Case Report: Novel pathogenic variant in autosomal recessive WNT10A-related odonto-onycho-dermal dysplasia

Odonto-onycho-dermal dysplasia (OODD) is a rare, autosomal recessive disorder caused by pathogenic variants in the WNT10A gene, which plays a crucial role in ectodermal tissue development, including teeth, hair, nails, and skin. We describe an 18-year-old male of Afghan origin, born to consanguineous parents, who was evaluated during an extensive genetic family screening for an unrelated ataxia variant. The patient reported long-standing dental abnormalities, sparse hair, nail defects and hyperhidrosis. Examination revealed severe hypodontia, atypical dental morphology, retained deciduous teeth, smooth tongue due to reduced lingual papillae and dysmorphic nail growth. Trio whole-genome sequencing identified a novel homozygous frameshift variant in WNT10A [c.694del, p. (Arg232GlyfsTer11)] in the index patient. Segregation within the trio confirmed heterozygosity in both parents, supporting the diagnosis of OODD. Our case expands the spectrum of WNT10A mutations causing OODD by identifying a novel causative variant and highlights the value of comprehensive genetic screening, especially in populations with high consanguinity for assessing incidental clinically relevant findings and diagnosing rare conditions.

Authors

Institutions

Publication Details

Journal
Frontiers in Genetics
Published
2026-03-26
DOI
https://doi.org/10.3389/fgene.2026.1750692
Primary Topic
Bone and Dental Protein Studies
Type
article
Field-Weighted Citation Impact
0.00
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Case Report: Novel pathogenic variant in autosomal recessive WNT10A-related odonto-onycho-dermal dysplasia

Theodorοs Mavridis, Chrysoula Koniari, Marianna Kalaszi, Ciaran Moore
Frontiers in Genetics
Bone and Dental Protein Studies
article

Case Report: Novel pathogenic variant in autosomal recessive WNT10A-related odonto-onycho-dermal dysplasia

Theodorοs Mavridis, Chrysoula Koniari, Marianna Kalaszi, Ciaran Moore
article en

Abstract

Odonto-onycho-dermal dysplasia (OODD) is a rare, autosomal recessive disorder caused by pathogenic variants in the WNT10A gene, which plays a crucial role in ectodermal tissue development, including teeth, hair, nails, and skin. We describe an 18-year-old male of Afghan origin, born to consanguineous parents, who was evaluated during an extensive genetic family screening for an unrelated ataxia variant. The patient reported long-standing dental abnormalities, sparse hair, nail defects and hyperhidrosis. Examination revealed severe hypodontia, atypical dental morphology, retained deciduous teeth, smooth tongue due to reduced lingual papillae and dysmorphic nail growth. Trio whole-genome sequencing identified a novel homozygous frameshift variant in WNT10A [c.694del, p. (Arg232GlyfsTer11)] in the index patient. Segregation within the trio confirmed heterozygosity in both parents, supporting the diagnosis of OODD. Our case expands the spectrum of WNT10A mutations causing OODD by identifying a novel causative variant and highlights the value of comprehensive genetic screening, especially in populations with high consanguinity for assessing incidental clinically relevant findings and diagnosing rare conditions.

Frontiers in GeneticsVol. 17
Tallaght University Hospital (IE), Eginition Hospital (GR), Dublin Dental University Hospital (IE)
Openalex Percentile: Top 5%
Bone and Dental Protein Studies
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.