| Literature DB >> 31479583 |
Alison Foster1,2, Anna Zachariou3, Chey Loveday3, Tazeen Ashraf4, Edward Blair5, Jill Clayton-Smith6,7, Huw Dorkins8, Alan Fryer9, Blanca Gener10, David Goudie11, Alex Henderson12, Melita Irving4, Shelagh Joss13, Vaughan Keeley14, Nayana Lahiri15, Sally Ann Lynch16, Sahar Mansour15,17, Emma McCann9, Jenny Morton2, Nicole Motton18, Alexandra Murray19, Katie Riches14, Deborah Shears5, Zornitza Stark20,21, Elizabeth Thompson22,23, Julie Vogt2, Michael Wright12, Trevor Cole2, Katrina Tatton-Brown3,15,17.
Abstract
Sotos syndrome is an overgrowth-intellectual disability (OGID) syndrome caused by NSD1 pathogenic variants and characterized by a distinctive facial appearance, an intellectual disability, tall stature and/or macrocephaly. Other associated clinical features include scoliosis, seizures, renal anomalies, and cardiac anomalies. However, many of the published Sotos syndrome clinical descriptions are based on studies of children; the phenotype in adults with Sotos syndrome is not yet well described. Given that it is now 17 years since disruption of NSD1 was shown to cause Sotos syndrome, many of the children first reported are now adults. It is therefore timely to investigate the phenotype of 44 adults with Sotos syndrome and NSD1 pathogenic variants. We have shown that adults with Sotos syndrome display a wide spectrum of intellectual ability with functioning ranging from fully independent to fully dependent. Reproductive rates are low. In our cohort, median height in adult women is +1.9 SD and men +0.5 SD. There is a distinctive facial appearance in adults with a tall, square, prominent chin. Reassuringly, adults with Sotos syndrome are generally healthy with few new medical issues; however, lymphedema, poor dentition, hearing loss, contractures and tremor have developed in a small number of individuals.Entities:
Keywords: Sotos syndrome; adult phenotype; overgrowth-intellectual disability syndrome
Mesh:
Year: 2019 PMID: 31479583 DOI: 10.1002/ajmg.c.31738
Source DB: PubMed Journal: Am J Med Genet C Semin Med Genet ISSN: 1552-4868 Impact factor: 3.359