Study contributions

Main results

Out of 2,147 grade 2 disability leprosy cases, the majority were male, of mixed race/skin color, multibacillary and borderline. The São Luís regional health unit showed a falling trend.

Implications for services

The results can guide strategies for the leprosy control program in the state, aiming at new approaches towards early diagnosis, treatment and prevention of disabilities.

Perspectives

Further studies are needed, such as spatial distribution of cases and detection rates of leprosy in children under 15 years of age, in order to gain a better understanding of the epidemiological profile of leprosy in Maranhão.

Keywords:
Leprosy; Epidemiology; Incidence; Degree of Physical Disability; Time Series Study

Abstract

Objective:

to describe the clinical and epidemiological characteristics of new cases of grade 2 disability leprosy and to analyze its trend in the state of Maranhão, from 2011 to 2020.

Methods:

this was a descriptive cross-sectional and ecological time-series study, using data from the Notifiable Health Conditions Information System. A descriptive analysis of the event was carried out according to the sociodemographic and clinical-laboratory characteristics of the cases. The temporal trend of event incidence was analyzed using Prais-Winsten regression.

Results:

of the 2,147 cases, 71.5% were male, 48.9% had up to 8 years of schooling, 66.5% were of mixed race/color, 95.5% had the multibacillary form, 58.8% were borderline, 32.3% had negative bacilloscopy at diagnosis. There was a stationary trend in the state and a falling trend in the São Luís Health Region (annual percentage change = -64.4%; 95% confidence interval: -73.7;-51.9).

Conclusion:

incidence trend was stable in the state of Maranhão and falling in São Luís.

Keywords:
Leprosy; Epidemiology; Incidence; Degree of Physical Disability; Time Series Study

Resumen

Objetivo:

describir las características clínicas y epidemiológicas de los nuevos casos de lepra con discapacidad física grado 2 y analizar su tendencia en el estado de Maranhão, de 2011 a 2020.

Métodos:

estudio transversal descriptivo y ecológico de serie temporal con datos del Sistema de Información de Agravamiento de Notificaciones. Se realizó un análisis descriptivo de características sociodemográficas y clínico-laboratoriales. La tendencia temporal de la incidencia de eventos se analizó mediante la regresión de Prais-Winsten.

Resultados:

de los 2.147 casos, 71,5% era masculino, 48,9% tenía hasta 8 años de escolaridad, 66,5% era de raza/piel parda, 95,5% de forma multibacilar, 58,8% dimorfa, 32,3% con baciloscopia negativa al diagnóstico. Hubo tendencia estacionaria en el estado y tendencia decreciente en la Región de Salud São Luís (variación anual = -64,4%; intervalo de confianza 95%: -73,7;-51,9).

Conclusión:

la tendencia de la incidencia se mantuvo estable en el estado de Maranhão y decreciente en São Luís.

Palabras-clave:
Lepra; Epidemiología; Incidencia; Grado de Discapacidad Física; Estudio de Series Temporales

INTRODUCTION

Leprosy is a chronic communicable disease; and it is neglected despite representing a public health problem in Brazil and worldwide. It is an infection of the skin and peripheral nerves, with granulomatous characteristics, and which has, as an etiological agent, the Mycobacterium Leprae bacillus.¹1. Kundakci N, Erdem C. Leprosy: a great imitator. Clin Dermatol. 2019;37(3):200-12. doi: 10.1016/j.clindermatol.2019.01.002
https://doi.org/10.1016/j.clindermatol.2... ^),(22. Maymone MBC, Laughter M, Venkatesh S, Dacso MM, Rao PN, Stryjewska BM, et al. Leprosy: clinical aspects and diagnostic techniques. J Am Acad Dermatol. 2020;83(1):1-14. doi: 10.1016/j.jaad.2019.12.080
https://doi.org/10.1016/j.jaad.2019.12.0... Also known as Hansen’s bacillus, it has an affinity for peripheral nerves and their infection can result in nerve damage with permanent physical disabilities, especially in the eyes, hands and feet, if there is no specific form of treatment.³3. Santos AR, Ignotti E. Prevenção de incapacidade física por hanseníase no Brasil: análise histórica. Cien Saude Colet. 2020;25(10):3731-44. doi: 10.1590/1413-812320202510.30262018
https://doi.org/10.1590/1413-81232020251...

Leprosy cases can be classified according to the number of skin patches or lesions and grade of disability (GD).⁴4. Ministério da Saúde (BR). Secretaria de Vigilância em Saúde. Departamento de Vigilância das Doenças Transmissíveis. Guia prático sobre a hanseníase. Brasília: Ministério da Saúde; 2017.^),(55. Thapa M, Kumaran MS, Narang T, Saikia UN, Sawatkar GU, Dogra S. A prospective study to validate various clinical criteriaused in classification of leprosy: a study from a tertiary care center in India. Int J Dermatol. 2018;57(9):1107-13. doi: 10.1111/ijd.14041
https://doi.org/10.1111/ijd.14041... A person with grade 2 disability (G2D) has significant impairment, visible in the eyes, hands and feet.³3. Santos AR, Ignotti E. Prevenção de incapacidade física por hanseníase no Brasil: análise histórica. Cien Saude Colet. 2020;25(10):3731-44. doi: 10.1590/1413-812320202510.30262018
https://doi.org/10.1590/1413-81232020251...

At the global level, in 2019, the rate of G2D leprosy diagnosis was 1.4 case/1 million inhabitants, reaching a total of 10,816 worldwide.⁶6. Organização Mundial da Saúde. Rumo à zero hanseníase: estratégia global de hanseníase 2021 - 2030. Genebra: Organização Mundial da Saúde; 2021. That was when the World Health Organization (WHO) established the goal of reducing rate of new cases with G2D to less than 1 case/1 million inhab., to be achieved by 2020.⁶6. Organização Mundial da Saúde. Rumo à zero hanseníase: estratégia global de hanseníase 2021 - 2030. Genebra: Organização Mundial da Saúde; 2021.

In Brazil, also in 2019, 2,351 new cases of G2D leprosy were reported, corresponding to an incidence rate of 11.2 cases/1 million inhabitants.⁸8. Instituto Brasileiro de Geografia e Estatística. Estimativas de População - EstimaPop [Internet]. Rio de Janeiro: Instituto Brasileiro de Geografia e Estatística; 2021 [citado 2021 Maio 2]. Disponível em: Disponível em: https://sidra.ibge.gov.br/pesquisa/estimapop/tabelas
https://sidra.ibge.gov.br/pesquisa/estim... In that year, the G2D incidence rate in the state of Maranhão was 31.5 new cases/1 million inhab., accounting for 223 cases of G2D leprosy.⁷7. Ministério da Saúde (BR). Secretária de Vigilância em Saúde. Boletim Epidemiológico Especial de Hanseníase [Internet]. Brasília: Ministério da Saúde ; 2021 [citado 2022 Mai 10]. Disponível em: Disponível em: https://www.gov.br/saude/pt-br/centrais-de-conteudo/publicacoes/boletins/epidemiologicos/especiais/2021/boletim-hanseniase-_-25-01.pdf
https://www.gov.br/saude/pt-br/centrais-... In view of data demonstrating late diagnosis of leprosy and active transmission of the disease,⁴4. Ministério da Saúde (BR). Secretaria de Vigilância em Saúde. Departamento de Vigilância das Doenças Transmissíveis. Guia prático sobre a hanseníase. Brasília: Ministério da Saúde; 2017.^),(77. Ministério da Saúde (BR). Secretária de Vigilância em Saúde. Boletim Epidemiológico Especial de Hanseníase [Internet]. Brasília: Ministério da Saúde ; 2021 [citado 2022 Mai 10]. Disponível em: Disponível em: https://www.gov.br/saude/pt-br/centrais-de-conteudo/publicacoes/boletins/epidemiologicos/especiais/2021/boletim-hanseniase-_-25-01.pdf
https://www.gov.br/saude/pt-br/centrais-... the need to understand G2D leprosy diagnosis is justified in Maranhão, throughout the 2010s, especially.

The objective of this research note was to describe the clinical and epidemiological characteristics of new cases of leprosy with G20 and to analyze its trend in the state of Maranhão, Brazil, from 2011 to 2020.

METHODS

This was both a descriptive cross-sectional study, aimed at the clinical-epidemiological characterization of new G2D leprosy cases, and also an ecological study, regarding the analysis of temporal trend in detection of new G2D cases in Maranhão, between 2011 and 2020. Maranhão comprises 217 municipalities and has a population of 7,153,262 inhab.⁸8. Instituto Brasileiro de Geografia e Estatística. Estimativas de População - EstimaPop [Internet]. Rio de Janeiro: Instituto Brasileiro de Geografia e Estatística; 2021 [citado 2021 Maio 2]. Disponível em: Disponível em: https://sidra.ibge.gov.br/pesquisa/estimapop/tabelas
https://sidra.ibge.gov.br/pesquisa/estim...

The ecological units analyzed were the state’s regional health units (RHU). The state of Maranhão is divided into 19 RHU: Açailândia, Bacabal, Balsas, Barra do Corda, Caxias, Chapadinha, Codó, Imperatriz, Itapecuru Mirim, Pedreiras, Pinheiro, Presidente Dutra, Rosário, Santa Inês, São João dos Patos, São Luís, Timon, Viana, and Zé Doca.⁹9. Secretaria do Estado de Saúde (MA). Unidades Regionais de Saúde [Internet]. São Luís: Secretaria do Estado de Saúde do Maranhão; 2021 [citado 2021 Dez 4]. Disponível em: Disponível em: https://www.saude.ma.gov.br/unidades-regionais-de-saude/
https://www.saude.ma.gov.br/unidades-reg...

The data used for the analysis were extracted from the Notifiable Health Conditions Information System (SINAN) on September 28, 2021, from database downloads in .cvs format files. The compulsory leprosy notification forms provide clinical and sociodemographic data, which were used as variables in this study.¹⁰10. Ministério da Saúde (BR). Secretaria de Vigilância em Saúde. Departamento de Vigilância Epidemiológica. O Sinan [Internet]. Brasília: Ministério da Saúde ; 2021 [citado 2021 Dez 10]. Disponível em: Disponível em: https://portalsinan.saude.gov.br/
https://portalsinan.saude.gov.br/... Leprosy cases with G2D at the time of diagnosis in the state of Maranhão in the period 2011-2020 were selected.

The variables selected for the descriptive analysis were:

a) sex (male; female);

b) age (at last birthday: 0-14; 15-29; 30-49; 50-59; 60 and over);

c) race/skin color (White; Black; Asian; mixed race; Indigenous; unknown);

d) schooling (illiterate; up to 8 years of schooling; over 8 years of schooling; not applicable; unknown);

e) clinical form (indeterminate; tuberculoid; borderline; virchowian; not classified; unknown);

f) operational class (multibacillary; paucibacillary);

g) bacilloscopy at diagnosis (positive; negative; not performed; unknown); and

h) number of lesions present (single lesion; 2-5; more than 5; not informed).

A descriptive analysis of case frequency distribution (absolute and percentage) according to sociodemographic and clinical-laboratory characteristics was performed. The new G2D leprosy case detection rate was obtained by dividing the number of new cases of G2D leprosy by the population residing in the area, in the same period, multiplied by 1 million.⁴4. Ministério da Saúde (BR). Secretaria de Vigilância em Saúde. Departamento de Vigilância das Doenças Transmissíveis. Guia prático sobre a hanseníase. Brasília: Ministério da Saúde; 2017. The trend in new G2D case detection was classified as rising, falling or stable, according to the Prais-Winsten regression coefficient value: a positive value indicates a rising trend; a negative value means a downward trend; and a null value or p-value without significance means a stable trend.¹¹11. Antunes JLF, Cardoso MRA. Using time series analysis in epidemiological studies. Epidemiol Serv Saude. 2015;24(3):565-76. doi: 10.5123/S1679-49742015000300024
https://doi.org/10.5123/S1679-4974201500... In order to test the statistical difference in the trend, annual percentage change (APC) was estimated, along with a 95% confidence interval (95%CI) and a 5% significance level. APC represents the average rate of change in disease incidence over a year, for each of the identified trends.¹¹11. Antunes JLF, Cardoso MRA. Using time series analysis in epidemiological studies. Epidemiol Serv Saude. 2015;24(3):565-76. doi: 10.5123/S1679-49742015000300024
https://doi.org/10.5123/S1679-4974201500... SPSS 24.0 software was used for statistical treatment of the data.

The study project was exempted from submission and appraisal by a Research Ethics Committee, since it was based exclusively on public domain data.

RESULTS

We assessed 2,147 cases of individuals diagnosed with leprosy and G2D treated in the state of Maranhão, within the proposed time frame.

Regarding case description, the majority were male (71.5%) and of mixed race/skin color (66.5%), in the multibacillary operational class (95.5%) and with the borderline clinical form (58.8%). Also noteworthy were the schooling of those affected with up to 8 years of complete study (48.9%), presence of more than 5 lesions (48.4%), age above 60 years (33.2%) and negative bacilloscopy at diagnosis (32.3%), as shown in Table 1.

The new G2D case detection rate was 35.2 cases/1 million inhab. in 2011, and 15.7/1 million inhab. in 2020 (Table 2). The new G2D case detection trend in the state proved to be stationary in the period studied (APC = -27.4%; 95%CI -53.3;13.0; p-value = 0.150) and in almost all the RHU : the exception was the São Luís RHU, where a falling G2D trend was found (APC = -64.4%; 95%CI -73.7;-51.9; p-value < 0.001).

DISCUSSION

Detection of new G2D leprosy cases showed a stable trend for state of Maranhão as a whole, while the São Luís RHU showed a falling trend. In the state of Maranhão, this form of infection has been hyperendemic. Besides causing serious physical limitations to the individual, G2D can revive stigmas and social prejudices associated with the disease for thousands of years.⁴4. Ministério da Saúde (BR). Secretaria de Vigilância em Saúde. Departamento de Vigilância das Doenças Transmissíveis. Guia prático sobre a hanseníase. Brasília: Ministério da Saúde; 2017.^),(66. Organização Mundial da Saúde. Rumo à zero hanseníase: estratégia global de hanseníase 2021 - 2030. Genebra: Organização Mundial da Saúde; 2021.^),(77. Ministério da Saúde (BR). Secretária de Vigilância em Saúde. Boletim Epidemiológico Especial de Hanseníase [Internet]. Brasília: Ministério da Saúde ; 2021 [citado 2022 Mai 10]. Disponível em: Disponível em: https://www.gov.br/saude/pt-br/centrais-de-conteudo/publicacoes/boletins/epidemiologicos/especiais/2021/boletim-hanseniase-_-25-01.pdf
https://www.gov.br/saude/pt-br/centrais-...

A higher proportion of leprosy cases with G2D was identified in males. A hypothesis has been raised that the higher frequency of late diagnosis of the disease in males is due to the lower number of men seeking care in health services.¹²12. Silva JSR, Palmeira IPP, Sá AMM, Nogueira LMV, Ferreira AMR. Fatores sociodemográicos associados ao grau de incapacidade física na hanseníase. Rev Cuid. 2018;9(3):2338-48. doi: 10.15649/cuidarte.v9i3.548
https://doi.org/10.15649/cuidarte.v9i3.5... Another hypothesis for this finding would be testosterone itself, the predominant male sex hormone, which stimulates the T helper 2 immune response, which is the main response in the multibacillary class, in which G2D is more predominant.¹³13. Nobre ML, Illarramendi X, Dupnik KM, Hacker MA, Nery JAC, Jerônimo SM, et al. Multibacillary leprosy by population groups in Brazil: lessons from an observational study. Plos Negl Trop Dis. 2017;11(02):e0005364. doi: 10.1371/journal.pntd.0005364
https://doi.org/10.1371/journal.pntd.000...

Leprosy cases with G2D were predominant in individuals aged over 60 years. Leprosy is a chronic disease and degree of disability is related to the time course of morbidity. In the elderly individual, therefore, the chances of physical disabilities and their severity are greater.¹⁴14. Souza CDF, Fernandes TRMO, Matos TS, Ribeiro Filho JM, Almeida GKA, Lima JCB, et al. Physical disability degree in the elderly population affected by leprosy in the state of Bahia, Brazil. Act Fisiátr. 2017;24(1):27-32. doi: 10.1016/j.abd.2019.01.011
https://doi.org/10.1016/j.abd.2019.01.01... ^),(1515. Sanchez MN, Nery JS, Pescarini JM, Mendes AA, Ichihara AY, Teixeira CSS, et al. Physical disabilities caused by leprosy in 100 million cohort in Brazil. BMC Infect Dis. 2021;21(1):290. doi: 10.1186/s12879-021-05846-w
https://doi.org/10.1186/s12879-021-05846...

This analysis showed a higher frequency of G2D cases in individuals with low schooling. There is evidence that lower levels of schooling contribute to a decrease in self-care and a reduction in the demand for care in health services, favoring the transmission of leprosy and the development of physical disabilities.¹⁶16. Moschioni C, Antunes CMF, Grossi MAF, Lambertucci JR. Risk factors for physical disability at diagnosis of 19,283 new cases of leprosy. Rev Soc Bras Med Trop. 2010;43(1):19-22. doi: 10.1590/S0037-86822010000100005
https://doi.org/10.1590/S0037-8682201000... ^)-(1818. Leano HAM, Araújo KMFA, Bueno IC, Niitsuma ENA, Lana FCF. Fatores socioeconômicos relacionados à hanseníase: revisão integrativa da literatura. Rev Bras Enferm. 2019;72(5):1474-85. doi: 10.1590/0034-7167-2017-0651
https://doi.org/10.1590/0034-7167-2017-0...

There was a higher proportion of cases classified as multibacillary and with the borderline clinical form. The multibacillary classification, which the borderline form falls into, is identified as a risk factor for physical disabilities. This aspect of multibacillary classification and diagnosis of physical disability points to late diagnosis.¹⁹19. Morais JR, Furtado EZL. Grau de incapacidade física de pacientes com hanseníase. Rev Enferm Ufpe On Line. 2018;12(6):1625-32. doi: 10.5205/1981-8963-v12i6a231049p1625-1632-2018
https://doi.org/10.5205/1981-8963-v12i6a... ^),(2020. Moreira RJO, Fernandes ALP, Dutra RP, Ferreira AGN, Santos Neto M, Santos LH. Fatores associados às incapacidades físicas da hanseníase: revisão integrativa. Rev Cereus. 2022;14(1):23-38. doi: 10.18605/2175-7275/cereus.v14n1p23-38
https://doi.org/10.18605/2175-7275/cereu...

Regarding bacilloscopy, although there were fewer positive results when compared to negative ones, the risk of cases of leprosy with positive bacilloscopy at diagnosis presenting physical disabilities related to leprosy is two times greater.²¹21. Silva JSR, Palmeira IP, Sá AMM, Nogueira LMV, Ferreira AMR. Variáveis clínicas associadas ao grau de incapacidade física na hanseníase. Rev Cuid . 2018;10(1):1-12. doi: 10.15649/cuidarte.v10i1.618
https://doi.org/10.15649/cuidarte.v10i1....

There was a higher proportion of G2D leprosy cases with more than 5 lesions. The presence of more than 5 lesions in a person with leprosy is a characteristic of the multibacillary operational classification, which is a risk factor for the development of physical disabilities due to the disease.⁴4. Ministério da Saúde (BR). Secretaria de Vigilância em Saúde. Departamento de Vigilância das Doenças Transmissíveis. Guia prático sobre a hanseníase. Brasília: Ministério da Saúde; 2017.^),(1919. Morais JR, Furtado EZL. Grau de incapacidade física de pacientes com hanseníase. Rev Enferm Ufpe On Line. 2018;12(6):1625-32. doi: 10.5205/1981-8963-v12i6a231049p1625-1632-2018
https://doi.org/10.5205/1981-8963-v12i6a... ^),(2020. Moreira RJO, Fernandes ALP, Dutra RP, Ferreira AGN, Santos Neto M, Santos LH. Fatores associados às incapacidades físicas da hanseníase: revisão integrativa. Rev Cereus. 2022;14(1):23-38. doi: 10.18605/2175-7275/cereus.v14n1p23-38
https://doi.org/10.18605/2175-7275/cereu...

There was stability in the detection trend of new cases of leprosy with grade 2 physical disability, in almost all RHU in the state of Maranhão; the only exception was the São Luís RHU, where this trend was found to be falling. The stationary trend in the state of Maranhão as a whole and the falling trend in São Luís can be attributed to the actions of the Family Health Strategy and its teams in the fight against leprosy, which has been in place since 1994.²²22. Penna MLF, Gross MAF, Penna GO. Country profile: leprosy in Brazil. Lepr Rev. 2013;84(4):308-15. From the year 2000 onwards, the effectiveness of the Family Health Strategy in Maranhão led to an increase in the number of diagnosed leprosy cases and, consequently, an increase in all related indicators, followed by a progressive drop in their rates.²²22. Penna MLF, Gross MAF, Penna GO. Country profile: leprosy in Brazil. Lepr Rev. 2013;84(4):308-15.

As limitations of this study, we highlight (i) possible underreporting of leprosy in the state of Maranhão and (ii) possible errors in filling out notification forms, given that the analysis includes only secondary data, which could compromise, mainly, the description of the clinical-epidemiological profile of the cases. Errors in filling out data are due to factors such as health worker lack of knowledge and overburdening, in addition to information system shortcomings.¹⁴14. Souza CDF, Fernandes TRMO, Matos TS, Ribeiro Filho JM, Almeida GKA, Lima JCB, et al. Physical disability degree in the elderly population affected by leprosy in the state of Bahia, Brazil. Act Fisiátr. 2017;24(1):27-32. doi: 10.1016/j.abd.2019.01.011
https://doi.org/10.1016/j.abd.2019.01.01...

We conclude that this study described the clinical and epidemiological profile of new cases of leprosy with G2D in the state of Maranhão, whereby there was a predominance of males, mixed race/skin color, age over 60 years, up to 8 years of schooling, multibacillary classification and borderline clinical form, as well as negative bacilloscopy at diagnosis. A stable trend was identified for G2D leprosy in the state, in general, while a falling trend was only found for the São Luís RHU. We recommended reinforcing active tracing aimed at achieving early diagnosis as the best approach to leprosy, in the sense of early diagnosis, treatment and prevention of disabilities.

REFERENCES

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