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