The Japanese journal of thoracic diseases
Online ISSN : 1883-471X
Print ISSN : 0301-1542
ISSN-L : 0301-1542
Three Cases of Granulomatous Angiitis (AGA)
Noriko HidakaHajime TakizawaTatsuo HisatomiTatsuo KosudaSumiki MiyachiEisuke OzawaIsao KitamuraKen-ichi TabuchiHaruko HinoMunekazu Nishiwaki
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1985 Volume 23 Issue 7 Pages 837-842

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Abstract
We report three cases of AGA with various manifestations. In the first case, a 19 year-old female first suffered from bronchial asthma and one year later developed systemic vasculitis showing symptoms of asymmetrical paresthesia, paresis in the extremities, skin eruptions and hypereosinophilia. The biopsy of the skin lesion revealed angiitis and extravascular granulomas. She underwent illectomy for multiple intestinal perforations, which were presumably due to angiitis. The second case was a 46 year-old male. Before the onset of vasculitis, attacks of bronchial asthma had recurred for seven years. He had complained of epigastric pain after meals and had lost six kilograms in weight one month before admission. Neurological examination disclosed paresthesia and hypesthesia, suggesting polyneuritis. The skin biopsy of livedo vasculitis revealed angiitis and perivascular eosinophil infiltration. In both cases, treatment by prednisolone produced marked improvement in symptoms without subsequent recurrence of the systemic disease. The third case was a 37 year-old female who had experienced rhinitis and PIE syndrome for a year before the onset of sytemic vasculitis which showed gastrointestinal, renal, dermatological and neulogical symptoms. Although treatment by prednisolone relieved symptoms, bronchial asthma first appeared on reduction of the steroid and vasculitis recurred one year later. Her clinical course was atypical for AGA because of the delayed onset of asthma. In her second attack of vasculitis, severe neurological involvements were manifest, including palsy of extrimities and hoarseness due to recurrent nerve palsy. Dermatologically livedo vasculitis and annular erythema were also salient features. The skin biopsy of livedo vasculitis on a lower limb revealed extravascular granulomas and vasculitis. Although this second attack was resistant to treatment, symptoms were gradually relieved on increase of steroid.
We reviewed 55 cases of AGA in the Japanese literature and summarized them with our three cases. The mean duration of asthma prior to the onset of vasculitis was four years. Initial symptoms involved asthma in most cases. Our third case was the only one reported in which systemic vasculitis preceded the onset of bronchial asthma.
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© by The Japanese Respiratory Society
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