Destitution, treatment adherence and survival of children with Burkitt lymphoma in a twinning programme in Northwest Cameroon.

BACKGROUND: Burkitt lymphoma (BL) is a curable childhood cancer. Treatment adherence is crucial for a good outcome, but is potentially problematic in low- and middle-income countries owing to parental financial constraints. AIMS: To investigate the association of destitution with treatment adherence and its effect on the survival of patients with BL. METHODS: Patients received free medical treatment from a twinning programme at two Cameroon Baptist hospitals. A destitution assessment questionnaire, based on socioeconomic status, was completed at diagnosis. Medical records were reviewed for treatment adherence and survival. Chi-squared and Fisher's exact tests were used to compare groups. Kaplan-Meier plots were used to calculate overall survival, and log-rank chi-squared tests when comparing survival rates between patient subgroups. Significance was measured at P < .05. RESULTS: The 225 children with BL had a mean age of 8.2 years (median 8.0) and the overall survival was 52%. The mean family destitution score was 56% on a linear scale. Few (8%) patients delayed treatment appointments. A quarter (25%) experienced more than a 1-week follow-up delay and 9.8% absconded within 1 year. The destitution score was not significantly associated with delay of treatment, but with delay in follow-up (P < .001). Guardian relationship (single mother) and patient's age were significantly associated with overall survival (P = .025). CONCLUSIONS: Though linked to poor follow-up, destitution was not significantly associated with absconding patients, poor outcome or poor adherence to treatment, probably due to comprehensive financial support from the international twinning programme. However, additional support for single mothers should be considered.

Insurance status impacts overall survival in Burkitt lymphoma.

The impact of insurance status on clinical outcomes in Burkitt (BL) and plasmablastic (PBL) lymphomas remains unknown. We used the National Cancer Database to examine insurance status' effect on overall survival (OS) in adults diagnosed with these lymphomas between 2004 and 2014. BL patients with private insurance had significantly better OS compared to those without. In patients aged <65 years, hazard ratios were 1.4 for uninsured status (95% confidence interval 1.2-1.7), 1.2 for Medicaid (95% CI 1.0-1.4), and 1.5 for Medicare (95% CI 1.2-1.9). For patients aged >65 years, hazard ratio for uninsured status was 8.4 (95% CI 2.5-28.3). Conversely, underinsured PBL patients experienced no difference in OS. Thus, expanding insurance-related access to care may improve survival in BL, for which curative therapy exists, but not PBL, where more effective therapies are needed. Our findings add to mounting evidence that adequate health insurance is particularly important for patients with curable cancers.

The cost effectiveness of treating Burkitt lymphoma in Uganda.

BACKGROUND: Perceptions of high cost and resource intensity remain political barriers to the prioritization of childhood cancer treatment programs in many low- and middle-income countries (LMICs). Little knowledge exists of the actual cost and cost-effectiveness of such programs. To improve outcomes for children with Burkitt lymphoma (BL), the most common childhood cancer in Africa, the Uganda Cancer Institute implemented a comprehensive BL treatment program in 2012. We undertook an economic evaluation of the program to ascertain the cost-effectiveness of BL therapy in a specific LIC setting. METHODS: We compared the treatment of BL to usual care in a cohort of 122 patients treated between 2012 and 2014. Costs included variable, fixed, and family costs. Our primary measure of effectiveness was overall survival (OS). Patient outcomes were determined through prospective capture and retrospective chart abstraction. The cost per disability-adjusted life-year (DALY) averted was calculated using the World Health Organization's Choosing Interventions That Are Cost-Effective (WHO-CHOICE) methodology. RESULTS: The 2-year OS with treatment was 55% (95% CI, 45% to 64%). The cost per DALY averted in the treatment group was US$97 (Int$301). Cumulative estimate of national DALYs averted through treatment was 8607 years, and the total national annual cost of treatment was US$834,879 (Int$2,590,845). The cost of BL treatment fell well within WHO-CHOICE cost-effectiveness thresholds. The ratio of cost per DALY averted to per capita gross domestic product was 0.14, reflecting a very cost-effective intervention. CONCLUSION: This study demonstrates that treating BL with locally tailored protocols is very cost-effective by international standards. Studies of this kind will furnish crucial evidence to help policymakers prioritize the allocation of LMIC health system resources among noncommunicable diseases, including childhood cancer.

Treatment of sporadic Burkitt lymphoma in adults, a retrospective comparison of four treatment regimens.

Burkitt lymphoma is an aggressive B cell malignancy accounting for 1-2% of all adult lymphomas. Treatment with dose-intensive, multi-agent chemotherapy is effective but associated with considerable toxicity. In this observational study, we compared real-world efficacy, toxicity, and costs of four frequently employed treatment strategies for Burkitt lymphoma: the Lymphome Malins B (LMB), the Berlin-Frankfurt-Münster (BFM), the HOVON, and the CODOX-M/IVAC regimens. We collected data from 147 adult patients treated in eight referral centers. Following central pathology assessment, 105 of these cases were accepted as Burkitt lymphoma, resulting in the following treatment groups: LMB 36 patients, BFM 19 patients, HOVON 29 patients, and CODOX-M/IVAC 21 patients (median age 39 years, range 14-74; mean duration of follow-up 47 months). There was no significant difference between age, sex ratio, disease stage, or percentage HIV-positive patients between the treatment groups. Five-year progression-free survival (69%, p = 0.966) and 5-year overall survival (69%, p = 0.981) were comparable for all treatment groups. Treatment-related toxicity was also comparable with only hepatotoxicity seen more frequently in the CODOX/M-IVAC group (p = 0.004). Costs were determined by the number of rituximab gifts and the number of inpatients days. Overall, CODOX-M/IVAC had the most beneficial profile with regards to costs, treatment duration, and percentage of patients completing planned treatment. We conclude that the four treatment protocols for Burkitt lymphoma yield nearly identical results with regards to efficacy and safety but differ in treatment duration and costs. These differences may help guide future choice of treatment.

The role of traditional healers in the diagnosis and management of Burkitt lymphoma in Cameroon: understanding the challenges and moving forward.

BACKGROUND: Burkittlymphoma(BL) is the most common childhood cancer in Cameroon with a reported incidence of 3 per 100,000 children under 15 years in the Northwest region. Treatment at three Baptist mission hospitals has a recorded cure rate of over 50%. Traditional medicine(TM) is recognized by the national health system, but its scope is undefined and entraps children with BL. The aim of this study was to investigate the attitudes and practices of parents and traditional healers (TH) towards TM in children with BL in order to develop recommendations for an integrative approach and improved access to life-saving treatment for children with BL. METHODS: This is a descriptive case series of children diagnosed with BL treated at Banso, Mbingo, and Mutengene Baptist Hospitals between 2003 and 2014. A questionnaire was used to obtain the following information: demographic information, religion, the rate of use of TM, reasons why guardians chose to use TM, the diagnoses made by the TH, treatment offered, and the type of payment requested, based on the accounts of patient caregivers. Data was analyzed using Center for Disease Control Epi Info 7. RESULTS: Three hundred eighty-seven questionnaires were completed by parents/guardians. 55% had consulted a TH, of whom 76.1% consulted the TH as first choice. Common diagnoses provided by TH included liver problem, abscess, witchcraft, poison, hernia, side pain, mushroom in the belly and toothache. Methods of management included massage, cuts, concoctions, and incantations. The fee for these services included chickens, farm tools, and cash ranging from 200FCFA (0.4USD) to 100,000FCFA(200USD). The choice of TM was based on accessibility, failed clinic/hospital attendance, recommendation of relatives, and belief in TM. CONCLUSIONS: TH are involved in BL management in Cameroon. TH are ignorant about BL, resulting in non-referral, and thus delay in diagnosis and treatment. Collaboration with TH could reduce late diagnosis and improve cure rates of BL and other childhood cancers.

The cost effectiveness of treating paediatric cancer in low-income and middle-income countries: a case-study approach using acute lymphocytic leukaemia in Brazil and Burkitt lymphoma in Malawi.

Approximately 90% of children with cancer reside in low-income and middle-income countries (LMIC) where healthcare resources are scarce and allocation decisions difficult. The cost effectiveness of treating childhood cancers in these settings is unknown. The objective of the present work was to determine cost-effectiveness thresholds for common paediatric cancers using acute lymphoblastic leukaemia (ALL) in Brazil and Burkitt lymphoma (BL) in Malawi as examples. Disability-adjusted life years (DALYs) prevented by treatment were compared to the gross domestic product (GDP) per capita of each country to define cost-effectiveness thresholds using WHO-CHOICE ('CHOosing Interventions that are Cost-Effective') guidelines. The case examples were selected due to the data available and because ALL and BL both have the potential to yield significant health gains at a low cost per patient treated. The key findings were as follows: the 3:1 cost/DALY prevented to GDP/capita ratio for ALL in Brazil was US $771,225; expenditures below this threshold were cost effective. Costs below US $257,075 (1:1 ratio) were considered very cost effective. Analogous thresholds for BL in Malawi were US $42,729 and US $14,243. Actual costs were far less. In Brazil, US $16,700 was spent to treat each patient while in Malawi total drug costs were less than US $50 per child. In summary, treatment of certain paediatric cancers in LMIC is very cost effective. Future research should evaluate actual treatment and infrastructure expenditures to help guide policymakers.

Socioeconomic constraints to effective management of Burkitt's lymphoma in south-eastern Nigeria.

This paper presents health outcomes and associated socioeconomic factors of 41 children admitted to a tertiary care institution in south-east Nigeria with Burkitt's lymphoma (BL) between 1987 and 2004. BL responds well to chemotherapy and does not pose a significant threat to health in industrialized nations. However, in resource-poor settings where it is endemic, socioeconomic factors significantly affect access to care for affected children, making this readily treatable condition a cause of considerable distress and early death in affected children. Half of the children reported in this paper presented with late stage disease. Although laboratory facilities were available, they were not accessible to all the children. Nearly a quarter of parents of these children could not afford the cost of confirmatory tests, and about a fifth (n = 8; 19.5%) of the children received no chemotherapy because of their parents' inability to pay. Only 21 of 41 children (51.2%) remained on treatment long enough (at least 12 weeks) to enable them to be confirmed either as short-term cure (n = 9; 64.3%), or as early relapse (n = 2; 4.9%). Owing to financial constraint, 13 of the parents (31.7%) withdrew their children against medical advice (n = 7; 17.1%) or left the hospital (n = 6; 14.6%). To address the challenge posed by these factors, we call for the establishment of a regional BL programme in Africa to help establish a critical mass of resources (human and material) to facilitate the development of an effective and accessible control programme in the region.

Malawi pilot study of Burkitt lymphoma treatment.

BACKGROUND: Burkitt lymphoma (BL) accounts for 50% of childhood cancer in Malawi. Lack of resources precludes the use of new successful treatment approaches such as the LMB 89 group B protocol, which cures >80% of children with stage III BL with high dose chemotherapy and matching supportive care. Our objective was to achieve a good cure rate in Murphy stage I-III BL with manageable toxicity in Malawi at a drug cost of <1000 US dollars per patient. PROCEDURE: The intensity and toxicity of the LMB 89 group B protocol was reduced and adapted to Malawi realities. All stages received the same treatment. Children with suspected BL in the period July 1997-November 1999 were subjected to abdominal ultrasound, a tumor biopsy and/or fine needle aspirate (FNA) and bone marrow (BM), cerebrospinal fluid (CSF), and peripheral blood examination. HIV seropositive children were excluded. Endpoints are projected event free survival (EFS) at minimum 1 year, blood and gastro-intestinal tract toxicity, and risk for and severity of infections. RESULTS: Forty-four children were eligible for treatment and analysis. Their median age was 7.2 years, M:F ratio 1.4:1 with 10 stage I, 5 stage II, and 29 stage III patients. Projected Kaplan-Meier EFS for all was 57% (CI 41-73) at 1 year with 90% EFS in stage I and 52% EFS in stage III. The survival curve remained stable at 500 days. Toxicity and delays in appropriate supportive care contributed to ten deaths during treatment. Local recurrent tumor caused five and CNS recurrence one death. Two children died from progressive disease. The incidence of severe (grade 3 and/or 4) hematologic toxicity varied from 13% to 36%, gastro-intestinal toxicity (GIT) from 2% to 17%, and infections from 7% to 41% per chemotherapy module. CONCLUSIONS: It is possible to administer less intense and less costly multiagent chemotherapy to children with BL in a developing society with acceptable EFS rates. Adequate supportive care of the at-times associated severe toxicity must be made available to better the results.