Who will guard the guardians? Cross-sectional study on prevalence of psychiatric morbidity, quality of life, and coping skills in caregivers of children with thalassemia major.

BACKGROUND: Patients of thalassemia major require frequent hospitalization. Caregivers are more affected than the patient themselves as they better appreciate the magnitude of illness and treatment, resulting in increased risks for psychiatric illness. AIMS AND
OBJECTIVES: The purpose of the study was to assess the prevalence of psychiatric morbidity in the caregivers of patients with thalassemia major. The study also examined the quality of life of the caregivers, their coping strategies, and its association with sociodemographic variables.
METHODOLOGY: A cross-sectional study with 100 caregivers, recruited by convenience sampling technique, attending the thalassemia daycare center, was carried out over 12 months in a tertiary care hospital. They were administered a semistructured proforma along with General Health Questionnaire 12 (GHQ 12), WHO-Quality of Life-BREF (WHO-QOL-BREF), and Coping Inventory for Stressful Situations 21 scale. The GHQ 12 was used for screening and those scoring three or more underwent a clinical psychiatric interview. Those who were diagnosed with psychopathology were ascribed diagnosis as per ICD-10. Descriptive analysis was done. Associations were studied using Fischer's exact test. Comparison of quality of life with blood transfusion variables was done using Mann-Whitney U test.
RESULTS: The prevalence of psychiatric morbidity amongst the caregivers was found to be 35% with depressive episode (22%) being the most common. Psychiatric morbidity was found to have a significant association with both, the frequency (P = 0.037) and total number of blood transfusions (P = 0.012). Coping was found to have a strong association with psychiatric morbidity (P = 0.001) and employment (P = 0.009).
CONCLUSIONS: Caregivers of children with thalassemia major face psychological burden like depression or anxiety, for which treatment is not sought. Improved psychological health of the caregivers will ensure better care of the child and guarantee better adherence to the treatment.

Introduction

Thalassemia major is the commonest single-gene disorder. Beta-thalassemia is a heterogeneous autosomal recessive hereditary disorder characterized by mutations in the gene coding for the β-subunits of hemoglobin, resulting in decreased or totally absent synthesis of globin β-chains.[1] Almost 12,000 children with thalassemia major are born in India every year.[2] Patients with thalassemia major often have severe microcytic and hypochromic anemia and hepatosplenomegaly. They usually come to medical attention within 2 years of birth. The vast majority of Indian population only has access to primary care facilities.[3] Without treatment, these children have severely compromised growth and a shortened life expectancy. Treatment includes a regular transfusion program and chelation therapy, which is aimed at decreasing transfusional iron overload, extending life expectancy into the third-to-fifth decade.[1]

A caregiver is someone, either the family member or the provider, who has the responsibility for meeting the physical and psychological needs of an infant, child, or dependent adult.[4] Thalassemia management program involves frequent hospitalization of the patients. This leads to feelings of emotional distress and fear among their caregivers resulting in a psychosocial burden.[5] Caregivers are often more affected than the patient themselves, because of better appreciation of the magnitude of illness and treatment resulting in increased risks for anxiety and mood disorders.[6]

Recent strides in medical management of thalassemia major have increased longevity without paying commensurate attention to the quality of life. Studies have shown that caregivers who cope less successfully to the child's illness tend to have a fatalistic and more negative attitude toward the ailment.[7] Researchers have classified coping skills into broadly three categories, namely, task oriented, avoidance oriented, and emotion oriented. While task-oriented coping involves problem analysis and employing immediate corrective actions, emotion-oriented coping includes becoming upset, anxious, and assigning blame. Avoidance-oriented coping may lead to disorders like substance abuse, gambling, and internet gaming disorder. Task-oriented coping is considered to be the most effective approach.[8]

With little attention given to the caregivers of patients with thalassemia major, it was necessary that this vulnerable population be studied for psychiatric morbidity, quality of life, and coping skills. There is a gap in available literature about the complex interplay of psychiatric morbidity of the caregivers, their quality of life, and coping skills as well as the way these together ultimately affect the patient, a lacuna which the current study undertakes to address. The aim of the study was to assess the prevalence and type of psychiatric morbidity in the caregivers of patients with thalassemia major. The study also examined the quality of life of the caregivers, their coping strategies, and its association with sociodemographic variables.

Methodology

Taking into consideration a previous study,[6] which had shown prevalence of 57%, and Cochran's formula (n = Z2PQ/e2) (where n = sample size, P = prevalence of the disease, Q = 1 − P, Z = Z value at confidence levels, and e = precision/margin of error), sample size was calculated to 10% margin of error and 95% confidence levels. Thus, 100 was taken as the sample size.

Ethical clearance was obtained from the Institutional Ethics Committee before the study commenced. By using convenience sampling technique, 100 caregivers of patients with thalassemia major, attending the thalassemia daycare center run by the Department of Pediatrics in a tertiary care hospital of a metropolitan city, were selected for the study. The inclusion criteria were age group of patients less than 12 years and age group of caregivers between 18 and 50 years. Caregivers of patients with other chronic physical illnesses in addition to thalassemia major were excluded from the study. Approval for the study was obtained from the Institutional Ethics Committee-I of Seth G.S. Medical College on 14th January, 2016 (Project No. EC/193/2015).

After taking their written informed consent, a semistructured proforma was used to document the sociodemographic data of the caregivers including the marital status, occupation, education, income, family type, and comorbid medical illnesses. Details about the patient including duration of illness, average number of blood transfusions per year, total blood transfusions till date, and the cost for the blood transfusions were also noted. The caregivers were subsequently administered.

WHO-Quality of Life-BREF Scale (WHO-QOL-BREF)

This is a brief version of the WHO's QOL scale. It was derived from the WHO-QOL-100 and measures the quality of life. The WHO-QOL-BREF questionnaire contains 2 items from general health and overall quality of life and 24 items of satisfaction which are divided into 4 domains: physical health with 7 items (DOM1), psychological health with 6 items (DOM2), social relationships with 3 items (DOM3), and environmental health with 8 items (DOM4).[9] A five-point Likert scale was used to rate the items. Cronbach's alpha values for the domains were 0.80 for environmental domain, 0.69 for social domain, 0.77 for psychological domain, and 0.84 for physical domain, demonstrating good internal consistency.[10]

Coping Inventory for Stressful Situations 21 Scale

The Coping Inventory for Stressful Situations 21 (CISS-21) is a measuring instrument that has been proved to possess good psychometric characteristics in samples consisting of adults. The CISS-21 has been developed to assess three coping strategies: task oriented, emotion oriented, and avoidance coping.[11] Task-oriented coping is considered as a healthy coping technique with avoidance and emotion-oriented coping coming under unhealthy coping skills.[8] The Likert scale responses can range from a score of 1 (almost never) to 5 (almost always). The responses to the seven items of each subscale are added together to obtain scores for the three coping strategies.[12] Internal consistency scores for CISS-21 range from 0.75 to 0.88.[13]

General Health Questionnaire 12 (GHQ 12) Scale

The GHQ is a screening tool, which helps to identify the severity of psychological distress an individual has experienced within the past few weeks. This scale focuses on breaks in normal functioning rather than on life-long traits.[14]

For this study, the GHQ scoring method (0–0–1–1) was used over the simple Likert scale scoring method (0–1–2–3), as this is believed to help eliminate biases which might result from some participants, who have a tendency to choose responses 1 and 4 or 2 and 3, respectively. The scores were obtained by adding all items on the scale from 0 to 12.

GHQ was used for screening and caregivers scoring 3 or more on GHQ 12 underwent a clinical psychiatric interview and those who were found to have psychopathology in the interview were ascribed diagnosis as per International Classification of Diseases—Classification of Mental and Behavioral Disorders-10 (ICD 10).

Statistical analysis

Graph Pad Instat 3.0 (San Diego, California) was used for all statistical analysis. Descriptive analysis in terms of mean and standard deviation for continuous variables and frequency with percentage for nominal and ordinal variables was used. Association between sociodemographic profile with psychiatric morbidity and coping skills was done using Fisher's exact test. Comparison of quality of life with blood transfusion variables was done using Mann–Whitney U test. A P value of <0.05 was considered indicative of statistical significance of results.

Results

During the duration of the study, 100 eligible patients meeting our inclusion criteria were examined in the thalassemia daycare center and their caregivers were recruited into the study. The response rate was 100%. The mean age of the caregivers was 34.29 years with males (49%) being marginally less than females (51%). There was an almost equal distribution of samples amongst the different age groups. Majority (66%) of the caregivers were employed while the remaining 34% were unemployed. Parents of the patients comprised 92% of the caregivers. Older siblings made up 6% of the caregivers while the remaining 2% caregivers were second-degree relatives of the patients.

The mean illness duration of patients was 5.62 years with the range of 1–11 years. Majority of the patients (79%) had to undergo blood transfusion more than once a month with a mean of 16.86 times a year. More than half (54%) of patients had undergone blood transfusions more than 100 times till date. The mean annual expenditure for the transfusion-related procedures was around Rs. 13,016.

The prevalence of psychiatric morbidity amongst the caregivers was 35% with 39% females having a psychiatric morbidity in contrast to 30% males. The prevalence of depressive episode was 22% which was the maximum amongst the psychiatric illnesses diagnosed [Table 1].

Table 1

Prevalence of psychiatric morbidity

Psychiatric morbidity	Number of caregivers (n=100)
Depressive episode	22 (22%)
Mixed anxiety and depressive disorder	6 (6%)
Panic disorder	3 (3%)
Mental and behavioral disorders due to use of alcohol	4 (4%)
None	65 (65%)
Psychiatric morbidity

Psychiatric morbidity was found to have a significant association with both, the frequency (P value = 0.037) as well as the total number of blood transfusions (P value = 0.012). No significant association was found between psychiatric morbidity and the duration of illness. The association between psychiatric morbidity and sociodemographic variables like age, gender, employment status, and type of family was found to be statistically insignificant [Table 2].

Table 2

Association of psychiatric morbidity with sociodemographic variables

Caregiver and Patient variables		Psychiatric morbidity		P value*
		Present	Absent
Age	18-32	10	20	1.000
	33-50	25	45
Gender	Males	15	34	0.406
	Females	20	31
Employment	Employed	22	44	0.662
	Unemployed	13	21
Type of family	Joint	20	40	0.675
	Nuclear	15	25
Frequency of blood transfusion	Monthly	3	18	0.037*
	More than monthly	32	47
Total number of blood transfusion	<100	10	36	0.012*
	>100	25	29
Duration of illness	<5 years	11	31	0.139
	>5 years	24	34

*Fischer’s exact test

There was no significant difference in the quality of life across all the four domains between caregivers of different age, gender, education, employment status, and type of family. There was significant association (P value = 0.046) between quality of life in the psychological domain and the total number of blood transfusions [Table 3].

Table 3

Quality of life with total blood transfusions

Domains WHO-QOL-BREF	Up to 100 blood transfusions till date		More than 100 blood transfusions till date		P value*
	Mean score	SD	Mean score	SD
Physical	72.38	12.30	68.15	17.76	0.314
Psychological	62.24	16.12	57.89	17.02	0.046*
Social	56.48	18.65	51.89	18.52	0.137
Environmental	55.09	13.84	50.78	12.72	0.108

SD=Standard deviation. *Mann–Whitney U test

The prevalence of caregivers employing task-oriented coping strategies was found to be 62%, followed by emotion-based coping strategies (26%) and avoidant-oriented coping strategies (12%). Coping had a strong association with psychiatric morbidity (P = 0.001) and employment status (P = 0.009) [Table 4]. There was a higher prevalence of unhealthy coping strategies among caregivers with psychiatric morbidity [Figure 1].

Table 4

Association of types of coping skills (healthy versus unhealthy) with sociodemographic variables

Caregiver variables		Coping skills		P value*
		Healthy	Unhealthy
Age	18-32	18	12	0.824
	33-50	44	26
Gender	Males	35	14	0.066
	Females	27	24
Employment	Employed	47	19	0.009*
	Unemployed	15	19
Type of family	Joint	38	22	0.834
	Nuclear	24	16
Psychiatric Morbidity	Present	14	21	0.001*
	Absent	48	17

*Fischer’s exact test

Figure 1

Comparison of coping techniques employed in caregivers with psychiatric morbidity and those without psychiatric morbidity

Discussion

There was an almost equal representation of both, males (49%) and females (51%). Women caregivers handled caregiving tasks like bathing, toileting, and dressing as compared to their male counterparts who were more likely to be helping with finances, arrangement of care, and other less burdensome tasks.

Almost two-thirds of the caregivers (66%) were employed at the time of interview. The employed caregivers had to decrease their working hours, take leave from their job for a day, or had to rearrange their work schedule as the transfusion timings coincided with the job timings. Similar findings were present in a study done in Bangalore.[6] The remaining 34% caregivers were unemployed. Thalassemia compounded their financial burden and the need for frequent travel to a tertiary care center made securing a job immensely difficult for them, thus highlighting the psychoeconomic impact thalassemia has on the caregivers of patients.

Blood transfusion

In addition to impacting their work schedule, the need for blood transfusions also led to increased financial expenditure. The expenditure on blood transfusion also included the cost of traveling, food, and transfusion-related procedures. Parents experienced financial and social instability in addition to physical and psychological fatigue shown in different studies.[151617]

It is observed that more than two-third of the families (70%) had to spend up to 20% of their annual income on the treatment of thalassemia.[18] This cost is beyond the reach of most people in a developing nation like India, where there is an urgent need to develop family-centered population-directed health-care facilities that are affordable and accessible.[19] Moreover, in thalassemia, the financial cost can be expected to increase as a result of additional children being born who will have similar disorders. However, when compared to other chronic childhood illnesses, significantly more financial burden is seen in caregivers of patients with cerebral palsy, cancer, renal disorders, epilepsy, and diabetes which is due to higher cost of drugs and more frequent hospital visits. This is in contrast to thalassemia groups who are offered free blood transfusions at government hospitals.[20]

Psychiatric morbidity

Using the ICD-10 criteria, the prevalence of psychiatric morbidity was found to be 35% amongst the caregivers. A higher prevalence of psychiatric morbidity (57%) was found amongst caregivers in a previous Indian study.[6] The prevalence of depressive episode was found to be in keeping with findings from previous studies.[2122] A study done by Shaligram et al.[6] in India (43%) and Aziz et al.[23] in Pakistan (71%) had reported a higher incidence of depressive disorders. In comparison to our study, a comparatively higher prevalence of anxiety disorders was noted in other studies.[616]

The finding of higher prevalence of psychiatric morbidity in female caregivers (39%) was echoed in an East European study done in Lithuania.[24] This can be attributed to the fact that mothers are more emotionally attached to the child, spend more time with them, and hence, are more vulnerable to have mental health consequences. They are also known to undertake a larger caregiver role as housewives in an Indian setting.[25]

Our results that highlighted a significant association between frequency of blood transfusions and psychiatric morbidity and higher prevalence of psychiatric morbidity in caregivers whose patients had higher frequency of blood transfusion indicated that it is the severity of the illness that predisposes to psychiatric morbidity in caregivers. The duration of illness did not have any significant association to psychiatric morbidity in caregivers.

Quality of life

There was no significant difference in the quality of life in all four domains (physical, psychological, social, and environmental) between caregivers of different age, gender, education, employment status, and type of family. This was in contrast to findings from a previous study in England which had reported a better quality of life among male caregivers as compared to female caregivers.[26] A study by Sajjadi et al.[27] had also concluded that the mothers of sick children experience considerable physical and psychological problems which, in turn, reduce their quality of life. Notably, a study by Beluci et al.[28] also noted that caregivers with higher educational status had a better quality of life in the environmental domain.

There was a significant difference in quality of life in the psychological domain amongst caregivers depending on the total number of blood transfusions of their patients with a higher number of total blood transfusions affecting the psychological quality of life. This finding was echoed in the study done by Sharma et al.[29] who also suggested that health-related quality of life is significantly affected in caregivers of children with thalassemia major on regular transfusion. Similar findings were also seen in caregivers of patients with stroke and terminal cancer, where they had a lower quality of life and depressive moods which affected their daily activities, vitality, sleeping, happiness, and cognitive functioning.[3031] There was significant association between quality of life in the psychological domain and the total number of blood transfusions further stressing that higher number of blood transfusions impact the psychological quality of life of the caregiver.

Coping skills

Majority patients used task-oriented coping techniques. A significant association was found between psychiatric morbidity in caregivers and type of coping strategies used which indicated that caregivers having psychiatric morbidity used unhealthy coping strategies. A study done by Motaharian et al.[32] also came to a similar conclusion. As thalassemia is a chronic illness, it acts as a source of stress for the caregivers. Most families successfully cope with their children's chronic conditions. However, some may be unable to cope due to the limited access to accurate information. Caregivers with unhealthy coping skills tend to experience psychological stress paving the way for psychopathological states like depression and anxiety. Some may even resort to substance use, excessive gambling, and social withdrawal which paves the way for further neuropathological states due to their avoidant coping strategies.

Our study had a few limitations. It was conducted at a single tertiary care center of a metropolitan city and so the sample consisted largely of caregivers belonging to the lower socioeconomic group. Confounding factors like deprivation of basic needs experienced by lower socioeconomic groups, major life events like the death of a child due to similar illness, and stressors at work were not taken into consideration. Longitudinal studies with control groups and a larger sample size that also takes into consideration behavioral assessment and prognosis after providing adequate caregiver counseling are future recommendations. Thalassemia pharmacoeconomics also needs to be studied in the Indian setting.

Conclusions

Caregivers of patients suffering from chronic health conditions like thalassemia had psychiatric comorbidities and suffered from depression or anxiety for which treatment was not sought. Majority of the caregivers were the parents of the patients. There was a higher prevalence of psychiatric morbidity in female caregivers as compared to male caregivers. Irrespective of the duration of illness, patients needing higher frequency and total number of blood transfusions had caregivers who developed psychiatric morbidity. Caregivers with psychiatric morbidity were found to employ more unhealthy coping strategies.

Adequate and prompt referrals are necessary for management of psychopathology arising due to stress in the caregivers. Consultant liaison will ensure that these caregivers are counseled regarding the nature of the illness, its treatment, and healthy coping strategies. Beneficial effects of caregiving, including feeling appreciated by the care recipient, should be emphasized upon. Improved psychological health of the caregivers will guarantee better care of the ill child and improved adherence to treatment.

Declaration of patient consent

The authors certify that appropriate patients’ consents was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.