Literature DB >> 31479583

The phenotype of Sotos syndrome in adulthood: A review of 44 individuals.

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.
© 2019 Wiley Periodicals, Inc.

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


  5 in total

1.  Drosophila NSD deletion induces developmental anomalies similar to those seen in Sotos syndrome 1 patients.

Authors:  Saeyan Choi; Bokyeong Song; Hyewon Shin; Chihyun Won; Taejoon Kim; Hideki Yoshida; Daewon Lee; Jongkyeong Chung; Kyoung Sang Cho; Im-Soon Lee
Journal:  Genes Genomics       Date:  2021-04-17       Impact factor: 1.839

Review 2.  Diagnostic Approach to Macrocephaly in Children.

Authors:  Andrea Accogli; Ana Filipa Geraldo; Gianluca Piccolo; Antonella Riva; Marcello Scala; Ganna Balagura; Vincenzo Salpietro; Francesca Madia; Mohamad Maghnie; Federico Zara; Pasquale Striano; Domenico Tortora; Mariasavina Severino; Valeria Capra
Journal:  Front Pediatr       Date:  2022-01-14       Impact factor: 3.418

3.  RNA Analysis and Clinical Characterization of a Novel Splice Variant in the NSD1 Gene Causing Familial Sotos Syndrome.

Authors:  Olatz Villate; Hiart Maortua; María-Isabel Tejada; Isabel Llano-Rivas
Journal:  Front Pediatr       Date:  2022-02-02       Impact factor: 3.418

4.  Cholesteatoma in Children with Sotos Syndrome.

Authors:  Colin Gerald Leonard; Sebastian Ranguis; Sharon Lynn Cushing; Susan Blaser; Adrian James
Journal:  J Int Adv Otol       Date:  2022-03       Impact factor: 1.316

Review 5.  Overgrowth Syndromes-Evaluation, Diagnosis, and Management.

Authors:  Joshua Manor; Seema R Lalani
Journal:  Front Pediatr       Date:  2020-10-30       Impact factor: 3.418

  5 in total

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