Keywords
hypospadias - spatial analysis - prevalence
Palabras clave
hipospadias - análisis espacial - prevalencia
Introduction
Hypospadias is a congenital malformation caused by an abnormal closure of the urethral folds during penile development, giving rise to a urethral opening on the ventral surface of the penis.[1]
[2]
[3] It is the second most common congenital malformation, after cryptorchidism.[1] Its prevalence has been described with results such as 20.9 per 10, 000 live births worldwide, between 10.6 and 34.7 per 10,000 live births in the European continent, 34.2 per 10,000 live births in North America, and a range between 1.5 and 8.6 per 10,000 live births for Latin American countries.[2]
[4] In Colombia, the prevalence of hypospadias has been described to be of 11.3 per 10,000 live births.[3]
[5] It is a multifactorial disease, as it has been associated with disruptions in hormonal homeostasis during embryogenesis, multiple environmental factors, and genetic predispositions.[6]
[7]
[8] The anatomical abnormalities of hypospadias have several long-term consequences on genitourinary function that carry a high Burden of Disease (BoD).[9] Early surgical intervention has been proven to be necessary for a favorable quality of life and reduction in disability.[10]
[11] Hypospadias is a disease with serious and long-lasting psychosocial repercussions.[12]
[13] In mid- and low-income countries, access to specialized care is limited. Understanding the geographical distribution of pediatric urology services is needed to improve access and allocate public health resources to improve areas with lack of coverage.
The Colombian Ministry of Health has developed the Integrated Social Protection Information System (SISPRO, in the Spanish acronym). One of the components of SISPRO is the Individual Health Record System (Registro Individual de Prestación de Servicios [RIPS, in the Spanish acronym). The purpose of the RIPS is to centralize health information, facilitating the knowledge of the health or disease status of the population across the regions by time period, insurance regimen, gender and age, among other characteristics. The RIPS is publicly available and is reported on a monthly basis by healthcare providers, health maintenance organizations, and healthcare professionals.
The aim of the present study is to describe the geographical distribution of access to healthcare of patients with hypospadias in Colombia.
Methods
Data Collection
For the present observational, retrospective study, data were collected from the RIPS from 2014 to 2018, according to the International Statistical Classification of Diseases and Health Problems 10th revision (ICD-10) related to hypospadias (Q540, Q541, Q542, Q543, Q548, Q549). These diagnoses were scrutinized with two diagnostic filters: confirmed new and confirmed repeated cases, excluding the type of diagnosis, diagnostic impression, and unspecified. To quantify the number of patients with this diagnosis, the function “people attended” was used, which includes each person only once, even if that person has been attended more than once during the previous 5 years.
Analyzed data included all registered male births during the study period in each geographic area, according to vital statistics of births and deaths reported by the National Administrative Department of Statistics (DANE, in the Spanish acronym). For geographical analysis, the geolocation data (latitude and longitude) from each department was extracted from the geoportal of the National Administrative Department of Statistics, using their capital cities as a geographical reference.
To determine the availability of a specialist, we identified all of the members of the Colombian Urological Society. We considered pediatric urologists those who have a dedication of > 70% of their time to seeing urology pediatric patients.
Statistical Analysis
The information was recorded in Microsoft Excel for MacOS (Microsoft Corp., Redmond, WA, USA). Satscan v9.6 for MacOS (Satscan) was used to analyze the database, where the georeferenced population distribution values were obtained according to the geographic coordinates. Using a Poisson model, a geographical distribution of hypospadias in Colombia was made. To visualize the results obtained, the software projected the outcome in a Google Earth map.
Results
Between January 2014 and December 2018, a total of 8,990 cases (“people attended”) of hypospadias were assessed in the Colombian territory. [Table 1] shows the number of cases seen per year, in each department. The geographical distribution in the national territory has areas with high evaluation rates. On average, the departments where most cases were evaluated during the study period were Bogotá, D.C., Antioquia and Valle del Cauca (2,196, 1,818 and 1,151 cases, respectively).
Table 1
Number of people attended in the Colombian territory between 2014 and 2018
|
Hypospadias cases assessment[*]
|
|
2014
|
2015
|
2016
|
2017
|
2018
|
|
|
DEPARTMENT
|
|
|
|
|
|
TOTAL
|
|
Amazonas
|
1
|
3
|
0
|
1
|
1
|
6
|
|
Antioquia
|
326
|
334
|
289
|
382
|
487
|
1818
|
|
Arauca
|
9
|
6
|
3
|
6
|
10
|
34
|
|
Archipiélago de San Andrés, Providencia y Santa Catalina
|
3
|
2
|
1
|
0
|
1
|
7
|
|
Atlántico
|
232
|
227
|
139
|
173
|
194
|
965
|
|
Bogotá, D.C.
|
478
|
416
|
379
|
472
|
451
|
2196
|
|
Bolívar
|
149
|
125
|
76
|
109
|
117
|
576
|
|
Boyacá
|
53
|
50
|
35
|
38
|
36
|
212
|
|
Caldas
|
54
|
49
|
52
|
28
|
62
|
245
|
|
Caquetá
|
17
|
10
|
15
|
15
|
25
|
82
|
|
Casanare
|
10
|
17
|
13
|
13
|
12
|
65
|
|
Cauca
|
31
|
38
|
44
|
25
|
32
|
170
|
|
Cesar
|
118
|
141
|
134
|
159
|
125
|
677
|
|
Chocó
|
12
|
20
|
17
|
28
|
21
|
98
|
|
Córdoba
|
94
|
93
|
88
|
81
|
87
|
443
|
|
Cundinamarca
|
144
|
150
|
150
|
157
|
182
|
783
|
|
Guaviare
|
2
|
2
|
2
|
0
|
1
|
7
|
|
Huila
|
55
|
43
|
39
|
49
|
62
|
248
|
|
La Guajira
|
33
|
31
|
26
|
46
|
56
|
192
|
|
Magdalena
|
89
|
78
|
47
|
65
|
85
|
364
|
|
Meta
|
36
|
21
|
22
|
21
|
27
|
127
|
|
Nariño
|
47
|
54
|
42
|
59
|
57
|
259
|
|
Norte de Santander
|
54
|
52
|
34
|
54
|
59
|
253
|
|
Putumayo
|
5
|
6
|
7
|
10
|
6
|
34
|
|
Quindio
|
29
|
20
|
13
|
21
|
26
|
109
|
|
Risaralda
|
90
|
75
|
37
|
54
|
67
|
323
|
|
Santander
|
128
|
144
|
122
|
109
|
156
|
659
|
|
Sucre
|
88
|
68
|
38
|
45
|
56
|
295
|
|
Tolima
|
54
|
44
|
36
|
32
|
48
|
214
|
|
Valle del Cauca
|
233
|
239
|
193
|
225
|
261
|
1151
|
|
Vaupés
|
3
|
2
|
0
|
0
|
0
|
5
|
|
Vichada
|
2
|
1
|
0
|
0
|
0
|
3
|
* Reported as n.
Spatial scan statistical analysis identified a total of three clusters, as shown in [Figure 1]. The area with the highest concentration of cases is shown in red. The geographic distribution showed increasing trends in areas close to the center of the country, especially in the municipalities of Bogotá, Cali, Ibagué, and Pereira, which agrees with the area where more specialists are available. Meanwhile, clusters in blue shows those municipalities below the average value of cases of hypospadias assessed for the years between 2014 and 2018. The analysis detected areas in the north region of the country, including Barranquilla, Cartagena, Sincelejo, and Monteria, where the number of children evaluated was lower compared with the center of the country. The same occurs for the area delimited by the second cluster, which encompasses the Amazon and Orinoquia regions.
Fig. 1 Spatial-temporal analysis. Identified hypospadias clusters with increasing trends in cases assessed (red) and clusters with decreasing trends (blue).
Discussion
Main Findings
The present study provides data about the geographical distribution of the cases of hypospadias seen in Colombia from 2014 to 2018. These results show that there is a greater concentration of evaluated cases in the center of the country, where major cities are located with possible better access to a subspecialized medical care. This highlights inequities in access to health services and surgical care opportunity among the different regions of the country. To our knowledge, there are 11 full-time pediatric urologists in Bogotá, 2 in Bucaramanga, and 1 in Cali. This explains why the care of hypospadias patients is concentrated in the center of Colombia, making the access to children from other regions difficult. The concentration of resources is inequitable. Specifically for hypospadias, there should be a national system that controls referrals and, probably, a system in which specialized teams go to remote areas and treat these patients may be a cost-effective system. The variety of congenital anomalies in pediatric urology added to the prevalence of these conditions worldwide and the lack of access to trained specialists highlights the critical need to consider pediatric urology as a global surgical health priority.[14]
Taking into account the DANE vital statistics for 2017, congenital abnormalities were among the main causes of mortality in children < 1 year old, causing 23.4% of the deaths in our country.[15] The “Ten-Year Public Health Plan 2012-2021” (Plan Decenal de Salud Pública) includes the surveillance of birth defects as a fundamental element to guide actions aimed at reducing the morbidity and mortality of these conditions, preventing the consequences that represent a high cost for the family and the state.[16] Although the notification of these birth defects has increased in recent years, there are still limitations regarding the registration of cases, mainly because data are based on births that occur in hospitals, without taking into account the percentage of births that occur in rural areas.
Contrast with Literature
During the last decade, the prevalence of hypospadias has remained constant in Colombia (6.8 per 10,000 newborns).[3] International guidelines have suggested that the ideal age for hypospadias correction surgery is between 6 and 18 months old.[17] The average age at the moment of surgery for hypospadias patients is 7.9 years old in Colombia. Its main reasons are lack of awareness of how to refer patients, delays on administrative processes during referral, and limited manpower of subspecialized pediatric urologists within the national territory.[18] It is necessary to continue monitoring these patients to identify complications that may require reintervention in a timely manner as well as re-evaluating the psychological repercussions associated with this condition and long-term results of surgical interventions.[19] In a health system with limited resources such as ours, these patients are not prioritized because this is not considered a life-threatening condition.[20] Nonetheless, prompt surgical treatment prevents a secondary disability. Delaying it may increase the BoD.[21]
[22] There are also indirect implications. At our system, having a patient with a congenital anomaly creates an economic burden to the family of the patients, since one of the guardians needs to stay at home and care for the child, reducing familial income and creating more limitations to healthcare access.[18] A study conducted by Poenaru et al[23] collected surgical records of 13 different congenital anomalies from a high-income setting (Canada) and a low-income country (Kenya). The objective was to calculate DALYs (Disability Adjusted Life Years) caused by delayed treatment, taking into account the weights of the disability as well as the ideal and real age of surgery. The results showed that delayed surgery were longer in Kenya for most of the procedures included in the analysis. The greatest delays were mainly in children with intestinal and urological conditions. Regarding hypospadias repair, African children had a greater delay in treatment when compared with Canadian children (50.0 months versus 17.2 months, p < 0.0001), which translated into higher accumulated DALYS. Similar findings were reported by other groups, like that of Yousef et al.,[20] that reported surgical delays of 2.1 years for the treatment of the most common congenital anomalies, being the longest for hypospadias and cryptorchidism. These conditions are associated with significant disability weights and, therefore, are responsible for more than 75,000 DALYs. According to our results, cases presented in regions far from the center of the country may not be treated in a timely manner, which increases the burden of the disease and DALYS. Timely surgery avoids years of potential morbidity and reduces the risk of lifelong disability. DALYS increase with each year of treatment delay, and even surgical results may be worse as the age of the children increases for some congenital anomalies.[24]
According to the Global Burden of Disease Study, ∼ 11% of the global BoD is attributable to surgical conditions, including congenital malformations.[25] Birth defects are a global health problem with limited access to surgical services, mainly in low- and middle-income countries (LMICs).[26] Congenital abnormalities of the urinary tract represent a large proportion of these conditions. In Colombia, the incidence of genitourinary tract anomalies has been estimated as 43 per 100,000 live births.[27] Based on this information, hypospadias is the most common congenital anomaly in the Colombian territory, and the lack of a well-distributed health system will pose a burden to the system if this and other congenital anomalies are not treated appropriately. The long-term implications should be taken into account, and efforts to improve subspecialized medical access need to be implemented. Although universal healthcare coverage is a highlight of the Colombian system, conditions like hypospadias need to be referred to and managed at highly specialized centers. A recent study conducted by Hanna et al.[28] found that 7.1 million people (∼ 15.1% of the population) in Colombia do not have timely healthcare access. The most affected populations are found in the Amazon and Orinoquia regions, which supports our results. If we consider that the prevalence rates of hypospadias remain stable, if not increasing, over time, there is hypothetically the possibility that roughly 87% of the patients with hypospadias will never be assessed by a subspecialist if the distribution of patient care is reflected based on our results.
Limitations
Among the limitations of the present study, it is important to mention the difficulty related to data reporting by the RIPS, such as the inadequate register of the cases. Additionally, we did not consider pediatric surgeons who also treat these patients. Despite this limitations, the present study offers an overview of the distribution of access to health services for patients with a pathology that can be disabling.
Conclusion
There is a greater concentration of cases evaluated in the center of the country, where the cities with better access to subspecialized medical care are located. This highlights inequalities in health services and the opportunity for surgical care among regions of the country. If we consider that the prevalence rates of hypospadias remain stable, ∼ 87% of patients with hypospadias will not be evaluated by a subspecialist.
