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Rare case of eosinophilic granulomatosis with polyangiitis in two patients with α-1-antitrypsin deficiency (PiSZ).
Moxey, Jordan Maureen; Low, Emma Victoria; Turner, Alice Margaret.
Affiliation
  • Moxey JM; University of Birmingham, Birmingham, UK.
  • Low EV; Department of Respiratory Medicine, Heart of England NHS Foundation Trust, Birmingham, UK.
  • Turner AM; Department of Inflammatory Research Facility, Institute of Inflammation and Ageing, Birmingham, UK.
BMJ Case Rep ; 20162016 Apr 26.
Article in En | MEDLINE | ID: mdl-27118743
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.
Subject(s)

Full text: 1 Database: MEDLINE Main subject: Phenotype / Granulomatosis with Polyangiitis / Alpha 1-Antitrypsin / Eosinophilic Granuloma / Alpha 1-Antitrypsin Deficiency / Eosinophilia / Lung Type of study: Prognostic_studies Limits: Aged80 / Female / Humans Language: En Journal: BMJ Case Rep Year: 2016 Type: Article

Full text: 1 Database: MEDLINE Main subject: Phenotype / Granulomatosis with Polyangiitis / Alpha 1-Antitrypsin / Eosinophilic Granuloma / Alpha 1-Antitrypsin Deficiency / Eosinophilia / Lung Type of study: Prognostic_studies Limits: Aged80 / Female / Humans Language: En Journal: BMJ Case Rep Year: 2016 Type: Article