Health-Related Quality of Life in Patients Living with Wilson Disease in Spain: A Cross-Sectional Observational Study

被引:1
|
作者
Marino, Zoe [1 ]
Berenguer, Marina [2 ]
Pena-Quintana, Luis [3 ,4 ]
Olveira, Antonio [5 ]
Miralpeix, Anna [1 ]
Sastre, Isabel [2 ]
Reyes-Dominguez, Ana [3 ]
Castillo, Pilar [5 ]
Garcia-Sola, Claudia [1 ]
Bono, Ariadna [2 ]
Romero, Miriam [5 ]
Perez-Sadaba, Francisco Javier [6 ]
Aceituno, Susana [6 ]
Anguera, Anna [7 ]
机构
[1] Univ Barcelona, Hosp Clin Barcelona, Ctr Invest Biomed Red Enfermedades Hepat & Digest, Liver Unit, Barcelona 08007, Spain
[2] Hosp Univ & Politecn La Fe, Ctr Invest Biomed Red Enfermedades Hepat & Digest, Dept Gastroenterol & Hepatol, Inst Invest Sanitaria IIS Fe, (MB ), (IS ), Valencia 46026, Spain
[3] Complejo Hosp Univ Insular Materno Infantil, Hepatoloy & Nutr Unit, Pediat Gastroenterol, Las Palmas Gran Canaria 35016, Spain
[4] Inst Salud Carlos III, Univ Palmas Gran Canaria, Ctr Invest Biomed Red Fisiopatol Obes & Nutr CIBE, Las Palmas Gran Canaria 35001, Spain
[5] Hosp La Paz, Dept Gastroenterol, Madrid 28046, Spain
[6] Outcomes 10 SLU, SA, Castellon de La Plana 12071, Spain
[7] AstraZeneca Rare Dis, Med Dept, Barcelona 08028, Spain
关键词
Wilson disease; health-related quality of life; real-world evidence; emotional status; adherence; VALIDATION; QUESTIONNAIRE; DIAGNOSIS; EQ-5D-5L; WORK;
D O I
10.3390/jcm12144823
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Wilson disease (WD) is a rare copper metabolism disorder caused by mutations in the ATP7B gene. It usually affects young individuals and can produce hepatic and/or neurological involvement, potentially affecting health-related quality of life (HRQoL). We assessed HRQoL in a cohort of Spanish patients with WD and evaluated disease impact on several domains of patients' lives, treatment adherence, drug preference and satisfaction, and healthcare resource utilisation in a cross-sectional, retrospective, multicentric, observational study. A total of 102 patients were included: 81.4% presented isolated liver involvement (group H) and 18.6% presented neurological or mixed involvement (group EH). Up to 30% of patients reported a deteriorated emotional status with anxiety and depression, which was greater in the EH subgroup; the use of neuropsychiatric drugs was high. Over 70% of the patients were satisfied with their current treatment but complained about taking too many pills, stating they would consider switching to another more patient-friendly treatment if available. The Simplified Medication Adherence Questionnaire revealed only 22.5% of patients were fully adherent to therapy, suggesting that alternative therapies are needed. This real-world study, even though is highly enriched with hepatic patients and mild disease, shows that WD impacts patients' HRQoL, especially in the emotional domain.
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页数:15
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