Literature DB >> 29249648

Congenital central hypoventilation syndrome: An overview of etiopathogenesis, associated pathologies, clinical presentation, and management.

Saher Zaidi1, Jason Gandhi2, Sohrab Vatsia3, Noel L Smith4, Sardar Ali Khan5.   

Abstract

Congenital central hypoventilation syndrome (CCHS), known colloquially as Ondine's curse, is a rare disorder characterized by impaired autonomic control of breathing during sleep from the loss of vagal input and diminished sensitivity of CO2 receptors in the medulla. CCHS correlates to the malformation of the neural crest located in the brainstem; this consequently affects the loss of sensitivity of CO2 chemoreceptors, bringing about hypoventilation during sleep. The primary cause of CCHS is the mutation of the paired-like homeobox PHO2XB gene, found in 90% of the patients. This mutation not only affects breathing but also drives neurological abnormalities such as autonomic and neurocognitive dysfunction. Though typically congenital, there have been late-onset (i.e., acquired) cases reported. It is vital for physicians and clinicians to be able to diagnose CCHS due to its similar presentation to other syndromes and disorders, which may cause it to be misdiagnosed and may account for its deleterious effects. CCHS can lead to a constellation of symptoms, and consideration of diseases that present concomitantly with CCHS affords us a better understanding of the etiology of this illness. Although a rare syndrome, we aim to review the current literature to emphasize the pathogenesis, etiology, clinical presentation, symptoms, diagnosis, and current treatment methods of CCHS for clinicians to better identify and understand this condition.
Copyright © 2017 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Congenital central hypoventilation syndrome; Diaphragm pacing; Hirschsprung's disease; Ondine's curse; Positive-pressure ventilation; Progestin; Sudden infant death syndrome

Mesh:

Substances:

Year:  2017        PMID: 29249648     DOI: 10.1016/j.autneu.2017.11.003

Source DB:  PubMed          Journal:  Auton Neurosci        ISSN: 1566-0702            Impact factor:   3.145


  10 in total

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3.  Noninvasive ventilation in a young infant with congenital central hypoventilation and 7-year follow-up.

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Review 5.  Research Advances on Therapeutic Approaches to Congenital Central Hypoventilation Syndrome (CCHS).

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9.  Respiratory Control by Phox2b-expressing Neurons in a Locus Coeruleus-preBötzinger Complex Circuit.

Authors:  Na Liu; Congrui Fu; Hongxiao Yu; Yakun Wang; Luo Shi; Yinchao Hao; Fang Yuan; Xiangjian Zhang; Sheng Wang
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10.  Central Hypoventilation: A Rare Complication of Wallenberg Syndrome.

Authors:  Keithan Sivakumar; Manveer Garcha; Dev Mehta; Megan C Leary; Hussam A Yacoub
Journal:  Case Rep Neurol Med       Date:  2018-03-21
  10 in total

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