title: Successful treatment with methylprednisolone pulse therapy for a life-threatening pulmonary insufficiency in a patient with chronic granulomatous disease following pulmonary invasive aspergillosis and Burkholderia cepacia infection.
abstract: A 14-year-old boy with X-linked chronic granulomatous disease developed severe invasive pulmonary aspergillosis. He was treated with itraconazole and amphotericin B. However, he deteriorated with progressive pulmonary lesions. Burkholderia cepacia was isolated from his bronchoalveolar lavage. Finally, he was given granulocyte transfusions. Following this procedure, his condition rapidly worsened leading to respiratory failure. His lung biopsy demonstrated organizing pneumonia at his right middle lobe. Then, a methylprednisolone pulse therapy was initiated together with the administration of appropriate antibiotics and adequate amounts of amphotericin B. Dramatically, his condition improved. Therefore, a methylprednisolone pulse therapy with appropriate antimicrobial drugs seems to be beneficial for severe pulmonary insufficiency in this type of patients.
authors.affiliation: Department of Pediatrics, Hokkaido University Hospital, Sapporo, Japan.
authors.forename: M
authors.initials: M
authors.lastname: Okano
authors.forename: M
authors.initials: M
authors.lastname: Yamada
authors.forename: M
authors.initials: M
authors.lastname: Ohtsu
authors.forename: N
authors.initials: N
authors.lastname: Kawamura
authors.forename: Y
authors.initials: Y
authors.lastname: Sakiyama
authors.forename: K
authors.initials: K
authors.lastname: Aoi
authors.forename: S
authors.initials: S
authors.lastname: Gandoh
authors.forename: M
authors.initials: M
authors.lastname: Fujita
authors.forename: K
authors.initials: K
authors.lastname: Kobayashi
mesh_terms: D000293:Adolescent; D000935:Antifungal Agents / Q000008:administration & dosage; D001228:Aspergillosis / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D019121:Burkholderia Infections / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D016956:Burkholderia cepacia / Q000302:isolation & purification*; D004359:Drug Therapy, Combination; D005500:Follow-Up Studies; D016469:Fungemia / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D005938:Glucocorticoids / Q000008:administration & dosage; D006105:Granulomatous Disease, Chronic / Q000150:complications / Q000175:diagnosis; D006801:Humans; D008172:Lung Diseases, Fungal / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D008297:Male; D008775:Methylprednisolone / Q000008:administration & dosage*; D011665:Pulmonary Valve Insufficiency / Q000188:drug therapy* / Q000209:etiology; D020551:Pulse Therapy, Drug; D016896:Treatment Outcome
chemical_list: D000935:Antifungal Agents; D005938:Glucocorticoids; D008775:Methylprednisolone