Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration
ISSN: 2167-8421 (Print) 2167-9223 (Online) Journal homepage: http://www.tandfonline.com/loi/iafd20
Caregiver burden and quality of life of patients with amyotrophic lateral sclerosis in India Priya Treesa Thomas, Manjusha G. Warrier, Arun Sadasivan, Bhuvaneshwari Balasubramanium, Veeramani Preethish-kumar, Saraswati Nashi, Kiran Polavarapu, Gopalkrishna Krishna, Seena Vengalil, Prakashi Rajaram & Atchayaram Nalini To cite this article: Priya Treesa Thomas, Manjusha G. Warrier, Arun Sadasivan, Bhuvaneshwari Balasubramanium, Veeramani Preethish-kumar, Saraswati Nashi, Kiran Polavarapu, Gopalkrishna Krishna, Seena Vengalil, Prakashi Rajaram & Atchayaram Nalini (2018): Caregiver burden and quality of life of patients with amyotrophic lateral sclerosis in India, Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration, DOI: 10.1080/21678421.2018.1482353 To link to this article: https://doi.org/10.1080/21678421.2018.1482353
Published online: 24 Jul 2018.
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Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration, 2018; 1–5
RESEARCH ARTICLE
Caregiver burden and quality of life of patients with amyotrophic lateral sclerosis in India
PRIYA TREESA THOMAS1, MANJUSHA G. WARRIER1, ARUN SADASIVAN1, BHUVANESHWARI BALASUBRAMANIUM1, VEERAMANI PREETHISH-KUMAR2,3 SARASWATI NASHI2, KIRAN POLAVARAPU2,3, GOPALKRISHNA KRISHNA2, SEENA VENGALIL2, PRAKASHI RAJARAM1 & ATCHAYARAM NALINI2
,
1
Department of Psychiatric Social Work, 2Department of Neurology, and 3Department of Clinical Neurosciences National Institute of Mental Health and Neuro Sciences, Bangalore, India
Abstract Aim: Amyotrophic lateral sclerosis (ALS) or motor neuron disease (MND) is a progressive degenerative disorder that can have significant debilitating impact. Few studies have explored living with ALS in the developing countries. The study aims to understand the relationship between functionality, quality of life, and caregiver burden in ALS in the sociocultural scenario in India. Methods: A cross-sectional descriptive study was performed among 30 persons with ALS and their caregivers (men ¼ 19; women ¼ 11) receiving treatment from a national quaternary referral care center for Neurological disorders in Southern India. All patients were diagnosed as Definite ALS according to El Escorial Criteria. The mean age at onset of illness was 51.6 years and mean duration of illness at presenting to hospital was 11 months. The caregivers were spouses, offspring, or siblings. Variables were assessed with ALS Functional Rating Scale Revised (ALSFRS- R), ALS Specific Quality of Life Scale (ALSSQOL-R) with the patients and Zarit Burden Interview (ZBI) with the caregiver. Results: Functionality and quality of life negatively correlated with caregiver burden. Caregiver burden was negatively associated with ‘‘negative emotional state’’ and ‘‘interaction of the patient with family and environment’’, sub domains in ALSQOL scale. No significant association was noted between caregiver burden and intimacy, religiosity as well as physical symptoms domains of quality of life. Conclusion: ALS patients and caregivers would benefit from structured care plan that is sensitive to the impact of the illness on the specific domains of quality of life as well as the deterioration in the neurological functioning. KEYWORDS: Caregiving, functionality, quality of life, caregiver burden, amyotrophic lateral sclerosis, developing countries
Introduction Among all neurological disorders, neurodegenerative diseases impose a substantial burden on the population throughout the world. Amyotrophic Lateral Sclerosis (ALS)/Motor Neuron Disease (MND) has a prevalence of 1–7 in 100,000 population in developed countries (1) while it is reported to be 4 in 100,000 population in India (2). The impact of ALS on the individual affected with the illness and their carers are multifaceted. Previous research has shown that distress and depression in ALS patient is not related to functionality (3). Quality of life in ALS patients are reported to be determined by various factors and not strictly by their functionality (4,5). Other factors influencing Quality of Life has been suggested to be
the individual’s inherent self-worth, coping strategies, adaptation, and perceived social support (4,6). As in other terminal illnesses, psychosocial impact of ALS on the care giver has been identified as a major component of how the patient lives with the illness (7). The carer’s perceptions, which may be positive or negative, can have an influence on their support for the patient. In India, there has been increasing shift in healthcare policy toward the non-communicable diseases such as cancer and stroke, with focus on establishing and developing community models of home-based palliative and rehabilitative care (8). Rare diseases such as ALS are underrepresented in such programs. The specialist neurological care is concentrated in selected urban areas and hence the journey of a patient suffering
Correspondence: Priya Treesa Thomas, Department of Psychiatric Social Work, National Institute of Mental Health and Neuro Sciences, Bangalore, India. E-mail:
[email protected] (Received 24 January 2018; revised 13 May 2018; accepted 21 May 2018) ISSN 2167-8421 print/ISSN 2167-9223 online ß 2018 World Federation of Neurology on behalf of the Research Group on Motor Neuron Diseases DOI: 10.1080/21678421.2018.1482353
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from ALS is marked by difficulty with accurate diagnosis, failure to access the care that is available and seeking disease modifying therapy and magical cure till the advanced stages of illness. There is limited possibility of accessing palliative care in ALS, since the existing facilities predominantly focus on cancer palliative care (9). Sociocultural factors and belief systems which emphasize the family system also plays a role in the care provision (10). There is no universal health insurance in India and limited public financing of specialist healthcare. Therefore, majority of the patients meet the cost of medical care, drugs, and other supportive services through ‘‘out-of-pocket’’ expenses by the patient or by the family members (9). Hence, the effect of the illness on the family as well as the role of family in the caregiving of persons with ALS is huge (5,11,12). Despite this understanding, carers’ responses are routinely under-explored. Though lower functional ability need not necessarily lead to low levels of individual quality of life, other illness-related, personality, and family factors have an impact. This study is an attempt to explore the relationship between functionality, quality of life of ALS patients, and the burden experienced by the caregivers in India.
Participants and methods Patients and caregivers Institute ethical clearance was obtained for conducting the study. Patients who sought treatment from a national Quaternary referral care center for neurology in South India were recruited for the study. In a year approximately 120 persons take treatment from the hospital for ALS both from inpatient and outpatient care in this setting. The individuals seeking care are evaluated and treated primarily as inpatients, and regularly followed up at a multidisciplinary outpatient special clinic for Neuro muscular disorders. In order to maximize the homogeneity of the group, 30 patients diagnosed as definite ALS under modified El Escorial criteria who received care between May and September 2016 who consented for the study were recruited for the study along with their caregivers. A detailed evaluation of the symptoms and the level of functionality was done by the neuromuscular specialist. Subsequently, the patients and caregivers were interviewed separately by the psychiatric social worker, interviews lasting for about 45 and 30 minutes respectively, for psychosocial assessment. Instruments Level of functionality of patients recruited for the study was assessed using revised ALS Functional Rating Scale (ALSFRS-R) (13). Their quality of life was assessed by ALS Quality of Life - (ALSQOL)
R Scale which consists of six sub domains—negative emotional state, interaction, religiosity, physical symptoms, and bulbar functions (14). Zarit Burden Interview (ZBI) (15) was administered with the caregivers to assess their subjective burden. Items are assessed on a 5-point Likert scale with scores ranging from 0 to 88, with higher scores being indicative of greater burden. The measure categorizes the caregiver burden as 0–21 (little or no burden), 21–40 (mild to moderate), 41–60 (moderate to severe), and 61–88 (severe). Along with these standard assessment tools, a structured interview schedule was prepared by the team to collect sociodemographic profile of the respondents. The illness details such as age at onset and duration of illness and the caregiver details like age of caregiver, relation of the caregiver to the patient and family details were collected. Statistical analysis The analysis of data was performed using R software. The sociodemographic variables, clinical findings, care giver, and family details collected using structured interview schedule were subjected to descriptive statistical analysis. The instruments administered with the patients (ALSFRS-R and ALSQOL-R) and with caregivers (ZBI) were subjected to basic descriptive analysis. The relationship between the functionality and quality of life of the patient and caregivers’ subjective experience of burden were analyzed using Pearson’s Correlation analysis. Results Descriptive statistics Demographic and clinical characteristics The majority of the patients were male (19, 63.3%). The age at onset of the illness was between 33 and 68 years. There were eight respondents who were 40 years or below. Duration of the illness varied from 2 months to 4 years. Eight of the respondents were homemakers, three were retired and others were in skilled employment. At the time of the study five of them continued to work. All the respondents were married and three of them were widowed. The caregivers (46.16 10.19) were younger than the patients (52.53 10.5). The majority (22, 73%) of the caregivers were spouses and many of them lived with their children (16, 53.3%). The demographic details are provided in Table 1. The disability levels as measured by ALSFRS were reported as mild (35.7 7.07). Five patients had severe functional impairment, 12 had moderate, and 13 had mild impairment. Quality of life perceived by the patients subjectively (5.4 1.4) agreed with the total QoL perceived in different domains (5.42 1.38). Overall caregiver burden
Caregiver burden and quality of life of patients with ALS Table 1. Sociodemographic details of ALS patients and caregivers. Variable
Table 4. Correlation of ALSFRS, ZBS, ALSQOL, and the illness variables.
Number Percentage
Gender Male Female Education Primary education Secondary Graduation and above No formal education Occupation Government/ private employee Self employed Home maker or retired Marital status Married Widow/widower Living status Living with spouse Living with spouse and children Living with spouse and other relatives Care giver’s relationship to the patient Spouse Sibling Parents or other relatives Children
19 11
63.3 36.7
7 13 7 3
23.3 43.3 23.4 10.0
9 10 11
30.0 33.3 36.7
27 3
90.0 10.0
5 16 9
16.7 53.3 30.0
22 1 5 2
73.3 3.3 16.7 6.7
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ALSFRS Zarit Burden Interview ALSQOL a
Age at the time of evaluation
Age at onset
Duration of illness
0.072 0.054 0.079
0.135 0.010 0.053
0.356 0.620a 0.371b
Correlation is significant at the 0.01 level (2-tailed). Correlation is significant at the 0.05 level (2-tailed).
b
score of ALSQOL significantly correlated with the functionality (r ¼ 0.53; p50.01). Family burden was significantly associated with overall quality of life of the patients (r¼ 0.41; p50.01) and negative emotional state (r¼ 0.50; p50.05). With the increasing duration of illness, care giving burden increases (Table 4). When the duration of illness is more, patients recognized their quality of life to be less (r ¼ 0.62; p50.01). There was no relation identified between age of the patient at the time of evaluation, age at onset and functionality and quality of life or burden. Discussion
Table 2. Functionality, quality of life and family burden in ALS. Instruments
Mean (standard deviation)
Range
35.7 (7.07) 5.4 (1.38) 12.9 (11.56)
18–47 1–10 0–58
ALSFRS (n ¼ 30) ALSQOL (n ¼ 30) ZBI (n ¼ 30)a a
Higher the score, greater the burden.
Table 3. Correlation between functionality and burden to quality of life and its specific domains. ALSFRS ALSQOL overall Negative emotional state Interaction Intimacy Religiosity Physical symptoms Bulbar functions
a
0.517 0.364b 0.199 0.236 0.349 0.236 0.525a
ZBI 0.411b 0.504a 0.236 0.169 0.098 0.129 0.166
a
Correlation is significant at the 0.01 level (2-tailed). Correlation is significant at the 0.05 level (2- tailed).
b
was mild (12.93 11). None of the caregivers reported experiencing severe burden. One caregiver reported moderate burden while the majority (80%) felt little or no burden (Table 2). Correlational analysis Functionality of the ALS patients and the burden perceived by caregivers were correlated to each domain of quality of life (Table 3). Bulbar function
The aim of this study was to find out the relationship between functionality and perceived quality of life and caregiver burden in ALS in a developing country. As per the authors’ knowledge, so far no study from India has explored the psychosocial aspects of illness severity, perceived family burden, and quality of life in ALS. While qualitative exploration of the caregiving experience of spouses have been undertaken in studies from different parts of the world (16–25), this study has adopted a quantitative assessment of the caregiving dimensions so that the relationship among the variables could be more clearly established. The age of the people diagnosed with ALS (M ¼ 52.53) and the gender distribution (63.3% were males) were similar to the other epidemiological studies in the Indian setting and worldwide (3,26,27). The spouses provided care to persons with ALS in the majority of our cases, which is similar to other caregiving studies in ALS (4–9,13). The family system in most communities in India is characterized by strict patriarchy, with one son inheriting the family house, who marries and stays in the family, and looks after the ailing parents. Today, Indian society is in a demographic transition with more and more families turning nuclear and parents living alone (17). Though more than half of the patients in this study are staying with their spouse and children, direct caregiving was found to be the responsibility of the able spouse. Support provision in the context of close relationships consists of two distinct components: emotional and instrumental support (18). In the absence of a structured healthcare support system,
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the instrumental support for care came from the offspring(s) in the form of helping with caregiving or household chores and errands, leading to significant out-of-pocket expenses and socioeconomic cost for the family as described in other studies (28–30). The living arrangement also meant that the decision making regarding care is made jointly, but in most cases, the decision is based on the financial viability. The majority of caregivers being family members, emotional support becomes an important area of focus for intervention. Previous studies have recommended the use of clinical interview to assess mental health concerns such as depression and anxiety in ALS, since findings in traditional depression inventories might be confounded by the physical symptoms in ALS (31). In the negative emotional domain of ALSQOL, 80% of our patients scored 56.23, indicating the need for further evaluation for depression, suicidal ideas, and anxiety. We found significant negative correlation between this domain of the ALSQOL and the caregiver’s perception on burden. Higher emotional scores represented better emotional state of the patient, and the family felt less burdened. This is supported by a longitudinal study on ALS caregiving that found that caregiver distress was predicted by psychosocial impact and emotional liability of the patient (32). As indicated in previous studies (10,23,33), interventions to enhance positive support system for the caregivers can consequently decrease their felt burden and improve the perceived quality of life of the patients. The spouse who provides care, who is also aging with the patient, requires special consideration from the healthcare team in coping with the experience of caregiving. This also becomes important since the caregiver burden increases with increase in the duration and progression of the illness (5). The reciprocal influence of negative emotions of the person with ALS and the family burden indicates the need for the early screening, periodic monitoring and appropriate psychosocial interventions in a person with ALS and their caregivers. Our findings add on to the emerging research on the need for and effectiveness of psychological interventions in ALS and caregivers (33–36). Studies with patients in Indian setting have found that belief in religion/spirituality and karma, having meaning of life and family support may act as protective factors (9,37). In India where caregiving happens at home, interventions tailor made to individual patient and family’s needs and contextualized to the unique sociodemographic and economic scenario has to be incorporated to the routine neurological care. In a collectivistic society like India, the communication of the diagnosis is more a family affair than individual and treating team have to recognize and negotiate collusion from family members.
The small number of participants and hospitalbased sample can be seen as limitations of this study. Hence, the group studied represents patients who are able to access care at a higher medical center. Since the mean duration of the illness in this study was 11 months and considering the younger age of the patient group, longitudinal studies that bring out the factors associated with the progression of illness would bring better understanding. Psychosocial aspects and caregiving experiences are better studied with qualitative methodology and could supplement the quantitative findings. In the relentlessly progressing illness like ALS, many of the dimensions of burden are influenced by the increasing practical caregiving demands. Recognition of this association from the time of diagnosis, periodic assessment, and supportive interventions through a multidisciplinary team with palliative approach is vital (38,39). Acknowledgements The authors would like to acknowledge the Indian Council of Medical Research for funding the study and thank the patients and caregivers who participated in the study
Declaration of interest The authors report no conflicts of interest.
ORCID Veeramani Preethish-kumar 0000-0003-1158-0971
http://orcid.org/
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