Literature DB >> 20445326

[Molecular analysis of two cases of severe congenital neutropenia].

Joonhong Park1, Myungshin Kim, Jihyang Lim, Yonggoo Kim, Bin Cho, Yeon Joon Park, Kyungja Han.   

Abstract

Severe congenital neutropenia is a rare hematological disease characterized by a selective decrease in circulating neutrophils, maturation arrest of granulocytic precursors at the promyelocyte stage, and recurrence of infections. A 2-month-old male infant (patient A) and a 14-month-old female child (patient B) were referred to our hospital due to severe neutropenia. Sequencing analysis of ELA2 and HAX1 genes was performed. Two single nucleotide polymorphisms of HAX1 gene were found. They were 5,104T-->G point mutation of exon 1 and 5,474A-->G point mutation of intron 1 in HAX1 gene. The mutation of ELA2 gene was not found. The patient A showed a good response to granulocyte colony-stimulating factor (G-CSF) treatment and the absolute neutrophil count recovered to 1,195/microL. But the patient B showed a partial response to G-CSF treatment and experienced several episodes of herpetic gingivostomatitis, oral ulcer, acute pharyngotonsillitis and otitis media during follow-up.

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Year:  2010        PMID: 20445326     DOI: 10.3343/kjlm.2010.30.2.111

Source DB:  PubMed          Journal:  Korean J Lab Med        ISSN: 1598-6535


  3 in total

Review 1.  Current insights into inherited bone marrow failure syndromes.

Authors:  Nack-Gyun Chung; Myungshin Kim
Journal:  Korean J Pediatr       Date:  2014-08-25

2.  Different clinical phenotypes in familial severe congenital neutropenia cases with same mutation of the ELANE gene.

Authors:  Hye-Kyung Cho; In Sang Jeon
Journal:  J Korean Med Sci       Date:  2014-02-27       Impact factor: 2.153

3.  Both Granulocytic and Non-Granulocytic Blood Cells Are Affected in Patients with Severe Congenital Neutropenia and Their Non-Neutropenic Family Members: An Evaluation of Morphology, Function, and Cell Death

Authors:  Lale Olcay; Şule Ünal; Hüseyin Onay; Esra Erdemli; Ayşenur Öztürk; Deniz Billur; Ayşe Metin; Hamza Okur; Yıldız Yıldırmak; Yahya Büyükaşık; Aydan İkincioğulları; Mesude Falay; Gülsüm Özet; Sevgi Yetgin
Journal:  Turk J Haematol       Date:  2018-07-24       Impact factor: 1.831

  3 in total

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