Rare case of eosinophilic granulomatosis with polyangiitis in two patients with α-1-antitrypsin deficiency (PiSZ)

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Two patients with alpha-1-antitrypsin deficiency PiSZ phenotype presented with eosinophilic granulomatosis with polyangiitis and pulmonary involvement, managed with steroids and azathioprine.

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Abstract

We present two cases of eosinophilic granulomatosis with polyangiitis occurring with α-1-antitrypsin deficiency, both PiSZ phenotype. The simultaneous occurrence of these two conditions has seldom been described in the literature, despite evidence of an association between α-1-antitrypsin deficiency and other forms of vasculitis. Both patients had pulmonary involvement and reported intermittent exacerbations of vasculitic symptoms. Both patients were managed on low-dose oral steroids and azathioprine remaining well with occasional exacerbations. It is important to consider whether there is an association between eosinophilic granulomatosis with polyangiitis and α-1-antitrypsin deficiency, as this may lead to more severe pulmonary symptoms during exacerbations. If a genetic association between the two conditions is found, clinicians should be aware of the possible need to screen for α-1-antitrypsin deficiency in appropriate patients.

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MeSH descriptors

alpha 1-Antitrypsin alpha 1-Antitrypsin Deficiency Eosinophilia Eosinophilic Granuloma Granulomatosis with Polyangiitis Lung Phenotype Aged, 80 and over alpha 1-Antitrypsin alpha 1-Antitrypsin alpha 1-Antitrypsin Deficiency Disease Management Eosinophilia Eosinophilia Eosinophilic Granuloma Eosinophilic Granuloma Eosinophils Eosinophils Female Genetic Testing

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europepmc
last seen: 2026-10-25T09:32:14.900964+00:00
pubmed
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