Cost–Benefit and Effectiveness of Newborn Screening of Congenital Hypothyroidism: Findings from a National Program in Iran

This Article

Citations


Article Information:


Group: 2010
Subgroup: Volume 8, Issue 1, Winter
Date: January 2010
Type: Original Article
Start Page: 1
End Page: 6

Authors:

  • SH Yarahmadi
  • Center for Disease Control, Ministry of Health and Medical Education, Tehran, IR.Iran
  • SJ Tabibi
  • Department of Health Services Management, Science and Research Branch , Islamic Azad University, Tehran, IR.Iran
  • KH Alimohammadzadeh
  • Department of Health Services Management, Science and Research Branch , Islamic Azad University, Tehran, IR.Iran
  • E Ainy
  • Safety Promotion and Injury Prevention Research Center, Tehran, IR.Iran
  • MM Gooya
  • Center for Disease Control, Ministry of Health and Medical Education, Tehran, IR.Iran
  • M Mojarrad
  • Research Institute for Endocrine Sciences, Shaheed Beheshti University of Medical Sciences, Tehran, IR.Iran
  • B Delgoshaei
  • Department of Health Services Management, Science and Research Branch , Islamic Azad University, Tehran, IR.Iran

      Correspondence:

      Affiliation: Department of Health Services Management, Science and Research Branch , Islamic Azad University
      City, Province: Tehran,
      Country: IR.Iran
      Tel:
      Fax:
      E-mail: Dr_khalil_AMZ@yahoo.com

Abstract:


In this study, the cost-benefit of a screening program based on the rial, the unit of currency used, was analyzed. The intelligence quotient (IQ), and height and weight were evaluated as indices for a population of children suffering from congenital hypothyroidism (CH). Materials and Methods: The total cost for the screening program, including hormone tests, diagnosis, medicine, treatments and care was identified and calculated up to the age of seventy years and this was compared to the costs related to training and caring for patients suffering from mental retardation, who had not been screened. The screening test was done using S&S filter paper and thyroid stimulating hormone (TSH) was determined by the ELISA test.The future costs and benefits with an annual rate of 3% discount of their current value was estimated. To evaluate the effectiveness of the screening program, IQs of 32 patients were identified and compared to 36 healthy children between 2004-2005. The IQs were evaluated according to "Good Enough" and "Proteus Maze" tests. Results: During 2008, 1165169 of 12489136 newborns (51% male, 49% female) underwent screening for CH, 92% cover-age of all newborns for that year. Four percent of recalled infants, with TSH>5 were subjected to diagnostic tests (T4, TSH, T3RU) and finally 2745 patients were identified. Benefit to cost ratios, based on a 3% annual discount rate, were 22, 41, 32, 34, 47 and 60 times lower, respectively. No significant differences were found with regard to diferences in IQ scores between cases and controls (cases: 105±19.3, controls: 111±19.4), height (cases: 106±0.7, controls: 102±4.9 cm) and weight (cases: 15.6±4.6, control 15.3±3.2 kg). Conclusion: The national Newborn Screening (NBS) program for CH has been successful and quite effective in Iran. The method not only has economical advantages but also reduces capital expenditures and preserves normal IQ of the patients under treatment and prevents mental retardation and growth complications.

Keywords: Newborn screening;Congenital hypothyroidism;Cost-benefit;Mental retardation

Manuscript Body:


Introduction

Congenital hypothyroidism (CH) is one of the most preventable causes of mental retardation1. National screening provides an opportunity for early detection and timely cure of newborns suffering from various genetic, glandular, vascular, hematological and lung diseases2. Screening for CH is regularly performed in most developed countries and in some of the third world countries as well3-7. Congenital hypothyroidism is a relatively common congenital disorder occurring in about 1 of 3000 to 1 of 4000 live births. Previous studies have reported a high incidence of CH in Iran8-10. In 2006, US preventive Services Tash Force (USPSTF) following a brief literature review reported the benefits of screening for CH continue to be well established. This update included a search for new and substantial evidence on the benefits and disadvantages of screening. The American Academy of Pediatrics (AAP) and the American Academy of Family Physicians recommend universal newborn screening for congenital hypothyroidism, and both in conjunction with the American Thyroid Association, and the Lawson Wilkins Pediatric Endocrine Society have recently published guidelines for screening and treatment for congenital hypothyroidism11,12. The cost-benefit and effectiveness of health services play important roles and have implications in the design and evaluation of health policies. The high incidence and prevalence of metabolic diseases, particularly congenital hypothyr-oidism (CH) and the damage caused thereby, such as mental retardation should considered as a critical issue and health priority in related policies. The object of this study was to evaluate the cost-benefit of the national newborn screening (NBS) program for CH. The intelligent quotient (IQ), height and weight were evaluated as indices to assess effectiveness of screening programs for children suffering from CH.

Materials and Methods

Total costs for screening program consisting of hormone tests, diagnosis, med-icine, treatments and caring were identified and calculated up to the age of seventy years and this was compared to the costs related to training and caring for patients suffering from mental retardation, in whom screening had not been applied. Excel was used to analyze data. The disability-adjusted life year (DALY) index was determinate by (DALY= YLL+YLD, YLL= N (0) * L (70), YLL=0 formula, standard life expectancy, incidence rate and aggrigation of disability according to the Dutch table were 70 years, 2.2 and 0.35 respectively. IQ score was determined from one of several different standardized tests, designed to assess intelligence. To evaluate the effectiveness of the screening program, IQs of 32 patients were identified and compared to 36 healthy children during 2004-2005; both groups were aged between 4-5 years and were of the same geographical area, social and eco-nomical classes; their IQs were evaluated using the "Good Enough" and "Proteus Maze" tests. SPSS software was employed to analyze the results. T-test was used to compare the mean values of the two groups, the cases and controls. Normal distribution and equal variance of the groups were assured by the application of “Kolmogorov-Smirnov" (KS) and "Leven" tests of evaluation.

Results

During 2008, 1165169 out of 12489136 newborns (51% males, 49% females) were subjected to the screening test, indicating 92.6% coverage of the total number of newborns for that year. Of recalled infants having TSH>5, 4.1% were subjected to diagnostic confirmation tests (T4, TSH, T3RU) and finally 2745 patients were identified. Incidence rate, coverage, number of recall and percent of recall were 2.38 per 1000, 92.6%, N: 46979 and 4.1 percent respectively. The DALY index was 53.9. In 2008, the total cost of screening, patient finding per each recalled newborn, during 70 years and mean of total screening identifying a patient in the program, medical cost, patient finding and medical care per care for each transient CH patient during 3 healthy and sick patients newborns during 70 years, and for each permanent CH patient years and are shown in table1.

Table 1. Total cost of Screening, patient finding per each recalled newborn, identification of a patient in program, medical care for each transient CH patient during 3 years and for each permanent CH patient during 70 years

Cost (Million Rials) $
Screening 2630 2.6
Cost of patient finding per each recalled newborn 95106 9.5
Cost of identifying a patient in program 12704554 1270
Total cost of medical care for each transient CH patient during 3 years 1425865 143
Total cost of medical care for each permanent CH patient during 70 years 3377778 338
Mean of total screening cost, patient finding and medical care per eachhealthy and patient newborns during 70 years 35868 3.6

 

Table 2 shows that benefit to cost ratio with the public sector day and night, 50% in the regard to education and care of patients with public sector and 50% in the private sector mental retardation. 100% in the public sector, day and night, 100% in private sector day 100% in the private sector, 50% in the public and night were 22, 41, 32, 34, 47 and 60 sector and 50% in the private sector, 100% in times lower respectively (Table 2).

Table 2. Proportion of benefit to cost ratios in congenital hypothyroidism screening Education and care of patients with mental retardation benefit / Cost (million Times Rials) lower

Education and care of patients with mental retardation benefit / Cost (millionRials) Times lower
100% in the public sector 923693/41649 22
100% in the private sector 1693801/41469 41
50% in the public sector and 50% in the private sector 1308747/41469 32
100% in the public sector day night 1407432/41469 34
50% in the public sector and 50% in the private sector day night 195031/41469 47
100% in the private sector day night 2493830/41469 60

Evaluation of the effectiveness of this groups in respect of IQ score are presented in program between the case and the control Table 3.

 

Table 3. Comparison of age, weight, height and IQ indexes in case and control group

Variables Cases Girls=14, Boys=18 Controls Girls=14, Boys=21 P value
Age(year) 4.9±1.4 5.0±3.5 NS
IQ(score) 111±19.4 105±19.3 NS
Weight(kg) 15.3±3.2 15.6±4.6 NS
Height(cm) 102±4.9 106±0.7 NS

 

In 2008 with the implementation of a screening program and identifying 2745 patients, the IQ score and DALY were CH in respective order of 98820 points and 1479555 years.

Discussion

The national NBS program for CH has been successful and quite effective in Iran. The method not only has economical advantages, and helps lower expenses, it also preserves the normal IQ of the patients under treatment and, last but not least, prevents mental retardation and growth complications. Other studies from Iran on national screening programs have documented a cost to benefit ratio of around 1 to 14 2. In 2008, of 46,979 infants recalled 2745 patients (6 %) were identified. In a similar research from the Zhejiang Province of China, of 6750 recalled newborns, 764 (approximately 11%) were screened13. In Scotland a study confirmed that after screening, the prevalence rate of disease in the first period (1979-1993) was 1 in 3655 live births, and in the second period (1994-2003), it reached 1 in 4363 live births14. Policy makers in accordance with the standards of America, interventions cost less than 50 to 60 dollars per quality-adjusted life year (QALY) is quite efficient15. National Institute for Health and Clinical Excellence (NICE) in the UK a reference laboratory for clinical eval-uation of interventions found that interven-tions amounting to less than 20 to 30000 pounds per QALY are acceptable16; the program conducted in Iran, was definitely more cost effective. A cost benefit ratio of 1:12 was documented for a screening program to identify congenital hypothyroidism, conducted in Layl of France17. Comprehensive screening programs to control congenital hypothyroidism (CH), a preventable form of mental retardation, are today seriously being considered by public health agencies18-20. A study form the UK documented congenital hypothyroidism screening programs to be a cost effective means for follow up of objectives21. A 1995 report from the USA declared the cost benefit of screening was 10 -fold, a finding which in line with previous reports.22-24 Using the first report doucument after implementation of the national screening program, in the current study, effectiveness of such screening programs was evaluated by IQ score and maintaining growth (height, weight and age) in identified and treated patients; non significant differences in IQ score average between aged 4 to 5 years in Esfahani children was observed 3. There was only one study in Iran which assessed children’s IQ score by Bender Gestalt and Ravn test in Mahdieh hospital of Tehran. Mean ± SD for case and control groups were (98±11) and (106±8)25. Treatment in all patients studied was begun a month earlier, the first dose of drug being adjusted according to previous studies (10-15 µg/kg) of infant’s body weight26,27. Several studies have shown a strong relationship between intelligence and early treatment and disease severity in patients with congenital hypothyroidism28-30. It seems that adequate doses of the drug is one of the causes for having normal intelligence quotientsin congenital hypothyroidism patients. Average date of treatment starting was 15.5 days after diagnosis; this finding was in line with previous reports31, 32. In line with our findings, one study showed that there was an inverse relation between age of patient at diagnosis and IQ score. Earlier diagnosis leads to higher IQ score. In this study, the patients who were treated at birth time (12 to 30 days after birth) had 15.7 higher average IQ scores compared to those treated 30 days after birth. In conclusion, he national NBS program for CH has been successful and quite effective in Iran. The method not only has economical implications which reduces capital costs, it also maintains the normal IQ of patients under treatment and, last but not least, prevents mental retardation and growth complications. Hence a suitably designed screening cost effective program opens new pathways for applications of such programs for other metabolic diseases.

Acknowledgement 
Authors express their gratitude to specialists from the ministry of health, and from the refer ence laboratory.

References: (34)

  1. Ordookhani A, Mirmiran P, Mohamadzadeh M, Hedayati M, Azizi F. A high prevalence of con-sanguineous and severe congenital hypothyroid¬ism in an Iranian population. J Pediatr Endocrinol Metab 2004; 17: 1201-9.
  2. Delavari AR, Yarahmadi SH, Mahdavi Hazaveh AR, Nouroozi Nejad A, Dini M. Cost- benefit Analysis of the neonatal Screening program im¬plementation for CH in Iran. DCC, Ministry of health and medical education, Tehran, I.R. Iran. Int J Endocrinol Metab 2006; 4: 84-87.
  3. Al-Maghamsi MS, Al-Hawsawi ZM, Ghulam GN,Okasha AM. Screening for congenital hypo-thyroidism in North West Region of Saudi Ara-bia. Saudi Med J 2002; 23: 1518-21.
  4. Delange F. Neonatal screening for congenital hy-pothyroidism: results and perspectives. Horm Res 1997; 48: 51-61.
  5. Elbualy M, Bold A, De Silva V, Gibbons U. Con¬genital hypothyroid screening: the Oman expe¬rience. J Trop Pediatr 1998; 44: 81-3.
  6. Sack J, Feldman I, Kaiserman I. Congenital hypo¬thyroidism screening in the West Bank: a test case for screening in developing regions. Horm Res 1998; 50: 151- 4.
  7. Yordam N, Calikoglu AS, Hatun S, Kandemir N,Oguz H, Tezic T, et al. Screening for congenital hypothyroidism in Turkey. Eur J Pediatr 1995; 154: 614-6.
  8. Hashemipour M, Amini M, Iranpour R, Sadri GH, Javaheri N, Haghighi S, et al. Prevalence of con¬genital hypothyroidism in Isfahan, Iran: results of a survey on 20,000 neonates. Horm Res 2004; 62: 79-83.
  9. Karamizadeh Z, Amirhakimi GH. Incidence of congenital hypothyroidism in Fars Province, Iran. Iran J Med Sci 1992; 17: 78-80 (Persian).
  10. Ordookhani A, Mirmiran P, Najafi R, Hedayati M, Azizi F. Congenital hypothyroidism in Iran. Indian J Pediatr 2003; 70: 625-8.
  11. U.S. Preventive Services Task Force. Screening for Congenital Hypothyroidism. Guide to Clinical Preventive Services, 2nd edition. Alexandria, VA:International Medical Publishing; 1996. p. 503-7.
  12. American Academy of Family Physicians. Policy Statement on Newborn Screening. Issue Brief 2006; 5: 1-12.
  13. Chen xx, yang RL, shi YH, cao Lp, zhou xL, Mao HQ, et al. Screening for congenital hypo-hyperthyrotrthyroidism in neonates of Zhejiang Province dur ing 1999-2004. Zhejiang Da Xue Xue Bao Yi Xue Ban 2005; 34: 304-7 (Chinese).
  14. Jones JH, Mackenzie J, Croft GA, Beaton S, Young D, Donaldson MD. Improvement in screening performance and diagnosis of congenit¬al hypothyroidism in Scotland 1979-2003. Arch Dis Child 2006; 91: 680-5
  15. Owens DK. Interpretation of Cost-Effectiveness Analyses. J Gen Intern Med 1998; 13: 716-7.
  16. Devlin N, Parkin D, editors. Does NICE have a cost effectiveness threshold and what other fac¬tors influence its decisions? A discrete choice analysis. London: Department of Economics, City University of London; 2003.
  17. Dhondt JL, Farriaux JP, Sailly JC, Lebrun T. Economic evaluation of cost- benefit ratio of neo¬natal screening procedure for phenylketunuria and hypothyroidism. J Inherit Metab Dis 1991; 14: 633-9.
  18. Gu X, Wang J, Ye J, Cheng X. A cost-benefit evaluation of neonatal screening for phenylketonuria and congenital hypothyroidism. Zhonghua Yu Fang Yi Xue Za Zhi 2000; 34: 147-9 (Chinese).
  19. Laberge C. Cost-benefit evaluation of neonatal thyroid screening: The Quebec Experience 1973¬1982. In: Dussault JH, Walker P, editors. Conge-nital Hypothyroidism. New York: Dekker 1983. p. 209-16.
  20. Layde PM, Von Allmen SD, Oakely GP Jr. Con-genital hypothyroidism control programs: A cost-benefit analysis. JAMA 1979; 241: 2290-2.
  21. Pollitt RJ, Green A, Mccabe, CJ, Biith A, Cooper NJ, Leonard JV, et al. Neonatal screening for in¬born errors of metabolism: cost, yield and out-come. Health Technol Assess 1997; 1: i-iv, 1-202.
  22. Mugarra Bidea I, Cabases Hita JM. Cost-benefit analysis of the early detection program for meta-bolic diseases in the Autonomous Basque Com-munity. Gac Sanit 1990; 4: 140-4 (Spanish).
  23. Elbualy M, Bold A, De Silva V, Gibbons U. Con-genital hypothyroid screening: the Oman expe rience. J Trop Pediatr 1998; 44: 81-3.
  24. Al-Maghamsi MS, Al-Hawsawi ZM, Ghulam GN, Okasha AM. Screening for congenital hypothyroidism in North West region of Saudi Arabia Saudi Med J 2002; 23: 1518-21.
  25. Azizi F, Afkhami M, Sarshar A, Nafarabadi M.Effects of transient neonatal hyperthyrotrthyroidism opinemia on intellectual quotient and psychomo-tor performance. Int J Vitam Nutr Res 2001; 71:70-3.
  26. Kaye CI; Committee on Genetics, Accurso F, LaFranchi S, Lane PA, Hope N, Sonya P, et al.Newborn Screening Fact Sheets. Pediatrics 2006;118; e934-963.
  27. American Academy of Pediatrics, Rose SR; Section on Endocrinology and Committee on Genetics, American Thyroid Association, Brown RS;Public Health Committee, Lawson Wilkins Pediatric Endocrine Society, Foley T, Kaplowitz PB, Kaye CI, et al. Update of newborn screening and therapy for congenital hypothyroidism. Pediatrics2006; 117: 2290-303.
  28. Glorieux J, Dussault JH, Morissette J, Desjardins M, Letarte J, Guyda M. Follow-up at ages 5 and 7 years on mental development in children with hy pothyroidism detected by Quebec Screening Program. J Pediatr 1985; 107: 913-5.
  29. Virtanen M, Santavuori P, Hirvonen E, Perheentupa J. Multivariate analysis of psychomotor de velopment in congenital hypothyroidism. Acta Paediatr Scand 1989; 78: 405-11.
  30. Fuggle PW, Grant DB, Smith I, Murphy G. Intelligence motor skills and behaviour at 5 years in early-treated congenital hypothyroidism. Eur J Pediatr 1991; 150: 570-4.
  31. Dubuis JM, Glorieux J, Richer F, Deal CL, Dussault JH, Van Vliet G. Outcome of severe congenital hypothyroidism: closing the developmental gap with early high dose levothyroxine treatment. J Clin Endocrinol Metab 1996; 81: 222–7.
  32. Bongers-Schokking JJ, Koot HM, Wiersma D, Verkerk PH, de Muinck Keizer-Schrama SM. Influence of timing and dose of thyroid hormone replacement on development in infants with congenital hypothyroidism. J Pediatr 2000; 136: 292– 7.
  33. Raymond J, LaFranchi SH. Fetal and neonatal thyroid function: review and summary of significant new findings. Curr Opin Endocrinol Diabetes Obes 2010; 1: 1-7.
  34. Kempers MJ, van der Sluijs Veer L, Nijhuis-van der Sanden RW, Lanting CI, Kooistra L, Wiedijk BM, et al. Neonatal screening for congenital hypothyroidism in the Netherlands: cognitive and motor outcome at 10 years of age. J Clin Endocrinol Metab 2007; 92: 919-24.