏原位移植后的 aHUS/TTP。移植后不久,尽管手术过程顺利,但患者出现严重的肾损伤和进行性意识改变,最终导致脑水肿、脑疝和死亡。她曾接受过一次他克莫司(FK506)和一次抗胸腺细胞球蛋白的治疗。包括因肾脏清除阿片类药物能力受损导致过度镇静,以及丹-诺病之前描述过的严重肌病在内的导致进行性嗜睡的原因已被排除,且患者的病情持续恶化。移植后早期的头部 CT 扫描显示没有脑水肿的迹象,但重复 CT 显示严重的脑水肿。根据尸检结果,弥漫性血栓性血管病,伴有溶血性贫血和裂体细胞的迹象,可能是导致她在更广泛的 aHUS/TTP 情况下恶化的原因。