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A newly found homozygous mutation in recombination activating gene 1 in a patient with leaky severe combined immunodeficiency disorder.
Salari, Fereshteh; Zaremehrjardi, Fatemeh; Arshi, Saba; Bemanian, Mohammad Hassan; Fallahpour, Morteza; Shokri, Sima; Seif, Farhad; Movahedi, Masoud; Nabavi, Mohammad.
Afiliación
  • Salari F; Department of Allergy & Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.
  • Zaremehrjardi F; Department of Allergy & Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.
  • Arshi S; Department of Allergy & Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.
  • Bemanian MH; Department of Allergy & Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.
  • Fallahpour M; Department of Allergy & Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.
  • Shokri S; Department of Allergy & Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.
  • Seif F; Academic Center for Education, Culture, and Research, Tehran University of Medical Sciences, Tehran, Iran.
  • Movahedi M; Neuroscience Research Center, Iran University of Medical Sciences, Tehran, Iran.
  • Nabavi M; Immunology Asthma, and Allergy Research Institute, Tehran University of Medical Sciences, Tehran, Iran. movahedm@sina.tums.ac.ir.
Mol Biol Rep ; 46(6): 6571-6575, 2019 Dec.
Article en En | MEDLINE | ID: mdl-31520268
The recombination activating genes, including RAG1 and RAG2, are essential for V(D)J somatic recombination in lymphocytes. Leaky severe combined immunodeficiency disorder (SCID) is characterized by normal or intermediate T cells and normal to absent B cells associated with partial T cell and B cell dysfunction. We present a newly found RAG1 deficiency in a 21-year-old boy with leaky SCID. Immunoglobulin levels, flow cytometry, and whole exome sequencing (WES) were evaluated. Flow cytometric analysis revealed a decreased number of CD3+, CD4+, and CD8+ T cells, and B cells whereas NK cell counts were normal. Immunoglobulin levels were also decreased. The WES revealed a newly found homozygous mutation of RAG1 gene (NM_000448: exon 2: c.C2275T). Atypical features, including leukopenia, candidiasis, and low lymphocyte counts in patients with late-onset combined immunodeficiency disorders (CID) such as leaky SCID due to RAG1 deficiency may result in misdiagnosis and inadequate therapy instead of adopting the curative hematopoietic stem cell transplantation in these patients.
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Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Inmunodeficiencia Combinada Grave / Proteínas de Homeodominio / Mutación Límite: Adult / Humans / Male Idioma: En Revista: Mol Biol Rep Año: 2019 Tipo del documento: Article País de afiliación: Irán

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Inmunodeficiencia Combinada Grave / Proteínas de Homeodominio / Mutación Límite: Adult / Humans / Male Idioma: En Revista: Mol Biol Rep Año: 2019 Tipo del documento: Article País de afiliación: Irán