Lamellar ichthyosis (LI) is an inherited autosomal recessive disorder of cornification. It was recently demonstrated to result from deleterious mutations in the transglutaminase 1 (TGM1) gene. However, the disease was shown to be genetically heterogeneous, since some families were found to be unlinked to TGM1. Homozygosity mapping on three consanguinous families originating from Morocco shows (i) absence of linkage with TGM1 and other regions of the genome containing genes involved in cornification, and (ii) location of a second disease-causing gene on chromosome 2q33-35. A maximum two-point lodscore of 7.60 was obtained with D2S157 for theta = 0. The analysis of recombination events places the gene within a 7-8 cM interval. Additional consanguinous pedigrees were also demonstrated to be unlinked both to TGM1 and to 2q33-35, suggesting the existence of at least a third disease-causing gene.

Download full-text PDF

Source
http://dx.doi.org/10.1093/hmg/5.4.555DOI Listing

Publication Analysis

Top Keywords

lamellar ichthyosis
8
chromosome 2q33-35
8
unlinked tgm1
8
disease-causing gene
8
mapping second
4
second locus
4
locus lamellar
4
ichthyosis chromosome
4
2q33-35 lamellar
4
ichthyosis inherited
4

Similar Publications

Background: Autosomal recessive congenital ichthyosis (ARCI) is a group of genetic skin disorders characterized by abnormal keratinization, leading to significant health issues and reduced quality of life. ARCI encompasses harlequin ichthyosis (HI), congenital ichthyosiform erythroderma (CIE), and lamellar ichthyosis (LI). While all ARCI genes are linked to LI and CIE, HI is specifically associated with severe mutations in the gene.

View Article and Find Full Text PDF

Collodion baby is a rare congenital condition marked by a parchment-like membrane covering the body, often leading to complications such as bilateral ectropion. This condition poses risks of exposure keratopathy and other ocular issues. We present a case series of five infants with congenital bilateral ectropion associated with collodion babies, all born prematurely.

View Article and Find Full Text PDF

A cellular disease model toward gene therapy of -dependent lamellar ichthyosis.

Mol Ther Methods Clin Dev

September 2024

Center for Regenerative Medicine "Stefano Ferrari", Department of Life Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Lamellar ichthyosis (LI) is a chronic disease, mostly caused by mutations in the gene, marked by impaired skin barrier formation. No definitive therapies are available, and current treatments aim at symptomatic relief. LI mouse models often fail to faithfully replicate the clinical and histopathological features of human skin conditions.

View Article and Find Full Text PDF
Article Synopsis
  • Autosomal recessive congenital ichthyosis (ARCI) is a rare skin condition primarily characterized by skin scaling and hair abnormalities, which are often overlooked regarding their impact on patients' quality of life.
  • This study involved clinical and trichoscopic examinations of 30 ARCI patients over three years, revealing that alopecia affects a significant number, with various patterns of hair loss documented among participants.
  • Notable trichoscopic findings included scaling and different hair types that correlate with the severity of ARCI, suggesting that hair health is a crucial component of this condition often underestimated in prior research.
View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!