Clinical implications ofELA2-,HAX1-, and G-CSF-receptor (CSF3R) mutations in severe congenital neutropenia

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G-CSF receptor (CSF3R) mutations in X-linked neutropenia evolving to acute myeloid leukemia or myelodysplasia.

X-linked neutropenia (XLN) is a rare form of Congenital Neutropenia (CN) caused by inherited gain-of-function mutations of WAS. Here we report 2 cases of the original L270P X-linked neutropenia kindred that evolved to MDS or AML, with acquisition of G-CSFR (CSF3R) mutations and monosomy 7. Thus, leukemic transformation with acquisition of CSF3R mutations and monosomy 7 is not restricted to clas...

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Significance of neutrophil elastase mutations versus G-CSF receptor mutations for leukemic progression of congenital neutropenia.

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HAX1 mutation in an infant with severe congenital neutropenia.

Severe congenital neutropenia (SCN) is a rare primary myelopoiesis disorder, characterized by persistent severe neutropenia and early-onset bacterial infections. Herein, we describe an 11-month-old male who was referred with recurrent cutaneous infections and chronic diarrhea. Serial complete blood counts indicated persistent neutropenia. Bone marrow aspiration of the patient demonstrated matur...

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In vivo expansion of cells expressing acquired CSF3R mutations in patients with severe congenital neutropenia.

Severe congenital neutropenia (CN) is a rare bone marrow failure syndrome with a high incidence of acute leukemia. In previous studies, we could show that point mutations in the gene for the granulocyte colony-stimulating factor (G-CSF) receptor CSF3R are a highly predictive marker for leukemic development in CN patients. To find out at which stage of hematopoietic development these mutations e...

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Novel HAX1 gene mutations associated to neurodevelopment abnormalities in two Italian patients with severe congenital neutropenia.

We read with interest the recent perspective article by Klein. 1 Genetic analysis in individuals with severe congenital neutropenia (SCN) indicates that 60% of cases were attributable to heterozygous mutation in ELA2 gene encoding neutrophil elastase. 2 Homozygous mutation in HAX1 gene has been identified in patients with autosomal recessive form of SCN (Kostmann syndrome). 3 Patients with ELA2...

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ژورنال

عنوان ژورنال: British Journal of Haematology

سال: 2009

ISSN: 0007-1048,1365-2141

DOI: 10.1111/j.1365-2141.2008.07425.x