NDLI: Reversal of pulmonary hypertension after diaphragm pacing in an adult patient with congenital central hypoventilation syndrome.

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Reversal of pulmonary hypertension after diaphragm pacing in an adult patient with congenital central hypoventilation syndrome.

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Reversal of pulmonary hypertension after diaphragm pacing in an adult patient with congenital central hypoventilation syndrome.

Content Provider	World Health Organization (WHO)-Global Index Medicus
Author	Morélot-Panzini, Capucine Gonzalez-Bermejo, Jesus Straus, Christian Similowski, Thomas
Description	Country affiliation: France Author Affiliation: Morélot-Panzini C ( Assistance Publique-Hôpitaux de Paris, Groupe Hospitalier Pitié-Salpêtrière Charles Foix, Service de Pneumologie et Réanimation Médicale, Paris, France. capucine.morelot@psl.aphp.fr)
Abstract	INTRODUCTION: Patients with the congenital central hypoventilation syndrome (CCHS) suffer from life-threatening hypoventilation when asleep, making them dependent on mechanical ventilation (MV) at night or during naps. State-of-art respiratory management consists of intermittent positive-pressure ventilation via a tracheotomy or mask. In some patients hypoventilation is permanent, in which case ventilatory support must be extended to the waking hours. Diaphragm pacing can prove useful in such situations. â © METHODS AND RESULTS: This report describes the case of a 26-year-old woman with CCHS in whom failure to achieve adequate MV led to life-threatening pulmonary hypertension (PH), with a systolic pulmonary artery pressure (PAP) of 80 mmHg and right ventricular hypertrophy, despite optimization of all possible measures and despite extensive therapeutic education efforts. Diaphragm pacing using laparoscopically implanted intradiaphragmatic phrenic nerve stimulation electrodes corrected alveolar hypoventilation and lastingly reversed PH (systolic PAP below 40 mmHg after 2 months, sustained after 2 years). Diaphragm pacing induced shoulder pain, however, involving the chronic use of analgesics. The pacing had to be stopped for tolerance reasons after two years, leading to PH worsening and the need for diurnal MV. â © CONCLUSIONS: Diaphragm pacing appears likely effective to restore alveolar ventilation and reverse PH in adult CCHS patients.
File Format	HTM / HTML
ISSN	03913988
Issue Number	6
Volume Number	36
e-ISSN	17246040
Journal	The International Journal of Artificial Organs
Language	English
Publisher	Sage Publication
Publisher Date	2013-06-25
Publisher Place	Italy
Access Restriction	One Nation One Subscription (ONOS)
Subject Keyword	Discipline Transplantation Diaphragm Innervation Electric Stimulation Therapy Hypertension, Pulmonary Therapy Hypoventilation Congenital Phrenic Nerve Physiopathology Sleep Apnea, Central Adult Analgesics Therapeutic Use Arterial Pressure Chronic Pain Drug Therapy Etiology Adverse Effects Instrumentation Familial Primary Pulmonary Hypertension Female Humans Diagnosis Hypertrophy, Right Ventricular Complications Neural Prostheses Pulmonary Artery Respiration, Artificial Shoulder Pain Time Factors Treatment Outcome Case Reports Journal Article Research Support, Non-u.s. Gov't
Content Type	Text
Resource Type	Case study Article
Subject	Medicine Biomaterials Bioengineering Biomedical Engineering

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