Literature DB >> 4095043

Short trachea, with reduced number of cartilage rings--a hitherto unrecognized feature of DiGeorge syndrome.

K Sein, T R Wells, B H Landing, C R Chow.   

Abstract

Adequate tracheas from 6 of 14 patients with DiGeorge syndrome (2 complete and 4 partial forms) were available for tracheal ring study. The number of tracheal rings in these patients was significantly low (mean +/- SD 14.7 +/- 1.4). This is consistent with an effect of deficient blood supply to the fetal cervical region, a mechanism that has been proposed to explain the defective development of thymus and parathyroid glands in DiGeorge syndrome. To our knowledge this lesion has not been described as one of the malformations found in the syndrome. In 8 of 14 (57%) patients with DiGeorge syndrome, common origin of the carotid arteries was found, and 4 of these 8 patients showed minor tracheal compression. These findings support the proposal that abnormal blood supply to the region of the third and fourth pharyngeal arches may play a role in the pathogenesis of DiGeorge syndrome.

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Year:  1985        PMID: 4095043     DOI: 10.3109/15513818509025905

Source DB:  PubMed          Journal:  Pediatr Pathol        ISSN: 0277-0938


  2 in total

Review 1.  Head and neck manifestations of 22q11.2 deletion syndromes.

Authors:  Tal Marom; Yehudah Roth; Abraham Goldfarb; Udi Cinamon
Journal:  Eur Arch Otorhinolaryngol       Date:  2011-08-23       Impact factor: 2.503

2.  Decreased embryonic retinoic acid synthesis results in a DiGeorge syndrome phenotype in newborn mice.

Authors:  Julien Vermot; Karen Niederreither; Jean-Marie Garnier; Pierre Chambon; Pascal Dollé
Journal:  Proc Natl Acad Sci U S A       Date:  2003-01-31       Impact factor: 11.205

  2 in total

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