Assessing the cost-effectiveness of integrated case management of Neglected Tropical Diseases in Liberia.

BACKGROUND: In 2017, Liberia became one of the first countries in the African region to develop and implement a national strategy for integrated case management of Neglected Tropical Diseases (CM-NTDs), specifically Buruli ulcer, leprosy, lymphatic filariasis morbidities, and yaws. Implementing this plan moves the NTD program from many countries' fragmented (vertical) disease management. This study explores to what extent an integrated approach offers a cost-effective investment for national health systems. METHODS: This study is a mixed-method economic evaluation that explores the cost-effectiveness of the integrated CM-NTDs approach compared to the fragmented (vertical) disease management. Primary data were collected from two integrated intervention counties and two non-intervention counties to determine the relative cost-effectiveness of the integrated program model vs. fragmented (vertical) care. Data was sourced from the NTDs program annual budgets and financial reports for integrated CM-NTDs and Mass Drug Administration (MDA) to determine cost drivers and effectiveness. RESULTS: The total cost incurred by the integrated CM-NTD approach from 2017 to 2019 was US$ 789,856.30, with the highest percentage of costs for program staffing and motivation (41.8%), followed by operating costs (24.8%). In the two counties implementing fragmented (vertical) disease management, approximately US$ 325,000 was spent on the diagnosis of 84 persons and the treatment of twenty-four persons suffering from NTDs. While 2.5 times as much was spent in integrated counties, 9-10 times more patients were diagnosed and treated. CONCLUSIONS: The cost of a patient being diagnosed under the fragmented (vertical) implementation is five times higher than integrated CM-NTDs, and providing treatment is ten times as costly. Findings indicate that the integrated CM-NTDs strategy has achieved its primary objective of improved access to NTD services. The success of implementing an integrated CM-NTDs approach in Liberia, presented in this paper, demonstrates that NTD integration is a cost-minimizing solution.

Stakeholder perspectives on an integrated package of care for lower limb disorders caused by podoconiosis, lymphatic filariasis or leprosy: A qualitative study.

BACKGROUND: Lower limb disorders including lymphoedema create a huge burden for affected persons in their physical and mental health, as well as socioeconomic and psychosocial consequences for them, their families and communities. As routine health services for the integrated management and prevention of lower limb disorders are still lacking, the 'Excellence in Disability Prevention Integrated across Neglected Tropical Diseases' (EnDPoINT) study was implemented to assess the development and delivery of an integrated package of holistic care-including physical health, mental health and psychosocial care-within routine health services for persons with lower limb disorders caused by podoconiosis, lymphatic filariasis and leprosy. METHODOLOGY/PRINCIPAL FINDINGS: This study was part of the first of three phases within EnDPoINT, involving the development of the integrated care package. Focus group discussions and key informant interviews were undertaken with 34 participants between January-February 2019 in Awi zone, Ethiopia, in order to assess the draft care package's feasibility, acceptability and appropriateness. Persons affected by lower limb disorders such as lymphoedema experience stigma, exclusion from families, communities and work as well as physical and financial hardship. Beliefs in disease causation inhibit affected persons from accessing care. Ignorance was a barrier for health care providers as well as affected persons. Training and education of affected persons, communities and caregivers is important in improving care access. It also requires time, space, materials and financial resources. Both top-down and grass roots input into service development are key, as well as collaboration across stakeholders including charities, community leaders and "expert patients". CONCLUSIONS/SIGNIFICANCE: This study highlighted the need for the EnDPoINT integrated care package and provided suggestions for solutions according to its three aspects of integrated care (integration into routine care; integration of mental health and psychosocial care; and integration of care across the three diseases), thereby giving support for its feasibility, acceptability and appropriateness.

Development of an integrated, holistic care package for people with lymphoedema for use at the level of the Primary Health Care Unit in Ethiopia.

BACKGROUND: Neglected Tropical Diseases (NTDs) are a group of several communicable and non-communicable diseases prevalent in tropical and subtropical areas. The co-endemicity of these diseases, the similarity of their clinical signs, and the need to maximize limited financial and human resources suggest the importance of adoptingan integratedapproach to their prevention and treatment. AIMS: This study describes the development of a comprehensive package of physical, mental health and psychosocial care for people with lower-limb lymphoedema caused bypodoconiosis, lymphatic filariasis (LF)or leprosy as part of the EnDPoINT program in Ethiopia. METHOD: The care package was developed using a mixed-methods approach, consisting of a literature review, situational analysis, Theory of Change (ToC) workshops, qualitative research, and additional workshops to fine-tune the draft care package. The care package was developed between March 2018 and January 2020 in Addis Ababa and the implementation research site, Awi zone in the North-West of Ethiopia. RESULTS: The holistic care package includes components implemented at three levels of the health care system:health organization, facility, and community. Sections of the care package are directed at strengthening capacity building, program management, community engagement, awareness-raising, stigma-reduction, morbidity management, disability prevention, follow-up visits, referral linkage, community-based rehabilitation, and monitoring and evaluation. CONCLUSIONS: The study developed a holistic integrated care package for lower limb disorder and co-morbid mental health problems caused by podoconiosis, LF or leprosy. The approach has the potential to significantly reduce lower limb disorder-associated morbidity, disability, and psychosocial problems. It also standardizes a scalable approach appropriate for the Ethiopian setting and, most likely, other countries where these NTDs are present.

The implementation of an integrated e-leprosy framework in a leprosy control program at primary health care centers in Indonesia.

BACKGROUND AND OBJECTIVE: The implementation of health information systems (HIS) could overcome obstacles in human resources and infrastructure at primary health care centers (PHCs). This study involved an e-Leprosy framework being integrated into the real setting of a leprosy control program in Indonesia. The objectives of this implementation study were to integrate e-Leprosy into a leprosy control program at 27 PHCs in Pekalongan District. Central Java Province, Indonesia to explore factors related the success or failure of such an implementation regarding the usability, involvement, and acceptance of e-Leprosy by PHC staff and to evaluate the effect of the implementation on leprosy patient attendance at PHCs. This paper is based on the Standards for Reporting Implementation Studies (StaRI) statement. METHOD: This study used mixed methods implementation research with longitudinal analysis and involved two groups of participants: Leprosy Surveillance Officers (LSOs), patients, and the relatives of patients. This study involved four phases consisting of preparation, baseline assessment, intervention, and evaluation. The qualitative study conducted focus group discussions and in-depth interviews. The e-Leprosy program automatically sent SMS reminders regarding leprosy treatment to the LSOs, patients, and patients' relatives every month. FINDINGS: This study determined that LSO had difficulties related to their workloads in PHCs while managing information and monitoring treatment and contact after release from treatment. The baseline assessment phase found that LSOs in Pekalongan District were unfamiliar with email but familiar using the internet. Overall, LSOs had a positive perception of the e-Leprosy program. The usability of this e-Leprosy program tended to increase over time, while acceptance of the e-Leprosy exhibited a significant relationship with computer and internet fluency (r = 0.48, p < 0.05) and age (r = 0.621, p < 0.01). The responsible patients correlated (r = 0.67, p < 0.01) with involvement in the e-Leprosy program. This study revealed that patient reminders increased on-time attendance by 13.9 % (p < 0.01 with OR = 2.41). CONCLUSION: Factors that should be considered during implementation HIS included the digital gap, PHC's staff workload, as well as the level of commitment and leadership in the health office.

Indications for Rituximab Use in an Integrated Health Care Delivery System.

BACKGROUND: Rituximab is a top-selling biologic that was first approved by the FDA in 1997 for a non-Hodgkin lymphoma orphan indication. It has since been approved for additional orphan indications, with rheumatoid arthritis as the only FDA-approved, nonorphan indication. Evidence suggests that rituximab is frequently used off-label, but information on its use over time and indications for use in the United States is limited. OBJECTIVE: To assess incident rituximab use over time in an integrated health care delivery system. METHODS: This was a cross-sectional, retrospective study. Data were collected from administrative databases and manual chart reviews. Patients who received their first rituximab infusion between October 1, 2009, and December 31, 2017, and who were not a part of a clinical trial were included. Indication for use (FDA-approved orphan/nonorphan, off-label) was determined. Proportions of use were assessed over time. Multivariable logistic regression modeling was performed to assess factors associated with receiving rituximab for an FDA-approved indication. RESULTS: A total of 1,674 patients were included. The majority (66.4%) of patients had an FDA-approved indication, with lymphoma being the most common approved indication (66.4%). The most common indication for off-label use was neurologic conditions (72.7%), predominantly demyelinating diseases. Off-label indication use increased from 1.2% in 2009 to 55.6% in 2017. Factors associated with rituximab use for an FDA-approved indication included increased age (adjusted odds ratio [AOR] = 1.05, 95% CI = 1.04-1.07) and increased burden of chronic disease (chronic disease score: AOR = 1.07, 95% CI = 1.02-1.12; Charlson Comorbidity Index score: AOR = 3.52, 95% CI = 3.03-4.10). CONCLUSIONS: Off-label use of rituximab grew dramatically over the course of the study. With the recent FDA approval of the rituximab biosimilar and its expected lower price, off-label use will likely continue to rise. Opportunities for cost savings and to ensure appropriate use of these medications should be evaluated. DISCLOSURES: This study was funded by Kaiser Permanente. All authors except Hansen are employed by Kaiser Permanente. Hansen has nothing to disclose. Preliminary results were presented at the Mountain States Conference for Residents and Preceptors in May 2019 in Salt Lake City, UT, and at an encore presentation October 2019 at the American College of Clinical Pharmacy Annual Meeting in New York, NY.

Assessing the feasibility of integration of self-care for filarial lymphoedema into existing community leprosy self-help groups in Nepal.

BACKGROUND: Lymphatic filariasis (LF) and leprosy are disabling infectious diseases endemic in Nepal. LF infection can lead to lymphoedema and hydrocoele, while secondary effects of leprosy infection include impairments to hands, eyes and feet. The disabling effects of both conditions can be managed through self-care and the supportive effects of self-help groups (SHGs). A network of SHGs exists for people affected by leprosy in four districts in Nepal's Central Development Region, however no such service exists for people affected by LF. The aim of this study was to determine the feasibility of integrating LF affected people into existing leprosy SHGs in this area. METHODS: A survey was conducted using a semi-structured questionnaire to elicit information on: (i) participant characteristics, clinical manifestation and disease burden; (ii) participants' knowledge of management of their condition and access to services; and (iii) participants' knowledge and perceptions of the alternate condition (LF affected participants' knowledge of leprosy and vice versa) and attitudes towards integration. RESULTS: A total of 52 LF affected and 53 leprosy affected participants were interviewed from 14 SHGs. On average, leprosy affected participants were shown to have 1.8 times greater knowledge of self-care techniques, and practiced 2.5 times more frequently than LF affected participants. Only a quarter of LF affected participants had accessed a health service for their condition, compared with 94.3% of leprosy affected people accessing a service (including SHGs), at least once a week. High levels of stigma were perceived by both groups towards the alternate condition, however, the majority of LF (79%) and leprosy (94.3%) affected participants stated that they would consider attending an integrated SHG. CONCLUSIONS: LF affected participants need to increase their knowledge of self-care and access to health services. Despite stigma being a potential barrier, attitudes towards integration were positive, suggesting that the SHGs may be a good platform for LF affected people to start self-care in this area. TRIAL REGISTRATION: This is not a registered trial.

Introducing leprosy post-exposure prophylaxis into the health systems of India, Nepal and Indonesia: a case study.

BACKGROUND: Leprosy has a wide range of clinical and socio-economic consequences. India, Indonesia and Nepal contribute significantly to the global leprosy burden. After integration, the health systems are pivotal in leprosy service delivery. The Leprosy Post Exposure Prophylaxis (LPEP) program is ongoing to investigate the feasibility of providing single dose rifampicin (SDR) as post-exposure prophylaxis (PEP) to the contacts of leprosy cases in various health systems. We aim to compare national leprosy control programs, and adapted LPEP strategies in India, Nepal and Indonesia. The purpose is to establish a baseline of the health system's situation and document the subsequent adjustment of LPEP, which will provide the context for interpreting the LPEP results in future. METHODS: The study followed the multiple-case study design with single units of analysis. The data collection methods were direct observation, in-depth interviews and desk review. The study was divided into two phases, i.e. review of national leprosy programs and description of the LPEP program. The comparative analysis was performed using the WHO health system frameworks (2007). RESULTS: In all countries leprosy services including contact tracing is integrated into the health systems. The LPEP program is fully integrated into the established national leprosy programs, with SDR and increased documentation, which need major additions to standard procedures. PEP administration was widely perceived as well manageable, but the additional LPEP data collection was reported to increase workload in the first year. CONCLUSIONS: The findings of our study led to the recommendation that field-based leprosy research programs should keep health systems in focus. The national leprosy programs are diverse in terms of organizational hierarchy, human resource quantity and capacity. We conclude that PEP can be integrated into different health systems without major structural and personal changes, but provisions are necessary for the additional monitoring requirements.

Public-private mix for TB and TB-HIV care in Lagos, Nigeria.

SETTING: Private and public tuberculosis (TB) treatment centres in Lagos State, Nigeria. OBJECTIVE: To assess the contribution of private health care providers to TB and TB-HIV (human immunodeficiency virus) case finding in Lagos State. DESIGN: A retrospective review of programme data submitted to the Lagos State TB and Leprosy Control Programme in 2011 by public, private for-profit (PFP) and private not-for-profit (PNFP) health care providers. RESULTS: A total of 8425 TB cases were notified by 31 private (11 PFP and 20 PNFP) and 99 public health facilities in Lagos State. Overall, the private facilities were responsible for 10.3% (866/8425) of the total TB cases notified. The proportion of TB patients tested for HIV was respectively 86.2%, 53.1% and 96.5% among public, PFP and PNFP facilities. Overall, 22.4% of the TB patients were HIV-positive. The HIV positivity rate among public, PFP and PNFP facilities was respectively 23.8%, 7.8% and 9.9%. Uptake of cotrimoxazole preventive therapy was respectively 69.6%, 25% and 38.2% among public, PFP and PNFP facilities, while that of antiretroviral therapy was respectively 23.8%, 8.3% and 9.1% in public, PFP and PNFP facilities. CONCLUSION: There is a need to scale up collaboration with the private sector, and particularly PNFP health providers.

Sustainability of integrated leprosy services in rural India: perceptions of community leaders in Uttar Pradesh.

As part of a community-based action research to reduce leprosy stigma, village committees were formed in 3 hyper endemic states of India. From a total of 10 village committees with nearly 200 members from Uttar Pradesh, a systematic random sample of 69 men and 23 women were interviewed in-depth regarding their views on sustainability of integrated leprosy services, as currently adopted. Their recommendations were also sought for further enhancement. Percentages were computed and compared for statistical significance using the z-normal test. The findings show that less than 50% of the respondents were confident that the present trend in voluntary early reporting for MDT and management of complications was adequate to sustain the integrated leprosy services. There were no differences by men or women members and they felt that lack of proper facilities, training and orientation of staff are most influencing factors. Many suggestions were given for improving the sustainability.

Public awareness on integration of leprosy services at primary health centres in Uttar Pradesh, India.

Leprosy services were integrated into the general health a decade ago but it seems that a majority of public are still ignorant of this development. Hence, a study was done in Uttar Pradesh, India to determine the awareness about integration and its relationships to various socio-demographic factors. A multistage representative random sample of 3000 persons was chosen in Faizabad district, selecting a sample of 3 villages each situated within 1 km, 1-3 km and beyond 3 km of a PHC. A systematic random sample of 10% of households was chosen from selected villages and an adult male and an adult female from each household interviewed by a qualified investigator. Data were computerized and cross- tabulated against distance from the PHC, sex, age, education and occupational status. Only 45.7% in Uttar Pradesh are aware of the availability of leprosy treatment facilities at PHC but most knew that MDT was free. A smaller proportion was also aware of other facilities such as ulcer dressing and treatment of complications. Family members and health workers and PHC were the main source of information. It is concluded that massive efforts are urgently needed to educate the rural public on integration.

Integration of leprosy in general health system vis-à-vis leprosy endemicity, health situation and socioeconomic development: observations from Chhattisgarh & Kerala.

This study looked at the integration of leprosy services in the GHS in context of health and socioeconomic situations using predefined indicators. It also looked at clients' perception of MDT services. The Indian states of Chhattisgarh and Kerala, which are at two extremes in leprosy endemicity, health situation and socioeconomic development, have been compared using predefined integration indicators related to the training of health workers, availability of MDT services, maintenance of MDT stock and involvement of Sub-centres in leprosy care. Data was collected by surveys of health facilities, sub-centres and communities in the two states, during 2006-2007. Information was collected by interviewing health personnel and clients, checking of records and on the spot observations using specifically designed formats. Results showed that integration is more inclusive in Chhattisgarh and has reached up to Sub-centre level. Both the community and health systems are sensitive and responsive to leprosy as it is perceived to be a major public health threat. But in Kerala, despite integration, it continues as a vertical programme with dependence on specialists and districts hospitals for diagnosis and treatment. MDT stock management is even poorer. Clients' perception towards MDT services are similar in both states.

Integration of leprosy into GHS in India: a follow up study (2006-2007).

In India leprosy services, were integrated into the General Health Services (GHS), in a phased manner, in different provinces, from 2001 to 2004. This study reports the findings from a follow-up operational research undertaken in 2006-2007, to assess the level of integration, on predetermined indicators related to: referral services, training of health functionaries, availability of diagnosis, treatment, MDT dispersal and counselling guidelines in health facilities, recording and reporting by GHS staff, MDT stock management and involvement of health sub-centres in different Indian provinces. Nine provinces, 18 districts, 88 health facilities and 108 sub-centres were selected, by using multistage stratified random sampling techniques. Reverse integration, as reflected by the training and deployment of vertical staff in GHS, was also assessed. Data was collected by medical officers experienced in leprosy, with the assistance of state health functionaries, and recorded on separate schedules for health facility and sub-centre levels. The study also touched on the issue of client perception towards MDT services by interviewing 149 under treatment/cured leprosy cases (who had completed treatment within the last year), in the community with the help of local interpreters. Results showed wide variations across the selected provinces in various parameters. District leprosy nuclei were understaffed in 12(66.7%) districts, and district hospitals were not working as referral institutions anywhere. The training status of medical officers and multi-purpose workers in leprosy was low in Andhra Pradesh (6.9 and 22.4%), Madhya Pradesh (26.3 and 14.5%), Rajasthan (19.7 and 40.9%) and Kerala (25.5 and 65.7%). MDT stock availability as per the National Leprosy Eradication Programme (NLEP) guidelines was not adequate in all provinces. Availability of patient counseling guidelines was nil/low in Kerala, Karnataka, West Bengal, Orissa, Rajasthan and Andhra Pradesh. The involvement of sub-centres, in case referral, recording and dispensing MDT was nil Kerala and Rajasthan and poor in Andhra Pradesh. Ninety percent of clients in Kerala and 38.0% in Andhra Pradesh and Madhya Pradesh did not get MDT in the nearest health facilities or sub-centres.

Integration of leprosy elimination into primary health care in orissa, India.

BACKGROUND: Leprosy was eliminated as a public health problem (<1 case per 10,000) in India by December 2005. With this target in sight the need for a separate vertical programme was diminished. The second phase of the National Leprosy Eradication Programme was therefore initiated: decentralisation of the vertical programme, integration of leprosy services into the primary health care (PHC) system and development of a surveillance system to monitor programme performance. METHODOLOGY/PRINCIPAL FINDINGS: To study the process of integration a qualitative analysis of issues and perceptions of patients and providers, and a review of leprosy records and registers to evaluate programme performance was carried out in the state of Orissa, India. Program performance indicators such as a low mean defaulter rate of 3.83% and a low-misdiagnosis rate of 4.45% demonstrated no detrimental effect of integration on program success. PHC staff were generally found to be highly knowledgeable of diagnosis and management of leprosy cases due to frequent training and a support network of leprosy experts. However in urban hospitals district-level leprosy experts had assumed leprosy activities. The aim was to aid busy PHC staff but it also compromised their leprosy knowledge and management capacity. Inadequate monitoring of a policy of 'new case validation,' in which MDT was not initiated until primary diagnosis had been verified by a leprosy expert, may have led to approximately 26% of suspect cases awaiting confirmation of diagnosis 1-8 months after their initial PHC visit. CONCLUSIONS/SIGNIFICANCE: This study highlights the need for effective monitoring and evaluation of the integration process. Inadequate monitoring could lead to a reduction in early diagnosis, a delay in initiation of MDT and an increase in disability rates. This in turn could reverse some of the programme's achievements. These findings may help Andhra Pradesh and other states in India to improve their integration process and may also have implications for other disease elimination programmes such as polio and guinea worm (dracunculiasis) as they move closer to their elimination goals.

Impact of integration on the profile of newly diagnosed leprosy patients attending a referral hospital in South India.

This article compares the clinical profile of new untreated leprosy patients attending a referral hospital (The Schieffelin Institute for Health Research & Leprosy Centre, formerly known as SLR&TC, Karigiri, South India, in post-integration period (2005-2007) with that during the pre-integration period (1995-1996). A total of 529 patients--259 in pre-integration and 270 in post-integration period--were seen at this hospital. The clinical data culled from records for the earlier period were compared with data gathered prospectively for the latter period and was analyzed using SPSS software. The results showed a significant increase in the mean age of registration, percent multibacillary (clinical criteria) and grade 2 diabilities in post-integration period. Increase in proportion of cases with grade 2 deformities is a matter of concern and suggests continued need for referral hospitals for their management and also population based overall assessment whether actual numbers with deformities have increased or it is peculiar to a tertiary care hospital where the cases with problems may be coming. As the proportion of bacteriological positive cases was not found to change, it is a positive sign of effective coverage in the post-integration scenario in this population.