Literature DB >> 8882111

Nager acrofacial dysostosis: management of a difficult airway.

R A Friedman1, E Wood, S M Pransky, A B Seid, D B Kearns.   

Abstract

Nager acrofacial dysostosis, first described by Nager and deReynier in 1948, is a rare syndrome characterized by mandibulofacial dysostosis with associated radial defects. The facial features include downward slanting palpebral fissures, absent eyelashes in the medial third of the lower lids, mandibular and malar hypoplasia, dysplastic ears with conductive deafness, and variable degrees of palatal clefting. Upper limb malformation is a constant feature of Nager syndrome and ranges from thumb hypoplasia to absence of the radial ray. The maxillo-mandibular hypoplasia and associated retroplaced tongue set the stage for early and significant upper airway obstruction. The craniofacial anomalies and associated trismus make emergent airway intubation challenging. We present a case of Nager syndrome with life threatening airway obstruction unresponsive to conservative management. This case and a review of the literature emphasize the importance of early tracheotomy for these patients.

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Year:  1996        PMID: 8882111     DOI: 10.1016/0165-5876(95)01304-0

Source DB:  PubMed          Journal:  Int J Pediatr Otorhinolaryngol        ISSN: 0165-5876            Impact factor:   1.675


  2 in total

Review 1.  Review of the Genetic Basis of Jaw Malformations.

Authors:  Mairaj K Ahmed; Xiaoqian Ye; Peter J Taub
Journal:  J Pediatr Genet       Date:  2016-10-12

2.  Clinical and mutation data in 12 patients with the clinical diagnosis of Nager syndrome.

Authors:  J C Czeschik; C Voigt; Y Alanay; B Albrecht; S Avci; D Fitzpatrick; D R Goudie; U Hehr; A J Hoogeboom; H Kayserili; P O Simsek-Kiper; L Klein-Hitpass; A Kuechler; V López-González; M Martin; S Rahmann; B Schweiger; M Splitt; B Wollnik; H-J Lüdecke; M Zeschnigk; D Wieczorek
Journal:  Hum Genet       Date:  2013-04-09       Impact factor: 4.132

  2 in total

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