Literature DB >> 8417743

Ear-nose-throat abnormalities in the CHARGE association.

D Morgan1, M Bailey, P Phelps, S Bellman, A Grace, R Wyse.   

Abstract

A comprehensive evaluation of the otolaryngological abnormalities in 50 patients with colobomata, heart defect, atresia of the choanae, retarded growth or development, genital hypoplasia, and ear anomalies or deafness (CHARGE) was performed. All the patients had ear abnormalities; 96% (48/50) had malformed pinnae, and 54% (27/50) had facial nerve palsies. Only 8% (4/50) had normal hearing, the commonest hearing defect being severe conductive or mixed loss. Eighty-four percent (42/50) of computed tomographic scans of the temporal bone were abnormal, the characteristic abnormality being the combination of a hypoplastic incus and absent semicircular canals. Eighty-six percent (43/50) of patients had upper airway abnormalities. Posterior choanal abnormalities occurred in 56% (28/50), and 42% (21/50) had retrognathia leading to intubation difficulties. Laryngotracheal abnormalities occurred in 38% (19/50), and 14% (7/50) required tracheostomies. Careful upper airway assessment is essential to avoid potentially lethal complications such as aspiration.

Entities:  

Mesh:

Year:  1993        PMID: 8417743     DOI: 10.1001/archotol.1993.01880130051006

Source DB:  PubMed          Journal:  Arch Otolaryngol Head Neck Surg        ISSN: 0886-4470


  18 in total

Review 1.  CHD associated with syndromic diagnoses: peri-operative risk factors and early outcomes.

Authors:  Benjamin J Landis; David S Cooper; Robert B Hinton
Journal:  Cardiol Young       Date:  2015-09-08       Impact factor: 1.093

2.  CHARGE syndrome: the phenotypic spectrum of mutations in the CHD7 gene.

Authors:  M C J Jongmans; R J Admiraal; K P van der Donk; L E L M Vissers; A F Baas; L Kapusta; J M van Hagen; D Donnai; T J de Ravel; J A Veltman; A Geurts van Kessel; B B A De Vries; H G Brunner; L H Hoefsloot; C M A van Ravenswaaij
Journal:  J Med Genet       Date:  2005-09-09       Impact factor: 6.318

3.  Absent semicircular canals in CHARGE syndrome: radiologic spectrum of findings.

Authors:  A K Morimoto; R H Wiggins; P A Hudgins; G L Hedlund; B Hamilton; S K Mukherji; S A Telian; H R Harnsberger
Journal:  AJNR Am J Neuroradiol       Date:  2006-09       Impact factor: 3.825

4.  Temporal bone computed tomography findings in bilateral sensorineural hearing loss.

Authors:  D E Bamiou; P Phelps; T Sirimanna
Journal:  Arch Dis Child       Date:  2000-03       Impact factor: 3.791

5.  CHARGE Syndrome.

Authors:  S K Jatana; K Venkatnarayan; Mng Nair
Journal:  Med J Armed Forces India       Date:  2011-07-21

6.  Imaging Modality of Choice for Pre-Operative Cochlear Imaging: HRCT vs. MRI Temporal Bone.

Authors:  Poornima Digge; Rajendra N Solanki; Dipali C Shah; Rajesh Vishwakarma; Sandeep Kumar
Journal:  J Clin Diagn Res       Date:  2016-10-01

7.  Tracheoesophageal fistula with esophageal atresia with absent external ear: a unusual association.

Authors:  Vijai D Upadhyaya; Ajay N Gangopadhyay; Punit K Srivastava; Zaheer Hasan; Shiv P Sharma
Journal:  Indian J Surg       Date:  2012-05-01       Impact factor: 0.656

8.  Phenotypic spectrum of CHARGE syndrome in fetuses with CHD7 truncating mutations correlates with expression during human development.

Authors:  D Sanlaville; H C Etchevers; M Gonzales; J Martinovic; M Clément-Ziza; A-L Delezoide; M-C Aubry; A Pelet; S Chemouny; C Cruaud; S Audollent; C Esculpavit; G Goudefroye; C Ozilou; C Fredouille; N Joye; N Morichon-Delvallez; Y Dumez; J Weissenbach; A Munnich; J Amiel; F Encha-Razavi; S Lyonnet; M Vekemans; T Attié-Bitach
Journal:  J Med Genet       Date:  2005-09-16       Impact factor: 6.318

9.  Mature middle and inner ears express Chd7 and exhibit distinctive pathologies in a mouse model of CHARGE syndrome.

Authors:  Elizabeth A Hurd; Meredith E Adams; Wanda S Layman; Donald L Swiderski; Lisa A Beyer; Karin E Halsey; Jennifer M Benson; Tzy-Wen Gong; David F Dolan; Yehoash Raphael; Donna M Martin
Journal:  Hear Res       Date:  2011-08-23       Impact factor: 3.208

10.  CHD7, the gene mutated in CHARGE syndrome, regulates genes involved in neural crest cell guidance.

Authors:  Yvonne Schulz; Peter Wehner; Lennart Opitz; Gabriela Salinas-Riester; Ernie M H F Bongers; Conny M A van Ravenswaaij-Arts; Josephine Wincent; Jacqueline Schoumans; Jürgen Kohlhase; Annette Borchers; Silke Pauli
Journal:  Hum Genet       Date:  2014-04-13       Impact factor: 4.132

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