Literature DB >> 28577347

Neuroradiographic findings in 22q11.2 deletion syndrome.

Lauren A Bohm1,2,3, Tom C Zhou1, Tyler J Mingo1, Sarah L Dugan4,5, Richard J Patterson6, James D Sidman1,2, Brianne B Roby1,2.   

Abstract

22q11.2 deletion syndrome (22q11.2DS) is a common genetic disorder with enormous phenotypic heterogeneity. Despite the established prevalence of developmental and neuropsychiatric issues in this syndrome, its neuroanatomical correlates are not as well understood. A retrospective chart review was performed on 111 patients diagnosed with 22q11.2DS. Of the 111 patients, 24 with genetically confirmed 22q11.2 deletion and brain MRI or MRA were included in this study. The most common indications for imaging were unexplained developmental delay (6/24), seizures of unknown etiology (5/24), and unilateral weakness (3/24). More than half (13/24) of the patients had significant radiographic findings, including persistent cavum septi pellucidi and/or cavum vergae (8/24), aberrant cortical veins (6/24), polymicrogyria or cortical dysplasia (4/24), inner ear deformities (3/24), hypoplastic internal carotid artery (2/24), and hypoplastic cerebellum (1/24). These findings reveal the types and frequencies of brain malformations in this case series, and suggest that the prevalence of neuroanatomical abnormalities in 22q11.2DS may be underestimated. Understanding indications for imaging and frequently encountered brain malformations will result in early diagnosis and intervention in an effort to optimize patient outcomes.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  22q11.2 deletion syndrome; DiGeorge syndrome; brain; developmental delay; magnetic resonance imaging; velocardiofacial syndrome

Mesh:

Year:  2017        PMID: 28577347     DOI: 10.1002/ajmg.a.38304

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  3 in total

1.  Haploinsufficiency of the HIRA gene located in the 22q11 deletion syndrome region is associated with abnormal neurodevelopment and impaired dendritic outgrowth.

Authors:  Marie-Laure Vuillaume; Dévina C Ung; Valerie E Vancollie; Tjitske Kleefstra; Binnaz Yalcin; Frédéric Laumonnier; Annick Toutain; Médéric Jeanne; Christel Wagner; Stephan C Collins; Sandrine Vonwill; Damien Haye; Nora Chelloug; Rolph Pfundt; Joost Kummeling; Marie-Pierre Moizard; Sylviane Marouillat
Journal:  Hum Genet       Date:  2021-01-08       Impact factor: 4.132

Review 2.  Neurological manifestation of 22q11.2 deletion syndrome.

Authors:  Michael Bayat; Allan Bayat
Journal:  Neurol Sci       Date:  2022-01-18       Impact factor: 3.307

3.  Chromosomal Microarray With Clinical Diagnostic Utility in Children With Developmental Delay or Intellectual Disability.

Authors:  Jin Sook Lee; Hee Hwang; Soo Yeon Kim; Ki Joong Kim; Jin Sun Choi; Mi Jung Woo; Young Min Choi; Jong Kwan Jun; Byung Chan Lim; Jong Hee Chae
Journal:  Ann Lab Med       Date:  2018-09       Impact factor: 3.464

  3 in total

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