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Successful cord blood transplantation after repeated transfusions of unmobilized neutrophils in addition to antifungal treatment in an infant with chronic granulomatous disease complicated by invasive pulmonary aspergillosis.
Shigemura, Tomonari; Nakazawa, Yozo; Yoshikawa, Kentaro; Hirabayashi, Koichi; Saito, Shoji; Kobayashi, Norimoto; Sakashita, Kazuo; Shiohara, Masaaki; Wada, Taizo; Shimodaira, Shigetaka; Agematsu, Kazunaga; Koike, Kenichi.
Afiliação
  • Shigemura T; Department of Pediatrics, Shinshu University School of Medicine, Matsumoto, Japan; Division of Hematology, Oncology, and Immunology, Nagano Children's Hospital, Azumino, Japan; Department of Pediatrics, School of Medicine, Institute of Medical, Pharmaceutical and Health Sciences, Kanazawa University, Kanazawa, Japan; Division of Transfusion Medicine, Cell Processing Center, Shinshu University Hospital; Department of Infection and Host Defense, Shinshu University, Graduate School of Medicine, Mat
Transfusion ; 54(3): 516-21, 2014 Mar.
Article em En | MEDLINE | ID: mdl-23808662
ABSTRACT

BACKGROUND:

Chronic granulomatous disease (CGD) is a rare inherited primary immunodeficiency that affects phagocytic cells. CGD patients are susceptible to fungal infections, especially Aspergillus infections. The management of life-threatening Aspergillus infections in CGD is particularly difficult because some infections cannot be eradicated with standard antifungal treatments and, hence, result in death. CASE REPORT A 2-week-old girl developed invasive pulmonary aspergillosis, which rapidly progressed to respiratory failure. Liposomal amphotericin B, micafungin, and voriconazole were not effective. At the age of 2 months, she was diagnosed with p67phox-deficient CGD. In addition to antifungal treatment, the patient received 21 granulocyte transfusions (GTX), which were obtained from 300- or 400-mL whole blood samples from healthy random donors who were not treated with granulocyte-colony-stimulating factor or dexamethasone. The median neutrophil count of the GTX was 1.88 × 10(8) /kg body weight. Rituximab was administered to reduce alloimmunization to human leukocyte antigens (HLA) after the eighth GTX, resulting in their absence of anti-HLA before and after cord blood transplantation (CBT). A marked improvement in her invasive pulmonary aspergillosis was achieved, although the first CBT was rejected. Complete hematopoietic recovery was obtained after the second CBT.

CONCLUSION:

Repeated GTX containing relatively low doses of neutrophils might be able to control severe Aspergillus infections in infants with CGD.
Assuntos

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Aspergilose / Doença Granulomatosa Crônica / Pneumopatias Fúngicas / Antifúngicos / Neutrófilos Limite: Female / Humans / Newborn Idioma: En Ano de publicação: 2014 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Aspergilose / Doença Granulomatosa Crônica / Pneumopatias Fúngicas / Antifúngicos / Neutrófilos Limite: Female / Humans / Newborn Idioma: En Ano de publicação: 2014 Tipo de documento: Article