Mandibular Distraction Osteogenesis in Monozygotic Twins With Structural-Dominant ALG8 Variants

Biallelic ALG8 mutations typically manifest as lethal multisystem congenital disorders of glycosylation (CDG-Ih). We report a unique case of monozygotic female twins presenting with Pierre Robin sequence (PRS) and orofacial-digital (OFD)-like features (cleft lip, tongue hamartomas, digital anomalies) without classic metabolic crises. Whole-exome sequencing identified compound heterozygous variants (c.140C>T and c.1349+1G>A). Severe airway obstruction was successfully managed through bilateral mandibular distraction osteogenesis (MDO), achieving complete respiratory weaning and oral feeding. This report provides critical evidence that specific ALG8 defects can manifest as a structural-dominant "metabolic ciliopathy," expanding the known phenotypic spectrum. Crucially for the craniofacial surgeon, this finding proves that MDO remains a safe, life-saving intervention even in complex genetic-metabolic backgrounds, providing a vital management precedent where theoretical risks of coagulation and bone metabolic issues might otherwise deter surgical intervention.

Authors

Institutions

Publication Details

Journal
Journal of Craniofacial Surgery
Published
2026-05-08
DOI
https://doi.org/10.1097/scs.0000000000012872
Primary Topic
Connective tissue disorders research
Type
article
Field-Weighted Citation Impact
0.00
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Mandibular Distraction Osteogenesis in Monozygotic Twins With Structural-Dominant ALG8 Variants

Weimin Shen, Xingyu Xia, Chuanzhi Wang, Jinlong Zou et al.
Journal of Craniofacial Surgery
Connective tissue disorders research
article

Mandibular Distraction Osteogenesis in Monozygotic Twins With Structural-Dominant ALG8 Variants

Weimin Shen, Xingyu Xia, Chuanzhi Wang, Jinlong Zou, Shigui Lu
article en

Abstract

Biallelic ALG8 mutations typically manifest as lethal multisystem congenital disorders of glycosylation (CDG-Ih). We report a unique case of monozygotic female twins presenting with Pierre Robin sequence (PRS) and orofacial-digital (OFD)-like features (cleft lip, tongue hamartomas, digital anomalies) without classic metabolic crises. Whole-exome sequencing identified compound heterozygous variants (c.140C>T and c.1349+1G>A). Severe airway obstruction was successfully managed through bilateral mandibular distraction osteogenesis (MDO), achieving complete respiratory weaning and oral feeding. This report provides critical evidence that specific ALG8 defects can manifest as a structural-dominant "metabolic ciliopathy," expanding the known phenotypic spectrum. Crucially for the craniofacial surgeon, this finding proves that MDO remains a safe, life-saving intervention even in complex genetic-metabolic backgrounds, providing a vital management precedent where theoretical risks of coagulation and bone metabolic issues might otherwise deter surgical intervention.

Journal of Craniofacial Surgery
Second Affiliated Hospital of Nanjing Medical University (CN)
Openalex Percentile: Top 5%
Connective tissue disorders research
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.