Literature DB >> 20499091

A novel NEMO gene mutation causing osteopetrosis, lymphoedema, hypohidrotic ectodermal dysplasia and immunodeficiency (OL-HED-ID).

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.

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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


  6 in total

1.  X-linked anhidrotic ectodermal dysplasia with immunodeficiency is caused by impaired NF-kappaB signaling.

Authors:  R Döffinger; A Smahi; C Bessia; F Geissmann; J Feinberg; A Durandy; C Bodemer; S Kenwrick; S Dupuis-Girod; S Blanche; P Wood; S H Rabia; D J Headon; P A Overbeek; F Le Deist; S M Holland; K Belani; D S Kumararatne; A Fischer; R Shapiro; M E Conley; E Reimund; H Kalhoff; M Abinun; A Munnich; A Israël; G Courtois; J L Casanova
Journal:  Nat Genet       Date:  2001-03       Impact factor: 38.330

2.  Incontinentia pigmenti in a surviving male is accompanied by hypohidrotic ectodermal dysplasia and recurrent infection.

Authors:  S Mansour; H Woffendin; S Mitton; I Jeffery; T Jakins; S Kenwrick; V A Murday
Journal:  Am J Med Genet       Date:  2001-03-01

Review 3.  Male cases of incontinentia pigmenti: case report and review.

Authors:  A E Scheuerle
Journal:  Am J Med Genet       Date:  1998-05-18

4.  A novel X-linked disorder of immune deficiency and hypohidrotic ectodermal dysplasia is allelic to incontinentia pigmenti and due to mutations in IKK-gamma (NEMO).

Authors:  J Zonana; M E Elder; L C Schneider; S J Orlow; C Moss; M Golabi; S K Shapira; P A Farndon; D W Wara; S A Emmal; B M Ferguson
Journal:  Am J Hum Genet       Date:  2000-10-24       Impact factor: 11.025

5.  Osteopetrosis, lymphedema, anhidrotic ectodermal dysplasia, and immunodeficiency in a boy and incontinentia pigmenti in his mother.

Authors:  Sophie Dupuis-Girod; Nadège Corradini; Smail Hadj-Rabia; Jean-Christophe Fournet; Laurence Faivre; Françoise Le Deist; Philippe Durand; Rainer Döffinger; Asma Smahi; Alain Israel; Gilles Courtois; Nicole Brousse; Stéphane Blanche; Arnold Munnich; Alain Fischer; Jean-Laurent Casanova; Christine Bodemer
Journal:  Pediatrics       Date:  2002-06       Impact factor: 7.124

Review 6.  Molecular aspects of hypohidrotic ectodermal dysplasia.

Authors:  Marja L Mikkola
Journal:  Am J Med Genet A       Date:  2009-09       Impact factor: 2.802

  6 in total
  15 in total

1.  Absence of an osteopetrosis phenotype in IKBKG (NEMO) mutation-positive women: A case-control study.

Authors:  Morten Frost; Michaela Tencerova; Christina M Andreasen; Thomas L Andersen; Charlotte Ejersted; Dea Svaneby; Weimin Qui; Moustapha Kassem; Allahdad Zarei; William H McAlister; Deborah J Veis; Michael P Whyte; Anja L Frederiksen
Journal:  Bone       Date:  2019-01-16       Impact factor: 4.398

Review 2.  Genetic lessons learned from X-linked Mendelian susceptibility to mycobacterial diseases.

Authors:  Jacinta Bustamante; Capucine Picard; Stéphanie Boisson-Dupuis; Laurent Abel; Jean-Laurent Casanova
Journal:  Ann N Y Acad Sci       Date:  2011-12       Impact factor: 5.691

3.  Prioritization of genes involved in endothelial cell apoptosis by their implication in lymphedema using an analysis of associative gene networks with ANDSystem.

Authors:  Olga V Saik; Vadim V Nimaev; Dilovarkhuja B Usmonov; Pavel S Demenkov; Timofey V Ivanisenko; Inna N Lavrik; Vladimir A Ivanisenko
Journal:  BMC Med Genomics       Date:  2019-03-13       Impact factor: 3.063

4.  Genetic Polymorphisms of Nuclear Factor-κB Family Affect the Bone Mineral Density Response to Zoledronic Acid Therapy in Postmenopausal Chinese Women.

Authors:  Wen-Jie Wang; Jin-Wei He; Wen-Zhen Fu; Chun Wang; Zhen-Lin Zhang
Journal:  Genes (Basel)       Date:  2022-07-27       Impact factor: 4.141

Review 5.  Genetics of lymphatic anomalies.

Authors:  Pascal Brouillard; Laurence Boon; Miikka Vikkula
Journal:  J Clin Invest       Date:  2014-03-03       Impact factor: 14.808

Review 6.  Genetics of Osteopetrosis.

Authors:  Eleonora Palagano; Ciro Menale; Cristina Sobacchi; Anna Villa
Journal:  Curr Osteoporos Rep       Date:  2018-02       Impact factor: 5.096

Review 7.  NF-κB signaling and bone resorption.

Authors:  Y Abu-Amer
Journal:  Osteoporos Int       Date:  2013-03-07       Impact factor: 4.507

Review 8.  Granulomatous skin lesions, severe scrotal and lower limb edema due to mycobacterial infections in a child with complete IFN-γ receptor-1 deficiency.

Authors:  Neslihan Edeer Karaca; Stephanie Boisson-Dupuis; Güzide Aksu; Jacinta Bustamante; Gulsen Kandiloglu; Nazan Ozsan; Mine Hekimgil; Jean-Laurent Casanova; Necil Kutukculer
Journal:  Immunotherapy       Date:  2012-11       Impact factor: 4.196

9.  Myeloid Deletion of Nemo Causes Osteopetrosis in Mice Owing to Upregulation of Transcriptional Repressors.

Authors:  Gaurav Swarnkar; Kyuhwan Shim; Amjad M Nasir; Kuljeet Seehra; Hung-Po Tim Chen; Gabriel Mbalaviele; Yousef Abu-Amer
Journal:  Sci Rep       Date:  2016-07-20       Impact factor: 4.379

Review 10.  Immunogenetics of the Ocular Anterior Segment: Lessons from Inherited Disorders.

Authors:  Jasmine Y Serpen; Stephen T Armenti; Lev Prasov
Journal:  J Ophthalmol       Date:  2021-06-28       Impact factor: 1.909

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