| Literature DB >> 23726861 |
Daniel Franzen1, Pierre A Krayenbuehl, Olivier Lidove, John-David Aubert, Frederic Barbey.
Abstract
Fabry disease (FD) is an X-linked lysosomal storage disorder caused by deficiency of alpha-galactosidase A, which leads to storage of sphingolipids in virtually all human cells and consequently to organ dysfunction. Pulmonary involvement is still debated. But, obstructive lung disease is up to ten times more prevalent in patients with FD compared to general public. Also, an accelerated decline in forced expiratory volume in one second (FEV1) over time was observed in these patients. Lysosomal storage of glycosphingolipids is considered leading to small airway disease via hyperplasia of the bronchiolar smooth muscle cells. Larger airways may become involved with ongoing disease process. There is no evidence for involvement of the lung interstitium in FD. The effect of enzyme replacement therapy on respiratory involvement remains to be determined in large, prospective controlled trials.Entities:
Keywords: Bronchiolar; Fabry disease; Lung; Obstructive; Pulmonary; Respiratory
Mesh:
Year: 2013 PMID: 23726861 DOI: 10.1016/j.ejim.2013.05.003
Source DB: PubMed Journal: Eur J Intern Med ISSN: 0953-6205 Impact factor: 4.487