| Literature DB >> 31863244 |
Federica Pulvirenti1, Maria Sangerardi2, Alessandro Plebani3, Annarosa Soresina3, Andrea Finocchi4, Claudio Pignata5, Emilia Cirillo5, Antonino Trizzino6, Alessandro Aiuti7, Maddalena Migliavacca7, Franco Locatelli8, Alice Bertaina8, Samuele Naviglio9, Maria Carrabba10, Marco De Carli11, Maria Grazia Foschino Barbaro12, Marco Gattorno13, Isabella Quinti1,14, Baldassarre Martire15.
Abstract
Chronic granulomatous disease (CGD) is a primary immunodeficiency characterized by life-threatening infections, inflammation, and autoimmunity with an impact on health-related quality of life (HRQoL). Few data are available for children, whereas no study has been conducted in adults. Here, we investigated HRQoL and emotional functioning of 19 children and 28 adults enrolled in Italian registry for CGD. PEDsQL and SDQ were used for children and their caregivers, and adults completed the SF-12 questionnaire. Mean scores were compared with norms and with patients affected by chronic diseases. Comparisons were made for CGD patients who underwent or not hematopoietic stem cell transplantation (HSCT). When compared with norms, CGD children exhibited higher difficulties in social/school areas, peer relationship, and conduct/emotional problems (< 5 years of age), as scored by proxies. Differently, CGD adults reported higher difficulties both in mental and physical area than norms. Only for children, clinical status had a damaging effect on psychosocial and school dimensions, whereas age had a negative impact on social areas. No significant difference was observed between patients treated or not with HSCT. When compared with patients affected by chronic diseases, CGD children and adults both displayed fewer physical disabilities. Differently, in mental scale adults scored lower than those with rheumatology diseases and had similar impairment in comparison with patients with diabetes mellitus and cancer. This study emphasized the impact of CGD on HRQoL since infancy and its decline in adulthood, with emotional difficulties occurring early. HRQoL impairment should be considered in clinical picture of CGD and pro-actively assessed and managed by clinicians.Entities:
Keywords: Chronic granulomatous disease; PEDsQL; SDQ; SF-12; burden of disease; emotional difficulties; health-related quality of life; hematopoietic stem cell transplantation
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Year: 2019 PMID: 31863244 DOI: 10.1007/s10875-019-00725-1
Source DB: PubMed Journal: J Clin Immunol ISSN: 0271-9142 Impact factor: 8.317