Javascript must be enabled to continue!
HSCT in Wiskott-Aldrich Syndrome
View through CrossRef
Introduction
Wiskott-Aldrich syndrome (WAS) is a rare genetic disorder primarily affecting males, characterized by a combination of immunodeficiency, thrombocytopenia, and eczema. This condition is caused by a mutation in the WASP gene, which is essential for the proper functioning of immune cells. HSCT remains the treatment of choice for severe forms of the disease, enabling the restoration of immune function. The aim of this study was to evaluate the outcomes after HSCT in children with Wiskott-Aldrich syndrome.
Methods
This is a retrospective descriptive study including children with WAS who underwent HSCT.
Results
Five boys with WAS were included. The age at diagnosis ranged from 1 month to 7 years, with a mean age of 31.2 months. Two patients received haploidentical grafts, and three received geno-identical grafts. Three patients received a protocol based on fludarabine and busulfan; one patient received busulfan, fludarabine, and antithymocyte globulin; and the last patient received fludarabine, thiotepa, and treosulfan. In vivo T depletion was based on post-transplant cyclophosphamide in the two patients receiving haploidentical HSCT. Post-transplant complications included bacterial infections in all patients, viral reactivation (CMV) in two patients, and acute graft-versus-host disease (GVHD) in two patients. Chronic GVHD was observed in one case. Furthermore, two patients developed thrombotic microangiopathy. All patients achieved engraftment with full donor chimerism. Finally, one patient out of five died at the age of 3 years and 5 months from extensive chronic GVHD. Four patients are alive and cured.
Conclusion
HSCT is a curative treatment in most patients with severe forms of WAS. However, post-transplant complications, including bacterial infections, viral reactivation, and GVHD, remain significant challenges in the management of these patients.
Rockefeller University Press
Title: HSCT in Wiskott-Aldrich Syndrome
Description:
Introduction
Wiskott-Aldrich syndrome (WAS) is a rare genetic disorder primarily affecting males, characterized by a combination of immunodeficiency, thrombocytopenia, and eczema.
This condition is caused by a mutation in the WASP gene, which is essential for the proper functioning of immune cells.
HSCT remains the treatment of choice for severe forms of the disease, enabling the restoration of immune function.
The aim of this study was to evaluate the outcomes after HSCT in children with Wiskott-Aldrich syndrome.
Methods
This is a retrospective descriptive study including children with WAS who underwent HSCT.
Results
Five boys with WAS were included.
The age at diagnosis ranged from 1 month to 7 years, with a mean age of 31.
2 months.
Two patients received haploidentical grafts, and three received geno-identical grafts.
Three patients received a protocol based on fludarabine and busulfan; one patient received busulfan, fludarabine, and antithymocyte globulin; and the last patient received fludarabine, thiotepa, and treosulfan.
In vivo T depletion was based on post-transplant cyclophosphamide in the two patients receiving haploidentical HSCT.
Post-transplant complications included bacterial infections in all patients, viral reactivation (CMV) in two patients, and acute graft-versus-host disease (GVHD) in two patients.
Chronic GVHD was observed in one case.
Furthermore, two patients developed thrombotic microangiopathy.
All patients achieved engraftment with full donor chimerism.
Finally, one patient out of five died at the age of 3 years and 5 months from extensive chronic GVHD.
Four patients are alive and cured.
Conclusion
HSCT is a curative treatment in most patients with severe forms of WAS.
However, post-transplant complications, including bacterial infections, viral reactivation, and GVHD, remain significant challenges in the management of these patients.
Related Results
Stem Cell Transplantation for Children with Sickle Cell Anemia: Factors Associated with Parent and Patient Interest
Stem Cell Transplantation for Children with Sickle Cell Anemia: Factors Associated with Parent and Patient Interest
Abstract
Abstract 1079
BACKGROUND:
Sickle cell disease (SCD) is the most common inherited blood disorder in the U...
Allogeneic Hematopoietic Stem Cell Transplant Versus Gene Therapy in Sickle Cell Disease: Updated Results from a Systematic Review
Allogeneic Hematopoietic Stem Cell Transplant Versus Gene Therapy in Sickle Cell Disease: Updated Results from a Systematic Review
Background: Patients with sickle cell disease (SCD) have reduced life expectancy and high morbidity, despite treatments such as hydroxyurea, transfusion, vaccination, and antibioti...
Lower neutrophil counts after hematopoietic stem cell transplant for sickle cell disease patients using donors with duffy-null phenotype
Lower neutrophil counts after hematopoietic stem cell transplant for sickle cell disease patients using donors with duffy-null phenotype
Abstract
Background: Individuals with the Duffy null phenotype (red blood cells that lack the Fya and Fyb antigens) have...
Outcomes Following Allogeneic Hematopoietic Stem Cell Transplantation For Metachromatic Leukodystrophy
Outcomes Following Allogeneic Hematopoietic Stem Cell Transplantation For Metachromatic Leukodystrophy
Abstract
Metachromatic Leukodystrophy (MLD) is a rare demyelinating disease caused by deficient lysosomal arylsulfatase-A (ARSA) activity and resulting pathologic su...
Relapsed Follicular Lymphoma Treatment - with or without Hematopoietic Stem Cell Transplant?
Relapsed Follicular Lymphoma Treatment - with or without Hematopoietic Stem Cell Transplant?
Abstract
BACKGROUND: The best treatment strategy in patients with relapsed Follicular Lymphoma (FL) remains controversial. The incorporation of rituximab (R) in the ...
High Expression of SPAG1 Is Associated with a Worse Clinical Outcome in Intermediate Risk Acute Myeloid Leukemia That Can be Partially Overcome By Hematopoietic Stem Cell Transplantation
High Expression of SPAG1 Is Associated with a Worse Clinical Outcome in Intermediate Risk Acute Myeloid Leukemia That Can be Partially Overcome By Hematopoietic Stem Cell Transplantation
Abstract
Introduction: Acute myeloid leukemia (AML) is a heterogeneous disease with variable responses to therapy and clinical outcomes. Cytogenetics and molecular a...
The Incidence and Risk Factors of the Euthyroid Sick Syndrome Soon after Allogeneic Hematopoietic Stem Cell Transplantation in Children.
The Incidence and Risk Factors of the Euthyroid Sick Syndrome Soon after Allogeneic Hematopoietic Stem Cell Transplantation in Children.
Abstract
Purpose: In this study, we analyzed the short term change of thyroid function, incidence and risk factors of thyroid dysfunction soon after allogeneic hemat...
Myeloablative Haploidentical Transplant as an Alternative to Matched Sibling Transplant for Peripheral T-Cell Lymphomas
Myeloablative Haploidentical Transplant as an Alternative to Matched Sibling Transplant for Peripheral T-Cell Lymphomas
The number of HLA-haploidentical allogeneic hematopoietic stem-cell transplantation (Haplo-HSCT) is increasing. Comparative studies about Haplo-HSCT versus allo-HSCT with HLA-match...

