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[异基因造血干细胞移植治疗施瓦茨曼-戴蒙德综合征:三例报告及文献复习]

[Allogeneic hematopoietic stem cell transplantation for Shwachman-Diamond syndrome: a report of three cases and literature review].

作者信息

Feng A H, Shi J M, Fu H R, Yu J, Zheng W Y, Zhu Y Y, Huang H, Zhao Y M

机构信息

Bone Marrow Transplantation Center, the First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310003, China Department of Hematology, Affiliated Hospital of Shandong Second Medical University, Weifang 261035, China.

Bone Marrow Transplantation Center, the First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310003, China.

出版信息

Zhonghua Xue Ye Xue Za Zhi. 2024 Jul 14;45(7):689-693. doi: 10.3760/cma.j.cn121090-20240107-00009.

Abstract

This study reports on three patients with Shwachman-Diamond syndrome (SDS) who underwent allogeneic hematopoietic stem cell transplantation (allo-HSCT) at the First Affiliated Hospital of Zhejiang University School of Medicine. Based on relevant literature, the clinical manifestations and genetic mutation characteristics of SDS were summarized, and the efficacy and timing of allo HSCT for such patients were explored. Three SDS patients were all male, with transplant ages of 32, 33, and 32 years old, respectively. All three patients were diagnosed in childhood. Case 1 presented with anemia as the initial clinical manifestation, which gradually progressed to a decrease in whole blood cells; Case 2 and 3 both present with a decrease in whole blood cells as the initial clinical manifestation. Case 1 and 3 have intellectual disabilities, while case 3 presents with pancreatic steatosis and chronic pancreatitis. All three patients have short stature. Three patients all detected heterozygous mutations in the SBDS: c.258+2T>C splice site. The family members of the three patients have no clinical manifestations of SDS. All three patients were treated with a reduced dose pre-treatment regimen (Fludarabine+Busulfan+Me-CCNU+Rabbit Anti-human Thymocyte Globulin). Case 1 and case 2 underwent haploid hematopoietic stem cell transplantation, while case 3 underwent unrelated donor hematopoietic stem cell transplantation. Case 1 was diagnosed with myelodysplastic syndrome transforming into acute myeloid leukemia before transplantation, but experienced early recurrence and death after transplantation; Case 2 is secondary implantation failure, dependent on platelet transfusion; Case 3 was removed from medication maintenance treatment after transplantation, and blood routine monitoring was normal.

摘要

本研究报告了3例在浙江大学医学院附属第一医院接受异基因造血干细胞移植(allo-HSCT)的施瓦赫曼-戴蒙德综合征(SDS)患者。基于相关文献,总结了SDS的临床表现和基因突变特征,并探讨了此类患者allo-HSCT的疗效及时机。3例SDS患者均为男性,移植年龄分别为32岁、33岁和32岁。3例患者均在儿童期确诊。病例1以贫血为初始临床表现,逐渐进展为全血细胞减少;病例2和3均以全血细胞减少为初始临床表现。病例1和3有智力障碍,病例3有胰腺脂肪变性和慢性胰腺炎。3例患者均身材矮小。3例患者均检测到SBDS基因杂合突变:c.258+2T>C剪接位点。3例患者的家庭成员均无SDS临床表现。3例患者均采用减低剂量预处理方案(氟达拉滨+白消安+司莫司汀+兔抗人胸腺细胞球蛋白)治疗。病例1和病例2接受单倍体造血干细胞移植,病例3接受非血缘供者造血干细胞移植。病例1在移植前诊断为骨髓增生异常综合征转化为急性髓系白血病,移植后早期复发死亡;病例2为继发性植入失败,依赖血小板输注;病例3移植后停用药物维持治疗,血常规监测正常。

相似文献

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Diagnosis, Treatment, and Molecular Pathology of Shwachman-Diamond Syndrome.施瓦赫曼-戴蒙德综合征的诊断、治疗及分子病理学
Hematol Oncol Clin North Am. 2018 Aug;32(4):687-700. doi: 10.1016/j.hoc.2018.04.006. Epub 2018 Jun 5.

本文引用的文献

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Hematopoietic Stem Cell Transplantation for Shwachman-Diamond Syndrome.施瓦赫曼-戴蒙德综合征的造血干细胞移植
Biol Blood Marrow Transplant. 2020 Aug;26(8):1446-1451. doi: 10.1016/j.bbmt.2020.04.029. Epub 2020 May 16.

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