exposures associated with pulmonary alveolar proteinosis
Kristin Cummings, Carl Reynolds,
Isabella Annesi-Maesano, John Balmes, David Fishwick, David Miedinger,
Nicola Murgia, Rajen Naidoo, Carrie Redlich, Torben Sigsgaard, Kjell
Toren, Denis Vinnikov, Paul Blanc
Respiratory Journal 2017 50: PA409; DOI:
alveolar proteinosis (PAP) is a rare, diffuse lung disease
characterized by alveolar filling with lipoproteinaceous material.
While most PAP is considered idiopathic, occupational etiologies, such
as silica exposure causing “silicoproteinosis,” are
Aim: To systematically review the occupational
contributions to PAP.
Methods: We identified and reviewed relevant publications
through January 2017 using PubMed and manual review of identified
papers’ references. We included publications that described
at least 10 cases of PAP and that also noted the number with
occupations involving likely exposure to various vapors, gases, dusts,
and/or fumes (VGDF) or with presumably occupational VGDF exposures. We
calculated the prevalence of occupational exposures generally and
silica exposure specifically.
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We included 24 publications with a total of 1413 cases (range: 11-241
cases/publication). The mean prevalence of occupational exposure was
29% (median: 30%, range: 0-59%). Thirteen publications specifically
addressed silica exposure; mean prevalence in those studies was 6%
(median: 6%, range: 0-22%). Studies that collected information about
exposure through questionnaire generally reported a higher prevalence
of exposure. Three publications included comparison to controls: one
found a higher prevalence of self-reported occupational inhalational
exposure in cases (34%) than controls (20%) (p=0.07); two were
histopathological investigations of the same cases that found more
birefringent particles (p<0.05) and higher inorganic particle
concentrations in case than control tissue.
Nearly one third of PAP in cases series is associated with work-related
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