Immunodeficiency, Centromeric Region Instability, and Facial Anomalies Syndrome (ICF) in a Boy with Variable Clinical and Immunological Presentations.


Journal

Iranian journal of allergy, asthma, and immunology
ISSN: 1735-5249
Titre abrégé: Iran J Allergy Asthma Immunol
Pays: Iran
ID NLM: 101146178

Informations de publication

Date de publication:
17 Apr 2021
Historique:
received: 16 05 2020
accepted: 08 12 2020
entrez: 27 4 2021
pubmed: 28 4 2021
medline: 10 11 2021
Statut: epublish

Résumé

Immunodeficiency, centromeric instability, and facial anomalies (ICF) syndrome is a rare primary immunodeficiency disorder characterized by recurrent infections and low immunoglobulin levels due to variable combined immunodeficiency, and centromeric region instability, and facial dysmorphism. We describe a 12-year-old boy with recurrent respiratory tract infections, facial anomalies, scoliosis, and psychomotor retardation. He had recurrent pneumonia with low serum IgG and IgM levels during infancy and preschool age. Later at the age of 10, he developed recurrent ear infections. An IgA and IgM deficiency was found accompanied by a normal B-cell and T-cell count as well as an impaired candida-induced T-cell proliferation. Further evaluations revealed a missense mutation in the DNMT3B gene on chromosome 20. Chromosomal analysis showed a sunburst multi-radial feature on chromosome 1, which is a hallmark of ICF syndrome. The genetic mutation and chromosomal abnormality along with clinical findings are compatible with the diagnosis of ICF syndrome. To the best of our knowledge, this is the first time that scoliosis is observed in an ICF patient. The additional variable clinical symptoms in the case were the presence of spastic gait as well as hypogammaglobulinemia with immunoglobulin isotype switch at different ages.

Identifiants

pubmed: 33904683

Substances chimiques

Immunoglobulins 0
DNA (Cytosine-5-)-Methyltransferases EC 2.1.1.37

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

249-254

Auteurs

Mohammad Hassan Bemanian (MH)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. mhbemanian@yahoo.com.

Saba Arshi (S)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. sabaarshi@yahoo.com.

Mohammad Nabavi (M)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. Mnabavi44@yahoo.com.

Mohammad Vafaee-Shahi (M)

Pediatric Growth and Development Research Center, Iran University of Medical Sciences, Tehran, Iran. dr.vafaeeshahi@yahoo.com.

Morteza Fallahpour (M)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. fallahpour.m@iums.ac.ir.

Sima Shokri (S)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. dr.shokri.83@gmail.com.

Afshin Rezaeifar (A)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. Rezaeifar.a@iums.ac.ir.

Hossein Shahzadi (H)

Department of Pediatric cardiology, Iran University of Medical Sciences, Tehran, Iran. hossein.shahzadi@yahoo.com.

Fatemeh Atashrazm (F)

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran. dr.atashrazm@gmail.com.

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Classifications MeSH