Abstract
We report the case of a 64-year-old woman with severe alveolar haemorrhage secondary to granulomatosis with polyangiitis (GPA), who was unknowingly treated with only penicillin and macrolide antibiotics, resulting in successful initial disease remission. The immunomodulatory effects of the macrolide antibiotic used could underlie this novel observation, which may shed new light on the pathogenesis and potential treatment of anti-neutrophil cytoplasm antibody (ANCA) associated vasculitis. The role of macrolides as an adjunct to conventional therapy in cases of AAV with respiratory involvement may be worthy of further investigation.
Highlights
Alveolar haemorrhage is one of the most serious manifestations of granulomatosis with polyangiitis (GPA), which can be rapidly fatal if untreated
The initial presentation of the case described is clearly consistent with a diagnosis of severe alveolar haemorrhage secondary to pulmonary GPA, and had the anti-neutrophil cytoplasm antibody (ANCA) result been available early on during the course of her disease, she would undoubtedly have received immunosuppressive treatment for ANCA associated vasculitis with pulmonary haemorrhage
We are unaware of any similar observations in the published literature. This observation could be explained in two different ways: first, that pulmonary vasculitis was provoked by an unidentified bacterial pathogen, the eradication of which eliminated the stimulus for persistent autoimmune damage; second, that the disease was not provoked by infection and remission resulted from an immunomodulatory effect of the antibiotics used
Summary
Alveolar haemorrhage is one of the most serious manifestations of GPA, which can be rapidly fatal if untreated. We present a case whose clinical course may be informative with regards pathogenesis and management of AAV. For ten months after her initial presentation, the patient remained entirely well, with normal blood pressure and renal function, and no respiratory symptoms.
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