Immunodeficiency, Centromeric Region Instability, and Facial Anomalies Syndrome (ICF) in a Boy with Variable Clinical and Immunological Presentations.
Centromere
Child
Chromosomal Instability
DNA (Cytosine-5-)-Methyltransferases
/ genetics
Face
/ abnormalities
Gait Disorders, Neurologic
/ blood
Humans
Immunoglobulins
/ blood
Immunologic Deficiency Syndromes
/ blood
Male
Mutation, Missense
Pelvis
/ abnormalities
Scoliosis
/ blood
DNA Methyltransferase 3B
Chromosomal instability
DNA methyltransferase 3B
Immunodeficiency
Scoliosis
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
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.
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