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Economic Valuation of the Global Burden of Cleft Disease Averted by a Large Cleft Charity

Authors:

Abstract

Background: This study attempts to quantify the burden of disease averted through the global surgical work of a large cleft charity, and estimate the economic impact of this effort over a 10-year period. Methods: Anonymized data of all primary cleft lip and cleft palate procedures in the Smile Train database were analyzed and disability-adjusted life years (DALYs) calculated using country-specific life expectancy tables, established disability weights, and estimated success of surgery and residual disability probabilities; multiple age weighting and discounting permutations were included. Averted DALYs were calculated and gross national income (GNI) per capita was then multiplied by averted DALYs to estimate economic gains. Results: 548,147 primary cleft procedures were performed in 83 countries between 2001 and 2011. 547,769 records contained complete data available for the study; 58 % were cleft lip and 42 % cleft palate. Averted DALYs ranged between 1.46 and 4.95 M. The mean economic impact ranged between USD 5510 and 50,634 per person. This corresponded to a global economic impact of between USD 3.0B and 27.7B USD, depending on the DALY and GNI values used. The estimated cost of providing these procedures based on an average reimbursement rate was USD 197M (0.7-6.6 % of the estimated impact). Conclusions: The immense economic gain realized through procedures focused on a small proportion of the surgical burden of disease highlights the importance and cost-effectiveness of surgical treatment globally. This methodology can be applied to evaluate interventions for other conditions, and for evidence-based health care resource allocation.
ORIGINAL SCIENTIFIC REPORT
Economic Valuation of the Global Burden of Cleft Disease
Averted by a Large Cleft Charity
Dan Poenaru
1
Dan Lin
2
Scott Corlew
3
ÓSocie
´te
´Internationale de Chirurgie 2015
Abstract
Background This study attempts to quantify the burden of disease averted through the global surgical work of a
large cleft charity, and estimate the economic impact of this effort over a 10-year period.
Methods Anonymized data of all primary cleft lip and cleft palate procedures in the Smile Train database were
analyzed and disability-adjusted life years (DALYs) calculated using country-specific life expectancy tables,
established disability weights, and estimated success of surgery and residual disability probabilities; multiple age
weighting and discounting permutations were included. Averted DALYs were calculated and gross national income
(GNI) per capita was then multiplied by averted DALYs to estimate economic gains.
Results 548,147 primary cleft procedures were performed in 83 countries between 2001 and 2011. 547,769 records
contained complete data available for the study; 58 % were cleft lip and 42 % cleft palate. Averted DALYs ranged
between 1.46 and 4.95 M. The mean economic impact ranged between USD 5510 and 50,634 per person. This
corresponded to a global economic impact of between USD 3.0B and 27.7B USD, depending on the DALY and GNI
values used. The estimated cost of providing these procedures based on an average reimbursement rate was USD
197M (0.7–6.6 % of the estimated impact).
Conclusions The immense economic gain realized through procedures focused on a small proportion of the surgical
burden of disease highlights the importance and cost-effectiveness of surgical treatment globally. This methodology
can be applied to evaluate interventions for other conditions, and for evidence-based health care resource allocation.
Introduction
Several metrics have been used in the measurement of
health interventions, including life expectancy, mortality
rates, disability-adjusted life years (DALYs), quality-
adjusted life years (QALYs), and others [13]. However, in
order to compare the societal impact of health care to other
sectors, a common measurement tool is needed. Economic
assessment is such a cross-sector ‘‘universal currency,’’ its
main drawback being the difficulty of measuring economic
benefit.
This study uses a framework derived from previous
work [411] to examine the economic value of the pro-
grams of a non-governmental organization. Smile Train
(ST) supports the surgical care of patients with cleft lip and
palate (CLP) in low- and middle-income countries
(LMICs). ST identifies and trains surgical teams in each
country and contracts to provide operative care for a pre-
determined fee per case [12]. This model is akin to the
&Scott Corlew
dscorlew@gmail.com
1
MyungSung Christian Medical Center, Addis Ababa,
Ethiopia and Montreal Children’s Hospital, Montreal, Canada
2
Rollins School of Public Health, Emory University, Atlanta,
GA, USA
3
2111 Riverview Drive, Murfreesboro, TN 37129, USA
123
World J Surg
DOI 10.1007/s00268-015-3367-z
specialty surgical hospital platform described by Shrime
et al. [13], but uses LMIC surgeons exclusively. Given that
CLP impacts the social, physical, and economic lives of
affected individuals, this study estimates the economic
impact of the ST work, providing a framework for exam-
ining the value of health interventions for cross-sector
comparison.
Methods
The approach was to estimate the economic productivity of
treated individuals, then derive the counterfactual of their
economic productivity had they not been beneficiaries of
the programs. The difference between these two figures, is
presented as the economic effect of the program. The dif-
ference in DALYs is also presented.
The database of all procedures performed in ST pro-
grams between 2001 and 2011 was utilized. Only primary
(first-time) CLP repair procedures were included. Country-
specific life expectancy (LE) values were used, rather than
a universal value as used by the Global Burden of Disease
(GBD) study [14].
The standard DALY formula was used for burden of
disease (BoD): DALYs =YLL (Years of Life Lost) ?
YLD (Years Lived with Disability). The YLL factor was
omitted in light of the small mortality of cleft lip and
palate.
The counterfactual BoD in DALYs potentially incurred
by each patient without the surgical intervention (which
includes the non-avertable as well as the avertable DALYs
[15]), was estimated thus:
BoDwithout intervention ¼DWuntreated ageoperation

þDWuntreated LEage at treatment

;
where DW
untreated
=disability weight for CLP untreated
(both DW
untreated
and DW
treated
from the GBD study [16]
and LE
age at treatment
=life expectancy at the age the
operation occurred, from the Standard West Level 26 Life
Table [17]).
DALYs actually incurred by each patient, or the
BoD
with intervention
, were then estimated in two ways. The
first used the GBD study method for calculating DALYs:
BoDwith intervention ¼DWuntreated ageoperation

þDWtreated LEage at operation

DALYs incurred with the intervention were also
estimated by the method published by McCord et al. [11]
and widely followed in the surgical literature [1821]. This
method multiplies the ideal impact of surgical intervention
by the risk of permanent disability without surgery (RPD)
and the estimate of residual disability (ERD) after the
operation. The latter factor aims to account for the residual
effects of the deformity, the possibility of post-operative
complications, and the possible need for further
procedures. In CLP, the RPD is 1.0, since disability is
expected in the absence of surgery in 100 % of cases, while
the resolution of disability (1-ERD) was estimated to be
Fig. 1 Diagram of age milestones and calculations used for estimating averted DALYs
World J Surg
123
between 75 and 94 % for cleft lip and between 25 and
74 % for cleft palate. For the purpose of this study, the two
factors were combined into a single ‘‘Effectiveness Factor’
(EF) of 0.8 for cleft lip and 0.5 for cleft palate, similar to
the previously used ‘‘probability of successful treatment’
(PST) for CLP [10]. This Effectiveness Factor postulates
that the operation, on average, resulted in resolution of
80 % of the residual disability for cleft lip and 50 % for
cleft palate patients. This EF was hence used in place of the
DW
treated
:
BoDwith intervention ¼ðDWuntreated AgeoperationÞ
þðDWuntreated 1EFðÞ
LEAge at operationÞ
The difference between DALYs without and with
surgical intervention represents the effect of the
intervention on the health status of the patient and,
globally, on alleviating BoD. Figure 1shows the
calculation markers in a schematic fashion.
DALYs were calculated with and without age weighting
as was done in the original GBD study [22], and with and
without 3 % future discounting.
These averted DALY values were then multiplied by the
GNI per capita for each country [23] to give the economic
value added to the national economy over the lifetime of
each patient. Using both the Atlas and Purchasing Power
Parity (PPP) methods, two economic totals for each
country were derived for each DALY calculation. The
methodologies used in this study for calculating DALYs
and the economic gain are shown diagrammatically in
Fig. 2.
Individual totals were summed to give an estimate of
economic value added for the entire program. This was
reported by year and for the entire period of the study.
A cost-effectiveness analysis (CEA) was performed as
previously reported for Smile Train [10]. As actual costs at
the hundreds of individual sites vary widely and were not
available, the set contributions per procedure paid by the
organization were used as a proxy.
Data analysis and simple descriptive statistics were
performed in Microsoft Excel
Ò
.
Results
Tables 1and 2show the results in very accessible format.
Between 2001 and 2011, 548,147 primary operations to
repair CLP were performed in 83 countries. Figure 3shows
the cases available for study and the patient characteristics.
Total averted DALYs achieved by method of calculation
and year are shown in Fig. 4. Table 1details the DALYs
averted by procedure and by the various calculation
methods. Using the GBD DW method, about one-third of
the averted BoD was due to cleft lip repairs and two-thirds
to cleft palate repairs; using the effectiveness factor
method, this was slightly greater than half, both reflecting
the greater disability associated with cleft palate.
Values derived for the economic gain from repair of
cleft lip and palate using the Atlas methodology for GNI
ranged between 3.0 billion and 10.7 billion USD and
between 7.9 billion and 27.7 billion USD using PPP. Using
the effectiveness factor, for each person undergoing cleft
lip repair, the average economic gain, calculated without
age weighting or discounting, was 9907 USD using the
Atlas method and 26,426 USD using PPP. For cleft palate,
these gains were 17,227 USD by Atlas and 44,064 using
PPP. Using the GBD method of calculating DALYs, these
figures were 10,362 USD Atlas and 27,639 USD PPP for
cleft lip and 32,216 USD Atlas and 82,405 USD PPP for
cleft palate. These data are detailed in Table 2. Figure 5
depicts the ranges of economic impact by method per year.
The overall cost for the interventions studied was esti-
mated at 197 million USD, which represents between 0.7
and 6.6 % of the estimated economic gain, depending on
the valuation method used. In terms of cost-effectiveness,
this represents between $40 and 135/DALY.
Discussion
This study is an effort to quantify the value of a global
surgical program in economic terms. Besides providing
economic data on the impact of surgical repair of CLP, the
methodology lends itself to similar calculations in other
specialties and programs.
The current study builds on several preceding reports of
economic modeling of interventions [4,6,8,9,24].
Methodologically, measuring the economic benefit of a
health intervention requires four data points: definition of
the health problem, definition of the intervention and its
probability of success, quantitative estimates of the change
due to the intervention, and a method of converting the
health effect into economic terms [5]. All of these require
significant assumptions and are subject to multiple
methodological approaches to address the assumptions.
These lead to wide confidence intervals, but barring
extensive, expensive, and lengthy direct studies of indi-
vidual economic productivity, such assumptions are
necessary.
While the anatomic clefts are the core problems com-
prising the defects studied, there are secondary issues:
hearing problems, increased rates of infection, difficulties
eating and corresponding malnutrition, and orthodontic
problems. These are addressed through the DWs from the
GBD study and the ‘‘effectiveness factor’’ (EF) method.
World J Surg
123
In regard to the interventions, the mean age of the
patients was quite advanced, reflecting a significant back-
log [10] and delayed access to surgical care in LMICs. The
preponderance of cleft lip repairs does not reflect the
expected relative incidence of CLP, thus pointing towards a
possible tendency to repair cleft lips but not palates. This
has been observed in low-resource settings [10] and may
even reflect a hidden mortality of cleft palate infants [25].
Finally, the gender distribution is skewed with a prepon-
derance of males, a potential reflection on gender inequity
issues in many low-resource settings.
The DALY data reflect the large BoD which is
avertable through surgical intervention even in a narrow
specialty. The 1–5 million DALYs averted over 10 years
are viewed in the context of an estimated 25 million
DALYs for overall surgical BoD in Africa [26], and the
mean averted DALYs per patient are similar to those
estimated for hydrocephalus and inguinal hernia [6,27].
The economic value of improved health resulting from
the surgical interventions was determined using GNI per
capita, based on the premise that each individual theoreti-
cally contributed an equal share. A first assumption was
that CLP affects equally all sectors of society regardless of
socioeconomic stratum, a reasonable assumption based on
current epidemiologic knowledge. A second assumption
was that DALYs account for all the social, psychological,
and secondary losses associated with CLP. Alternative
econometric methodologies such as labor productivity,
Table 1 Averted burden of disease in DALYs per patient and total
0.0 3.0 3.1
Sum of averted DALYs using Eff factor
Cleft palate 1,749,252 759,125 921,451
Cleft lip 1,607,513 704,900 834,406
Total 3,356,765 1,464,025 1,755,857
Sum of averted DALYs using GBD DW treated versus untreated
Cleft palate 3,271,329 1,419,662 1,723,233
Cleft lip 1,681,327 737,268 872,721
Total 4,952,656 2,156,930 2,595,954
Average of averted DALYs per pt using Eff factor
Cleft palate 7.61 3.30 4.01
Cleft lip 5.06 2.22 2.63
Total 6.13 2.67 3.21
Average of averted DALYs per pt using GBD DW treated versus untreated
Cleft palate 14.22 6.17 7.49
Cleft lip 5.29 2.32 2.75
Total 9.04 3.94 4.74
Fig. 2 Diagram of sensitivity analysis methodologies used in
calculating the economic value of interventions
World J Surg
123
willingness to pay, value of a statistical life, or direct
income studies may be the focus of future studies.
The GNI data indicate a very substantial economic
impact, in keeping with other reports on surgery in limited
resource settings. Using a similar methodology, Alkire
et al. estimated the economic impact of treating CLP in
Sub-Saharan Africa (SSA) to USD 252–441 million, while
Warf et al. projected the economic impact of surgically
treating hydrocephalus in SSA to around 1 billion USD [6].
Cost-effectiveness analysis (CEA) is significantly lim-
ited in this context by the assumptions necessary in the
modeling, but is a natural extension of studies of economic
Table 2 Economic gain per patient and total by method of calculation
Atlas 0.0 PPP 0.0 Atlas 3.0 PPP 3.0 Atlas 3.1 PPP 3.1
Average economic gain per pt using DW untreated versus treated (USD)
CP 32,216 82,405 13,693 35,303 16,478 42,629
CL 10,362 27,639 4,391 11,843 5102 13847
Total 19,538 50,634 8297 21,693 9878 25,932
Atlas 0.0 PPP 0.0 Atlas 3.0 PPP 3.0 Atlas 3.1 PPP 3.1
Average economic gain using effectiveness factor (USD)
CP 17,227 44,064 7322 18,877 8811 22,795
CL 9907 26,426 4198 11,323 4878 13,239
Total 12,981 33,832 5510 14,495 6529 17,251
Sum of 0.0 Atlas
economic gain
Sum of 0.0 PPP
economic gain
Sum of 3.0 Atlas
econ gain
Sum of 3.0 PPP
econ gain
Sum of 3.1 Atlas
econ gain
Sum of 3.1 PPP
econ gain
Sum of economic gain using DW untreated versus treated (USD)
CP 7,409,516,181 18,952,646,713 3,149,314,664 8,119,377,845 3,789,848,882 9,804,409,158
CL 3,292,831,886 8,782,991,330 1,395,432,488 3,763,409,139 1,621,160,945 4,400,167,793
Total 10,702,348,067 27,735,638,043 4,544,747,152 11,882,786,984 5,411,009,827 14,204,576,950
Sum of 0.0 EF Atlas
econ gain
Sum of 0.0 EF PPP
econ gain
Sum of 3.0 EF Atlas
econ gain
Sum of 3.0 EF PPP
econ gain
Sum of 3.1 EF Atlas
econ gain
Sum of 3.1 EF PPP
econ gain
Sum of economic gain using effectiveness factor (USD)
CP 3,962,032,958 10,134,401,368 1,684,008,536 4,341,611,764 2,026,516,416 5,242,635,452
CL 3,148,268,535 8,397,396,589 1,334,169,598 3,598,186,299 1,549,988,025 4,206,989,694
Total 7,110,301,493 18,531,797,956 3,018,178,134 7,939,798,063 3,576,504,442 9,449,625,14
548,147 cases in database
378 cases from Palesnian Territories excluded 2° to Life Table data not available
547,769 for burden of disease analysis
4396 cases from Myanmar and Somalia 2° to no World Bank economic data for these years
543,373 cases for economic study
No PPP data for Argenna, and Atlas data only through 2006, so Argenna cases aer 2006 used 2006
GNI/cap data; Argenna cases excluded from PPP analysis.
Djibou data only available through 2005, so Djibou cases aer 2005 used 2005 GNI/cap data
543,373 cases for study
38% female 62% male
58% cle lip 42% cle palate
average age 5.56 average age 6.80
Fig. 3 Cases available for
study and Patient characteristics
World J Surg
123
impact. A frequent metric of CEA is the $/DALY. The
current estimate of $40–135/DALY compares favorably
with a previous report from Smile Train of $70–134/DALY
for CLP repairs [10] and with other CLP CEA studies
ranging between USD 29–285/DALY [4,28,29].
Limitations
There are several limitations to the current study, some of
which have been alluded to above. First, the sample is not
random—the locale of the interventions, both nationally
and regionally, reflects strategic and logistic choices within
one large non-governmental organization. Absent data for
some countries compound this effect, though it affects a
very small proportion of the entire dataset.
DALY calculations rely on DWs which are notoriously
difficult to estimate accurately [30] and on subjective
effectiveness factors. In the face of multiple competing
strategies such as age weighting and future discounting, the
authors’ only recourse was to offer ranges rather than
precise values. One specific additional limitation of our
study was the assumption that mortality from CLP is
negligible, which in some low-resource settings may not be
true [25]. Higashi et al. found that in addition to a small
mortality attributable to the cleft deformity, there also was
a higher all-cause mortality in unrepaired than repaired
clefts throughout life [31]. Any mortality associated with
cleft deformities would serve to increase our estimates of
economic benefit to repair. The uncertainty is also exac-
erbated by the econometric estimates, which include two
alternative methodologies (Atlas and PPP) yielding dis-
parate results.
This study does not purport to evaluate complications,
quality of care, externalities, or any other measures of the
specific procedures performed. It also does not specifically
account for the costs of the procedures; our cost-effec-
tiveness analysis relied on the average reimbursement rates
paid by the organization for each intervention.
There are other reasons for believing that our economic
estimates, while impressively high, are still underestimates.
It is well-recognized that school attendance, cognitive
development, and social development during childhood
have significant effects on adult productivity [32], and this
is not included in the DALY estimates. The Smile Train
work also includes a number of secondary procedures
which increase the economic impact of this surgical
program.
Conclusions
The current study documents the tremendous economic
value of a scaled surgical program. Whether the actual
contribution to the global economy is closer to 3 billion or
30 billion USD, it highlights the significant economic and
public health value of vertical surgical interventions like
CLP repair in low-resource settings. For a donor cost under
200 million USD, a 15- to 150-fold increase was added to
the economies of the 83 beneficiary countries. Moreover,
the clinical effect of these surgical interventions is a per-
manent one, not requiring ongoing therapy or expense to
maintain or renew the gain. Congenital anomalies such as
CLP are the ultimate ‘‘poverty trap’’ as described by
Banerjee and Duflo [33], as without resolution of these
issues, there is little opportunity for the individual who
otherwise has the same potential as any other person to
break out of her/his economic state.
While the current study is limited to a small area of
surgical care, similar studies can be undertaken in other
specialties. Such studies would add to the growing body of
evidence supporting the value and cost-effectiveness of
-
1,00,000
2,00,000
3,00,000
4,00,000
5,00,000
6,00,000
7,00,000
8,00,000
9,00,000
2001 2002 2003 2004 2005 2006 2007 2008 2009 2010 2011
DALYs
GBD DALYs 0,0
GBD DALYs 3,0
GBD DALYs 3,1
EF DALYs 0,0
EF DALYs 3,0
EF DALYs 3,1
Fig. 4 Averted BoD through cleft lip and palate surgery by method
and year
-
1,00,00,00,000
2,00,00,00,000
3,00,00,00,000
4,00,00,00,000
5,00,00,00,000
6,00,00,00,000
7,00,00,00,000
2001 2002 2003 2004 2005 2006 2007 2008 2009 2010 2011
GBD 0,0 Atlas
GBD 0,0 PPP
EF 0,0 Atlas
EF 0,0 PPP
Fig. 5 Economic impact by year and calculation method without
age weighting or discounting
World J Surg
123
surgical care as a primary health intervention globally. This
should inform advocacy efforts for resource allocation in
the funding of health care globally.
Acknowledgments The authors express their appreciation to
MacKinnon Engen of The Smile Train for making the database
available and facilitating our analysis.
References
1. Lopez AD, Mathers CD, Ezzati M, Jamison DT, Murray CJL
(2006) Global burden of disease and risk factors. Oxford
University Press and The World Bank
2. Murray CJL, Salomon JA, Mathers C (2000) A critical exami-
nation of summary measures of population health. Bull WHO
78(8):981–994
3. Gold MR, Stevenson D, Fryback DG (2002) HALYS AND
QALYS AND DALYS, OHMY: similarities and differences in
summary measures of population. HealthAnnu Rev Public Health
23:115–134
4. Corlew DS (2010) Estimation of impact of surgical disease
through economic modeling of cleft lip and palate care. World J
Surg 34(3):391–396
5. Corlew D (2013) Economic modeling of surgical disease: a
measure of public health interventions. World J Surg
37:1478–1485. doi:10.1007/s00268-012-1796-5
6. Warf BC, Alkire BC, Bhai S, Hughes C, Schiff SJ, Vincent JR,
Meara JG (2011) Costs and benefits of neurosurgical intervention
for infant hydrocephalus in sub-Saharan Africa. J Neurosurg
Pediatr 8(5):509–521
7. Tan-Torres Edejer T, Baltussen R, Adam T, Hutubessy R,
Acharya A, Evans DB, Murray CJL, eds (2003). WHO Guide to
cost—effectiveness analysis. Geneva, pp 1–239
8. Alkire B, Hughes CD, Nash K, Vincent JR, Meara JG (2011)
Potential economic benefit of cleft lip and palate repair in sub-
Saharan Africa. World J Surg 35(6):1194–1201. doi:10.1007/
s00268-011-1055-1
9. Alkire BC, Vincent JR, Burns CT, Metzler IS, Farmer PE, Meara
JG (2012) Obstructed labor and caesarean delivery: the cost and
benefit of surgical intervention. PLoS ONE 7(4):e34595
10. Poenaru D (2013) Getting the Job Done: analysis of the impact
and effectiveness of the smiletrain program in alleviating the
global burden of cleft disease. World J Surg 37(7):1562–1570.
doi:10.1007/s00268-012-1876-6
11. McCord C, Chowdhury Q (2003) A cost effective small hospital
in Bangladesh: what it can mean for emergency obstetric care. Int
J Gynaecol Obstet 81(1):83–92
12. Smile Train: leading children’s charity. www.smiletrain.org.
Accessed March 31, 2014
13. Shrime MG, Sleemi A, Ravilla TD (2015) Charitable platforms in
global surgery: a systematic review of their effectiveness, cost-
effectiveness, sustainability, and role training. World J Surg
39:10–20. doi:10.1007/s00268-014-2516-0
14. Murray CJL, Lopez A (1996) A comprehensive assessment of
mortality and disability from disease, injures and risk factors in
1990 and projected to 2020. In The global burden of disease.
Harvard University Press, Cambridge, pp 1–51
15. Bickler S, Ozgediz D, Gosselin R, Weiser T, Spiegel D, Hsia R,
Dunbar P, McQueen K, Jamison D (2010) Key concepts for
estimating the burden of surgical conditions and the unmet need
for surgical care. World J Surg 34(3):374–380. doi:10.1007/
s00268-009-0261-6
16. Lopez A (2006) Global burden of disease and risk factors. (Lopez
AD, Mathers CD, Ezzati M, Jamison DT, Murray CJL, Eds.
Oxford University Press & The World Bank, Library. New York
17. World Health Organization (2015) Standard Life Tables.
Who.int/healthinfo/bodreferencesstandardlifetable.xls. Accessed
Jan 21
18. Shillcutt SD, Clarke MG, Kingsnorth AN (2010) Cost-effec-
tiveness of groin hernia surgery in the Western Region of Ghana.
Arch Surg (Chicago, IL: 1960) 145(10):954–961
19. Gosselin RA, Maldonado A, Elder G (2010) Comparative cost-
effectiveness analysis of two MSF surgical trauma centers. World
J Surg 34(3):415–419. doi:10.1007/s00268-009-0230-0
20. Gosselin RA, Thind A, Bellardinelli A (2006) Cost/DALY
averted in a small hospital in Sierra Leone: what is the relative
contribution of different services? World J Surg 30(4):505–511.
doi:10.1007/s00268-005-0609-5
21. Wu VK, Poenaru D (2013) Burden of surgically correctable dis-
abilities among children in the Dadaab Refugee Camp. World J
Surg 37(7):1536–1543. doi:10.1007/s00268-012-1899-z
22. Murray CJL, Lopez AD, Jamison DT (1994) The global burden
of disease in 1990: summary results, sensitivity analysis and
future direction. Bull WHO 72(3):495–509
23. World Bank website: http://databank.worldbank.org/data/views/
reports/tableview.aspx. Accessed Jan 21, 2015
24. Hughes CD et al (2012) The Clinical and economic impact of a
sustained program in global plastic surgery: valuing cleft care in
resource-poor settings. PRS. doi:10.1097/PRS.0b013e3182
54b2a2
25. Wilson J, Hodges A (2011) Cleft lip and palate surgery carried
out by one team in Uganda: where have all the palates gone?
Cleft Palate Craniofac J 49(3):1–7
26. Ozgediz D, Riviello R (2008) The ‘‘other’’ neglected diseases in
global public health: surgical conditions in sub-Saharan Africa.
PLoS Med 5(6):e121
27. Shillcutt SD, Sanders DL, Teresa Butro
´n-Vila M, Kingsnorth AN
(2013) Cost-effectiveness of inguinal hernia surgery in north-
western Ecuador. World J Surg 37(1):32–41. doi:10.1007/
s00268-012-1808-5
28. Alkire B, Vincent J, Meara J (2014) Benefit-cost analysis of a
cleft lip and palate surgical subspecialty hospital in India. In
DCP3 draft, pp 1–17
29. Moon W, Perry H, Baek R-M (2012) Is international volunteer
surgery for cleft lip and cleft palate a cost-effective and justifiable
intervention? A case study from east Asia. World J Surg
36:2819–2830. doi:10.1007/s00268-012-1761-3
30. Gosselin R, Ozgediz D, Poenaru D (2013) A square peg in a
round hole? challenges with DALY-based ‘‘burden of disease’
calculations in surgery and a call for alternative metrics. World J
Surg 37(11):2507–2511. doi:10.1007/s00268-013-2182-7
31. Higashi H, Barendregt JJ, Kassebaum NJ, Weiser TG, Bickler
SW, Vos T (2015) The burden of selected congenital anomalies
amenable to surgery in low and middle-income regions: cleft lip
and palate, congenital heart anomalies and neural tube defects.
Arch Dis Child 100:233–238
32. Bloom DE and Canning D (2008), Population health and eco-
nomic growth, Working paper 24, Commission on Growth and
Development, The World Bank
33. Banerjee AV, Duflo E (2012) Poor economics: a radical
rethinking of the way to fight global poverty, Public Affairs
World J Surg
123
... As part of their quality standards, Smile Train requests partners to collect and share data [41]. Using the data for outcomes evaluations, cost-effectiveness analysis, and other scientific publications contributes to the local health systems and supports the need for further expansion of surgical programs [43][44][45][46][47][48][49]. ...
... As a testament to the attention cleft organizations have drawn to the global need for investing in surgical care, one quarter of the resources for surgery raised by NGOs comes through cleft organizations [53,54]. Just as importantly, these funds have been spent in cost-effective ways, making the case for investing in surgery [43,[47][48][49]55] (Fig. 42.4). ...
... BCG vaccine for tuberculosis prevention48 Vaccines for tuberculosis, disptheria, pertussis, tetanus, polio, and measles48 Bednets for malaria prevention48 Aspirin and β blocker for ichaemic heart disease 48 ...
Chapter
The provision of cleft care in low-resource settings has seen massive improvements and innovation in the last half century as providers attempt to access all children while providing high-quality care. In attempts to understand the challenges to patients and practitioners, it is evident that the parts of the world with the greatest barriers to receiving and providing cleft care are also the areas with the weakest surgical health systems (Fig. 42.1) [1]. In order to provide sustainable solutions that secure safe, timely, and affordable care for all cleft patients, the surgical systems of low- and middle-income countries (LMICs) must be concomitantly strengthened. Just as cleft surgery has taken dramatic steps forward, so too has “global surgery” which seeks to improve health outcomes and equity for all people in need of surgical care [2]. By aligning the goals of cleft providers with the global surgery efforts, surgical systems can be strengthened to provide a foundation on which comprehensive cleft care can thrive.
... The occurrence of a cleft does not result in an anatomic problem alone. Secondary issues such as speech and hearing problems, increased susceptibility to dental caries, difficulty eating-with associated malnutrition, and orthodontic problems also occur (13,14). The handicaps associated with CL/P are medical and social. ...
... Cleft lip and palate (CLP) cause significant disability, economic hardship, and psychosocial stress worldwide. [1][2][3][4] Disease incidence varies between 3.4 and 22.9 per 10,000 births, and low-and middle-income countries (LMICs) register the majority of CLP cases. 5 Paradoxically, LMICs lack the human resources, infrastructure, health policies, funding, and information needed to meet local CLP treatment needs. ...
Article
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Cleft lip and palate (CLP) comprise over 90% of the world's congenital anomalies and cause significant disability worldwide, while disproportionally burdening low- and middle-income countries (LMICs). Research can help inform strategies that reduce disparities in accessing CLP care. We performed a scientometric analysis of CLP research in LMICs to identify influential contributors and themes. Methods: The authors searched seven citation databases accessed via Web of Science, from inception to March 2, 2021. Social network analysis was done using VOSviewer. The Kruskal-Wallis test and linear regression were used. Results: In total, 1561 articles authored by 6414 researchers affiliated with 2113 organizations in 119 countries were included. Most authors (n = 6387, 99.6%) had published two or more articles. The USA (454 articles), Brazil (211 articles), China (175 articles), and India (127 articles) published the most. The most prolific institutions were the University of Sao Paulo (94 articles), the University of Pittsburgh (57 articles), and the University of Iowa (55 articles). Marazita ML (33 articles), Shi B (27 articles), and Murray JC (22 articles) had the highest number of publications. An estimated 510 articles (32.7%) were focused on epidemiology, 240 (15.4%) on management, and 54 (3.5%) on global plastic surgery for CLP. Conclusions: LMICs are disproportionally burdened by CLP, but research is limited and often produced by high-income countries. This study elucidates partnership and health system strengthening opportunities to improve LMIC research capacity and ultimately informs the management and outcomes for patients with CLP.
... The occurrence of a cleft does not result in an anatomic problem alone. Secondary issues such as speech and hearing problems, increased susceptibility to dental caries, difficulty eating-with associated malnutrition, and orthodontic problems also occur (13,14). The handicaps associated with CL/P are medical and social. ...
Article
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Objective: This scoping review aims to identify the various areas and current status of the application of artificial intelligence (AI) for aiding individuals with cleft lip and/or palate. Introduction: Cleft lip and/or palate contributes significantly toward the global burden on the healthcare system. Artificial intelligence is a technology that can help individuals with cleft lip and/or palate, especially those in areas with limited access to receive adequate care. Inclusion Criteria: Studies that used artificial intelligence to aid the diagnosis, treatment, or its planning in individuals with cleft lip and/or palate were included. Methodology: A search of the Pubmed, Embase, and IEEE Xplore databases was conducted using search terms artificial intelligence and cleft lip and/or palate. Gray literature was searched using Google Scholar. The study was conducted according to the PRISMA- ScR guidelines. Results: The initial search identified 458 results, which were screened based on title and abstracts. After the screening, removal of duplicates, and a full-text reading of selected articles, 26 publications were included. They explored the use of AI in cleft lip and/or palate to aid in decisions regarding diagnosis, treatment, especially speech therapy, and prediction. Conclusion: There is active interest and immense potential for the use of artificial intelligence in cleft lip and/or palate. Most studies currently focus on speech in cleft palate. Multi-center studies that include different populations, with collaboration amongst academicians and researchers, can further develop the technology.
Article
Objective The objective of this paper is to conduct a systematic review that summarizes the cost-effectiveness of cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) based on existing literature. Design We searched eleven electronic databases for articles from January 1, 2000 to December 29, 2020. This study is registered in PROSPERO (CRD42020148402). Two reviewers independently conducted primary and secondary screening, and data extraction. Setting All CL/P cost-effectiveness analyses in LMIC settings. Patients, Participants In total, 2883 citations were screened. Eleven articles encompassing 1,001,675 patients from 86 LMICs were included. Main Outcome Measures We used cost-effectiveness thresholds of 1% to 51% of a country's gross domestic product per capita (GDP/capita), a conservative threshold recommended for LMICs. Quality appraisal was conducted using the Joanna Briggs Institute (JBI) checklist. Results Primary CL/P repair was cost-effective at the threshold of 51% of a country's GDP/capita across all studies. However, only 1 study met at least 70% of the JBI criteria. There is a need for context-specific cost and health outcome data for primary CL/P repair, complications, and existing multidisciplinary management in LMICs. Conclusions Existing economic evaluations suggest primary CL/P repair is cost-effective, however context-specific local data will make future cost-effectiveness analyses more relevant to local decision-makers and lead to better-informed resource allocation decisions in LMICs.
Objective To evaluate the surgical rehabilitation of cleft lip and/or palate by the Brazilian public health system. Methods Retrospective, analytical and comparative ecological study, with information on hospital procedures performed on individuals with cleft lip and/or palate in Centers authorized by the Brazilian public health system, between the years 2008 and 2020. The information was collected in databases Ministry of Health data. Results Between 2008 and 2020, there was an increase of 8 (36.4%) qualified Centers in Brazil, currently having 30 Centers in 100% of the geographic regions. The surgical procedures performed totaled 68,716; with multiple surgeries being the most frequent. Complete cleft lip and palate was the most frequent type in hospital admissions. The public financial resources invested in the surgical rehabilitation of cleft lip and palate in the qualified Lip and Palate Malformation Treatment Centers were US$ 39,693 million, making an average value per procedure of US$ 577.64. Conclusions In Brazil, public health system performed and financed, over the years 2008 and 2020, an important volume of surgical procedures for cleft lip and/or palate, which presented a polarization in the Southeast region but with a slight tendency expansion to other regions of the country. The most performed surgical procedures were multiple surgeries and mostly for individuals with cleft lip and palate. The amounts paid showed a heretogeneous distribution in the national territory. Level of evidence Level 5: Report containing program evaluation data.
Article
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Introduction: There is a significant unmet need for cleft repair in low-income countries. The procedure is challenging due to limited access, small fragile flaps, and the depth at which sutures need to be placed. The aim of this analysis is to review available literature pertaining to cleft lip and palate repair training, with a focus on those applicable to low-income countries. Methodology: Literature searches of Medline and EMBase were performed to identify studies of cleft lip and/or palate training. Terms including "cleft," "lip," "palate," and "training" were searched. Literature published in English from inception to May 2021 in which the full text was available was considered for inclusion. Studies were included on the basis that they included interventions such as virtual/online training, simulation training, courses, fellowships, and/or hospital-based training. Studies that met this criterion were further assessed using a validated scoring tool then the applicability of each training strategy for low-income countries reviewed. Results: Nineteen studies were included in this analysis. Of these 4 studies pertained to online training, 11 studies illustrated cleft models and simulation training, while 4 studies highlighted the role of charity led training achieved through direct hospital teaching and fellowship programs. The training strategies described have been summarised and presented herein in a narrative format. The simplicity, fidelity, and training benefit of palatoplasty models has also been described. Conclusions: Cleft lip and palate repair training should be accessible to surgeons in low-income countries. Direct practical experience has an important role in training to perform cleft procedures. Affordable, simple simulation models that resemble human tissue are essential so that surgeons can safely practice skills between initial training and operating on a living child.
Article
Objective: There are many adults with cleft lip deformities in developing countries. This is due to the lack of public awareness, social stigma, distance from the health center, and parents' financial condition. Lip repair under local anesthesia is safe, cost-effective and would be beneficial for the underprivileged population. Design: A retrospective cohort study with follow-up of 1 to 8 years. Setting: Academic Hospital. Patients/participants: Cleft lip repair was performed in 252 patients of age more than 12 years from 2012 to 2019. Patients with cleft palate, cardiopulmonary disease, who did not consent for the procedure while awake were excluded. Interventions: Cleft lip surgery done under local anesthesia. Main outcome measures: Outcome measures were patients' self-satisfaction and comments of peer. Results: Two hundred fifty-two primary cleft lip operations were done in 168 male and 84 female patients. The mean age was 23.62 years, and the mean weight was 49.66 kg. Unilateral was 227, Bilateral cleft lip 25. The postoperative period was uneventful. No case of wound dehiscence or wound infection was observed. Patients were discharged on the same day, except the ones who traveled a long distance. Conclusion: Cleft lip repair in adults under local anesthesia is safe and cost-effective.
Chapter
Visiting educator trips and tele-mentoring increase surgical capacity in low- and middle-income countries.
Article
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Objective To quantify the burden of selected congenital anomalies in low and middle-income countries (LMICs) that could be reduced should surgical programmes cover the entire population with access to quality care. Design Burden of disease and epidemiological modelling. Setting LMICs from all global regions. Population All prevalent cases of selected congenital anomalies at birth in 2010. Main outcome measures Disability-adjusted life years (DALYs). Interventions and methods Surgical programmes for three congenital conditions were analysed: clefts (lip and palate); congenital heart anomalies; and neural tube defects. Data from the Global Burden of Disease 2010 Study were used to estimate the combination of fatal burden that could be addressed by surgical care and the additional long-term non-fatal burden associated with increased survival. Results Of the estimated 21.6 million DALYs caused by these three conditions in LMICs, 12.4 million DALYs (57%) are potentially addressable by surgical care among the population born with such conditions. Neural tube defects have the largest potential with 76% of burden amenable by surgery, followed by clefts (59%) and congenital heart anomalies (49%). Sub-Saharan Africa and South Asia have the greatest proportion of surgically addressable burden for clefts (68%), North Africa and Middle East for congenital heart anomalies (73%), and South Asia for neural tube defects (81%). Conclusions There is an important and neglected role surgical programmes can play in reducing the burden of congenital anomalies in LMICs.
Article
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This study was designed to propose a classification scheme for platforms of surgical delivery in low- and middle-income countries (LMICs) and to review the literature documenting their effectiveness, cost-effectiveness, sustainability, and role in training. Approximately 28 % of the global burden of disease is surgical. In LMICs, much of this burden is borne by a rapidly growing international charitable sector, in fragmented platforms ranging from short-term trips to specialized hospitals. Systematic reviews of these platforms, across regions and across disease conditions, have not been performed. A systematic review of MEDLINE and EMBASE databases was performed from 1960 to 2013. Inclusion and exclusion criteria were defined a priori. Bibliographies of retrieved studies were searched by hand. Of the 8,854 publications retrieved, 104 were included. Surgery by international charitable organizations is delivered under two, specialized hospitals and temporary platforms. Among the latter, short-term surgical missions were the most common and appeared beneficial when no other option was available. Compared to other platforms, however, worse results and a lack of cost-effectiveness curtailed their role. Self-contained temporary platforms that did not rely on local infrastructure showed promise, based on very few studies. Specialized hospitals provided effective treatment and appeared sustainable; cost-effectiveness evidence was limited. Because the charitable sector delivers surgery in vastly divergent ways, systematic review of these platforms has been difficult. This paper provides a framework from which to study these platforms for surgery in LMICs. Given the available evidence, self-contained temporary platforms and specialized surgical centers appear to provide more effective and cost-effective care than short-term surgical mission trips, except when no other delivery platform exists.
Article
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In recent years, surgical providers and advocates have engaged in a growing effort to establish metrics to estimate capacity for surgical services as well the burden of surgical diseases in resource-limited settings. The burden of disease (BoD) studies have established the disability-adjusted life year (DALY) as the primary metric to measure both disability and premature mortality. Nonetheless, DALY-based approaches present methodological challenges, some of which are unique to surgical conditions, not fully addressed through the multiple iterations of the BoD studies, including the most recent study. This paper examines these challenges in detail, including issues around age-weighting and discounting, and estimates of disability-weights for specific conditions. Surgical burden measurements of specific conditions, or through the assessment of hospital wards as platforms for service delivery, still have unresolved methodological hurdles. The 2010 BoD study addresses some of these issues, but many questions still remain. Other methods estimating surgical prevalence, backlogs in treatment, and disability incurred by delays in care may provide more practical approaches to disease burden that can be useful tools for clinicians and health advocates. These approaches warrant further exploration in LMICs and these debates require active engagement by surgical providers and advocates globally.
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Background: Surgery is increasingly recognized as a means to reduce the morbidity and mortality of disabling impairments in resource-limited environments. We sought to estimate the burden of surgically correctable disabling impairments and the cost-effectiveness of their treatment among children in a large refugee camp. Methods: This is a chart review of all patients aged 0-18 years from Dadaab Refugee Camp (Kenya) treated at a facility primarily responsible for providing pediatric surgical care in the region. Total disability-adjusted life years (DALYs) averted were calculated using life expectancy tables and established or estimated disability weights. A sensitivity analysis was performed using various life expectancy tables. Delayed averted DALYs caused by delay in care were also estimated. Inpatient costs were collected to perform a cost-effectiveness analysis. Results: Between 2005 and 2011 a total of 640 procedures were performed on 341 patients. The median age at surgery was 4.6 years, and 33 % of the children treated were female. Only 13.5 % of surgeries estimated as required for common congenital surgical conditions were actually performed. The total number of DALYs averted ranged from 4,136 to 9,529 (6.4-14.8 per patient), depending on the calculation method used. Cost-effectiveness analysis resulted in values of $40-$88 per DALY. Conclusions: The burden of pediatric surgical disabling impairments in refugee camps is substantial. Surgical intervention to address this burden is both feasible and cost-effective. Such intervention can significantly decrease the burden of disability among children affected by armed conflicts.
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Background: The study measured the success of SmileTrain, the largest cleft charity globally, in alleviating the global burden of disease (GBD). It was done by estimating averted disability-adjusted life years (DALYs) and delayed averted DALYs because of the global backlog in cleft procedures. Methods: Anonymized data for all procedures in the SmileTrain global database were analyzed by age, sex, country, region, and surgery type. DALYs averted were calculated using life expectancy tables and established and estimated disability weights. The cost-effectiveness analysis used mean SmileTrain procedural disbursement figures. Sensitivity analysis was performed using various cleft incidence rates, life expectancy tables, and disability weights. Results: During 2003-2010 a total of 536,846 operations were performed on 364,467 patients-86 % in Southeast Asia and the western Pacific region. Procedure numbers increased yearly. Mean age at primary surgery-6.2 years (9.8 years in Africa)-remained fairly constant over time in each region. Globally, 2.1-4.7 million DALYs were averted through the operations at a total estimated cost of US$196 M. Mean DALYs per patient were 3.8-9.0, and mean cost per DALY was $72-$134. Total delayed GBD due to advanced age at surgery was 191,000-457,000 DALYs. Conclusions: Despite an unparalleled number of surgeries performed and yearly increase by one charity, the unmet and delayed averted cleft GBD remains significant in all regions. Large geographic disparities reflect varied challenges regarding access to surgery. Cleft surgeries are cost-effective interventions to reduce the global burden of disease (GBD). Future challenges include increased collaboration among cleft care providers and a focus on remote global areas by building infrastructure and local training.
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Background: Cost-effectiveness of tension-free inguinal hernia repair at a private 20-bed rural hospital in Esmeraldas Province, Ecuador, was calculated relative to no treatment. Methods: Lichtenstein repair using mosquito net or polypropylene commercial mesh was provided to patients with inguinal hernia by surgeons from Europe and North America. Prospective data were collected from provider, patient, and societal perspectives, with component costs collected on site and from local supply companies or published literature. Patient outcomes were forecasted using disability adjusted life years (DALYs) averted. Uncertainty in patient-level data was evaluated with Monte-Carlo simulation. Results: Surgery was provided to 102 patients with inguinal hernias of various sizes. Local anesthesia was used for 80 % of operations during the first mission, and spinal anesthesia was used for 89 % in the second mission. Few complications were observed. An average 6.39 DALYs (3,0) were averted per patient (95 % confidence interval: 6.22-6.84). The average cost per patient was US$499.33 (95 % CI: US$490.19-$526.03) from a provider perspective, US$118.79 (95 % CI: US$110.28-$143.72) from a patient perspective, and US$615.46 (95 % CI: US$603.39-$650.40) from a societal perspective. Mean cost-effectiveness from a provider perspective was US$78.18/DALY averted (95 % CI: US$75.86-$85.78) according to DALYs (3,0) averted using the West Life Table level 26, well below the Ecuadorian per-capita Gross National Income (US$3,850). Results were robust to all sensitivity analyses. Conclusions: Inguinal hernia repair was cost-effective in western Ecuador through international collaboration.
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"Billions of government dollars, and thousands of charitable organizations and NGOs, are dedicated to helping the world's poor. But much of the work they do is based on assumptions that are untested generalizations at best, flat out harmful misperceptions at worst. Banerjee and Duflo have pioneered the use of randomized control trials in development economics. Work based on these principles, supervised by the Poverty Action Lab at MIT, is being carried out in dozens of countries. Their work transforms certain presumptions: that microfinance is a cure-all, that schooling equals learning, that poverty at the level of 99 cents a day is just a more extreme version of the experience any of us have when our income falls uncomfortably low. Throughout, the authors emphasize that life for the poor is simply not like life for everyone else: it is a much more perilous adventure, denied many of the cushions and advantages that are routinely provided to the more affluent"--
Article
The measurement of the burden of disease and the interventions that address that burden can be done in various units. Reducing these measures to the common denominator of economic units (i.e., currency) enables comparison with other health entities, interventions, and even other fields. Economic assessment is complex, however, because of the multifactorial components of what constitutes health and what constitutes health interventions, as well as the coupling of those data to economic means. To perform economic modeling in a meaningful manner, it is necessary to: (1) define the health problem to be addressed; (2) define the intervention to be assessed; (3) define a measure of the effect of the health entity with and without the intervention (which includes defining the counterfactual); and (4) determine the appropriate method of converting the health effect to economics. This paper discusses technical aspects of how economic modeling can be done both of disease entities and of interventions. Two examples of economic modeling applied to surgical problems are then given.