Lethal Congenital Contracture Syndrome 11: A Case Report and Literature Review.
Miriam PotronyAntoni BorrellNarcís MasollerAlfons NadalLeonardo Rodriguez-CarunchioKarmele Saez de GordoaJuan Francisco Quesada-EspinosaJosé Luis Villanueva-CañasMontse PautaMeritxell JodarIrene MadrigalCelia BadenasMaria Isabel Alvarez-MoraLaia Rodriguez-RevengaPublished in: Journal of clinical medicine (2022)
Lethal congenital contracture syndrome 11 (LCCS11) is caused by homozygous or compound heterozygous variants in the GLDN gene on chromosome 15q21. GLDN encodes gliomedin, a protein required for the formation of the nodes of Ranvier and development of the human peripheral nervous system. We report a fetus with ultrasound alterations detected at 28 weeks of gestation. The fetus exhibited hydrops, short long bones, fixed limb joints, absent fetal movements, and polyhydramnios. The pregnancy was terminated and postmortem studies confirmed the prenatal findings: distal arthrogryposis, fetal growth restriction, pulmonary hypoplasia, and retrognathia. The fetus had a normal chromosomal microarray analysis. Exome sequencing revealed two novel compound heterozygous variants in the GLDN associated with LCCS11. This manuscript reports this case and performs a literature review of all published LCCS11 cases.
Keyphrases
- copy number
- case report
- genome wide
- early onset
- endothelial cells
- single cell
- dna methylation
- pulmonary hypertension
- magnetic resonance imaging
- pregnant women
- preterm infants
- minimally invasive
- preterm birth
- systematic review
- gene expression
- radiation therapy
- binding protein
- sentinel lymph node
- lymph node
- amino acid
- rectal cancer
- meta analyses
- case control
- electronic health record
- protein protein
- data analysis
- chemotherapy induced