The burden of Huntington’s disease: A prospective longitudinal study of patient/caregiver pairs.
Parkinsonism & Related Disorders. 2022-10-01; 103: 77-84
DOI: 10.1016/j.parkreldis.2022.08.023
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Youssov K(1), Audureau E(2), Vandendriessche H(3), Morgado G(4), Layese R(5), Goizet C(6), Verny C(7), Bourhis ML(8), Bachoud-Lévi AC(9).
Author information:
(1)Département d’Études Cognitives, École normale supérieure, PSL University,
75005, Paris, France; Université Paris-Est Créteil, Faculté de médecine, 94000,
Créteil, France; Inserm U955, Institut Mondor de Recherche Biomédicale, Équipe
E01 NeuroPsychologie Interventionnelle, 94000, Créteil, France; Assistance
Publique Hôpitaux de Paris (AP-HP), Centre de référence Maladie de Huntington,
Service de Neurologie, Hôpital Henri Mondor-Albert Chenevier, 94000, Créteil,
France.
(2)Université Paris-Est Créteil, Faculté de médecine, 94000, Créteil, France;
Assistance Publique Hôpitaux de Paris (AP-HP), Unité de Recherche Clinique (URC
Mondor), Service de Santé Publique, Hôpital Henri Mondor-Albert Chenevier, 94000,
Créteil, France; Inserm U955, Institut Mondor de Recherche Biomédicale, CEpiA
(Clinical Epidemiology and Ageing) Team, France.
(3)Département d’Études Cognitives, École normale supérieure, PSL University,
75005, Paris, France; Université Paris-Est Créteil, Faculté de médecine, 94000,
Créteil, France; Inserm U955, Institut Mondor de Recherche Biomédicale, Équipe
E01 NeuroPsychologie Interventionnelle, 94000, Créteil, France.
(4)Inserm, Centre d’Investigation Clinique 1430, AP-HP, Hôpital Henri Mondor,
Créteil, France.
(5)Assistance Publique Hôpitaux de Paris (AP-HP), Unité de Recherche Clinique
(URC Mondor), Service de Santé Publique, Hôpital Henri Mondor-Albert Chenevier,
94000, Créteil, France.
(6)Centre de Reference Maladies Rares « Neurogénétique », Centre de compétence
Maladies Rares « Maladie de Huntington », Service de Génétique Médicale, CHU
Bordeaux and Univ. Bordeaux, Equipe « Neurogénétique Translationnelle – NRGEN »,
INCIA CNRS UMR5287 Université Bordeaux, Bordeaux, France.
(7)Service de Neurologie, Centre de référence Huntington, Laboratoire Mitolab UMR
CNRS 6015 – Inserm 1083, 49933, CHU d’ANGERS, France.
(8)Inserm, Centre d’Investigation Clinique 1430, AP-HP, Hôpital Henri Mondor,
Créteil, France; Inserm U955, Institut Mondor de Recherche Biomédicale, CEpiA
(Clinical Epidemiology and Ageing) Team, France.
(9)Département d’Études Cognitives, École normale supérieure, PSL University,
75005, Paris, France; Université Paris-Est Créteil, Faculté de médecine, 94000,
Créteil, France; Inserm U955, Institut Mondor de Recherche Biomédicale, Équipe
E01 NeuroPsychologie Interventionnelle, 94000, Créteil, France; Assistance
Publique Hôpitaux de Paris (AP-HP), Centre de référence Maladie de Huntington,
Service de Neurologie, Hôpital Henri Mondor-Albert Chenevier, 94000, Créteil,
France. Electronic address: .
BACKGROUND: Caregiver burden is widely recognized in Huntington’s disease, but
little is known about the factors determining its evolution over time in the
absence of longitudinal studies. Our objective was to identify typical patterns
of caregiver burden level and evolution using both patients’ and caregivers’
characteristics over a one-year period to identify potential levers for
alleviation.
METHODS: We conducted a prospective multicenter longitudinal study in
caregiver/patient pairs in Huntington’s disease (NCT02876445) between March 2011
and May 2015. Caregiver data were derived from two questionnaires at one-year
interval on perceived burden (Zarit Burden Interview), social environment and
support. Caregiver data were linked to clinical and demographic data from
patients included in the Biomarker study (NCT01590589). Unsupervised clustering
analysis was performed using self-organizing maps.
RESULTS: 105 caregiver/patient pairs were included in the analysis. We identified
four clusters. Of the two clusters of patients with advanced disease, cluster A
was characterized by high levels of irritability and obsessive-compulsive
behaviors, with high and increasing burden (N = 30; 29%), cluster B, the more
apathetic group, with low and decreasing burden (N = 22; 21%). Clusters C
(N = 27; 26%) and D (N = 26; 25%) were composed of patients in earlier stages,
associated with a stable burden in group C but a notably increasing one in group
D driven by patients’ depression scores increase.
CONCLUSIONS: Our results revealed the dynamics of caregiver burden over time in
Huntington’s disease, combining the stage of the disease, the severity of the
patients’ decline, psychiatric and behavioral disorders, and their evolution over
time.
Copyright © 2022 The Authors. Published by Elsevier Ltd.. All rights reserved.
Conflict of interest statement: Declaration of competing interests No conflict of interest.