Document Detail

Diaphragmatic pacing in children with congenital central alveolar hypoventilation syndrome.
MedLine Citation:
PMID:  7604375     Owner:  NLM     Status:  MEDLINE    
BACKGROUND: The purposes of this study were to study congenital central alveolar hypoventilation syndrome (CCAHS), to report the experience at our institution with this disorder, and to describe the surgical technique of diaphragmatic pacing. Three patients are in our diaphragmatic pacing program. They all have CCAHS, documented by means of lack of respiratory drive to hypercapnia and normal peripheral nerve and muscle studies. Two patients have associated Hirschsprung's disease. METHODS: The ages of patients at insertion of bilateral phrenic nerve pacemakers were 1, 2, and 5 years. A bilateral axillary thoracotomy in the third interspace was used in two patients, and anterior thoracotomy was used in one for insertion of electrodes on the phrenic nerves. Counterincisions in both flanks were used in all patients for insertion of the receivers in subcutaneous pockets. RESULTS: In all patients pacing was commenced within 1 week of the surgical procedure, because no primary pacemaker failures occurred. One patient has experienced no failure of the equipment and no infectious complications at 4 years. A second patient has had the pacemakers for 6 months without complications. The third patient underwent placement of bilateral pacemakers in 1984. In this patient a 10-year experience has resulted in subcutaneous implant failure on two separate occasions; also the presence of a staphylococcal empyema necessitated the temporary removal of one phrenic nerve electrode for 6 months, with successful reinsertion. All patients now receive mechanical assisted ventilation when sleeping and phrenic nerve pacing when awake. CONCLUSIONS: Pediatric surgeons should be aware of CCAHS because it may be treated with surgically implanted electrodes that allow for pacing of the diaphragm. The technique has an acceptable complication rate, and it can greatly decrease the impact of the disease on the lifestyle and activity of the patient. CCAHS also may be associated with Hirschsprung's disease.
H Flageole; V R Adolph; G M Davis; J M Laberge; L T Nguyen; F M Guttman
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Surgery     Volume:  118     ISSN:  0039-6060     ISO Abbreviation:  Surgery     Publication Date:  1995 Jul 
Date Detail:
Created Date:  1995-08-07     Completed Date:  1995-08-07     Revised Date:  2010-03-24    
Medline Journal Info:
Nlm Unique ID:  0417347     Medline TA:  Surgery     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  25-8     Citation Subset:  AIM; IM    
Department of Surgery, Montreal Children's Hospital/McGill University, Quebec, Canada.
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MeSH Terms
Artificial Organs*
Child, Preschool
Diaphragm / innervation*
Electric Stimulation
Phrenic Nerve
Retrospective Studies
Sleep Apnea Syndromes / surgery*

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

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