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Economic burden of specific learning disability: A prevalence-based cost of illness study of its direct, indirect, and intangible costs S Karande1, S D'souza1, N Gogtay2, M Shiledar1, R Sholapurwala11 Department of Pediatrics, Seth G.S. Medical College and K.E.M. Hospital, Parel, Mumbai, India 2 Department of Clinical Pharmacology, Seth G.S. Medical College and K.E.M. Hospital, Parel, Mumbai, India
Correspondence Address: Source of Support: None, Conflict of Interest: None DOI: 10.4103/jpgm.JPGM_413_18
Keywords: Attention-deficit hyperactivity disorder, cost of illness, dyslexia, specific learning disorders, students
Specific learning disability (SpLD) are a group of neurodevelopmental disorders characterized by severe and persistent difficulties in learning to efficiently read (“dyslexia” or “SpLD1”), write (“dysgraphia” or “SpLD2”), and/or perform mathematical calculations (“dyscalculia” or “SpLD3”), despite conventional instruction, intact hearing and vision, adequate motivation, and sociocultural opportunity.[1],[2] These afflicted students have poor school performance (PSP),[1],[2] anxiety,[3] social maladaptation,[4] and their problems generate significant parental stress.[5] From the societal perspective, it is important to evaluate the economic impacts [”cost of illness” (COI)] associated with SpLD and identify interventions that can reduce the burden of this invisible disability. We conducted the present study with the primary objective of evaluating the economic burden of SpLD imposed on families of afflicted students, on the healthcare provider, and on society. A secondary objective was to assess the impact of variables on the economic burden imposed on families of afflicted students.
Ethics The present study was approved by the institutional ethics committee. The study protocol is registered with the clinical trials registry of India (CTRI/2017/09/009814). The accompanying parent or legal guardian signed an informed consent form to participate in the study. Additionally, all students signed an assent form prior to enrolment. Confidentiality was maintained using unique identifiers. Design, setting, and sample size The present cross-sectional single-arm questionnaire-based descriptive COI study was conducted at the learning disability clinic of a public medical college in Mumbai, a megacity in western India over a period of 10 months, from December 2014 to September 2015. The prevalence of SpLD in India has been reported to be 3–10% among student populations.[6] In the present study, we assumed that 10% of students would have SpLD. With a 95% confidence level and 5% precision, the Daniel's formula [7] yielded a sample size of 138. Inclusion criteria and enrolment process The study population (recruited by non-probability sampling) comprised students aged ≥8 years who were diagnosed with SpLD (“one or more of these three disabilities,” viz., SpLD1 ± SpLD2 ± SpLD3) and were studying in English medium schools. Students with probable SpLD who were studying in non-English (vernacular) medium schools in our city could not be included for non-availability of standardized educational tests for confirming their diagnosis. A total of 138 students were recruited. No exclusion criteria that would preclude participation were used among students who met the inclusion criteria. Diagnosis of SpLD Only children ≥8 years of age were included in the study as a conclusive diagnosis of SpLD cannot be made before that age.[8] Each student had undergone standard recommended psycho-educational evaluation before the diagnosis of SpLD was confirmed.[8] Hearing and visual hearing deficits of >40% were ruled out by an otolaryngologist and an ophthalmologist, respectively. The counselor ruled out whether any environmental deprivation due to poor home or school environment, or any emotional problem was primarily responsible for a child's PSP. Wechsler Intelligence Scale for Children-Revised (M.C. Bhatt's Indian adaptation) was used to determine whether a student's global intelligence quotient score was average or above average (≥85).[8],[9] Using a locally developed and validated English curriculum-based test, the special educator conducted an educational assessment in specific areas of learning, namely, basic learning skills, reading comprehension, oral expression, listening comprehension, written expression, mathematical calculation, and mathematical reasoning.[10] Based on this test, an academic underachievement of up to 2 years below the student's actual school grade placement or chronological age led to a diagnosis of SpLD.[8],[10] In our clinic and hospital all consultations and tests were conducted free of cost. However, some parents who wished to do their child's audiometry and visual testing and occupational therapy assessment from a private clinic were allowed to do so. Using information from the child's parents and teachers, diagnosis of co-occurring attention-deficit/hyperactivity disorder (ADHD), if present, was made by ascertaining that student's specific behaviors met the required Diagnostic and Statistical Manual of Mental Disorders-IV-revised (DSM-IV-R) criteria.[11] Up to 40–46% of children with SpLD have associated ADHD which is characterized by persistent hyperactivity, impulsivity, and inattention and this comorbidity further impairs their learning.[12],[13] Data collection In the present COI study, data collection of costs were carried out from the “societal” perspective, namely both from the “afflicted families” (viz. the parent/guardian) and from the “healthcare provider's” perspectives.[14],[15] To collect data, our study followed the “prevalence-based retrospective” approach which measures the COI in the present and the past in a given year.[14],[15] Data collection of costs from the “afflicted families” perspective To estimate the costs from the “afflicted families” perspective, the “bottom-up” approach (“person-based”) was followed which assigns costs to individuals with the health condition of interest.[14],[15] A structured questionnaire was used to interview the parent/guardian to collect data related to three types of treatment costs of the afflicted students, viz. (1) “direct costs (medical and non-medical),”[14],[15] (2) “indirect costs,”[14],[15] and (3) “intangible losses.”[14],[15] For documenting the “direct costs (medical and non-medical)” and “indirect costs” of individual students the parent/guardian were asked to state the costs incurred since the time their PSP had been noticed. (1) “Direct costs”: (a) “Direct medical costs”: The direct medical costs (defined as the medical care expenditures incurred for diagnosis, treatment, and rehabilitation)[14],[15] of the students included all out-of-pocket payments for: (i) out-patient registration paper of our clinic, (ii) audiometry (including otolaryngologist's consultation charges, travelling expenses for doing audiometry), (iii) vision testing (including ophthalmologist's consultation charges, travelling expenses for doing vision testing, cost of spectacles/contact lenses if prescribed), (iv) psycho-educational testing (including clinical psychologist and special educators' charges, travelling expenses for doing testing), (v) remedial education, which is the cornerstone of treatment of SpLD [1],[2],[8] (including remedial teacher's charges and travelling expenses); one-to-one hourly remedial sessions with a special educator/remedial teacher twice or thrice weekly for a few years are necessary to achieve academic competence,[1],[8] (vi) counseling (including counselor's consultation charges, travelling expenses), (vii) occupational therapy, if availed, for students having SpLD2 (including occupational therapist's consultation charges, travelling expenses), (viii) psychiatric evaluation, if done (including psychiatrist's consultation charges, travelling expenses), (ix) medications, including complementary therapies such as ayurveda, homeopathy, if availed, and (x) other investigations [electroencephalogram, magnetic resonance imaging (MRI)/computed tomography (CT) brain, blood tests (e.g., vitamin B12/folic acid/thyroid hormone levels)], if done. (b) “Direct non-medical costs”: The direct non-medical costs [14],[15] of the students included all out-of-pocket payments for transportation costs for travelling to the learning disability clinic. (2) “Indirect costs”: Indirect costs included: (i) “loss of earnings” (average annual loss of wages due to absenteeism), (ii) “productivity losses” (average annual loss of income due to loss of job) for parent/guardian, as valued by the human capital method,[14],[15] and (iii) costs of “tuition classes,” including its travelling expenses. In our city, many parents due to lack of knowledge about SpLD prefer tuitions (both before and even after diagnosis of their child's disability) taken from a private regular teacher which is not the therapy for their child's disability,[1],[8] and (iv) costs of “academic enhancement measures,” namely books/compact discs bought to improve student's scholastic performance or costs of recreational activities classes (e.g., dance/swimming/art/craft) to channelize the student's energies into academics. (3) “Intangible costs”: Intangible costs data were collected by documenting the willingness-to-pay (WTP) value using the contingent valuation technique, a recommended method in COI studies that adopts a societal perspective.[14],[15] The parent/guardian would be provided with an initial bid (namely, their per capita income to minimize the starting bid bias) and asked whether they would like to pay this amount of money as a one-time payment for a remedy that would cure their child's disability. If the parent/guardian subjects answered positively (negatively), then the amount was increased (decreased) (i.e., doubled or halved) until respondents declined (accepted) the specified amount. Data related to seven variables: (i) age, (ii) gender of student, (iii) number of sibling(s), (iv) address (whether staying in Mumbai or outside Mumbai), (v) socioeconomic status, (vi) absence or presence of comorbid ADHD, and (vii) duration of PSP were noted using a supplementary questionnaire. Socioeconomic strata was determined by the Kuppuswamy's socioeconomic scale.[16],[17] Data collection of costs from the “healthcare provider's” perspective The direct medical costs from the “healthcare provider's” perspective,[14],[15] i.e., the “learning disability clinic costs” was computed by calculating the expenditure to run it, namely; “overt expenditure”: (i) salaries paid to the secretary, medical officer, counselor, clinical psychologist, and special educator, (ii) clinic's landline telephone bill expenses, (iii) stationary and photocopying expenses for maintaining patient records and for psycho-educational tests material, and (iv) the on-going “covert expenditure” (namely, electricity, water, and building maintenance expenses) during the period of study. All the costs were documented in Indian Rupees (INR) (1 US$ = ~66 INR in 2015). Data analysis Estimation of economic burden of SpLD from the “afflicted families” perspective First, the “direct costs” and “indirect costs” for every student were estimated as a simple sum of all their components under the individual costs. Second, every student's “total costs” was estimated by adding his/her “direct costs” and “indirect costs.” Third, the “direct costs,” “indirect costs,” and “total costs” of all 138 students were estimated. Fourth, the “average annual total costs” of every student was computed by dividing his/her “total costs” by the number of years the PSP had been noticed. Fifth, the “average annual total costs” of all 138 students were added to estimate the “average annual total costs of SpLD” for afflicted families. Sixth, the “intangible costs” of SpLD were estimated from the data collected. Estimation of economic burden of SpLD from the “healthcare provider's” perspective By extrapolating the “learning disability clinic costs” calculated over the study period of 10 months to 12 months the “average annual learning disability clinic costs” was estimated. Estimation of economic burden of SpLD from the “societal” perspective The “average annual total costs” for afflicted families was added to the “average annual learning disability clinic costs” to estimate “average annual total costs of SpLD” for society. The “average annual total costs of SpLD” per student was calculated by dividing the “average annual total costs of SpLD” for society by the number of participants (n = 138). Cost data are usually skewed, as was the case in the present study. Thus, we have presented the median and the ranges of all the cost components, including for the intangible costs. Since the outcomes (“dependent”) variables (direct costs, indirect costs, total costs, and intangible costs) are expressed as numerical data; the impact of the seven variables (“predictors”) on economic burden was analyzed by using regression. The dependent variables were first assessed for normality. Based on whether they were normal or not normally distributed, we decided to apply either linear or quantile regression as appropriate. We also assessed whether the predictor variables (that were quantitative) had a linear relationship with the dependent variable and chose only those that had a linear relationship for the regression analysis. The data were analyzed using the Statistical Package for Social Sciences, Version 17.0 for Windows (Chicago, USA). The statistical significant level was a P value <0.05 (two-tailed) for all analyses.
Characteristics of students enrolled in the study Of the 138 students enrolled, 32 (23.2%) students were “newly diagnosed” cases of SpLD and remaining 106 (76.8%) were “known” cases who had been diagnosed earlier elsewhere; or with us and were following up for re-certification of their disability for X or XII standard board examinations. No parent/guardian or student declined consent/assent for participation in the study. Of the informants: 79 (57.3%) were the students' mothers, 58 (42.0%) were the fathers, and 1 (0.7%) was the guardian. The mean age of all the 138 SpLD students was 14.6 years [standard deviation (SD) 2.2, range 8.2–18.4]; and of the 32 newly diagnosed SpLD students was 13.2 years (SD 2.1, range 8.3–16.1). The overall boy to girl's ratio was 1.9:1. Other details of clinical and sociodemographic characteristics (“variables”) are shown in [Table 1].
Economic burden of SpLD from the “afflicted families” perspective Direct costs ”Direct costs” estimated of all 138 students was INR 5,936,053 (five million, nine hundred thirty-six thousand, fifty-three); (median 14,195; range 180–476,120). The components of the “direct costs” are shown in [Table 2]a. Expenditure availed on remedial education comprised 64.39% of the “direct costs.” Of the 106 “known” cases: 33 (31.1%) had not undergone any remedial education; 45 (42.5%) had discontinued remedial education prematurely (mean 7.8 months, SD 7.1, range 1–24) after spending INR 695,604 (six hundred ninety-five thousand, six hundred four); (median 8,000; range 880–72,800) on it; 14 (13.2%) had completed at least 36 months (mean 43.7 months, SD 12.1, range 36–72) of remedial education and then discontinued it after spending INR 1,514,790 (one million, five hundred fourteen thousand, seven hundred ninety); (median 91,200; range 18,000–273,520) on it. Only 14/106 (13.2%) students were receiving it currently (mean 49.3 months, SD 27.9, range 6–96) and had spent INR 1,611,716 (one million, six hundred eleven thousand, seven hundred sixteen); (median 112,440; range 6,816–300,000) on it.
Indirect costs ”Indirect costs” estimated of all 138 students was INR 29,261,220 (twenty-nine million, two hundred sixty-one thousand, two hundred twenty); (median 87,000; range 0–2,786,000). The components of the “indirect costs” are shown in [Table 2]a. Expenditure availed on tuitions comprised 61.61% of the “indirect costs.” Only three known SpLD students were not attending tuition classes; of which only one was currently receiving remedial education since 18 months and had already spent INR 118,800 (one hundred eighteen thousand, eight hundred) on it; namely, INR 6,600/month. One student had received remedial education for 8 months and stopped after spending INR 18,560 (eighteen thousand, five hundred sixty) on it; namely, INR 2,320/month. One student had received neither remedial education nor tuitions. “Productivity losses” comprised 37.66% of the “indirect costs.” Fifteen previously working mothers had left their job to look after their child's studies after their disability had been diagnosed. Total costs ”Total costs” estimated of all 138 students was INR 35,197,273 (thirty-five million, one hundred ninety-seven thousand, two hundred seventy-three); (median 128,380; range 6,770–2,861,710). “Indirect costs” comprised 83.1% of the “total costs.” Average annual total costs The duration of PSP noticed by parent/guardian in their children is shown in [Table 2]b. The mean duration of PSP in the 138 students was 3.2 years (SD 1.9, range 1.0–9.0). Maximum students (37; 26.8%) had an “average annual total costs” in the range INR 50,001–100,000 [Table 2]c. The “average annual total costs” estimated was INR 10,356,720 (ten million, three hundred fifty-six thousand, seven hundred twenty); (median 45,995; range 5,290–523,730). Intangible costs Only 95 (68.8%) parents/guardian participated to estimate the “intangible costs” of SpLD. Of the remaining 43 (31.2%) parents who declined to participate, their reasons were as follows: 27 (19.6%) were not willing to pay any money for a remedy that would improve their child's disability; 6 (4.4%) felt that their child's disability cannot be improved; 6 (4.4%) refused to believe that their child had any disability; 2 (1.4%) said that they could not afford to pay anything; and 2 (1.4%) gave no reason for not participating. “Intangible costs” estimated of 95 students was INR 42,295,000 (forty-two million, two hundred ninety-five thousand); (median 200,000; range 8,000–4,000,000) as shown in [Table 2]d. Economic burden of SpLD from the “healthcare provider's” perspective The “average annual learning disability clinic costs” were estimated to be INR 2,169,888 (two million, one hundred sixty-nine thousand, eight hundred eighty-eight) as shown in [Table 2]d; of which the “overt expenditure” to run the clinic was found to be INR 729,888 (seven hundred twenty-nine thousand, eight hundred eighty-eight; 33.6%); namely (i) expenditure on salaries INR 672,000 (six hundred seventy-two thousand, 31.0%), (ii) landline telephone bill expenses INR 6,000 (six thousand, 0.2%], and (iii) stationary and photocopying expenses INR 51,888 (fifty-one thousand, eight hundred eighty-eight, 2.4%). The “covert expenditure” (namely, electricity, water, and maintenance expenses calculated as INR 1,800 per square feet per year, as per hospital data) was INR 1,440,000 (one million, four hundred forty thousand; 66.4%). Economic burden of SpLD from the “societal” perspective The “average annual total costs of SpLD” were estimated to be INR 12,526,608 (twelve million, five hundred twenty-six thousand, six hundred eight); and the “average annual total costs of SpLD” per student was estimated to be INR 90,773 (ninety thousand, seven hundred seventy-three) as shown in [Table 2]d. Impact of variables on economic burden of SpLD Impact on direct costs of SpLD As shown in [Table 3]a, only “duration of PSP” was the significant predictor of the direct costs (P = 0.001). In other words, for every month's increase in the duration of PSP, higher was the family expenditure by INR 804 (eight hundred four).
Impact on indirect costs of SpLD As shown in [Table 3]b, “duration of PSP” was the only significant predictor of “indirect costs.” For every monthly increase in the “duration of PSP” higher was the family's expenditure by INR 2,114 (two thousand one hundred fourteen) on “indirect costs” (P = 0.004). Impact on total costs of SpLD As shown in [Table 3]c, “duration of PSP” was the only significant predictor of “total costs.” For every monthly increase in the “duration of PSP” higher was the family's expenditure by INR 4,636 (four thousand six hundred thirty-six) on “total costs” (P < 0.001). Impact on intangible costs of SpLD As shown in [Table 3]d, “socioeconomic status” was the only significant predictor of “intangible costs.” Higher the “socioeconomic status” of the student's family, lower was their “intangible costs” by INR 130,000 (one hundred thirty thousand) (P = 0.003).
A COI study is useful to inform the economic burden of a disability, and aid the decision making for health resource allocation.[14],[15] The present study has documented that the economic burden of SpLD in the city of Mumbai is huge for the afflicted families, the healthcare provider, and for society at large. “Indirect costs” far outweighed “direct costs” of SpLD (83.1% versus 16.9%). Although, only 68.8% of the parents participated in estimating the hypothetical “intangible costs” of SpLD; it was higher than the “total costs” of SpLD. Furthermore, quantile regression analysis revealed that “longer duration of PSP” was an independent predictor of increased “direct, indirect, and total costs”; and “higher socioeconomic status” was an independent predictor of lower “intangible costs.” We cannot compare the present study with previous work because there is not any. A PubMed search using the medical subject headings (MeSH) words “economic burden” and “specific learning disorders” did not reveal any study which has evaluated the economic burden of SpLD in students. What is the importance of the present study?First, our study shows that in our city SpLD continues to be diagnosed late (mean age 13.2 years). Ideally, SpLD should be diagnosed when the afflicted student is in primary school so that there is adequate time to avail adequate remedial education to achieve academic competence.[1],[8] Second, our study shows that parents of students with SpLD wastefully spend a humungous amount on “tuition classes.” Almost all (96.4%) students, both before and after their diagnosis of SpLD, were attending “tuition classes” conducted by private regular teachers. Third, our study shows that a very few (13.2%) of the known students with SpLD complete 3 years of remedial education; while an overwhelming majority (73.6%) either does not undergo any remedial education or quits it prematurely. It is likely that the high costs of remedial education (approximately INR 36,000–108,000 annually) in our city could be one of the main reasons for this poor compliance. Currently, very few schools have resource rooms for remedial education within their premises and most parents have to avail remedial education from private special educators. Fourth, “productivity losses” due to SpLD are far from modest. Almost 11% of mothers quit their job after their child was diagnosed with SpLD resulting in loss of income for their families. Fifth, “intangible costs” far exceed the “total costs” due to SpLD. Many parents are willing to pay exceedingly high amounts (median, INR 200,000) for a hypothetical cure for their child's disability. “Intangible costs” reflect parental anguish in dealing with their child's disability and need to be documented for a comprehensive estimation of economic burden of a disability.[14],[15] Sixth, we found that higher socioeconomic status of the family was predictive of lower intangible costs. Perhaps parents from high socioeconomic status are not overwhelmed by their child's PSP; as their financial clout allows them to plan for their child's future irrespective of his/her academic competency (e.g., joining the family business or getting a capitation seat in higher education). What can be done to reduce the economic burden of SpLD? All “regular” classroom teachers should be sensitized to suspect and trained to screen for this disability early when the child is in primary school. A validated check list to be used by a regular teacher for a student with PSP studying in class standard III or IV is available in the public domain.[1] School managements should become proactive to set up resource rooms and employ special educators to ensure that these children receive regular and affordable remedial education.[1] Counseling parents would help improve their knowledge about SpLD and reduce wasteful expenditure on tuitions.[1],[18] The inclusion of SpLD in the “Rights of Persons with Disabilities” (RPWD-2016) bill, is a recent encouraging step in the betterment of care for students with SpLD.[19],[20] Ample funds from the Government of India's flagship program Sarva Shiksha Abhiyan (“Education for All” movement) are now available to train school teachers about this disability, set up resource rooms in every school, and learning disability centers in every medical college all over the country.[19] The strengths of the present study include adequate sampling size, high participation, and high response rates. The present study has its limitations. First, we could not assess the economic burden of SpLD in afflicted students who were studying in non-English (vernacular) medium schools in our city. Most of these go undetected for non-availability of standardized educational tests.[8] Second, students with SpLD from the lower socioeconomic strata of society were very few in our study population. Most of these study in poor-quality schools and go undetected because the school authorities lack awareness of SpLD.[8] Third, the non-probability sampling of the present study may have led to a recruitment bias in our findings. Fourth, the study results are subjected to recall bias of participating parent/guardian. However, we do not believe that these limitations adversely affect the utility of our results. Both due to the limitations as outlined above and the general paucity of data on economic burden of SpLD, there is a need for such studies to be carried out in clinics situated all over the country/world.
SpLD is a cost-intensive disability (intangible > indirect > direct costs). Tuitions, which are not the therapy for SpLD, are the most costly component of indirect costs; and remedial education is the most costly component of direct costs. To reduce the enormous economic burden of SpLD, all primary school students with PSP should be screened for SpLD to aid its early diagnosis. Afflicted students should be offered affordable remedial education within their schools. Parents should be counseled to reduce wasteful expenditure on tuitions. Acknowledgements We thank all the students who participated in the present study and their parents/guardians. Financial support and sponsorship The Learning Disability Clinic at Seth G.S. Medical College and K.E.M. Hospital is partially funded by a research grant from the Tata Interactive Systems, Mumbai. Conflict of interest Dr. Sunil Karande is the Editor of the Journal of Postgraduate Medicine.
[Table 1], [Table 2], [Table 3]
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