An anadysplasia-like, spontaneously remitting spondylometaphyseal dysplasia secondary to lamin B receptor (LBR) gene mutations: further definition of the phenotypic heterogeneity of LBR-bone dysplasias.

TitleAn anadysplasia-like, spontaneously remitting spondylometaphyseal dysplasia secondary to lamin B receptor (LBR) gene mutations: further definition of the phenotypic heterogeneity of LBR-bone dysplasias.
Publication TypeJournal Article
Year of Publication2015
AuthorsSobreira, N, Modaff, P, Steel, G, You, J, Nanda, S, Hoover-Fong, J, Valle, D, Pauli, RM
JournalAm J Med Genet A
Volume167A
Issue1
Pagination159-63
Date Published2015 Jan
ISSN1552-4833
KeywordsAdolescent, Adult, Child, Child, Preschool, Female, Humans, Infant, Infant, Newborn, Male, Mutation, Osteochondrodysplasias, Phenotype, Pregnancy, Radiography, Receptors, Cytoplasmic and Nuclear, Spine
Abstract

We describe a boy who has an anadysplasia-like spondylometaphyseal dysplasia. By whole exome sequencing he was shown to have compound heterozygous mutations of LBR that codes for the lamin B receptor. He shares many similarities with a case previously described, but in whom the early natural history could not be established [Borovik et al., 2013]. Thus, in addition to Greenberg dysplasia (a perinatal lethal disorder), homozygosity or compound heterozygosity of mutations in LBR can result in a mild, spontaneously regressing bone dysplasia.

DOI10.1002/ajmg.a.36808
Alternate JournalAm. J. Med. Genet. A
PubMed ID25348816
PubMed Central IDPMC4882113
Grant ListT32 GM007471 / GM / NIGMS NIH HHS / United States
T32 GM007814 / GM / NIGMS NIH HHS / United States
U54 HG006542 / HG / NHGRI NIH HHS / United States