TY - JOUR
T1 - A novel POC1A variant in an alternatively spliced exon causes classic SOFT syndrome: clinical presentation of seven patients
AU - Al-Kindi, Adila
AU - Al-Shehhi, Maryam
AU - Westenberger, Ana
AU - Beetz, Christian
AU - Scott, Patrick
AU - Brandau, Oliver
AU - Abbasi-Moheb, Lia
AU - Yüksel, Zafer
AU - Bauer, Peter
AU - Rolfs, Arndt
AU - Grüning, Nana Maria
PY - 2020/1/1
Y1 - 2020/1/1
N2 - Biallelic pathogenic variants in POC1A are ultra rare. They have been reported in 13 families as causing either Short stature, Onychodysplasia, Facial dysmorphism, and hypoTrichosis (SOFT) syndrome, or a milder partially overlapping phenotype, variant POC1A-related syndrome. This pleiotropic effect is likely precipitated by the variant’s location and respective affected protein domain. Here, we describe seven patients from two consanguineous Omani families with classic SOFT syndrome and a novel homozygous POC1A variant (c.64G>T; p.(Val22Phe)), which is the first one described for the alternative exon 2. This result refines the POC1A mutational spectrum relevant for exertion of the described pleiotropic effect. Furthermore, six of our patients experienced recurrent mild to severe respiratory difficulties that have not been previously reported for SOFT syndrome and may be an underdiagnosed or a genotype-specific complication that warrants attention in future studies. Thus, our study unravels new aspects of the genotype-phenotype correlation suggested by previous reports.
AB - Biallelic pathogenic variants in POC1A are ultra rare. They have been reported in 13 families as causing either Short stature, Onychodysplasia, Facial dysmorphism, and hypoTrichosis (SOFT) syndrome, or a milder partially overlapping phenotype, variant POC1A-related syndrome. This pleiotropic effect is likely precipitated by the variant’s location and respective affected protein domain. Here, we describe seven patients from two consanguineous Omani families with classic SOFT syndrome and a novel homozygous POC1A variant (c.64G>T; p.(Val22Phe)), which is the first one described for the alternative exon 2. This result refines the POC1A mutational spectrum relevant for exertion of the described pleiotropic effect. Furthermore, six of our patients experienced recurrent mild to severe respiratory difficulties that have not been previously reported for SOFT syndrome and may be an underdiagnosed or a genotype-specific complication that warrants attention in future studies. Thus, our study unravels new aspects of the genotype-phenotype correlation suggested by previous reports.
UR - http://www.scopus.com/inward/record.url?scp=85075440102&partnerID=8YFLogxK
U2 - 10.1038/s10038-019-0693-2
DO - 10.1038/s10038-019-0693-2
M3 - Journal articles
C2 - 31767933
AN - SCOPUS:85075440102
SN - 1434-5161
VL - 65
SP - 193
EP - 197
JO - Journal of Human Genetics
JF - Journal of Human Genetics
IS - 2
ER -