Psychosocial impact at the time of a rare disease diagnosis

被引:9
作者
Benito-Lozano, Juan [1 ,2 ]
Arias-Merino, Greta [1 ]
Gomez-Martinez, Mario [1 ]
Arconada-Lopez, Beatriz [3 ]
Ruiz-Garcia, Begona [4 ]
Posada de la Paz, Manuel [1 ,5 ]
Alonso-Ferreira, Veronica [1 ,6 ]
机构
[1] Inst Salud Carlos ISCIII 3, Inst Rare Dis Res IIER, Madrid, Spain
[2] Univ Nacl Educ Distancia UNED, Madrid, Spain
[3] Federac Espanola Enfermedades Raras, Spanish Federat Rare Dis FEDER, Madrid, Spain
[4] IMSERSO, State Reference Ctr Assistance People Living Rare, Ctr Referencia Estatal Atenc Personas Enfermedades, Burgos, Spain
[5] Undiagnosed Dis Network Int UDNI, Madrid, Spain
[6] Ctr Invest Biomed Red Enfermedades Raras CIBERER, Madrid, Spain
来源
PLOS ONE | 2023年 / 18卷 / 07期
关键词
D O I
10.1371/journal.pone.0288875
中图分类号
O [数理科学和化学]; P [天文学、地球科学]; Q [生物科学]; N [自然科学总论];
学科分类号
07 ; 0710 ; 09 ;
摘要
Over half of all persons with rare diseases (RDs) in Spain experience diagnostic delay (DD) but little is known about its consequences. This study therefore aimed to analyze the psychological impact of obtaining a diagnosis of an RD, and to ascertain what social determinants are influenced and what the personal consequences are, according to whether or not patients experienced DD. Data were obtained from a purpose-designed form completed by persons registered at the Spanish Rare Diseases Patient Registry. The following were performed: a descriptive analysis; a principal component analysis (PCA); and logistic regressions. Results revealed that while searching for a diagnosis, people who experienced DD were more in need of psychological care than those diagnosed in less than one year (36.2% vs 23.2%; p = 0.002; n = 524). The PCA identified three principal components, i.e., psychological effects, social implications, and functional impact. Reducing DD would improve psychological effects, such as irritability (OR 3.6; 95%CI 1.5-8.5), frustration (OR 3.4; 95%CI 1.7-7.1) and concentration on everyday life (OR 3.3; 95%CI 1.4-7.7). The influence of the social implications and functional repercussions of the disease was greater in persons with DD (scores of 22.4 vs 20 and 10.6 vs 9.4, respectively) in terms of the difficulty in explaining symptoms to close friends and family (3.3 vs 2.9), and loss of independence (3.3 vs 2.9). In conclusion, this is the first study to analyze the psychosocial impact of diagnosis of RDs in Spain and one of few to assess it in the patients themselves, based on data drawn from a purpose-designed form from a national registry open to any RD. People affected by RDs who underwent DD experienced greater psychosocial impact than did those who were diagnosed within the space of one year.
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页数:17
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