ABSTRACT.
Children who live in leprosy-endemic areas are susceptible to infection due to early and frequent exposure to Mycobacterium leprae. Indonesia is on the verge of eliminating this disease (prevalence rate < 1/10,000 population), but pediatric leprosy continues to occur in low-endemic areas. This study aimed to evaluate pediatric leprosy over a decade in a tertiary hospital in Surabaya, Indonesia. A retrospective study of leprosy in children under 15 years old between 2010 and 2019 was conducted in the Morbus Hansen Division, Outpatient Clinic at Dr. Soetomo Hospital in Surabaya, Indonesia. Seventy pediatric leprosy cases were identified between 2010 and 2019, consisting of 58 multibacillary (MB)-type cases and 12 paucibacillary (PB)-type cases. Slit skin smear (SSS) was positive in 26 cases. There were two cases of grade-2 disability and 15 cases of leprosy reaction (erythema nodosum leprosum) in children at the time of diagnosis. There was an insignificant decline in the number of pediatric leprosy cases in the last 10 years. Cases and disabilities in children were found in some leprosy pocket areas even though the national elimination rate has been achieved. MB infections, disability, and treatment defaults were common problems in pediatric leprosy.
INTRODUCTION
Hansen’s disease (HD), or commonly known as leprosy, is an infectious disease caused by Mycobacterium leprae1 and M. lepromatosis,2 transmitted through droplets and close contact with infected individuals.3 The peripheral nerves engagement and cutaneous lesions are the primary indications that might worsen without early detection and treatment due to the nervous system’s involvement and its association with acute disability.1 Leprosy occurs in children and adults. The clinical symptoms vary according to the immunosuppressive conditions or other immune system disorders.4 Moreover, pediatric leprosy cases are an important indicator to assess leprosy transmission. To mitigate this disease, the World Health Organization (WHO) had launched The Global Leprosy Strategy (2016–2020) to achieve zero pediatric leprosy cases and second-degree disability by 2020.5 Health education and information dissemination about pediatric leprosy are still highly recommended to reduce the severity of the disease and increase early detection of the infection.
Indonesia ranks second in pediatric leprosy cases after India.6 Despite the global decrease in the number of children affected by leprosy in 2018, Indonesia had more cases from 1.755 to 1.861 cases, where 33 cases were accompanied by a grade-2 disability. Indonesia succeeded in achieving leprosy elimination rates in 2000, although East Java, the province with the highest number of leprosy patients in the country, managed to achieve the elimination rate much later in 2017. The proportion of affected children in East Java was under 5% between 2009 and 2016. Meanwhile, Surabaya was still an endemic leprosy city in 2017, recording pediatric leprosy cases at 8%.7
Various case studies reported cases of multibacillary (MB) in children, leprosy reactions, disability, and defaulted treatment in the pediatric leprosy.7 However, only a few reported pediatric leprosy cases in tertiary hospitals in Indonesia in the last 10 years. Thus, this study investigated the demography, clinical epidemiology, and any related problems of pediatric leprosy cases at Dr. Soetomo Hospital, Surabaya, East Java.
MATERIALS AND METHODS
This retrospective study was conducted using medical records of under-15-year-old leprosy patients registered in the Morbus Hansen Division of Dr. Soetomo Hospital, a tertiary teaching hospital and a national reference hospital for East Indonesia. The data were collected from January 2010 to December 2019. After a thorough examination, the information on gender, race, age, residency, leprosy contacts, types of leprosy, total cases of leprosy, and the clinical features of leprosy was extracted from the records, in consideration of the complete history and physical examinations of patients.
The leprosy type was classified according to the hospital’s diagnosis and therapy guidelines, such as the presence of symptoms and cardinal signs, including 1) the presence of patches on the skin accompanied by numbness, 2) enlargement of peripheral nerves, and 3) a routine slit skin smear (SSS) test. Based on the leprosy diagnosis, the cases were categorized as paucibacillary (PB) or MB. A patient was classified as PB if he/she had at most five (≤ 5) skin patches with or without one to two thickened major cutaneous nerves. By contrast, patients with at least six (≥ 6) skin patches and/or more than two (> 2) thickened nerves with infiltrations, with/without papules or nodules, and smear-positive cases were classified as MB.1 Finally, the collected data were analyzed using the Statistical Package for Social Sciences (SPSS) version 16.0 (IBM, USA). The demography and clinical epidemiology information were presented descriptively.
RESULTS
Table 1 presents the annual pediatric leprosy cases compared with adult cases in Dr. Soetomo Hospital between 2010 and 2019. A total of 70 new pediatric leprosy cases were identified out of the 1.319 cases within a decade. The lowest percentage of pediatric leprosy cases was recorded in 2016 (3%), whereas the highest rate was recorded in 2012 (7.74%).
Table 1.
Child and adult leprosy case comparison in 2010–2019 at Dr. Soetomo Hospital, Indonesia
| Year | Pediatric leprosy | Adult leprosy | Grand total | Pediatric leprosy rate (%) | ||||
|---|---|---|---|---|---|---|---|---|
| PB | MB | Total | PB | MB | Total | |||
| 2010 | 1 | 5 | 6 | 12 | 135 | 147 | 153 | 3.92 |
| 2011 | 1 | 6 | 7 | 12 | 122 | 134 | 141 | 4.96 |
| 2012 | 0 | 11 | 11 | 9 | 122 | 131 | 142 | 7.75 |
| 2013 | 0 | 5 | 5 | 3 | 136 | 139 | 144 | 3.47 |
| 2014 | 3 | 6 | 9 | 8 | 109 | 117 | 126 | 7.14 |
| 2015 | 3 | 6 | 9 | 10 | 128 | 138 | 147 | 6.12 |
| 2016 | 0 | 3 | 3 | 5 | 93 | 98 | 101 | 2.97 |
| 2017 | 3 | 6 | 9 | 6 | 122 | 128 | 137 | 6.57 |
| 2018 | 0 | 4 | 4 | 1 | 119 | 120 | 124 | 3.22 |
| 2019 | 1 | 6 | 7 | 1 | 96 | 97 | 104 | 6.73 |
| Total | 12 | 58 | 70 | 67 | 1,182 | 1,249 | 1,319 | 5.31 |
MB = multibacillary; PB = paucibacillary.
Our study showed that the cases were more frequently observed in boys. However, the PB type was more common among girls (Table 2). The youngest patient was 4 years old and the oldest one was 14 years old. The results showed that leprosy contacts in one house were detected in 25 cases, consisting of 20 MB-type and 5 PB-type.
Table 2.
Demographic and epidemiological characteristics of pediatric leprosy cases (N = 70)
| PB (N = 12) | MB (N = 58) | |
|---|---|---|
| Gender | ||
| Male | 5 (41.7%) | 37 (63.8%) |
| Female | 7 (58.3%) | 21 (36.2%) |
| Age | ||
| 4–9 years old | 5 | 13 |
| 10–14 years old | 7 | 45 |
| Leprosy Contact | ||
| Yes | 5 | 20 |
| No | 7 | 38 |
MB = multibacillary; PB = paucibacillary.
Table 3 demonstrates the clinical features of the cases. Long clinical symptoms (exceeding 24 months) were found in both categories, two cases in PB-type and 12 in MB-type. Besides, peripheral nerve enlargement was mostly observed in the ulnar nerve (two cases in PB-type and 15 in MB-type). The SSS examination was positive in 26 MB-type cases. Additionally, grade 1 deformity was found in both types (one PB-type and eight MB-type), while grade 2 deformity on early diagnosis was found in MB-type (two cases). Apart from that, type 1 (seven cases) and type 2 (eight cases), leprosy reactions were identified only in MB-type. Moreover, two PB patient and 20 MB patients did not get adequate treatment. Meanwhile, 10 patients were treated at health primary care.
Table 3.
Clinical characteristics of pediatric leprosy cases (N = 70)
| PB (N = 12) | MB (N = 58) | |
|---|---|---|
| Duration of symptoms | ||
| ≤ 24 months | 10 | 46 |
| 2 | 12 | |
| Peripheral nerve enlargement | ||
| N. Auricularis magnus | 2 | 7 |
| N. Ulnaris | 2 | 15 |
| N. Peroneus | 1 | 6 |
| N. Tibialis posterior | 0 | 5 |
| Slit skin smear | ||
| Positive 1 | 0 | 6 |
| Positive 2 | 0 | 9 |
| Positive 3 | 0 | 7 |
| Positive 4 | 0 | 4 |
| Negative | 12 | 32 |
| Disability | ||
| Grade 1 | 1 | 8 |
| Grade 2 | 0 | 2 |
| Reaction | ||
| Type 1 reaction | 0 | 7 |
| Type 2 reaction | 0 | 8 |
| Therapy | ||
| Complete | 9 | 29 |
| Incomplete | 2 | 20 |
| Others* | 1 | 9 |
MB = multibacillary; PB = paucibacillary.
*Others: 10 patients were treated at the Health Center of origin.
DISCUSSION
This study presented an overview of the current pediatric leprosy situation as a progress assessment toward a world free of leprosy. Although there was a case number decrease in the last decade, a plateau in pediatric leprosy cases indicated a continuous, active transmission of this disease within the community. However, since the data used in this study were collected from a tertiary hospital, they were not a true reflection of the country’s actual pediatric leprosy situation. Case detection studies in the field indicated a higher prevalence and proportion of pediatric leprosy than the recorded prevalence.8
Furthermore, the proportion of pediatric leprosy cases increased in the last 10 years, where 3.9% of cases were recorded in 2010 and then increased to 6.7% in 2019. Nevertheless, these values were considerably lower than in India, with 9.6% (2010) and 11.2% (2019) of child cases in tertiary hospitals.9,10 In addition, individual immune responses and genetic factors could influence the occurrence of leprosy in children. Most of the affected children were between 10 and 14 years of age, indicating that this disease was more common in older children. This is similar to other previously conducted studies, where older children (11–14 years) dominated in the recorded cases, accounting for up to 70.3%9 and 71.2%.10 This observation may be attributed to the incubation period being linked to close and long-term contact.11 However, the youngest leprosy patient in the selected hospital was 4 years old.
Moreover, there were conflicting findings in terms of gender, where several studies reported that males were more susceptible to leprosy than females1 and others reported varying results.9 This gender tendency can be related to the cultural and socio-economic factors in the study area, where women tend to delay in seeking treatment11 and use nonmedical therapy in the long term.1 Various studies showed the existence of gender differentiation in the immune response to M. leprae infection. One of the reasons is that men experience environmental exposure more often than women. In addition, sex hormones also play a role in the host’s immune response to various infections. However, most of the studies were conducted on adult patients or postpubertal children. Thus, gender differentiation in children has not been well explained.12,13
It was observed that household contact history was recorded in 35% of patients (five PB patients and 20 MB patients). However, their contacts’ leprosy type could not be found in the children’s medical records. The presence of close contacts within the community increases the risk of leprosy transmission by four times and the risk becomes nine times higher in-home contact.14 Therefore, children living in households with infected contacts face a higher risk because of the closer proximity with the primary source of leprosy transmission.
The number of MB-type cases was 58, much larger than the PB-type, which was 12 cases. The number of MB cases in East Java shows a higher number when compared with previous studies. In reports of pediatric leprosy at a tertiary hospital in India, the number of MB cases was 51.7% and 52.5%, respectively.6,15
MB-type leprosy in children requires special attention because of its higher bacterial index (BI), increased risk of transmission, and associated with deformity and disability. This condition is linked to nerve enlargement, which is usually observed at the ulnar nerve. Peripheral nerve enlargement is threatening because it contributes to the disability. Furthermore, the high number of MB cases in this study might be linked to the hospital setting; more MB cases were reported in studies conducted at tertiary and referral hospitals in India.16,17
Early detection and multidrug treatment (MDT) are the basis to control leprosy as recommended by WHO.5 In this study, 14 patients had experienced leprosy symptoms for 24–36 months before properly diagnosed. Grade-2 disability in children was found in 2 MB cases, while grade 1 disability was found in 1 PB case and 8 MB cases. The patients with grade 2 disability were both 10 years old. One had the symptoms for more than 24 months while the other had it for more than 4 months with the BI of +6 and +1, respectively. On the other hand, seven cases in grade 1 disability had complaints for more than 24 months. This is in accordance with Darlong et al.,18 who stated that disability could occur in children and was more frequently in MB type with positive BI.
There were 15 cases (21.4%) accompanied by type 1 or type 2 reactions. Leprosy reactions are not uncommon in children; a similar study at the Tertiary Hospital reported the incidence of leprosy reactions in children varying from 18.6% to 33.9%.6,15
Slit skin smear examination remains as one of the relevant tools routinely carried out to diagnose leprosy in local hospitals, although a microscopic examination is no longer compulsory. A total of 27 positive BI results were obtained upon the SSS examination, varying from absence to +4. Positive Acid-Fast Bacteria (AFB) indicated that MB cases in children could be accompanied by a high bacterial load, making them a potential transmission source. Most importantly, the SSS and microscopic examination are applicable because of their simplicity, low cost, and almost 100% specificity if carried out by skilled laboratory personnel.19
We found 22 (31.4%) children who experienced defaulted treatment. This observation is higher than several studies that reported the defaulted treatment rates in children ranging from 10% to 20%14 due to the prolonged treatment and difficult-to-consume drug preparation, especially for younger children.14 This situation is dangerous, especially for MB cases, because incomplete therapy can result in the therapeutic dose not being achieved, which leads to drug resistance. Therefore, this condition could cause therapeutic failure and potentially disrupts the leprosy control program.20,21
Despite reaching an elimination rate, this program should continue in Surabaya for continuous monitoring. Based on the study at Dr. Soetomo Hospital, Indonesia’s hope to achieve the WHO target of a leprosy-free world by 2020 calls for more substantial effort. The limitations of this study were the usage of medical records as data rather than direct observation and only one tertiary hospital was involved in this study.
CONCLUSION
Pediatric leprosy was still found in East Java; however, the prevalence of leprosy decreased at the national level. Our study provided an overview of the clinical and bacteriological characteristics in cases of pediatric leprosy and observed that the number of child cases fluctuated in the last 10 years. This condition shows that leprosy transmission still occurs in the community. In addition, high MB-type cases, disability, and leprosy reactions are still present in children. Health education and information dissemination about pediatric leprosy are highly recommended to reduce the disease’s severity and increase early detection.
This project is a part of Indonesia’s leprosy study, especially in the northern coast of Java as an area with highly reported leprosy cases. In addition to studies in pediatric groups, a similar study will also be conducted in adult and geriatric leprosy patients in Indonesia.
ACKNOWLEDGMENTS
We would like to thank the Indonesian Endowment Fund for Education (LPDP), staff, and residents in Dr. Soetomo Hospital.
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