Abstract
Cleft lip and/or cleft palate are a common group of birth defects that further classify into syndromic and non-syndromic forms. The syndromic forms are usually accompanied by additional physical or cognitive abnormalities. Isolated cleft palate syndromes are less common; however, they are associated with a variety of congenital malformations and generally have an underlying genetic etiology. A single report in 2019 described a novel syndrome in three individuals, characterized by cleft palate, developmental delay and proliferative retinopathy due to a homozygous non-sense mutation in the LRRC32 gene encoding glycoprotein A repetitions predominant (GARP), a cell surface polypeptide crucial for the processing and maturation of transforming growth factor β (TGF-β). We describe a patient who presented with cleft palate, prenatal and postnatal severe growth retardation, global developmental delay, dysmorphic facial features and progressive vitreoretinopathy. Whole exome sequencing (WES) revealed a very rare homozygous missense variant in the LRRC32 gene, which resulted in substitution of a highly conserved isoleucine to threonine. Protein modeling suggested this variant may negatively affect GARP function on latent TGF-β activation. In summary, our report further expands the clinical features of cleft palate, proliferative retinopathy and developmental delay syndrome and emphasizes the association of LRRC32 pathogenic variants with this new syndrome.
| Original language | English |
|---|---|
| Article number | 859034 |
| Journal | Frontiers in Pediatrics |
| Volume | 10 |
| DOIs | |
| State | Published - 17 May 2022 |
Bibliographical note
Publisher Copyright:Copyright © 2022 Hexner-Erlichman, Fichtman, Zehavi, Khayat, Jabaly-Habib, Izhaki-Tavor, Dessau, Elpeleg and Spiegel.
Funding
We thank the family for their cooperation. We thank Cindy Cohen for professional language editing.
Keywords
- GARP protein
- LRRC32 gene
- TGF-β
- cleft palate
- retinopathy
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