Literature DB >> 10982485

Asymptomatic laryngeal malformations are common in patients with Pallister-Hall syndrome.

F Ondrey1, A Griffith, C Van Waes, S Rudy, K Peters, L McCullagh, L G Biesecker.   

Abstract

Pallister-Hall syndrome (PHS) comprises hypothalamic hamartoma, polydactyly, pituitary dysfunction, laryngotracheal cleft, imperforate anus, and other anomalies. Some patients with PHS have a bifid epiglottis, a rare malformation. Greig cephalopolysyndactyly syndrome (GCPS) comprises polydactyly with craniofacial malformations without the PHS malformations. Both disorders are caused by mutations in the GLI3 gene. Laryngoscopy on 26 subjects with PHS showed that 15 had a bifid or cleft epiglottis (58%) and none of 14 subjects with GCPS had a cleft epiglottis. The malformed epiglottis was asymptomatic in all of the prospectively evaluated subjects. One additional PHS subject was found to have bifid epiglottis and a posterior laryngeal cleft on autopsy. We conclude that bifid epiglottis is common in PHS but not GCPS. Posterior laryngeal clefts are an uncommon manifestation of PHS and are identified only in severely affected patients. The diagnosis of a bifid epiglottis should prompt a thorough search for other sometimes asymptomatic anomalies of PHS to provide better medical care and recurrence risk assessment for affected individuals and families.

Entities:  

Mesh:

Year:  2000        PMID: 10982485     DOI: 10.1002/1096-8628(20000904)94:1<64::aid-ajmg13>3.0.co;2-d

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  9 in total

1.  Molecular and clinical analyses of Greig cephalopolysyndactyly and Pallister-Hall syndromes: robust phenotype prediction from the type and position of GLI3 mutations.

Authors:  Jennifer J Johnston; Isabelle Olivos-Glander; Christina Killoran; Emma Elson; Joyce T Turner; Kathryn F Peters; Margaret H Abbott; David J Aughton; Arthur S Aylsworth; Michael J Bamshad; Carol Booth; Cynthia J Curry; Albert David; Mary Beth Dinulos; David B Flannery; Michelle A Fox; John M Graham; Dorothy K Grange; Alan E Guttmacher; Mark C Hannibal; Wolfram Henn; Raoul C M Hennekam; Lewis B Holmes; H Eugene Hoyme; Kathleen A Leppig; Angela E Lin; Patrick Macleod; David K Manchester; Carlo Marcelis; Laura Mazzanti; Emma McCann; Marie T McDonald; Nancy J Mendelsohn; John B Moeschler; Billur Moghaddam; Giovanni Neri; Ruth Newbury-Ecob; Roberta A Pagon; John A Phillips; Laurie S Sadler; Joan M Stoler; David Tilstra; Catherine M Walsh Vockley; Elaine H Zackai; Touran M Zadeh; Louise Brueton; Graeme Charles M Black; Leslie G Biesecker
Journal:  Am J Hum Genet       Date:  2005-02-28       Impact factor: 11.025

2.  Mutational analysis of NOG in esophageal atresia and tracheoesophageal fistula patients.

Authors:  Andrew J Murphy; Yina Li; Joshua B Pietsch; Chin Chiang; Harold N Lovvorn
Journal:  Pediatr Surg Int       Date:  2011-11-15       Impact factor: 1.827

Review 3.  What you can learn from one gene: GLI3.

Authors:  L G Biesecker
Journal:  J Med Genet       Date:  2006-06       Impact factor: 6.318

4.  Congenital Bilateral Saccular Cysts and Bifid Epiglottis: Presentation and Management.

Authors:  Rawan Arif; Talal Al-Khatib; Razan Daghistani; Maher Shalabi
Journal:  Indian J Otolaryngol Head Neck Surg       Date:  2015-12-19

Review 5.  Developmental basis of trachea-esophageal birth defects.

Authors:  Nicole A Edwards; Vered Shacham-Silverberg; Leelah Weitz; Paul S Kingma; Yufeng Shen; James M Wells; Wendy K Chung; Aaron M Zorn
Journal:  Dev Biol       Date:  2021-05-21       Impact factor: 3.582

6.  Cilia-mediated Hedgehog signaling controls form and function in the mammalian larynx.

Authors:  Jacqueline M Tabler; Maggie M Rigney; Gordon J Berman; Swetha Gopalakrishnan; Eglantine Heude; Hadeel Adel Al-Lami; Basil Z Yannakoudakis; Rebecca D Fitch; Christopher Carter; Steven Vokes; Karen J Liu; Shahragim Tajbakhsh; Se Roian Egnor; John B Wallingford
Journal:  Elife       Date:  2017-02-13       Impact factor: 8.140

7.  Pallister-Hall syndrome diagnosed in a young man after an acute adrenal crisis.

Authors:  Anis Grassa; Meriem Yazidi; Jihene Marrakchi; Chaima Bel Hadj Sliman; Ibtissem Oueslati; Melika Chihaoui
Journal:  Clin Case Rep       Date:  2022-08-22

8.  Pallister-Hall syndrome with orofacial narrowing and tethered cord: a case report.

Authors:  Femia Hayek
Journal:  J Med Case Rep       Date:  2018-11-29

9.  Bifid epiglottis, high-arched palate, and mental disorder in a patient with Pallister-Hall syndrome.

Authors:  Masanori Tsukamoto; Takashi Hitosugi; Hitoshi Yamanaka; Takeshi Yokoyama
Journal:  Indian J Anaesth       Date:  2018-10
  9 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.