|Ahead of print publication
A retrospective clinico-epidemiological study of leprosy cases treated at a tertiary care hospital in Western Maharashtra
Deepak Vashisht1, Prerna Shankar2, Vikas Pathania1, Surabhi Sharma2, Sunmeet Sandhu3, Ruby Venugopal1
1 Department of Dermatology, Command Hospital, Pune, Maharashtra, India
2 Department of Community Medicine, Armed Forces Medical College, Pune, Maharashtra, India
3 Department of Dermatology, Armed Forces Medical College, Pune, Maharashtra, India
|Date of Submission||29-May-2020|
|Date of Decision||17-Aug-2020|
|Date of Acceptance||21-Sep-2020|
Station Health Organization, Pune - 411 040, Maharashtra
Source of Support: None, Conflict of Interest: None
Background: Leprosy is a chronic communicable disease caused by Mycobacterium Leprae. Despite the multidrug regimen against this formidable pathogen for nearly four decades, leprosy remains a public health scourge. The World Health Organization has intensified its efforts to eliminate leprosy by launching “Global Leprosy Strategy 2016–2020. Notwithstanding, India had accounted for 60% of new cases globally in 2016. Aims: The aim of this study is to describe the clinical and epidemiological spectrum of leprosy patients encountered at a tertiary care center in Western Maharashtra. Settings and Design: Record-based, retrospective, descriptive study. Methods: Case records of leprosy patients treated at a tertiary care hospital over 10 years were studied. Two hundred and thirteen cases that fulfilled the World Health Organization's 1998 case definition of leprosy and whose case records were replete with a basic demographic, case history, examination, and treatment details were included in the study. Statistical Analysis: Data were compiled in MS Excel and analyzed with the SPSS statistical software version 20. Results: Majority (87.3%) cases were multibacillary leprosy. A significant number of patients had borderline tuberculoid leprosy (71.5%). Meantime taken for the diagnosis, i.e., time taken from the onset of symptoms to diagnosis was 271.74 days. Contact tracing could be elicited in only 1.4% of cases. A light-colored numb patch was the most common clinical presentation in 79% of patients. 96.7% of patients had peripheral nerve thickening, of which, the ulnar nerve was the most frequently involved (93.5%). Ninety-nine (46.5%) cases had documented leprosy reactions. Grade 2 disability accounted for 23% cases with claw hand as the most common deformity in17.4%. Conclusions: The present study provides an insight into disease burden as well as the effectiveness of health services at a tertiary care hospital in Western Maharashtra. The study also highlights the importance of early diagnosis and management of leprosy and reactions, thereby minimizing deformities and disabilities.
Keywords: Deformity, epidemiology, eradication, Hansen's disease, leprosy
|How to cite this URL:|
Vashisht D, Shankar P, Pathania V, Sharma S, Sandhu S, Venugopal R. A retrospective clinico-epidemiological study of leprosy cases treated at a tertiary care hospital in Western Maharashtra. Med J DY Patil Vidyapeeth [Epub ahead of print] [cited 2021 Jun 12]. Available from: https://www.mjdrdypv.org/preprintarticle.asp?id=316414
| Introduction|| |
Leprosy, also known as Hansen's disease, is a chronic infectious disease caused by Mycobacterium leprae. The disease mainly affects the skin, the peripheral nerves, mucosal surfaces of the upper respiratory tract, and the eyes. M. leprae is a tardy multiplier and the incubation period of the disease is 5 years on an average. Efforts to eliminate leprosy have been intensified with the Global Leprosy Strategy 2010–2020. According to the global health observatory data repository, India had 120334 new cases detected in 2018. The new case detection rate in the Indian scenario shows >10/100,000 population which is on an alarming side. The current prevalence rate corresponds to 0.66/10,000. The main challenge in achieving this goal is the presence of highly endemic pockets in a few countries, including India. The prolonged incubation period and late onset of symptoms postinfection have been cited as the main reasons for delayed diagnosis. In addition, the stigma, prejudice, and discrimination against leprosy patients are still prevalent in India. The delay in the diagnosis predisposes the patient to inevitable morbidity in the form of deformities and disabilities, thus perpetuating a vicious cycle.
Current statistics show that leprosy continues to be a public health problem in India despite free treatment and continuous efforts of the government. India and Brazil account for the maximum burden of disease. National Leprosy Eradication Programme (NLEP) in India is working toward the goal of leprosy eradication. However, despite the deployment of well thought out strategies, there are the pockets of high prevalence in India. India achieved a prevalence rate of <1/10,000 population at the national level on January 1, 2006, yet there are pockets of high prevalence in a few states. Previous studies in the state of Maharashtra have concluded an ineffective outreach leaving disease hotspots across the region. Reasons implicated for such inconsistency are a high migration rate and a dense tribal population devoid of effective health-care infrastructure. The current scenario demands collaboration at various geographical regions, health-care sectors, public and private sectors to address the imminent resurfacing of new leprosy cases.
The present study aims to give an insight into the clinical and epidemiological profile of leprosy patients at a tertiary care center in Western Maharashtra.
| Methods|| |
This study was conducted at a tertiary care center in Western Maharashtra with a mixed urban and rural population. Case records of leprosy patients treated at a tertiary care hospital over 10 years from 2008 to 2018 were studied retrospectively. All cases that fulfilled the case definition of leprosy (WHO 1998) were included. Case records which were complete concerning basic demographic data, history (onset, detection, and contact history), examination details (lesion count, nerve involvement, deformities, and reactionary episodes), treatment details (the type of multidrug treatment), and laboratory investigations (slit skin smears and histopathology) were included in the study. Ethical approval for the study was obtained from the Institutional Review Board (letter No. IECS No. IEC/2018/58 dated August 07, 2018). The data were compiled in MS Excel fFormat and analyzed using the Statistical Package for the Social Sciences (SPSS) version 20 (SPSS Inc. Released 2018, Chicago, IL, USA) using the appropriate statistical technique.
| Results|| |
A total of 213 cases of leprosy with age ranging from 22 to 79 years (mean age 35.0 ± 10.28 years) were treated during the period and included in the present study. The maximum disease burden (77.5%) was seen in the age group of 21–40 years [Figure 1]. The study population consisted of 154 (72.3%) males and 59 (27.7%) females. Male: female ratio was 2.6:1 history of contact was elicited in 03 cases (1.4%).
The most common clinical presentation was a light-colored numb patch in 79% of patients followed by raised lesions of erythema nodosum leprosum (ENL) and weakness of limbs in 08% cases each [Figure 2]. Epidemiologically, there was a steady increase in the number of cases, peaking in 2013 thereafter plateauing till 2016 and a declining trend subsequently [Figure 3]. Ninety-seven (45.5%) of the patients presented with one to three skin lesions, and ≥50 lesions were observed in 10.8% cases [Table 1]. Two hundred and six (96.7%) cases had thickening of nerves with mononeuropathy in 43 (20.8%), whereas 07 cases had no thickened nerves. The most common thickened nerve was the ulnar nerve (93.5%) followed by the median nerve (44%) and radial nerve (38.4%) in upper limbs and superficial peroneal nerve (47%) in lower extremities. Bilateral ulnar nerve involvement was seen in 58.3% of cases.
One hundred and eighty-six (87.3%) cases were multibacillary (MB) leprosy as per the WHO case definition of 1988. Paucibacillary to the MB case ratio was 1:6.9. [Figure 4] shows the clinical spectrum of cases seen in the study. Borderline tuberculoid (BT) was the most common pole of leprosy in 153 (71.5%) cases. Pure neuritic leprosy (PNL) and histoid leprosy constituted 10% and 1.4% cases, respectively. Meantime taken for the diagnosis of leprosy from the onset of symptoms was around 271.74 days with the longest duration of disease for indeterminate leprosy with 592.50 (199.5) days [Table 2].
|Figure 4: Clinical spectrum of leprosy cases in study population. (a) Hansen's disease (Borderline tuberculoid). (b) Borderline Leprosy showing central clearing (Borderline borderline). (c) Hansens disease (Borderline Lepromatous). (d) Lepromatous Leprosy showing facial infiltration with madarosis. and (e) Histoid Leprosy|
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|Table 2: Distribution of cases as per pole of leprosy, bacteriological index, reactions and time taken for diagnosis (n=213)|
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Ninety-nine (46.5%) cases had documented leprosy reactions. [Figure 5] shows the spectrum of lepra reactions seen in the study. Type 1 reaction (T1R) seen in 79 (79.8%) cases was more common than Type 2 reaction (T2R) seen in 20 (20.2%) patients [Table 2]. Among patients with T1R, 22 (27.9%) cases had more than 1 episodes of T1R during the treatment period. All these 22 patients were initially managed with tapering doses of oral steroids for ≤12 weeks and >20 weeks of oral steroids in subsequent T1R with no recurrence. Remaining 57 cases with initial T1R without any recurrence was managed with tapering doses of oral steroids (started @ 1 mg/kg/day) with a duration of >20 weeks. Of the 20 cases with T2R, 17 (85%) had ENL at initial presentation while 3 (15%) developed ENL within 60 days of treatment initiation of which 02 cases were of histoid leprosy. Eighteen (90%) patients with ENL were managed with thalidomide and 02 with oral steroids with mean treatment duration for the reaction of 240 ± 53.8 days. Five cases had recurrent ENL lesions during the entire treatment period.
|Figure 5: Clinical spectrum of lepra reactions in study population. (a) Type II Lepra Reaction (erythema nodosum leprosum). (b) Type II Lepra reaction (erythema nodosum leprosum necroticans). (c) Type I lepra reaction (cutaneous). (d) Type I lepra reaction (Reaction Hand)|
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Grade 2 disability was seen in 49 (23%) cases. [Figure 6] shows various deformities seen in the study. Claw hand was the most common deformity encountered in 37 (17.4%) followed by foot drop in 10 (4.7%) cases, trophic ulcer, and resorption of digits in one case each [Table 3].
|Figure 6: Clinical spectrum of deformities of leprosy in the study population. (a) Claw Hand deformity. (b) Trophic ulcers over digits. (c) Trophic ulcer over first metatarsal|
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Investigations recorded in the study were slit skin smears for bacillary index (BI), histopathological examination (HPE) with staining for acid-fast bacilli lepra (AFB [L]). Initial BI recorded was as given in [Table 2]. The most common presentation in HPE was consistent with BT leprosy in 133 (62.6%) cases. AFB (L) was present in 35 (16.4%) cases on modified Fite-Faraco staining in all cases of LL, BL, and histoid leprosy. Five patients of BT with no AFB (L) on HPE had BI of 1+ [Table 2].
All 213 cases were administered by MB multi-drug therapy (MDT). Two hundred and six (97.6%) cases received three-drug MDT (MB) (rifampicin 600 mg monthly, clofazimine 50 mg daily, and dapsone 100 mg daily) during entire treatment period while 07 (2.4%) cases developed adverse drug reactions to dapsone (dapsone hypersensitivity syndrome: 4, hemolytic anemia: 02, Drug-induced liver injury to Dapsone: 1) which required it to be stopped and continued on 02 drug MDT with rifampicin and clofazimine subsequently. The mean duration of treatment was 29.08 ± 19.4 months.
| Discussion|| |
It is indeed ironic that despite the recognition of causative organisms in 1873 and advances in investigations and treatment modalities, leprosy continues to be a public health problem in India. India continues to account for 60% of new cases reported globally each year and is among 22 “global priority countries.” As stated earlier, in a vast country like India, effective contact tracing, and a heterogeneous platform for accessing health-care facilities remains the primary challenge in the eradication of leprosy. This is further compounded by inequitable distribution of cases, with 76 districts having a prevalence of <1/10,000 while approximately 39 districts are having a prevalence of >2–5/10,000.
The mean age of our patients was 35 years. The majority of the patients (77.5%) were between 21 and 40 years of age which corroborates with other studies.,, Male: female ratio of 2.6:1 is similar to reported literature.,, Contact history was positive in 03 cases only which was much lesser than earlier reports of 25%–35%.,, Mahajan et al. reported a positive family history in 2.43% cases. The majority (79%) cases presented a hypopigmented hyperesthetic patch followed by ENL and motor weakness similar to previous studies., Mahajan et al. in their study had reported 90.57% out of 329 patients presenting with patches.
Following the earlier studies, BT leprosy was the most common pole of leprosy constituting 153 (71.5%) patients followed by lepromatous leprosy 22 (10.3%) patients,, though some studies have reported BL or LL, as the most common spectrum also.,,, High proportion of BT cases in our study can be attributed to relatively easier diagnosis and early suspicion of patients as patches in this spectrum are well defined, easily noticeable, and have a sensory loss which leads a patient to report earlier than BL/LL spectrum where clinical symptoms are generally absent. There was no case of tuberculoid pole in our study. Since the introduction of MDT, the borderline spectrum has been reported more than the polar forms which were seen more in the dapsone era. The proportion of PNL of 10.3% in our study is much higher than previously reported of 4.6% by Mahajan et al. and 5.5% by Dongre et al. in this region of India., Histoid leprosy was diagnosed in 03 (1.4%) patients similar to observations of Kaur et al.(1.8%) and Mahajan et al. (2.7%).,
A total of 186 (87.3%) patients out of 213 patients were classified as MB leprosy which is far more than the national level at 51.27% and globally average reported of 61%. This was concordant with other recent Indian studies.,,,, This indicates that despite intense efforts at the national level and effective MDT, there remain inaccessible pockets, where leprosy remains undiagnosed or is diagnosed late. These subsets of patients are more prone to deformities and handicaps. A high proportion of MB cases in our study can be explained by the fact that this being a tertiary care center, severe cases with reactions and disabilities were referred which could not have been effectively managed at the peripheral health center. In addition, patients also tend to hide their diagnosis due to the associated stigma. In any case, this requires a detailed and proper investigation at the community level to catch hidden and missed cases which are the major reservoir of infection.
Thickened nerves were observed in 96.7% of patients; mononeuropathy was present in 20.8% patients corresponding well with previous literature;,, nevertheless, few studies have reported a lower proportion of thickened nerves., The ulnar nerve was the most common thickened nerve.,,,, Examination of thickened nerves can have a significant intra-observer bias, the likelihood of detection of thickened nerves varied from 20% to 96% in a study conducted by the Department of Clinical Neurology, University of Oxford, UK. This can explain the variation in the literature about the reporting of thickened nerves in leprosy.
Leprosy reactions were documented in 99 (46.5%) cases. Type-1 reaction 79 (79.8%) was more common than Type-2 reaction 20 (20.2%). This was contradictory to the majority of the previous studies where T2R was more common.,,,,, This variation may have probably resulted in owing to the majority of our patients presenting with BT leprosy 153 (71.5%) in which T1R are more commonly seen.
Twenty-two patients with a recurrent episode of T1R were managed initially with <12 weeks of the tapering dose of oral steroids (starting dose varying between 0.75 mg/kg/day and 1 mg/kg/day), whereas remaining 57 cases with a single episode of T1R were managed with steroids (starting dose @ 1 mg/kg/day) with the duration of steroids >20 weeks similar to a study by Rao et al. where 46% patients required additional steroids subsequently and 20 weeks course was significantly better than 12 weeks course of steroid. Out of the 20 patients with T2R, 17 (8%) cases had ENL as the initial presentation. 05 (25%) of these 20 cases had recurrent ENL episodes during the treatment period. The literature review showed recurrent ENL episodes varied from 39% to 77%.
One of the targets of the Global Leprosy Strategy (2016–2020) is the reduction of new leprosy cases with Grade 2 disability (G2D) to <1 case per million population. G2D was seen here in 49 (23%) cases which was much higher than the NLEP report for the year 2015–2016 with a disability rate of 4.46% and WHO Global leprosy update of 2015 with 3.8% for the reporting year 2016. Similar to this study, numerous studies also report higher G2D rates.,,,, On the one hand, G2D indicates the level of awareness about the symptomatology of leprosy and health-seeking behavior in the society, and on the other hand, the capacity of the health system to manage leprosy at an early stage, before the disabilities set in. However, it indicates the delay in the detection of leprosy cases presently.
Although this study had 153 (71.5%) cases of BT as per Ridley Jopling Classification, the most common presentation in HPE was consistent with BT leprosy in 133 (62.6%) cases only. This clinicopathological discordance could be explained by the non-representative site of skin biopsy not corresponding with clinical diagnosis.
All the 213 patients received the MB MDT with mean treatment duration of 29.08 ± 19.4 months. All the 213 patients who underwent treatment for leprosy were subjected to repeat skin biopsy before deciding onto the stoppage of MDT and treatment was terminated on the evidence of histopathological resolution of leprosy. Thus, prolonged duration of treatment can be explained by the time taken for the histopathology clearance in these MB cases. Seven patients developed dapsone-induced adverse drug reaction which required it to be stopped and were continued remaining rifampicin and clofazimine. There were no treatment defaulters.
The changes in population demography, internal migration, and merging of urban-rural boundaries in developing countries are a few of the many factors attributed to the rise in leprosy cases recently. In the present study, only 1.4% of cases revealed contact history. This could be explained due to prolonged and variable incubation periods as well as ever-increasing migratory population. This scenario has also been highlighted in preceding Indian studies, making it difficult to implement universal policies to eliminate leprosy.,
Another major factor highlighted by this study is a delay in diagnosis which leads to the progression of the disease, reactionary episodes, and consequent sequelae in the form of permanent deformities leading to preventable deformities and disabilities. The major reason cited for this could be a suboptimal awareness and stigmatization of the disease in the community. Toward this objective, robust information, education, and communication model can go a long way in ensuring that the stigma and discrimination against leprosy patients in the community do not hamper the detection of new cases as well as the follow-up of existing cases. Thus, the case for early detection and effective management of leprosy cases in the community. This can be achieved by strengthening of the health system at the center and state level to align it with the vertical and effective implementation of the Leprosy eradication programme. As highlighted in this study, a three-pronged strategy as advocated by the NLEP in 2016–2017 needs to be employed to effectively eradicate leprosy. These include decreasing the time lag between first symptom and diagnosis, universal health coverage with a focus on children, women, and underserved populations like displaced and migrant population and contact tracing and surveillance.
This study has certain limitations. First, being a tertiary care center, the majority of the cases referred were of the severe spectrum which might explain a higher proportion of MB cases and G2D. Second, it was a retrospective study based on hospital records of patients limited to only those cases who reported directly to us or were referred. Larger population-based studies are required to better understand the current situation and plan remedial measures.
| Conclusions|| |
The present study provides an insight into disease burden as well as the effectiveness of health services in a tertiary care hospital in Western Maharashtra. The high rate of MB cases (87.3%) and a higher proportion of G2D (23%) in this study highlight the concern on the effectivity of the NLEP. One of the key reasons cited for the increase in leprosy cases is the delay in diagnosis and management of leprosy and reactions leading to persistent neuritis and subsequent deformities and disabilities. This requires an integrated approach with not only the health-care support effectively but also increased awareness about leprosy signs and symptoms and self-reporting among the community members to reach the targets of “Global Leprosy Strategy 2016–2020.”
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
[Table 1], [Table 2], [Table 3]