| Literature DB >> 25899773 |
Hao Liu1, Sarah L Sawyer2,3, Monika Gos4, David Grynspan5, Kheirie Issa6, Raveena Ramphal7, Carmen Rotaru8, Jacek Majewski9, Kym M Boycott2,3, Gail Graham2,3, Matthew Bromwich10.
Abstract
Fibrodysplasia ossificans progressiva (FOP) is a rare genetic disorder characterized by congenital malformations of the great toes and progressive heterotopic ossification of connective tissue that begins during the first decade of life. Our patient presented with intrauterine growth retardation, respiratory distress, neonatal onset soft tissue masses, bilateral hallux valgus, and congenital anomalies of the thyroid and uterus. She was initially diagnosed with atypical infantile myofibromatosis based on clinical and pathological findings. She underwent whole-exome sequencing (WES) as part of the FORGE study to identify the gene for infantile myofibromatosis; however a de novo dominant mutation in ACVR1 (NM_001105.4:c.617G>A) revised the diagnosis to FOP. This patient highlights the utility of WES as an early diagnostic tool in the investigation of patients with unusual presentations of rare diseases, thereby providing clinicians with accurate molecular diagnoses and the opportunity to tailor clinical management to improve patient care.Entities:
Keywords: fibrodysplasia ossificans progressiva; hallux valgus; heterotopic ossification; whole exome sequencing
Mesh:
Substances:
Year: 2015 PMID: 25899773 DOI: 10.1002/ajmg.a.36969
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802