Pharmacoeconomic Evaluation of Costs of Myelomeningocele and Meningocele Treatment and Screening

Figures & data

Figure 1 PRISMA flow diagram of literature search and selection for articles included in this systematic review. *Studies not eligible excluded based on their abstracts. **Abstracts, conference papers, reviews, systematic reviews, posters, protocols, and letters to the editors were excluded.

Table 1 An Overview of All the Economic Studies on the Meningocele Included in This Review

No	1	2	3	4	5	6
Reference	Gollogly et al, 2008^Citation15	Davis et al, 2014^Citation21	Roux et al, 2007^Citation22	Lorber et al, 1972^Citation23	Oucheng et al, 2010^Citation19	Liptak et al, 1985^Citation24
Country	Cambodia	Guatemala	Cambodia	UK	Cambodia	USA
Year of Publication	2008	2014	2007	1972	2010	1985
Study Type	Cost description	Cost effectiveness analysis	Cost description	Cost description	Cost description	Cost description
Population	128 patients	1 patient with thoracic meningocele/4 years old; 6 lipomyelomeningocele patients	Forty-five children and seven young adults with frontoethmoidal meningocoeles (43 cases with nasoethmoidal MC	36 infants	257 frontoethmoidal meningoencephalocele	5 patients
Discount rate (Cost)	N.R	N.R	N.R	N.R	N.R.	N.R.
Intervention	Computerized tomography scanning cost vs magnetic resonance imaging.	Full cost of surgery in Guatemala	Full cost of surgery	Inpatient cost	Full cost of surgery in Cambodia	Hospital cost for shunt revision
Comparator/s	Cost of surgery	Full cost of surgery at the University of Michigan	N.R	N.R	N.R	N.R
Intervention duration	Within the period of 4 years	Week-long annual pediatric neurosurgical visit	4 weeks/year for 3 years	1 June 1962–5 September 1964	2004–2009	Children born since 1973 with meningocele at Strong Memorial Hospital (SMH) and the Birth Defects Center (BDC), Rochester
Outcomes	US$100 (computerized tomography scanning), and US$200 (magnetic resonance);US$300–US$500 (cost per operation)	Complete mission costs were $53,152, for a cost effectiveness of $385 per DALY averted	Complete cost of operation was US $380.	Thus the total inpatient cost would be of the order of £750,000 or £7500 per patient	The total cost of each operation was US $380.	The mean hospital cost for shunt revision (excluding physician’s fees) was $ 4543.
Perspective	Governmental	Healthcare system	Healthcare system	Governmental	Healthcare system	Healthcare system
ICER/ QALY/QALD/DALY	The net gain of 10 DALYs was reported for each surgery.	Thoracic meningocele repair was reported to have a 4.3 DALYs.	N.R	N.R	N.R	N.R

Table 2 An Overview of All the Economic Studies on the Myelomeningocele Included in This Review

No	1	2	3	4	5	6	7
Reference	Packer et al, 2021^Citation25	Werner et al, 2012^Citation20	Lim et al, 2019^Citation26	Sin et al, 2007^Citation27	Harris et al, 2016^Citation28	Pham et al, 2013^Citation29	Santos et al, 2017^Citation30
Country	USA	USA	USA	USA	USA	Australia	Africa
Year of Publication	2021	2012	2019	2000	2016	2013	2017
Study Type	Cost-effectiveness; Decision tree	Cost-effectiveness	Cost-effectiveness	Cost description analysis	Cost description analysis	Cost description analysis	Cost description analysis
Population	Theoretical cohort of 500 patients with prenatally diagnosed MMC	Hypothetical cohort of either neonates undergoing post natal surgery, or fetus undergoing prenatal repair between 19 and 26 weeks’ gestation	42 patients	88 MMC patients	1279 patients	23 patients	20 MMC patients
Discount rate	3%	N.R	N.R	N.R	N.R	N.R.	N.R
Intervention	Fetoscopic surgical approach	Prenatal MMC repair	Ventriculoperitoneal shunting (VPS)	Total cost for the hospitalization	Total cost for the hospitalization	Costs of surgical intervention	Cost of a brain ultrasound
Comparator/s	Open repair of MMC	Postnatal MMC repair	Endoscopic third ventriculostomy (ETV), and ETV with choroid plexus cauterization (ETV/CPC)	N.R	N.R	N.R.	Cost of computed tomography scan
Intervention duration	N.R	Soon after birth or between 19 and 26 weeks gestation	January 2012-January 2015	Patient admissions for the year 2000	MMC patients for the year 2000, 2003, 2006, and 2009	2007–2009	February 2016-August 2016
Outcomes	The total cost of fetoscopic approach was $29,156,613 versus $25,608,238 for the open repair approach	Prenatal MMC repair is cost effective and cost saving (saves $2066778per 100 cases repaired) compared to post natal repair	VPS was more cost-effective than ETV/CPC	The average cost was $59,889.00 ($9657.00–$419,890.00)	Total cost of hospitalization for MMC patients remained almost stable from 42,843 dollars in 2003 to 46,749 dollars in 2009.	The average cost of shunt insertion in MMC patients was $50,186.	The cost of a brain ultrasound (11 USD), vs computed tomography scan (110 USD)
Perspective	Maternal	Healthcare system	Healthcare system	Hospital	N.R.	Healthcare system	Healthcare system
ICER/QALY/DALY	Fetoscopic MMC surgical approach was cost-effective with an ICER of $1029/QALY	ICER= $4273/QALY gained (at the minimal postnatal repair cost of $5882). ICER=$35,531/QALY gained (at the maximum prenatal repair cost of $75,787)	VPS had an ICER of $76,620.32 vs ETV/CPC with an ICER of $112,762.44	N.R	N.R	N.R	N.R
No	8	9	10	11	12	13
Reference	Kshettry et al, 2014^Citation31	Bea et al, 1994^Citation32	Hubbard et al, 2019^Citation33	Liptak et al, 1985^Citation24	Cartwright et al, 2019^Citation34	Werner et al, 2012^Citation35
Country	USA	Spain	Haiti	USA	USA	USA
Year of Publication	2014	1994	2019	1985	2019	2012
Study Type	Cost description analysis	Cost description analysis	Cost description analysis	Cost description analysis	Cost effectiveness	Cost effectiveness
Population	860 MMC surgical cases	1500 MMC patients	38 MMC patients	62 MMC patients	13 MMC patients	100 MMC patients
Discount rate	N.R.	N.R.	N.K	N.R	N.R	N.R.
Intervention	Cost of hospitalization for MMC repair	Cost of admission in hospital	Cost of neurosurgical procedure	Hospital cost for shunt revision	Cost of the occlusive method	Prenatal MMC repair
Comparator/s	N.R	N.R.	N.R	N.R	Cost of the drip method	Postnatal MMC repair
Intervention duration	2007–2010	1986–1988	2008–2015	Children born since 1973 with MMC at Strong Memorial Hospital (SMH) and the Birth Defects Center (BDC), Rochester	Immediately after delivery	Soon after birth or during pregnancy
Outcomes	The total cost of MMC repair in hospital was $77.2 million dollars/year.	The cost of admissions in Spanish hospital over the past 15 years was US$44,297,070.	The net economic benefit for neurosurgical intervention was $2.5- $5.5 million	The mean hospital cost for shunt revision (excluding physician’s fees) was $ 4543.	Costs of supplies per patient for the drip method was ($4.84), higher than the occlusive method ($0.88). The occlusive technique was more cost effective.	Prenatal MMC repair is cost effective. For 100 fetal MMC repairs there is a QALY of 64, and a cost savings of $3,135,557.
Perspective	Neurosurgical	Healthcare system	Neurosurgical	Healthcare system	Hospital	Healthcare system
ICER/QALY/DALY	N.R	N.R.	The cost per DALY was $86- $245	N.R	N.R	ICER is $48,993 per QALY gained

Gollogly J, Oucheng N, Lauer G. Frontoethmoidal meningoencephalocoele repair in Cambodia: outcomes and cost comparisons. Trop doctor. 2008;38(3):167–170. doi:10.1258/td.2007.070124

 PubMed Web of Science ®Google Scholar

Davis MC, Than KD, Garton HJ, et al. Cost effectiveness of a short-term pediatric neurosurgical brigade to Guatemala. World Neurosurg. 2014;82(6):974–979. doi:10.1016/j.wneu.2014.08.038

 PubMed Web of Science ®Google Scholar

Roux F-E, Lauwers F, Oucheng N, et al. Treatment of frontoethmoidal meningoencephalocele in Cambodia: a low-cost procedure for developing countries. J Neurosurg. 2007;107(1 Suppl):11–21. doi:10.3171/PED-07/07/011

 PubMed Web of Science ®Google Scholar

Lorber J. Spina bifida cystica. Results of treatment of 270 consecutive cases with criteria for selection for the future. Arch Dis childhood. 1972;47(256):854–873. doi:10.1136/adc.47.256.854

 PubMed Web of Science ®Google Scholar

Oucheng N, Lauwers F, Gollogly J, et al. Frontoethmoidal meningoencephalocele: appraisal of 200 operated cases. Journal of Neurosurgery. Pediatrics. 2010;6(6):541–549. doi:10.3171/2010.9.PEDS1043

 PubMed Web of Science ®Google Scholar

Liptak GS, Masiulis BS, McDonald JV, et al. Ventricular shunt survival in children with neural tube defects. Acta neurochirurgica. 1985;74(3–4):113–117. doi:10.1007/BF0141879

 PubMed Web of Science ®Google Scholar

Packer CH, Hersh AR, Caughey AB. Fetoscopic compared with open repair of myelomeningocele: a 2-delivery cost-effectiveness analysis. Am J Clin Exp Obstet Gynecol. 2021;3(6):100434. doi:10.1016/j.ajogmf.2021.100434

 Google Scholar

Werner EF, Han CS, Burd I, et al. Evaluating the cost-effectiveness of prenatal surgery for myelomeningocele: a decision analysis. Ultrasound Obstet Gynecol. 2012;40(2):158–164. doi:10.1002/uog.11176

 PubMed Web of Science ®Google Scholar

Lim J, Tang AR, Liles C, et al. The cost of hydrocephalus: a cost-effectiveness model for evaluating surgical techniques. J Neurosurg Pediatr. 2023;23(1):109–118. doi:10.3171/2018.6.PEDS17654

 Google Scholar

Sin AH, Rashidi M, Caldito G, et al. Surgical treatment of myelomeningocele: year 2000 hospitalization, outcome, and cost analysis in the US. Childs Nerv Syst. 2007;23(10):1125–1127. doi:10.1007/s00381-007-0375-9

 PubMed Web of Science ®Google Scholar

Harris DA, Cherian J, LoPresti M, et al. Trends in epidemiology and hospitalization utilization for myelomeningocele repair from 2000 to 2009. Childs Nerv Syst. 2016;32(7):1273–1279. doi:10.1007/s00381-016-3091-5

 PubMed Web of Science ®Google Scholar

Pham ACQ, Fan C, Owler BK, et al. Treating pediatric hydrocephalus in Australia: a 3-year hospital-based cost analysis and comparison with other studies. J Neurosurg Pediatr. 2013;11(4):398–401. doi:10.3171/2013.1.PEDS12233

 PubMed Web of Science ®Google Scholar

Santos MM, Rubagumya DK, Dominic I, et al. Infant hydrocephalus in sub-Saharan Africa: the reality on the Tanzanian side of the lake. J Neurosurg Pediatr. 2017;20(5):423–431. doi:10.3171/2017.5.PEDS1755

 PubMed Web of Science ®Google Scholar

Kshettry VR, Kelly ML, Rosenbaum BP, et al. Myelomeningocele: surgical trends and predictors of outcome in the United States, 1988–2010. J Neurosurg Pediatr. 2014;13(6):666–678. doi:10.3171/2014.3.PEDS13597

 PubMed Web of Science ®Google Scholar

Bea Muñoz M, Diaz Llopis I, Martinez Agullo E, et al. A multicentre study of the hospital care of 1500 patients with myelomeningocele. Paraplegia. 1994;32(8):561–564. doi:10.1038/sc.1994.88

 PubMedGoogle Scholar

Hubbard ZS, Shah AH, Ragheb M, et al. Economic benefit of neurosurgical intervention for infant hydrocephalus in Haiti. J Neurosurg Pediatr. 2019:1–9. doi:10.3171/2019.4.PEDS18563

 PubMed Web of Science ®Google Scholar

Cartwright CC, Igbaseimokumo U, Olsen S, et al. A comparison of dressing techniques for presurgical closure of myelomeningocele in the Neonate. J Neurosci Nurs. 2019;51(5):217–220. doi:10.1097/JNN.0000000000000461

 PubMed Web of Science ®Google Scholar

Werner EF, Lipkind H, Copel JA, et al. A cost effective analysis of prenatal surgery for myelomeningocele. Presented at the 30th Annual Meeting of the Society for Maternal-Fetal Medicine; February 9–12; 2012; Dallas, TX.

 Google Scholar