Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study

YJPSU-59487; No of Pages 20 Journal of Pediatric Surgery xxx (xxxx) xxx

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Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study☆ Doulia Hamad, Yasmine Yousef, Natasha G. Caminsky, Elena Guadagno, Viet Anh Tran, Jean-Martin Laberge, Sherif Emil, Dan Poenaru ⁎ Division of Pediatric General and Thoracic Surgery, The Montreal Children's Hospital, McGill University Health Centre, Montreal, Quebec, Canada

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Article history: Received 12 June 2018 Received in revised form 2 November 2019 Accepted 5 November 2019 Available online xxxx Key words: Global surgery Pediatric surgery Mortality Pediatric surgical workforce Congenital disease Low- and middle-income countries

a b s t r a c t Purpose: Low- and middle-income countries (LMICs) have only 19% of the global surgical workforce yet see 80% of worldwide deaths from noncommunicable diseases. We aimed to interrogate the correlation between pediatric surgical workforce density (PSWD) and survival from pediatric surgical conditions worldwide. Methods: A systematic review of online databases identified outcome studies for key pediatric surgical conditions (gastroschisis, esophageal atresia, intestinal atresia, and typhoid perforation) as well as PSWD data across lowincome (LICs), middle-income (MICs), and high-income countries (HICs). PSWD was expressed as the number of PSs/million children under 15 years of age and we correlated this to surgical outcomes for our case series. Results: PSWD ranged between zero (Burundi, The Gambia, and Mauritania) and 125.2 (Poland) across 86 countries. Outcomes for at least one condition were obtained in 61 countries: 50 outcomes in HICs, 52 in MICs and 8 in LICs. The mean survival in our case series was 42.3%, 69.4% and 91.6% for LICs, MICs, and HICs, respectively. A PSWD ≥4 PSs/million children under 15 years of age significantly correlated to odds of survival ≥80% (OR 16.8, p b 0.0001, 95% CI 5.66–49.88). Specifically in the studied LICs and MICs, increasing the PSWD to 4 would require training 1427 additional surgeons. Conclusion: Using a novel approach, we have established a benchmark for the scale-up of pediatric surgical workforce, which may support broader efforts to reduce childhood deaths from congenital disease. Levels of evidence: 2c – Outcomes Research. © 2019 Elsevier Inc. All rights reserved.

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Material and methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.1. Systematic review . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.2. Statistical analysis . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.2.1. Overview . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.2.2. Calculating survival . . . . . . . . . . . . . . . . . . . . . . 1.2.3. Calculating PSWD . . . . . . . . . . . . . . . . . . . . . . . 1.2.4. Plotting survival as a function of PSWD . . . . . . . . . . . . . 1.2.5. Establishing the critical PSWD required for case series survival ≥80% 1.2.6. Establishing the PSWD scale-up required among countries studied . Results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1. Systematic reviews. . . . . . . . . . . . . . . . . . . . . . . . . . . 2.2. Critical PSWD and survival . . . . . . . . . . . . . . . . . . . . . . . Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Conclusions . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

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Abbreviations: COSECSA, College of Surgeons of East, Central and Southern Africa; DALYs, Disability-adjusted life years; HDI, Human Development Index; HICs, High-income countries; LCoGS, Lancet Commission on Global Surgery; LICs, Low-income countries; LMICs, Low-and middle-income countries; MICs, Middle-income countries; PSs, Pediatric surgeons; PSWD, Pediatric surgical workforce density; SAO, Specialist surgeons, anesthesiologists and obstetricians. ☆ Presented at the 49th annual meeting of the Canadian Association of Pediatric Surgeons, Banff, CANADA, October 5–October 7, 2017. ⁎ Corresponding author at: Division of Pediatric General and Thoracic Surgery, The Montreal Children's Hospital, 1001 Decarie Blvd, Room B04.2028, Montreal, Quebec H4A 3J1, Canada. Tel.:+1 514 412 4497; fax: +1 514 412 4289. E-mail address: [email protected] (D. Poenaru). https://doi.org/10.1016/j.jpedsurg.2019.11.001 0022-3468/© 2019 Elsevier Inc. All rights reserved.

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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Acknowledgements . . . . . . . . . . . . . . . . . . . . . . . Appendix A. Search methodology – surgical mortality . . . . . . Appendix B. Search methodology – surgical workforce . . . . . Appendix C. Retained publications – pediatric surgeon workforce. Appendix D. Retained publications – pediatric survival . . . . . Appendix E. Nonlinear regression using PROC NLIN . . . . . . . References . . . . . . . . . . . . . . . . . . . . . . . . . . .

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There is a wide disparity between surgical need and access to safe and appropriate surgical care in low- and middle-income countries (LMICs) [1, 2]. This gap is most evident in the surgical care of children in LMICs, where children constitute upwards of 50% of the population [3]. Recent work by the Lancet Commission on Global Surgery (LCoGS) has shown a positive correlation between a greater density of specialist surgeons, anesthesiologists and obstetricians (SAO) per capita and increased maternal survival, encouraging the definition of clear benchmarks and targets for workforce scale-up in a number of countries [4]. While an overall deficit in healthcare provider workforce is therefore evident in LMICs, the relationship between pediatric surgical workforce deficit and childhood mortality remains undefined. Though childhood deaths from infectious diseases have decreased in the last decade, neonatal mortality has proportionally increased, most prominently in LMICs [5]. Due to multiple factors, LMICs are disproportionately burdened with congenital diseases compared to high-income countries (HICs), suffering from both a higher incidence and a reduced capacity to respond to them [6]. With 8% of disability-adjusted life years (DALYs) worldwide caused by neonatal disorders and 13% of all surgical DALYs contributed by congenital anomalies and perinatal conditions, there is significant incentive to identify deficiencies in the pediatric surgical workforce in LMICs [7]. To address the current burden of pediatric surgical need in LMICs, earlier literature suggested that over 7500 pediatric surgeons (PSs) are needed in Africa and Asia alone [8], or approximately 1 PS/200,000 children under 15 years of age [9]. However, these estimates were established using a theoretical projection of the need for pediatric surgical workers based on current workforce numbers in HICs. This study aims to identify, using actual reported outcome and workforce data, the critical pediatric surgical workforce density (PSWD) associated with a ≥ 80% survival rate for several key surgically correctable congenital malformations and acquired pathologies.

1. Material and methods 1.1. Systematic review Five index pediatric surgical conditions were initially examined in this study: gastroschisis, esophageal atresia, intestinal atresia, typhoid intestinal perforation, and the category of neonatal bowel obstruction. These conditions were agreed upon via expert consensus based on known burden of disease in LMICs as well as frequent poor survival reported in the literature [6, 7, 10, 11]. In order to identify the mortality of our five conditions of interest, the following nine databases were searched from 2000 to May 12, 2017: Medline (Ovid), Embase (Ovid), Cochrane (Wiley), PubMed (NLM), Africa-Wide (Ebsco), Biosis (Ovid), Global Health (Ovid), LILACs (Latin-American and Caribbean Center on Health Sciences Information) and Web of Science (Thomson Reuters), with no language restrictions. The search strategy used text words found in the title, abstract or keyword fields, and relevant subject indexing to retrieve articles pertaining to mortality or survival rates in the neonatal population for gastroschisis, esophageal atresia, bowel obstruction or intestinal atresia

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as well as typhoid intestinal perforation in the pediatric population, with modifications to search terms as necessary. We included any study reporting survival to 30 days postoperatively or until hospital discharge. Of note, a large proportion of studies reported compound outcomes over several years gathered through retrospective chart reviews. Neonatal bowel obstruction was subsequently excluded from further analysis given the small number of identified studies as well as the significant heterogeneity of the conditions described under this category. Subsequent analyses were therefore carried out with only four conditions of interest. In order to identify the pediatric surgical workforce worldwide, the following databases were searched from January 1, 2000 until April 28, 2017: Medline (Ovid), Embase (Ovid), PubMed (NLM), Africa-Wide (Ebsco), LILACs (Latin-American and Caribbean Center on Health Sciences Information) and Web of Science (Thomson Reuters), with no language restrictions. The WHO Emergency and Essential Surgical Care (EESC) Global database was also interrogated. The search strategy used text words found in the title, abstract or keyword fields, and relevant subject indexing to retrieve articles pertaining to the pediatric surgical workforce/manpower, with modifications to search terms as necessary. Surgical specialties within the global health context for all age groups were also interrogated. The retained citations were screened by abstract and title by two independent reviewers, and subsequently compared and pooled. Citations retained by both were then read in full to extract the required information. This systematic review was registered in the PROSPERO international prospective register of systematic reviews (CRD42017065435). See appendices A through D for full details on search methodology and retained publications.

1.2. Statistical analysis 1.2.1. Overview Statistical analyses and graphic creation were carried out using Excel© 2011 and SAS. The data of survival for our case series versus PSWD was initially represented as a Gaussian probability density function, which was subsequently integrated to represent the cumulative distribution function. We solved for a numerical solution through multiple iterations to find the best curve fit and identified on this curve the critical PSWD associated with ≥80% survival in this case series. The odds of ≥80% survival in countries with this critical density compared to the odds of ≥80% in countries not achieving the critical density were subsequently calculated using Fisher's Exact Test.

1.2.2. Calculating survival The reported mortality rate from each study was retained and converted to a survival rate (1 – mortality). Only one survival outcome per condition per country was retained in order to avoid duplicate data. When several publications reported outcomes for the same country, the most recent publication and/or the publication with the largest data set was prioritized. Whenever possible, national registries and cohort studies were preferred over single-center or single-surgeon reported outcomes.

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

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Fig. 1. Systematic review PRISMA flow diagram – survival.

1.2.3. Calculating PSWD To determine the PSWD, we identified the most recent census of PSs working in each country from any published literature, and by querying local medical/surgical association websites whenever available. PSs were defined as general surgeons with dedicated formal training in pediatric surgery and appropriate certification as specialist surgeons from their country's regulating bodies. Of note, for the current study, general surgeons operating on children without formal training, as well as pediatric surgical subspecialists such as pediatric orthopedic, cardiac, or plastic surgeons were not included whenever possible. Local general surgery fellows and residents, as well as foreign or volunteer PSs participating in humanitarian initiatives were also excluded. We then identified the number of children under 15 years of age in the countries of interest by referring to most recent data available from the CIA Factbook [13]. Income level was determined based on the World Bank 2016 online Income Level Data Files [14].

The PSWD for each country was then calculated as the number of PSs/million children under 15 years of age. This was then mapped using JSFiddle© online to modify the freely available code shared by Google GeoCharts, as well as the ISO Online Browsing Platform for country codes [15–17].

1.2.4. Plotting survival as a function of PSWD A Gaussian probability density function was initially plotted using change in survival as a function of PSWD, followed by integration of the probability function in order to yield a cumulative distribution function of survival in this case series as a function of PSWD. A cumulative distribution function was deemed the most appropriate model as it depicts how the addition of each pediatric surgical worker (increase in xvariable) will correspond to an increase in survival (y-variable) up to a possible maximum of 100%. Addition of further surgeons past this critical point will not affect survival.

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

Records iden gh database searching (n = 1,695)

Addi onal records iden fied through other sources (associa on websites) (n = 4)

Iden

on

4

Eligibility

Screening

Records a er duplicates removed (n = 1,515) (n = 1511 from database)

Records screened (n = 1,515) (n = 1511 from database)

Records excluded (n = 1,430)

Full-text ar ssessed for eligibility (n = 85) (n = 81 from database)

Full-text ar xcluded, with reasons (n = 70)

Included

Studies included in e synthesis (n = 15) (n = 11 from database)

Studies included in quan synthesis (n = 15) (n = 11 from database)

Fig. 2. Systematic review PRISMA flow diagram – surgical workforce.

Mean and standard deviation of the cumulative distribution function were subsequently optimized so as to minimize the absolute error between the projected function and existing data, thus establishing a best-fit curve depicting the relationship between PSWD and survival. The 95% confidence intervals of this cumulative distribution function were also represented graphically to illustrate the error of our model. 1.2.5. Establishing the critical PSWD required for case series survival ≥80% We set a threshold for acceptable survival of 80% based on a reasonably attainable survival for all conditions in HICs. We then computed

the number of surgeons based on mean case series survival rates, using survival rate as the dependent variable. We fit the non-linear model choosing starting points for the mean and variance, and through multiple iterations, obtained a convergence with final values of mean = 1.6844 and variance = 8.5210. This process was repeated removing extreme values for PSWD N70 and once again the mean and variance remained the same at model convergence. The residual sum of squares after convergence was 4.5329. Odds ratio to establish the significance of the association were calculated using Fisher's Exact Test. (Appendix E).

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

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Fig. 3. Global PSWD for children under 15 years of age in 86 countries. A, PSWD by country. B, PSWD by income for 86 countries. Legend: PSWD is expressed as number of PSs/million children under 15 years of age. A, PSWD for each of the 86 countries identified via the systematic review protocol. PSWD is represented using a colorized scale. B, Average PSWD by income level as defined by the WHO.

Table 1 Survival outcomes by condition and income. Survival Condition

LICs (n = 8)

MICs (n = 52)

HICs (n = 50)

Gastroschisis Esophageal atresia Intestinal atresia Perforated typhoid enteritis

6.0% (±8.7%) 28.0%⁎ 73.9% (±20.8%) 72.3% (±1.8%)

56.9% (±27.4%) 61.8% (±30.4%) 84.0% (±14.6%) 91.5% (±9.7%)

94.8% (±7.2%) 86.7% (±11.7%) 95.0% (±4.4%) ⁎⁎

For each condition, survival (expressed as % of all cases) and standard deviation were calculated across the range of low, middle- and high-income countries. ⁎ Only one country was retained for this category. ⁎⁎ No studies were retained for this category.

1.2.6. Establishing the PSWD scale-up required among countries studied To determine the number of additional PSs required for a mean survival of ≥80% among our case series for the four conditions studied, the overall annual incidence of each condition was identified and the number of incident cases per country was calculated using the annual birth rate [13, 18–23]. The following formulas were used: • Current mean annual survival for case series per condition = [current mean survival (%)] × [projected incident cases] • Expected annual survival per condition = 0.8 × projected incident cases

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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Fig. 4. PSWD and mean survival for case series by income. A, Mean Survival for case series by income. B, Mean survival for case series by condition across income levels.

• PSWD necessary for ≥80% case series survival = [(Current PSWD × expected mean annual survival)] / [current annual survival] The required workforce scale-up was estimated by calculating the difference in the number of PSs between the existing density and the critical PSWD as follows: • Required pediatric surgical workforce scale-up = [Projected PSWD associated with ≥80% survival in case series] – [Current PSWD]. Of note, only the countries which did not meet the critical PSWD identified in the present work were included in the calculation of the scale-up. 2. Results 2.1. Systematic reviews We identified 8368 articles in the primary search for pediatric surgical outcomes of interest and 1695 articles in the primary search for

pediatric surgical workforce. After title and abstract screening by two independent reviewers, followed by careful review of the selected articles, 123 and 15 articles were ultimately retained for outcomes and surgical workforce, respectively (Figs. 1-2; Appendices A-D). The PSWD data was obtained for a total of 86 countries: 31 HICs, 35 middle-income countries (MICs), and 20 low-income countries (LICs). The total census was 10,707 PSs. Three countries (Burundi, The Gambia and Mauritania) had no PSs at the time of this review. Recent data presented by the College of Surgeons of East, Central and Southern Africa (COSECSA) at the GICS III meeting in Vellore, India additionally indicated that South Sudan did not have any PSs [24]. In contrast, Poland held the highest PSWD, namely 125 PSs/million children under 15 years of age (Fig. 3). The average PSWD by income was 32.8, 9.4, and 0.5 PSs/ million children under 15 years of age in HICs, MICs, and LICs respectively. The overall survival by income for this case series, pooling 110 reported outcomes from 61 individual countries was 91.8% (± 9.5%) in HICs, 69.4% (± 27.7%) in MICs, and 42.3% (± 34.9%) in LICs, a trend that was generally retained within individual conditions (Table 1, Fig. 4).

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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B

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Fig. 5. Mean survival for case series for all conditions based on the systematic review of children under 15 years of age. Fig. 5.1 depicts a scatter plot of PSWD vs mean survival in case series. Fig. 5.2 depicts PSWD vs. Survival Rate with our Predicted Survival Rate (CDF) in Case Series. Fig. 5.3 depicts Critical PSWD vs Mean Survival in Case Series with Reference Lines, Convergence lines identifying Critical Density of PSWD corresponding with 80% survival for this case series and Reference Lines for upper and lower limits of Predicted Survival Rate. Legend: Survival outcomes for case series correlated to PSWD across all 48 countries and all conditions are represented graphically as a cumulative distribution function. A critical PSWD of 4.14 PSs/million children under 15 years of age was associated with ≥80% survival for this case series. Odds of survival ≥80% if ≥4 PSWD: OR 16.8, 95% CI 5.64–48.1. (CDF: cumulative distribution function. STDEV: standard deviation). n = 94.

2.2. Critical PSWD and survival We subsequently plotted survival outcomes of the selected pediatric surgical conditions against PSWD for 48 countries for which both variables were available, translating into 94 discrete values. We found that a survival ≥ 80% in our case series was significantly associated with a minimum of 4 PSs/million children under 15 years of age; a country with a PSWD at or above this threshold would be up to 16.8 times more likely to have survival outcomes ≥ 80% for the index conditions studied (OR 16.8, p b 0.0001, 95% CI 5.66–49.88) (Fig. 5 and supplemental Fig. 6). Among the countries for which both outcomes and pediatric surgeon census was available, 18 did not reach the critical PSWD. In this specific subset, increasing PSWD to this optimal value would require training approximately 1427 additional surgeons. This group of countries represents about 755.5 million children under 15 years of age. Following the critical density of 4 PS/million children under 15 years of age, there would be a need for approximately 3023 pediatric surgeons in total (including currently practicing surgeons) in

these selected countries. However current data collected in this study identifies only 1596 PS currently practicing in these areas, and therefore a need to nearly double the current pediatric surgical workforce in these countries. On a more global scale, there are approximately 1742 billion children living in LMICs [25]. Scaling-up to a critical density of 4 PS/million children under 15 years of age globally using this figure would represent a need for approximately 6967 additional PS in LMICs as a whole (4 PS/ million × 1.742 billion children under 15). 3. Discussion To our knowledge, this is the first study examining the relationship between PSWD and childhood mortality from surgical conditions on a global scale. Recent studies have estimated that while 11% of the global burden of disease is surgical, significant gaps exist between population needs and access to surgical, anesthesia, and obstetrical care in LMICs [1, 2, 26]. It is also estimated that a potential 77.2 million DALYs are avertable by scaling up SAO care, as well as 1.5 million deaths (6–7% of all deaths) avertable yearly in LMICs through provision of essential

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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surgeries [27]. These same authors also identified a deficit of up to 9 general surgeons/100,000 people in LMICs compared to the USA, echoed by other recent estimates as well [4, 28]. With over one fifth of surgical DALYs stemming from untreated congenital anomalies and perinatal conditions [7], there is a significant need to clarify how gaps in pediatric surgical workforce and recorded surgical outcomes both co-exist and correlate in LMICs and support well-defined goals for the scaling up of pediatric surgical workforce. Accordingly, our study documents in our case series the significant disparity in survival outcomes after pediatric surgical procedures globally across the human development index (HDI) spectrum, with an average 40% difference in survival rate for most conditions between lowand high- income countries. The survival vs. PSWD scatter plots are remarkably similar for the conditions examined, although numbers are small for typhoid perforations (Fig. 4), likely because typhoid perforation disappears as the HDI increases. Only intestinal obstruction did not follow the same distribution, explicable by the heterogeneity of this category given the variety of underlying causes for obstruction. In addition, neonatal bowel obstruction was not included in the final analyses since in LICs it typically comprises term infants with Hirschsprung's disease and anorectal malformations, while in HICs this category commonly includes sick infants with meconium ileus/meconium peritonitis, necrotizing enterocolitis, and other neonatal conditions/syndromes. Of note, our review did not include a detailed analysis of gestational ages or patient co-morbidities. In contrast with previous literature depicting a logarithmic relationship between PSWD and survival [4], we chose to represent the relationship of survival for our case series and PSWD as a cumulative distribution function, since the increasing PSWD in any given country can only add to the current surgical survival, and raise it at best to a maximum of 100%. We subsequently elected to set the benchmark for survival at approximately 80% based on a reasonably attainable survival for all conditions in HICs. Although there remains room for improvement in the care of the conditions of interest across all income levels, the HICs included in this review represent the highest feasible standard of accessible population-wide care in the present day. Solving for the best fit of the cumulative distribution function of mean survival for our case series versus PSWD subsequently led us to the critical density of 4 PSs/million children under 15 years of age, corresponding to a ≥ 80% survival for our case series. When examining the PSWD, significant variability was observed depending on country's HDI; none of the LICs (0/20), 40.0% (14/35) of the MICs, and 96.9% (30/31) of the HICs studied currently reach this critical density of 4 PSs/million children under 15 years of age. Comparing these numbers to the literature, this critical density identified is in keeping with earlier estimates by Chirdan et al. stating a need for approximately 1 PS/200,000 children under 15 years of age [9]. We further observe that in 2015, the LCoGS set a target goal of at least 20–40 SAO providers/100,000 people globally [4, 28]. This however remains a distant goal for a large proportion of LMICs, as shown by O'Flynn et al. who estimated a current workforce of 0.53 surgeons/100,000 people in a subset of East African countries [4, 28]. Indeed, LMICs employ only 19% of the global surgical workforce despite being home to roughly 84% of the world's population [7, 29–31]. Importantly, children under 15 years of age represent 28% of the population in LMICs and 17% of the population in HICs [32, 33]. Based on this age distribution, we can extrapolate that our estimated target of 4 PSs/million children under 15 years of age would become a target of approximately 1.1 and 0.7 PSs/million population in LMICs and HICs respectively. This is a figure comparable to the LCoGS projections when taking into account the proportion of SAO providers who would be pediatric surgeons. A significant strength of this present work, in contrast to previous estimates, is the use of a systematic review to extract workforce and outcome data directly from local LIC, MIC, and HIC centers that have reported in the literature. In addition, through a cumulative distribution function, we are the first to characterize the relationship between PSWD

and survival for the four conditions of interest. Up to this point, studies have primarily calculated pediatric surgical workforce needs using existing ratios of surgeons per capita in HICs. These models are however limited as the demographic make-up of each population as well as the national infrastructure within which physicians practice varies significantly across national income levels. By rooting our analysis in the countries' own best available data describing workforce and outcomes, we believe to have better, though still imperfectly, described the way these two variables interact. We can therefore express more robustly the need to scale up surgical workforce in LMICs to a minimum of 4 PSs/million children under 15 years of age, or 1.1 PSs/million capita. Scaling up the surgical workforce in LMICs, including training and retaining surgical personnel, remains complex and challenging. Recent figures presented by the COSECSA in the context of the Global Initiative for Children's Surgery show little to no change in pediatric surgical workforce across a number of East African countries [24]. Moreover, many LMICs rely on external training programs for their surgical subspecialist workforce, which increases the likelihood of specialist migration to countries with better working conditions [4, 8]. Nonetheless, in recent years several organizations such as the West African College of Surgeons, the Pan-African Academy of Christian Surgeons, and in particular COSECSA, have suggested innovative solutions to the issue of surgeon workforce retention after training [34]. Unfortunately, the pediatric surgical workforce is not addressed specifically in that report. It is therefore hoped that the current study will help inform further strategic planning of surgical workforce scale-up in LMICs. In addition, our results reinforce the strong correlation between HDI and density of PSs for any given country, as already noted by Lalchandani et al. [35]. It is however difficult to accurately capture the number of healthcare professionals performing pediatric surgery in any given country. In several resource-constrained areas of the world, surgical task shifting (non-surgeon physicians and at times nonphysician healthcare personnel taking on the tasks of a specialist surgeon) complicates these measurements [36]. Several reports mention surgical task shifting as an answer to chronic shortages of PSs [37–40] in as many as 33% of LMICs [36]. Moreover, many other surgical specialists besides general PSs routinely operate on children – neurosurgeons, plastic surgeons, urologists, and so forth. Several other factors influencing PSWD, such as local definition of subspecialties, pediatric referral patterns, and the overarching structure of the healthcare system including regionalization of care, may further complicate accurate estimates. The actual number of active surgeons caring for children is therefore very difficult to ascertain, and thus we have intentionally limited this study to standard pediatric general surgical procedures treated by general PSs. Moreover, the literature on pediatric surgical outcomes and corresponding national workforce in LMICs remains limited, potentially misrepresenting the true survival rate of rare conditions [5, 41]. The studies identified originate from academic centers with institutional outcomes often above the national average, thereby probably overestimating the true survival rate for those countries at the population level. In particular, due to the high birth rate outside hospital settings in LMICs and the geographic and logistic limitations in access to specialized care [4], the proportion of children who die before reaching appropriate care is estimated to be significant and remains unaccounted for. Based on all these reasons, our results may overestimate the true survival outcomes reported for LMICs. Our study also does address the morbidity related to pediatric surgical conditions and the toll on patient's caregivers and families. Deriving metrics from these yet to be fully characterized, but nonetheless crucial processes, would have been very difficult to achieve. Another set of noteworthy limitations to our study stems from the PSWD being only one of many factors influencing outcomes in pediatric surgery. Timely access to intensive care units staffed by neonatologists, safe pediatric anesthesia care, access to intensive resources such as total parenteral nutrition and mechanical ventilation, expert nursing care,

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

and management by a multidisciplinary team are crucial factors to improving survival for many conditions [42, 43]. However, we do not currently have the means of measuring these elements on a global scale. Therefore, while the PSWD may be a suitable surrogate measure for a nation's overall pediatric surgical capacity, scaling it up as suggested is unlikely to significantly impact pediatric surgical mortality unless it is accompanied by investments in neonatal, intensive, and supportive care. It is therefore important to interpret and apply our conclusions with these caveats in mind. Interestingly however, we have witnessed several instances where the advent of a fully trained PS in a resourcepoor setting led to a broad scaling up of children's care, with anesthesiologists, pediatricians, and nurses seeking additional training, usually with the support of hospital administrators and/or government officials.

9

high-income countries, and scaling up the workforce to a critical number of PSs as suggested in our study may contribute, in conjunction with additional systemic and structural factors, to improving children's survival. We have attempted to set rough benchmarks for the development of global pediatric surgical capacity over the coming decades. Further studies are needed to refine these benchmarks by clarifying the role of non-surgical providers, other surgical subspecialists, and allied healthcare professionals on surgical mortality, as well as estimating the impact of broader capacity indicators such as nursing, intensive care, and advanced nutritional support, towards the overarching goal of improving childhood survival globally. Supplementary data to this article can be found online at https://doi. org/10.1016/j.jpedsurg.2019.11.001.

4. Conclusions The present work is the first of its kind to explore the relationship between PSWD and mortality from key pediatric surgical conditions on a global scale. Significant disparities exist in both PSWD and childhood mortality from surgical conditions across low-, middle- and

Acknowledgements We would like to thank Dr. John Sampalis for his support in the revision of this manuscript.

Appendix A. Search methodology – surgical mortality

Surgical Mortality Appendix - Databases Searched Medline [Ovid] (May 12, 2017) Ovid MEDLINE(R) and Epub Ahead of Print, In-Process & Other Non-Indexed Citations, Ovid MEDLINE(R) Daily b1946 to PresentN 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 21

exp Gastroschisis/ (gastrosch* or gastro-sch*).tw,kf. Esophageal Atresia/ ((esophag* or oesophag* or endoesophag* or intraesophag* or tracheoesophag*) adj3 (atres* or atretic* or atroph*)).tw,kf. exp Esophagus/ and (atres* or atretic* or atroph*).tw,kf. exp Intestinal Obstruction/ ((bowel* or gastro* or intes* or digest* or colon* or gastric* or ileus or ileiti* or duoden* or jejun*) adj5 (obstruc* or block* or atres* or atretic* or atroph*)).tw,kf. 1 or 2 or 3 or 4 or 5 or 6 or 7 exp Infant, Newborn/ Intensive Care Units, Neonatal/ Intensive Care, Neonatal/ Fetus/ (newborn* or new-born* or neonat* or neo-nat* or nicu or infan* or prematur* or pre-matur* or postmatur* or post-matur* or preemie* or preterm or pre-term or postneonat* or perinatal or peri-natal or prenatal or pre-natal or low birth weight or VLBW or LBW or natality or antenatal or ante-natal or fetus* or fetal or foetus or foetal).jw,tw,kf. 9 or 10 or 11 or 12 or 13 8 and 14 Typhoid Fever/ and Intestinal Perforation/ ((typhoid* or typhus*) adj5 (perforat* or intes*)).tw,kf. 16 or 17 exp pediatrics/ or exp adolescent/ or exp child/ or exp infant/ (newborn* or new-born* or neonat* or neo-nat* or nicu or infan* or prematur* or pre-matur* or postmatur* or post-matur* or preemie* or preterm or pre-term or postneonat* or perinatal or peri-natal or prenatal or pre-natal or low birth weight or VLBW or LBW or natality or antenatal or ante-natal or fetus* or fetal or foetus or foetal or child* or adolesc* or paediatr* or pediatr* or baby* or babies* or toddler* or kid or kids or boy* or girl* or juvenile* or teen* or youth* or pubescen* or preadolesc* or prepubesc* or preteen or tween).jw,tw,kf. 19 or 20 18 and 21 exp Mortality/ mo.fs. exp Death/ (mortalit* or death* or die or died or dies or dying or dead or fatal* or demise).tw,kf. Survival Analysis/ surviv*.tw,kf. 23 or 24 or 25 or 26 or 28 15 and 29 22 and 29 30 or 31 Animals/ not (Animals/ and Humans/) ((animals or animal or canine* or cat or cats or dog or dogs or feline or hamster* or mice or monkey or monkeys or mouse or murine or pig or pigs or piglet* or porcine or primate* or rabbit* or rats or rat or rodent* or sheep*) not (human* or patient*)).ti. 32 not (33 or 34) remove duplicates from 35 #19 NOT #20

1093 2127 3299 4039 1394 44472 43990 75256 561788 11822 5029 75667 1073934 1317496 13336 354 522 611 3277083 2693445 4254968 282 335650 502247 137353 1462414 117792 947820 2379247 3419 158 3571 4365866 1938401 3427 3355 426

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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Appendix B. Search methodology – surgical workforce

Surgical Workforce (Scoping) Appendix - Databases Searched Medline [Ovid] (April 28, 2017) Ovid MEDLINE(R) and Epub Ahead of Print, In-Process & Other Non-Indexed Citations, Ovid MEDLINE(R) Daily b1946 to PresentN 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 #7 #6 #5 #4 #3 #2 #1

Health Manpower/ 12119 ma.fs. 62533 (manpower* or man-power* or workforc* or work-forc* or under-doctor* or underdoctor* or underserv* or under-serv*).tw,kf. 34859 1 or 2 or 3 99571 exp Surgeons/ 3742 Specialties, Surgical/ 3399 (surgeon* or surgical).tw,kf. 924375 5 or 6 or 7 926245 4 and 8 4013 exp pediatrics/ or exp adolescent/ or exp child/ or exp infant/ 3270260 (newborn* or newborn* or neonat* or neonat* or infan* or child* or adolesc* or paediatr* or pediatr* or baby* or babies* or toddler* or kid or kids or boy* or girl* 2167112 or juvenile* or teen* or youth* or pubescen* or preadolesc* or prepubesc* or preteen or tween).tw,kf. (pediatr* or paediatr*).jw. 515671 10 or 11 or 12 3946735 9 and 13 635 ((surgeon* or surgical) adj5 (specialist* or subspecialist* or specialization* or subspecialization* or global* or international* or world* or humanitarian)).tw,kf. 6599 ((surgeon* or surgical) adj3 (aid* or assistan*)).tw,kf. 3222 15 or 16 9775 4 and 17 371 *Specialties, Surgical/ma 112 18 or 19 466 exp world health organization/ 31439 ((WHO or lancet or unicef) adj (commission or index or global* or survey* or situational or tool* or checklist* or check-list* or surg* or indicator* or safe*)).tw,kf. 1363 exp societies, medical/ 66558 Advisory Committees/ 8438 Global Health/ 38313 ((global* or international* or world* or humanitarian) adj1 health).tw,kf. 64593 21 or 22 or 23 or 24 or 25 or 26 189837 9 and 27 324 14 or 20 or 28 1239 remove duplicates from 29 1213 limit 30 to yr="2000 -Current" 874 3,017 TS=((surgeon* or surgical) NEAR/3 (aid* or assistan*)) TS=((specialist* or subspecialist* or specialization* or subspecialization* or global* or international* or world* or humanitarian*) NEAR/5 (surgeon* or 5,726 surgical)) 239 #3 AND #4 TS=(newborn* or newborn* or neonat* or neonat* or infan* or child* or adolesc* or paediatr* or pediatr* or baby* or babies* or toddler* or kid or kids or boy* 2,532,376 or girl* or juvenile* or teen* or youth* or pubescen* or preadolesc* or prepubesc* or preteen or tween) 1,470 #2 AND #1 718,168 TS=(surgeon* or surgical) 50,261 TS=(manpower* or man-power* or workforc* or work-forc* or under-doctor* or underdoctor* or underserv* or under-serv*)

Appendix C. Retained publications – pediatric surgeon workforce

Scoping Review – Workforce No.

Country

Year published

Year of census

Pediatric surgeon no.

Authors

1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18

Austria Bangladesh Belgium Benin Brazil Bulgaria Burkina Faso Burundi Cambodia Cameroon Central African Republic China Colombia Côte d'Ivoire Croatia Cuba Cyprus Czech Republic

2017 2016 2017 2016 2016 2017 2016 2016 2016 2014 2014 2015 2016 2016 2017 2016 2017 2017

2014-2015 2015 2014-2015 2015 2015 2014-2015 2015 2015 2000 2011 2011 N/A 2014 2015 2014-2015 2010 2014-2015 2014-2015

67 161 19 2 721 44 8 0 24 3 1 1850 46 30 42 93 8 69

Parigi, G. B., et al.[1] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] O'Flynn, E., et al. [3] Krishnaswami, S., et al. [2] Hoyler, M., et al. [4] Krishnaswami, S., et al. [2] Hoyler, M., et al. [4] Lalchandani, P. and J. C. Dunn [5] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] Parigi, G. B., et al. [1] Parigi, G. B., et al. [1]

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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11

(continued) No.

Country

Year published

Year of census

Pediatric surgeon no.

Authors

19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 73 74 75 76 77 78 79 80 81 82

Denmark Ecuador Egypt England Eritrea Ethiopia Finland Gabon Germany Ghana Greece Guinea Hong Kong Hungary India Indonesia Iran Iraq Ireland Italy Japan Kenya Luxemburg Madagascar Malawi Malaysia Mali Mauritania Mozambique Myanmar Netherlands Nicaragua Niger Nigeria Norway Pakistan Papua New Guinea Peru Philippines Poland Portugal Puerto Rico Rwanda Saudi Arabia Senegal Sierra Leone Singapore Slovakia South Africa South Korea Spain Sri Lanka Sweden Switzerland Taiwan Tanzania Thailand The Gambia Togo Turkey Uganda USA Zambia Zimbabwe

2017 2016 2016 2015 2011 2016 2003 2016 2015 2016 2003 2016 2000 2017 2016 2016 2016 2016 2015 2017 2003 2016 2017 2016 2016 2016 2016 2016 2016 2016 2017 2016 2016 2016 2017 2016 2006 2016 2016 2003 2003 2016 2016 2016 2016 2016 2000 2017 2016 2000 2003 2016 2003 2003 2015 2016 2016 2017 2016 2011 2016 2016 2016 2016

2014-2015 2015 2009 N/A N/A 2015 N/A 2008 N/A 2015 N/A 2015 N/A 2014-2015 2015 2000 2000 2000 N/A 2014–2015 N/A 2015 2014-2015 2014 2015 2000 2008 2015 2015 2000 2014-2015 2000 2015 2015 2014-2015 2015 2006 2000 2014 N/A N/A 2015 2015 2000 2015 2014 N/A 2014-2015 2015 N/A N/A 2015 N/A N/A N/A 2015 2015 N/A 2015 2005 2015 2015 2015 2015

10 91 120 338 1 7 95 3 250 11 11 6 17 59 1001 20 64 75 4 440 636 16 3 2 3 17 1 0 2 8 35 57 4 87 15 170 3 102 46 710 75 8 1 50 15 1 8 31 35 36 349 15 100 51 61 6 166 0 3 86 7 1250 1 3

Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Lalchandani, P. and J. C. Dunn [5] Calisti, A., et al. [6] O'Flynn, E., et al. [3] Driller, C. and A. M. Holschneider [7] Krishnaswami, S., et al. [2] Lalchandani, P. and J. C. Dunn [5] Krishnaswami, S., et al. [2] Driller, C. and A. M. Holschneider [7] Krishnaswami, S., et al. [2] Saing, H.[8] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Lalchandani, P. and J. C. Dunn [5] Parigi, G. B., et al. [1] Driller, C. and A. M. Holschneider [7] O'Flynn, E., et al. [3] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] O'Flynn, E., et al. [3] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] O'Flynn, E., et al. [3] Krishnaswami, S., et al. [2] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] Kevau, I. and D. A. Watters [9] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Driller, C. and A. M. Holschneider [7] Driller, C. and A. M. Holschneider [7] Krishnaswami, S., et al. [2] O'Flynn, E., et al. [3] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Saing, H. [8] Parigi, G. B., et al. [1] Krishnaswami, S., et al. [2] Saing, H. [8] Driller, C. and A. M. Holschneider [7] Krishnaswami, S., et al. [2] Driller, C. and A. M. Holschneider [7] Driller, C. and A. M. Holschneider [7] Lalchandani, P. and J. C. Dunn [5] Krishnaswami, S., et al. [2] Krishnaswami, S., et al. [2] Toobaie, A., et al. [10] Krishnaswami, S., et al. [2] Durak, H. I. and A. Avanoglu [11] O'Flynn, E., et al. [3] Krishnaswami, S., et al. [2] O'Flynn, E., et al. [3] O'Flynn, E., et al. [3]

Association Websites Queried Item No.

Country

Year

Pediatric surgeon no.

Source

1 2 3 4

Argentina Australia Canada France

2017 2017 2017 2011

141 67 77 270

Asociación Civil Argentina de Cirugía Pediátrica [12] Royal Australian College of Surgeons [13] Canadian Medical Association [14] Société Française de Chirurgie Pédiatrique [15]

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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References 1. Parigi, G.B., et al., European Census on Pediatric Surgery. European Journal of Pediatric Surgery, 2017. 04: p. 04. 2. Krishnaswami, S., B.C. Nwomeh, and E.A. Ameh, The pediatric surgery workforce in low- and middle-income countries: problems and priorities. Seminars in Pediatric Surgery, 2016. 25(1): p. 32-42. 3. O'Flynn, E., et al., The Specialist Surgeon Workforce in East, Central and Southern Africa: A Situation Analysis. World Journal of Surgery, 2016. 40(11): p. 2620-2627. 4. Hoyler, M., et al., Shortage of doctors, shortage of data: a review of the global surgery, obstetrics, and anesthesia workforce literature. World Journal of Surgery, 2014. 38(2): p. 269-80. 5. Lalchandani, P. and J.C. Dunn, Global comparison of pediatric surgery workforce and training. Journal of Pediatric Surgery, 2015. 50(7): p. 1180-3. 6. Calisti, A., et al., Promoting major pediatric surgical care in a low-income country: a 4-year experience in Eritrea. World Journal of Surgery, 2011. 35(4): p. 760-6. 7. Driller, C. and A.M. Holschneider, Training in pediatric surgery–a comparison of 24 countries in Europe and other countries around the world.[Erratum appears in Eur J Pediatr Surg. 2003 Dec;13(6):434]. European Journal of Pediatric Surgery, 2003. 13(2): p. 73-80. 8. Saing, H., Training and delivery of pediatric surgery services in Asia. Journal of Pediatric Surgery, 2000. 35(11): p. 1606-11. 9. Kevau, I. and D.A. Watters, Specialist surgical training in Papua New Guinea: the outcomes after 10 years. ANZ Journal of Surgery, 2006. 76(10): p. 937-41. 10. Toobaie, A., et al., Pediatric surgical capacity in Africa: Current status and future needs. Journal of Pediatric Surgery, 2017. 29: p. 29. 11. Durak, H.I. and A. Avanoglu, Factors influencing the choice of pediatric surgery as a medical career among Turkish pediatric surgeons and residents. Turkiye Klinikleri Journal of Medical Sciences, 2011. 31(2): p. 450-454. 12. Asociacion Civil Argentina de Cirugia Pediatrica (ACACIP). Historia. 2017 July 16, 2017]; Available from: http://acacip.org.ar/institucion-Nosotros-Historia.php. 13. Royal Australian College of Surgeons, Find a Surgeon - Directory 2017, Royal Australian College of Surgeons: Web. 14. Canadian Medical Association (CMA), Number of physicians by specialty and age, Canada, 2017, in CMA Masterfile, C.M. Association, Editor. 2017. 15. Société Française de Chirurgie Pédiatrique. Accréditations. 2011 2011 [cited 2017 July 16]; Available from: https://www.chirpediatric.fr/organisme-accreditation-dpc-cnce.html.

Appendix D. Retained publications – pediatric survival Gastroschisis Year Published

Years Data Collected

Survival (%)

1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26

No.

Argentina Australia Barbados Brazil Canada Chile China Colombia Côte d'Ivoire Czech Republic Denmark Egypt France Germany Greenland India Iran Italy Jamaica Malawi Mexico Netherlands Nigeria Norway Poland Portugal

Country

2011 2016 2016 2016 2017 2013 2014 2010 2016 2004 2017 2011 2016 2017 2017 2017 2012 2017 2017 2016 2016 2017 2016 2016 2007 2016

2009-2011 1992-2009 1993-2012 1995-2010 2003-2015 1996-2010 2000-2012 1998-2006 2012-2014 1995-2002 1997-2009 1998-2005 2009-2014 2007-2012 1989-2005 2008-2016 2005-2007 2002-2013 2006-2015 2012-2014 2008-2013 2009-2013 2012-2014 1993-2008 1998-2004 2002-2011

81.8% 97.0% 71.5% 85.1% 98.6% 100.0% 60.9% 82.0% 0.0% 94.0% 95.8% 82.7% 97.2% 100.0% 82.0% 60.0% 20.0% 94.7% 22.6% 0.0% 73.8% 100.0% 25.0% 100.0% 95.9% 92.3%

Palermo, M. S. F. [1] Pszczola, R., et al. [2] Singh, K. and A. Kumar [3] Miranda da Silva Alves, F., et al. [4] Youssef, F., et al. [5] Nazer, H. J., et al [6] Du, L., et al. [7] Toro, M. N. H., et al. [8] Ford, K., et al. [9] Rygl, M., et al. [10] Risby, K., et al. [11] Hashish, A. A. E. and E. Elhalaby [12] Tosello, B., et al. [13] Dingemann, C., et al. [14] Bugge, M., et al. [15] Rattan, K. N., et al. [16] Askarpour, S., et al. [17] Briganti, V., et al. [18] Marshall Niles, S. G., et al. [19] Ford, K., et al. [9] Mendez-Martinez, S., et al. [20] Fleurke-Rozema, H., et al. Ford, K., et al. [9] Champion, V., et al. [21] Bulhak-Guz, H., et al. [22] Valente, L., et al. [23]

Authors

27 28 29 30 31 32 33 34 35 36 37 38 39

Romania Serbia South Africa Spain Sweden Taiwan Thailand Turkey Uganda Ukraine United Kingdom USA Zimbabwe

2013 2016 2016 2016 2015 2011 2011 2012 2016 2001 2016 2016 2015

2001-2010 2001-2015 2012-2014 2004-2014 2006-2014 1990-2010 1986-2009 2000-2010 2014-2015 1996-2000 2012-2014 2005-2013 2013

37.0% 89.2% 71.0% 96.3% 94.2% 81.0% 32.1% 65.5% 2.0% 55.0% 100.0% 96.6% 16.0%

Tarca, E. and S. G. Aprodu [24] Marinovic, V. M., et al. [25] Ford, K., et al. [9] Dore Reyes, M., et al. [26] Mesas Burgos, C., et al. [27] Chen, I. L., et al. [28] Niramis, R., et al. [29] Erdogan, D., et al. [30] Wesonga, A. S., et al. [31] Liaturyns'ka, O. V. and M. Kurochkin [32] Ford, K., et al. [9] Youssef, F., et al. [5] Apfeld, J. C., et al. [33]

Intestinal Atresia No. 1 2 3 4 5 6 7

Country Argentina Australia Barbados Bulgaria Canada China Czech Republic

Year Published

Years Data Collected

Survival (%)

2000 2008 2014 2006 2017 2012 2008

N/A 1992-2003 1993-2012 2000-2004 2004-2014 2009-2010 1991-2006

100.0% 92.0% 86.7% 80.0% 97.8% 100.0% 90.9%

Authors Giuseppucci, J. C. and A. Martinez [34] Walker, K., et al. [35] Kumar, A. and K. Singh [36] Ionkov, A. [37] Zani, A., et al. [38] Li, B., et al. [39] Rouskova, B., et al. [40]

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

13

(continued) No.

Country

Year Published

Years Data Collected

Survival (%)

Authors

8 9 10 11 12 13

Egypt India Iran Italy Mexico Nepal

2016 2017 2015 2012 2013 2012

2008-2016 2012-2015 2002-2010 1992-2009 2007-2012 2004-2008

75.0% 69.6% 76.9% 95.4% 86.7% 59.2%

El-Asmar [41] Mohan, M. J., et al. [42] Ghafouri-Taleghani, F., et al. [43] Calisti, A., et al. [44] Gutierrez-Carrillo, M. P., et al. [45] Williams, O. M., et al. [46]

14 15 16 17 18 19 20 21 22 23 24 25

Netherlands Nigeria Pakistan Poland Réunion Saudi Arabia South Africa South Korea Spain Turkey United Kingdom USA

2009 2014 2010 2008 2009 2007 2007 2012 2013 2016 2013 2017

1971-2004 2007-2012 2007-2009 1986-2006 2000-2007 1993-2005 2002-2005 2001-2010 1995-20011 2009-2014 2005-2012 2012

89.0% 78.3% 57.5% 100.0% 100.0% 88.6% 100.0% 98.9% 97.6% 100.0% 94.5% 97.1%

Stollman, T. H., et al. [47] Ezomike, U. O., et al. [48] Kamal, A., et al. [49] Baglaj, M., et al. [50] Harper, L., et al. [51] Al-Salem, A. H. [52] Banieghbal, B. and P. G. Beale [53] Lee, S. H., et al. [54] Delgado Alvira, R., et al. [55] Savran, B., et al. [56] Bishay, M., et al. [57] Erickson, T., et al.[58]

Esophageal atresia No.

Country

Year Published

Years Data Collected

Survival (%)

1

Algeria

2015

2001-2011

60.8%

2 3 4 5 6 7

Argentina Australia Austria Cameroon Canada China

2009 2015 2017 2010 2017 2012

2002-2006 1999-2010 1975-2011 2005-2010 2000-2014 2007-2012

100.0% 100.0% 95.8% 10.0% 100.0% 94.0%

8

Colombia

2003

1997-2001

83.0%

9 10 11 12

Czech Republic Finland France Germany

2011 2013 2017 2017

1992-2008 1991-2011 1998-2013 2013-2014

90.0% 98.0% 81.0% 92.0%

13 14 15 16 17 18 19 20

Ghana Iceland Indonesia Iran Ireland Italy Japan Korea

2016 2004 2015 2016 2007 2013 2009 2014

2007-2014 1963-2002 2007-2012 2010-2014 1977-2004 1981-2012 1980-2005 2008-2013

40.3% 73.0% 16.0% 95.8% 81.0% 85.0% 83.50% 96.0%

21

Malaysia

2013

2000-2009

77.0%

22

Nigeria

2013

1997-2008

10.0%

23 24

Pakistan Poland

2013 2009

2007-2012 2005-2008

52.7% 87.0%

25

Portugal

2011

2000-2009

85.1%

26 27 28 29 30 31 32 33 34 35 36

Saudi Arabia Senegal Serbia Spain Sweden Switzerland Taiwan Thailand Turkey United Kingdom USA

2013 2015 2015 2015 2016 2004 2006 2013 2016 2016 2016

1992-2011 2010-2013 1999-2009 2008-2014 1994-2013 1973-1999 1994-2003 2003-2010 2008-2013 2006-2014 1997-2009

79.1% 28.0% 71.7% 50.0% 94.0% 78.0% 60.0% 80.3% 75.0% 90.0% 96.0%

Authors Bouguermouh, D. Salem, A.[59] Cannizzaro, C. et al [60] Shah, R. et al [61] Friedmacher, F. et al. [62] Tambo, F. F. et al. [63] Zani, A. et al. [64] Huang, J. et al. [65] Mejia Sarasti, F. J. & Medina Mejia, J. F. [66] Kalousova, J. et al. [67] Koivusalo, A. I. et al. [68] Sfeir, R. S. et al. [69] Trobs, R. B. et al. [70]

Osei-Nketiah, S. et al. [71] Gunnarsdottir, A. et al. [72] Millano, L. et al.[73] Hiradfar, M. et al. [74] Sri Paran, T. et al. [75] Cassina, M. et al. [76] Okamoto, T. et al. [77] Lee, S. et al. [78] Narasimman, S. et al. [79] Nwosu, J. N. & Onyekwulu, F. A. [80] Nasar, G. N. [81] Patkowsk, D. et al. [82] Pissarra, S. & Guimaraes, H. [83] Al-Salem, A. H. et al. [84] Fall, M. et al. [85] Vukadin, M. et al. [86] Munoz-Garcia, M. et al. [87] Donoso, F. et al. [88] Tonz, M. et al. [89] Yang, C. F. et al. [90] Niramis, R. et al. [91] Karakus, S. C. et al. [92] Ogundipe, E et al. [93] Sayari, A. J. et al. [94]

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

14

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

Perforated typhoid enteritis No. 1

Country Benin

2 3 4 5 6 7 8 9 10

Central African Republic Cote d'Ivoire Ghana India Mauritius Nigeria Pakistan Taiwan Turkey

Year Published

Years Data Collected

Survival (%)

Authors

2013

2009-2011

73.6%

Caronna, R. et al. [95]

2002

2000-2010

71.0%

Serengbe, G.B., et al. [96]

2001 2009 2005 2005 2014 2012 2006 2002

1993-2012 2003-2015 1996-2010 2000-2012 1998-2006 2012-2014 1995-2002 1997-2009

94.0% 87.4% 100.0% 100.0% 80.0% 75.0% 100.0% 95.2%

Kouame, B.D. et al. [97] Abantanga, F.A. [98] Walia, M. et al. [99] Issack, M.I. [100] Talabi, A.O. et al. [101] Jamro, B. [102] Chang, Y.T. et al. [103] Onen, A. et al. [104]

Neonatal bowel obstruction No.

Country

Year Published

Years Data Collected

Survival (%)

1 2 3 4 5 6 7 8 9 10 11 12 13

Austria Brazil Egypt Ethiopia India Iraq Italy Malawi Mexico Netherlands Nigeria Norway Pakistan

2013 2008 2015 2016 2016 2000 2011 2016 2009 2011 2009 2002 2011

2001-2009 1993-2001 2014-2015 2011-2013 2003-2016 1986-1996 1992-2008 2012-2014 N/A 1984-2007 2006-2008 1985-2000 2010-2011

95.3% 94.0% 78.8% 86.7% 77.5% 78.0% 81.8% 100.0% 95.1% 100.0% 74.6% 89.7% 97.0%

14 15 16 17 18 19 20

Saudi Arabia South Korea Spain Trinidad and Tobago Tunisia Turkey USA

2008 2016 2006 2009 2011 2016 2017

1996-2005 1995-2014 1990-2003 1999-2006 2000-2009 2009-2014 2006-2016

97.4% 96.6% 85.8% 72.7% 92.0% 100.0% 88.1%

Authors Wolfgang, R., et al. [105] Miranda, M. E., et al. [106] Wella, H. L. and S. M. M. Farahat [107] Soressa, U., et al. [108] Kumar, P., et al. [109] Nasir, G. A., et al. [110] Paradiso, V. F., et al. [111] Shah, M., et al. [112] Baeza-Herrera, C., et al. [113] Karimi, A., et al. [114] Osifo, O. D. and J. C. Okolo[115] Brantberg, A., et al. [116] Soomro, B. A., et al. [117] Mustafawi, A. R. and M. E. Hassan [118] Kim, J. Y., et al. [119] Bustos Lozano, G., et al. [120] Anatol, T. I. and S. Hariharan [121] Faouzi, N., et al. [122] Savran, B., et al. [56] Lau, P. E., et al. [123]

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Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

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Appendix E. Nonlinear regression using PROC NLIN

The NLIN procedure Dependent variable survival rate Method: Gauss-Newton Iterative Phase Sum of squares Iter

MU

0 1 2 3 4 5 6 7 8 Estimation summary

0.1000 0.2494 0.7913 1.6145 1.6871 1.6845 1.6844 1.6844 1.6844

SIGMA2 0.3000 1.5464 6.0322 8.2372 8.3891 8.4926 8.5148 8.5199 8.5210

8.4774 7.0314 5.0771 4.5362 4.5329 4.5329 4.5329 4.5329 4.5329

Method

Gauss-Newton

Iterations R PPC(SIGMA2) RPC(SIGMA2) Object Objective Observations Read Observations Used Observations Missing Note:

8 7.408E-6 0.000031 0.000135 1.314E-9 4.532891 94 94 0 An intercept was not specified for this model Approx Pr N F F Value

Source

DF

Model Error Uncorrected Total Corrected Total Parameter

2 92 94 93 Estimate

Sum of squares

Mean square

59.8125 4.5329 64.3453 6.7087

29.9062 0.0493

Approx SE

606.98

b.0001

Approximate 95% confidence limits

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

17

(continued) Parameter

Estimate

Approx SE

Approximate 95% confidence limits

MU SIGMA2 Approximate correlation matrix

1.6844 8.5210

0.3504 3.9315

0.9885 0.7128

MU SIGMA2

2.3802 16.3292

MU

SIGMA2

1.0000000 -0.4277105

-0.4277105 1.0000000

NOTE: Convergence criterion met.

PSWD required based on survival rate SR

PROBIT_SR

PSWD_REQUIRED

0.5 0.6 0.7 0.8 0.9 1.0

0.00000 0.25335 0.52440 0.84162 1.28155 8.20954

1.6844 2.4239 3.2151 4.1411 5.4253 25.6487

Predicted survival rate and its 95% confidence interval

Country Cameroon Indonesia Malawi (Queen Elizabeth Hospital, Blantyre) Benin Uganda (Mulago Hospital, Kampala) Central African Republic Zimbabwe (Harare Children's Hospital) Ghana Ghana Nigeria Malaysia South Africa South Africa (George Mukhari Academic Hospital, Pretoria) Senegal Pakistan Pakistan Pakistan India India India (PGIMS) Iran Iran Iran (Imam Khoemini Hospital, Ahvaz Jundishapur University of Medical Sciences, Ahvaz) Cote d'Ivoire Côte d'Ivoire (Centre Hospitalier Universitaire, Treichville) Egypt Egypt (Tanta University Hospital) Ireland Colombia Colombia (Hospital Universitario San Vicente de Paul, in Medellin) Turkey Turkey Turkey Turkey (Sami Ulus Maternity and Children's Hospital) South Korea Saudi Arabia Saudi Arabia China China China (Xinhua Hospital, Shanghai) Denmark (Odense University Hospital) Netherlands Netherlands (2 centres) Nigeria

Real survival rate Predicted survival Condition PSWD (%) rate (%)

Lower limit

Upper limit

EA EA GS TP GS TP GS EA TP EA EA IA GS EA EA IA TP IA TP GS EA IA GS

0.3 0.3 0.3 0.4 0.4 0.5 0.5 1.1 1.1 1.1 2 2.3 2.3 2.5 2.6 2.6 2.6 2.9 2.9 2.9 3.3 3.3 3.3

10 16 0 73.6 2 71 16 40.3 87.4 10 77 100 71 28 52.7 57.5 75 69.6 100 60 95.8 76.9 20

31.8 31.8 31.8 33.0 33.0 34.2 34.2 42.1 42.1 42.1 54.3 58.4 58.4 61.0 62.3 62.3 62.3 66.1 66.1 66.1 71.0 71.0 71.0

18.0 18.0 18.0 19.5 19.5 21.0 21.0 30.7 30.7 30.7 45.5 49.9 49.9 52.7 54.0 54.0 54.0 57.6 57.6 57.6 62.0 62.0 62.0

45.5 45.5 45.5 46.5 46.5 47.5 47.5 53.4 53.4 53.4 63.1 66.8 66.8 69.3 70.6 70.6 70.6 74.7 74.7 74.7 80.0 80.0 80.0

TP GS IA GS EA EA GS EA IA TP GS IA EA IA EA IA GS GS IA GS IA

3.4 3.4 3.8 3.8 3.8 4 4 4.3 4.3 4.3 4.3 5.3 6.7 6.7 7.9 7.9 7.9 10.8 12.4 12.4 12.4

94 0 75 82.7 81 83 82 75 100 95.2 65.5 98.9 79.1 88.6 94 100 60.9 95.8 89 100 78.3

72.2 72.2 76.6 76.6 76.6 78.6 78.6 81.5 81.5 81.5 81.5 89.2 95.7 95.7 98.3 98.3 98.3 99.9 100.0 100.0 100.0

63.0 63.0 66.9 66.9 66.9 68.8 68.8 71.4 71.4 71.4 71.4 79.7 89.2 89.2 94.6 94.6 94.6 99.5 99.9 99.9 99.9

81.3 81.3 86.2 86.2 86.2 88.5 88.5 91.5 91.5 91.5 91.5 98.7 102.3 102.3 102.1 102.1 102.1 100.3 100.1 100.1 100.1

(continued on next page)

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D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

(continued) Real survival rate Predicted survival Condition PSWD (%) rate (%)

Country Nigeria Nigeria (National Hospital, Abuja) Argentina Argentina Argentina (National Hospital Professor Alejandro Posadas) Canada Canada Canada Thailand Thailand (Queen Sirikit National Institute of Child Health, College of Medicine, Rangsit University, Bangkok) Brazil (Maternidade Otto Cirne do Hospital das Clínicas da Universidade Federal de Minas Gerais ) Norway (Haukeland University Hospital, Bergen) France France France (2 large level III NICUs) Taiwan Taiwan Taiwan (Kaohsiung Chang Gung Memorial Hospital) USA USA USA Germany Germany (patients covered by Techniker Krankenkasse insurance) UK UK United Kingdom (King’s Centre for Global Health, London) Australia Australia Australia Japan Switzerland Bulgaria Czech Republic Czech Republic Czech Republic (Medical Faculty Charles University and Faculty Hospital Motol, Prague) Portugal Portugal (Centro Hospitalar São João) Spain Spain Spain (Hospital Universitario La Paz, Madrid) Italy Italy Italy (Division of Obstetrics and Gynecology "San Camillo", Rome) Austria Sweden Sweden (Astrid Lindgren’s Children’s Hospital, Stockholm) Finland Poland Poland Poland (Instytutu Centrum Zdrowia Matki Polki, Lodzi)

Lower limit

Upper limit

TP GS EA IA GS EA GS IA EA

12.4 12.4 13 13 13 14.1 14.1 14.1 14.2

80 25 100 100 81.8 100 98.6 97.8 80.3

100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0

99.9 99.9 100.0 100.0 100.0 100.0 100.0 100.0 100.0

100.1 100.1 100.0 100.0 100.0 100.0 100.0 100.0 100.0

GS GS

14.2 15.4

32.1 85.1

100.0 100.0

100.0 100.0

100.0 100.0

GS EA IA GS EA TP GS EA GS IA EA GS EA IA GS EA GS IA EA EA IA EA IA GS EA GS EA IA GS EA IA GS EA EA GS EA EA IA GS

15.8 19.8 19.8 19.8 19.8 19.8 19.8 20.5 20.5 20.5 24.1 24.1 30.1 30.1 30.1 31 31 31 38.7 41.3 42.4 42.9 42.9 42.9 44.7 44.7 46.6 46.6 46.6 51.8 51.8 51.8 54.8 58.6 58.6 105.2 125.2 125.2 125.2

100 81 100 97.2 60 100 81 96 96.6 97.1 92 100 90 94.5 100 100 97 92 83.5 78 80 90 90.9 94 85.1 92.3 50 97.6 96.3 85 95.4 94.7 95.8 94 94.2 98 87 100 95.9

100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0

100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0

100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0 100.0

PSWD – Frequency Table Based on PSWD = 4 PS4

SurvivalRate80

Count

Y Y N N

Y N Y N

56 10 7 21

Please cite this article as: D. Hamad, Y. Yousef, N.G. Caminsky, et al., Defining the critical pediatric surgical workforce density for improving surgical outcomes: a global study, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.001

D. Hamad et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

19

PSWD – odds ratio and its 95% confidence interval based on PSWD = 4 The FREQ procedure Table of PS4 by SurvivalRate80 PS4

SurvivalRate80

Frequency Percent Row Pct Col Pct N N

21 22.34 75.00 67.74 10 10.64 15.15 32.26 31 32.98

Y

Total Statistic

DF

chi-Square Likelihood ratio chi-square Continuity adj. chi-square Mantel-Haenszel chi-square Phi coefficient Contingency coefficient Cramer's V Fisher's exact test

1 1 1 1

Y

Total

7 7.45 25.00 11.11 56 59.57 84.85 88.89 63 67.02 Value

28 29.79

31.8591 31.5631 29.2089 31.5202 0.5822 0.5031 0.5822

Cell (1,1) Frequency (F) Left-sided Pr b= F Right-sided Pr N= F Table Probability (P) Two-sided Pr b= P Odds ratio and relative risks

66 70.21

94 100.00 Prob b.0001 b.0001 b.0001 b.0001

21 1.0000 b.0001 b.0001 b.0001

Statistic

Value

95% Confidence limits

Odds ratio Relative risk (Column 1) Relative risk (Column 2) Sample size = 94

16.8000 4.9500 0.2946

5.6579 2.6905 0.1539

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