Literature DB >> 2663410

[Nightly home artificial respiration in juvenile Pompe's disease with pulmonary hypertension and right cardiac insufficiency].

C Budde-Steffen1, L Grävinghoff, M Albani, H H Hellwege, A Kohlschütter.   

Abstract

A 17-year-old girl with type II glycogen storage disease (Pompe's) developed severe right-heart failure as a result of pulmonary hypertension due to, predominantly nocturnal, hypoventilation. At night the partial pressure of oxygen was only 30-50 mmHg, pCO2 70-100 mmHg. After persistent nightly intermittent positive pressure ventilation blood gases as well as electrocardiographic and echocardiographic findings have now--after 12 months--become normal and the patient has been completely restored to a normal life. This case demonstrates that life expectancy and quality of patients with chronic forms of Pompe's disease (and probably also with other chronic neuromuscular diseases) can be markedly improved by nightly artificial ventilation which reduces nocturnal hypoventilation and resulting pulmonary hypertension.

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Year:  1989        PMID: 2663410     DOI: 10.1055/s-2008-1066727

Source DB:  PubMed          Journal:  Dtsch Med Wochenschr        ISSN: 0012-0472            Impact factor:   0.628


  2 in total

Review 1.  The natural course of non-classic Pompe's disease; a review of 225 published cases.

Authors:  Léon P F Winkel; Marloes L C Hagemans; Pieter A van Doorn; M Christa B Loonen; Wim J C Hop; Arnold J J Reuser; Ans T van der Ploeg
Journal:  J Neurol       Date:  2005-08       Impact factor: 4.849

2.  Treatment of pulmonary hypertension in patients with connective tissue disease and interstitial lung disease.

Authors:  Shikha Mittoo; Thomas Jacob; Andrea Craig; Zoheir Bshouty
Journal:  Can Respir J       Date:  2010 Nov-Dec       Impact factor: 2.409

  2 in total

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