Severe multifocal form of buruli ulcer after streptomycin and rifampin treatment: comments on possible dissemination mechanisms.

Buruli ulcer (BU), a disease caused by Mycobacterium ulcerans, leads to the destruction of skin and sometimes bone. Here, we report a case of severe multifocal BU with osteomyelitis in a 6-year-old human immunodeficiency virus (HIV)-negative boy. Such disseminated forms are poorly documented and generally occur in patients with HIV co-infection. The advent of antibiotic treatment with streptomycin (S) and rifampin (R) raised hope that these multifocal BU cases could be reduced. The present case raises two relevant points about multifocal BU: the mechanism of dissemination that leads to the development of multiple foci and the difficulties of treatment of multifocal forms of BU. Biochemical (hypoproteinemia), hematological (anemia), clinical (traditional treatment), and genetic factors are discussed as possible risk factors for dissemination.

Family relationship, water contact and occurrence of Buruli ulcer in Benin.

BACKGROUND: Mycobacterium ulcerans disease (Buruli ulcer) is the most widespread mycobacterial disease in the world after leprosy and tuberculosis. How M. ulcerans is introduced into the skin of humans remains unclear, but it appears that individuals living in the same environment may have different susceptibilities. OBJECTIVES: This study aims to determine whether frequent contacts with natural water sources, family relationship or the practice of consanguineous marriages are associated with the occurrence of Buruli ulcer (BU). DESIGN: Case control study. SETTING: Department of Atlantique, Benin. SUBJECTS: BU-confirmed cases that were diagnosed and followed up at the BU detection and treatment center (CDTUB) of Allada (Department of the Atlantique, Benin) during the period from January 1st, 2006, to June 30th, 2008, with three matched controls (persons who had no signs or symptoms of active or inactive BU) for age, gender and village of residence per case. MAIN OUTCOMES MEASURED: Contact with natural water sources, BU history in the family and the practice of consanguineous marriages. RESULTS: A total of 416 participants were included in this study, including 104 cases and 312 controls. BU history in the family (p<0.001), adjusted by daily contact with a natural water source (p = 0.007), was significantly associated with higher odds of having BU (OR; 95% CI = 5.5; 3.0-10.0). The practice of consanguineous marriage was not associated with the occurrence of BU (p = 0.40). Mendelian disorders could explain this finding, which may influence individual susceptibility by impairing immunity. CONCLUSION: This study suggests that a combination of genetic factors and behavioral risk factors may increase the susceptibility for developing BU.

Setting up a national reference laboratory for Buruli ulcer: the case of Benin.

OBJECTIVE: To report the experience of Benin, where Buruli ulcer (BU) is endemic, in the implementation of diagnostic laboratory services. METHODS AND RESULTS: There has been a gradual introduction of biologic diagnostic activities for BU comprising (1) training of a laboratory technician in a highly experienced reference laboratory; (2) acquiring indispensable laboratory start-up materials; (3) progressive development of diagnostic laboratory activities; (4) regular external quality assessment with an experienced reference laboratory and (5) decentralization of activities to various clinical diagnostic and treatment centres for BU in Benin. CONCLUSION: Setting up a reference laboratory for BU is a continuous process, which necessitates motivated personnel and the cooperation of an experienced external reference laboratory.