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

Zhonghua Xue Ye Xue Za Zhi

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

Published: July 2024

AI Article Synopsis

  • This study examines three male patients with Shwachman-Diamond syndrome (SDS) who received allogeneic hematopoietic stem cell transplantation (allo-HSCT) at a hospital in China.
  • All three patients, diagnosed in childhood, presented with various clinical symptoms like anemia, blood cell decrease, and intellectual disabilities, along with specific genetic mutations in the SBDS gene.
  • Despite undergoing different types of stem cell transplants and receiving a reduced intensity pre-treatment regimen, outcomes varied significantly, with one patient experiencing early recurrence of leukemia and another having secondary failure, while the third showed positive post-transplant recovery.

Article 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.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC11388119PMC
http://dx.doi.org/10.3760/cma.j.cn121090-20240107-00009DOI Listing

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