Ardissino Gianluigi, Salardi Stefania, Berra Silvia, Colussi Giacomo, Cugno Massimo, Zecca Marco, Giglio Fabio, Peccatori Jacopo, Diral Elisa, Tel Francesca, Clivio Alberto, Tedeschi Silvana
Center for HUS Control, Prevention and Management, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milano, Italy.
Laboratory of Medical Genetics, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milano, Italy.
Biol Blood Marrow Transplant. 2017 Sep;23(9):1580-1582. doi: 10.1016/j.bbmt.2017.05.013. Epub 2017 May 15.
Hematopoietic stem cell transplant-related thrombotic microangiopathy (HSCT-TMA) is a severe complication whose pathophysiology is unknown. We describe 6 patients in which the disease was associated with complement regulatory gene abnormalities received from their respective donors. It is suggested that mutated and transplanted monocyte-derived cells are responsible for production of abnormal proteins, complement dysregulation, and, ultimately, for the disease. This observation might have important drawbacks as far as HSCT-TMA pathophysiology and treatment are concerned.
造血干细胞移植相关血栓性微血管病(HSCT-TMA)是一种严重并发症,其病理生理学尚不清楚。我们描述了6例该疾病与从各自供体获得的补体调节基因异常相关的患者。提示突变并移植的单核细胞衍生细胞负责异常蛋白的产生、补体失调,最终导致该疾病。就HSCT-TMA的病理生理学和治疗而言,这一观察结果可能具有重要意义。