| Literature DB >> 20499091 |
Catherine M L Roberts1, Janet E Angus, Ian H Leach, Elizabeth M McDermott, David A Walker, Jane C Ravenscroft.
Abstract
Genetic conditions are increasingly recognised as a cause of multisystem diseases in children. We report a 6-year-old boy with hypohidrotic ectodermal dysplasia, immunodeficiency, osteopetrosis and lymphoedema, associated with a novel mutation in the NF-κβ essential modulator (NEMO) gene. He is the longest surviving of three reported boys with these clinical features. Hypohidrotic ectodermal dysplasia, a congenital disorder of teeth, hair and eccrine sweat glands is most commonly inherited as an X-linked recessive trait. Associated immunodeficiency (HED-ID) may give rise to serious infections in early life. Mutations in the NEMO gene give rise to a heterogeneous group of disorders, including the X-linked dominant disorder incontinentia pigmenti. This is characterised by typical skin changes leading to linear pigmentary change and variable associated features; in males, prenatal death usually occurs. Our patient, like one if the previous cases and all of their mothers, demonstrates features of incontinentia pigmenti.Entities:
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Year: 2010 PMID: 20499091 DOI: 10.1007/s00431-010-1206-7
Source DB: PubMed Journal: Eur J Pediatr ISSN: 0340-6199 Impact factor: 3.183