Volume 83, Issue 2
  • ISSN: 0002-9637
  • E-ISSN: 1476-1645



Buruli ulcer (BU), a disease caused by , 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.


Article metrics loading...

The graphs shown below represent data from March 2017
Loading full text...

Full text loading...



  1. Lagarrigue V, Portaels F, Meyers WM, Aguiar J, , 2000. Buruli ulcer: risk of bone involvement! A propos of 33 cases observed in Benin. Méd Trop 60: 262266 [Google Scholar]
  2. Portaels F, Zinsou C, Aguiar J, Debacker M, de Biurrun E, Guédénon A, Josse R, Lagarrigue V, Silva MT, Steunou C, Meyers WM, , 2003. Les atteintes osseuses de l'ulcère de Buruli: à propos de 73 cas. Bull Seances Acad R Sci Outre Mer 49: 161190. [Google Scholar]
  3. World Health Organization Portaels F, Johnson P, Meyers WM, , 2001. Buruli ulcer. Diagnosis of Mycobacterium ulcerans disease . , eds. A Manual for Health Care Providers. Geneva: WHO/CDS/CPE/GBUI/2001.4. [Google Scholar]
  4. Walsh DS, Portaels F, Meyers WM, , 2008. Buruli ulcer (Mycobacterium ulcerans infection). Trans R Soc Trop Med Hyg 102: 969978.[Crossref] [Google Scholar]
  5. Debacker M, Aguiar J, Steunou C, Zinsou C, Meyers WM, Guédénon A, Scott JT, Dramaix M, Portaels F, , 2004. Mycobacterium ulcerans disease (Buruli ulcer) in rural hospital, southern Benin, 1997–2001. Emerg Infect Dis 10: 13911398.[Crossref] [Google Scholar]
  6. Portaels F, Silva MT, Meyers WM, , 2009. Buruli ulcer. Clin Dermatol 27: 291305.[Crossref] [Google Scholar]
  7. Johnson RC, Ifebe D, Hans-Moevi A, Kestens L, Houessou R, Guédénon A, Meyers WM, Portaels F, , 2002. Disseminated Mycobacterium ulcerans disease in an HIV-positive patient: a case study. AIDS 16: 17041705.[Crossref] [Google Scholar]
  8. Toll A, Gallardo F, Ferran M, Gilaberte M, Iglesias M, Gimeno JL, Rondini S, Pujol RM, , 2005. Aggressive multifocal Buruli ulcer with associated osteomyelitis in an HIV-positive patient. Clin Exp Dermatol 30: 649651.[Crossref] [Google Scholar]
  9. Ouattara D, Meningaud JP, Saliba F, , 2002. Multifocal forms of Buruli ulcer: clinical aspects and management difficulties in 11 cases. Bull Soc Pathol Exot 95: 287291 [Google Scholar]
  10. Eddyani M, Fraga AG, Schmitt F, Uwizeye C, Fissette K, Johnson C, Aguiar J, Sopoh G, Barogui Y, Meyers WM, Pedrosa J, Portaels F, , 2009. Fine needle aspiration, an efficient sampling technique for bacteriological diagnosis of non ulcerative Buruli ulcer. J Clin Microbiol 47: 17001704.[Crossref] [Google Scholar]
  11. World Health Organization, 2004. Provisional guidance on the role of specific antibiotics in the management of Mycobacterium ulcerans disease (Buruli ulcer). Geneve: WHO/CDS/CPE/GBUI.10. [Google Scholar]
  12. World Health Organization, 2008. Buruli ulcer: progress report, 2004–2008. Wkly Epidemiol Rec 83: 145146. Available at: http://whqlibdoc.who.int/hq/2001/WHO_CDS_CPE_GBUI_2001.3.pdf. Accessed April 4, 2008. [Google Scholar]
  13. Portaels F, Johnson C, Aguiar J, Meyers WM, Debacker M, , 2008. Etude de 106 cas d'ulcères de Buruli avec atteintes osseuses traités à Zagnanado, Bénin. Bull de l'ALLF 23: 4850. [Google Scholar]
  14. Pszolla N, Sarkar MR, Strecker W, Kern P, Kinzl L, Meyers WM, Portaels F, , 2003. Buruli ulcer: a systemic disease. Clin Infect Dis 37: 7882.[Crossref] [Google Scholar]
  15. Silva MT, Portaels F, Pedrosa JR, , 2009. Pathogenetic mechanisms of the intracellular parasite Mycobacterium ulcerans leading to Buruli ulcer. Lancet Infect Dis 9: 699710.[Crossref] [Google Scholar]
  16. Guarner J, Bartlett J, Whitney EAS, Raghunathan PL, Stienstra Y, Asamoa K, Etuaful S, Klutse E, Quarshie E, van der Werf TS, van der Graaf WT, King CH, Ashford DA, , 2003. Histopathologic features of Mycobacterium ulcerans infection. Emerg Infect Dis 9: 651656.[Crossref] [Google Scholar]
  17. Limbos P, Bretey J, Jadin J, Brutsaert P, , 1961. Sur un cas d'abcès à Mycobacterium fortuitum . Ann Soc Belg Med Trop 2: 127132. [Google Scholar]
  18. Wang CT, Sun JS, Hou SM, , 2000. Mycobacterial infection of the upper extremities. J Formos Med Assoc 99: 710715. [Google Scholar]
  19. Mauvieux L, , 2005/2006. Les hyperéosinophilies. Université Louis Pasteur – Faculté de Médecine - 2005/2006 - DCEM3 - Module 17 - Maladies du Sang et Transfusion- Item 311. Available at: http://www-ulpmed.u-strasbg.fr/medecine/cours_en_ligne/e_cours/module_17/item_311.pdf. Accessed June 4, 2010. [Google Scholar]
  20. Schütte D, Pluschke G, , 2009. Immunosuppression and treatment-associated inflammatory response in patients with Mycobacterium ulcerans infection (Buruli ulcer). Expert Opin Biol Ther 9: 187200.[Crossref] [Google Scholar]
  21. Schütte D, Um-Boock A, Mensah-Quainoo E, Itin P, Schmid P, Pluschke G, , 2007. Development of highly organized lymphoid structures in Buruli ulcer lesions after treatment with rifampicin and streptomycin. PLoS Negl Trop Dis 1: e2.[Crossref] [Google Scholar]
  22. O'Brien DP, Robson ME, Callan PP, McDonald AH, , 2009. “Paradoxical” immune-mediated reactions to Mycobacterium ulcerans during antibiotic treatment: a result of treatment success, not failure. Med J Aust 191: 564566. [Google Scholar]
  23. Kestens L, Seddiki N, Bohjanen PR, , 2009. Immunopathogenesis of immune reconstitution disease in HIV patients responding to antiretroviral therapy. Curr Opin HIV AIDS 3: 419424.[Crossref] [Google Scholar]
  24. Lawn DS, Lockwood DN, , 2007. Leprosy after starting antiretroviral treatment: an increasingly reported clinical problem but not a serious public health risk. BMJ 334: 217218.[Crossref] [Google Scholar]
  25. Corti M, Villafañe MF, Ambroggi M, Sawicki M, Gancedo E, , 2007. Soft tissue abscess and lymphadenitis due to Mycobacterium avium complex as an expression of immune reconstitution inflammatory syndrome after a second scheme of highly active antiretroviral therapy: case report. Rev Inst Med Trop Sao Paulo 49: 267270.[Crossref] [Google Scholar]
  26. Singal A, Mehta S, Pandhi D, , 2006. Immune reconstitution inflammatory syndrome in an HIV seropositive leprosy patient. Lepr Rev 77: 7680. [Google Scholar]
  27. Fock RA, Vinolo MA, Crisma AR, Nakajima K, Rogero MM, Borelli P, , 2008. Protein-energy malnutrition modifies the production of interleukin-10 in response to lipopolysaccharide (LPS) in a murine model. J Nutr Sci Vitaminol (Tokyo) 54: 371377.[Crossref] [Google Scholar]
  28. Rodriguez L, Gonzalez C, Flores L, Jimenez-Zamudio L, Graniel J, Ortiz R, , 2005. Assessment by flow cytometry of cytokine production in malnourished children. Clin Diagn Lab Immunol 12: 502507. [Google Scholar]
  29. Schaible UE, Kaufmann SH, , 2007. Malnutrition and infection: complex mechanisms and global impacts. PLoS Med 4: e115.[Crossref] [Google Scholar]
  30. Funada H, Machi T, Matsuda T, , 1991. Disseminated mycobacteriosis in patients with severe hematologic disorders. Kansenshogaku Zasshi 65: 12971303.[Crossref] [Google Scholar]
  31. Skinsnes LK, Higa LH, , 1976. The role of protein malnutrition in the pathogenesis of ulcerative “Lazarine” leprosy. Int J Lepr Other Mycobact Dis 44: 346358. [Google Scholar]
  32. Stienstra Y, Van Der Werf TS, Oosterom E, Nolte IM, van der Graaf WT, Etuaful S, Raghunathan PL, Whitney EA, Ampadu EO, Asamoa K, Klutse EY, te Meerman GJ, Tappero JW, Ashford DA, van der Steege G, , 2006. Susceptibility to Buruli ulcer is associated with the SLC11A1 (NRAMP1) D543N polymorphism. Genes Immun 7: 185189.[Crossref] [Google Scholar]
  33. Mulder AA, Boerma RP, Barogui Y, Zinsou C, Johnson RC, Gbovi J, van der Werf TS, Stienstra Y, , 2008. Healthcare seeking behaviour for Buruli ulcer in Benin: a model to capture therapy choice of patients and healthy community members. Trans R Soc Trop Med Hyg 102: 912920.[Crossref] [Google Scholar]
  34. Kibadi K, Stragier P, Muyembe-Tamfum JJ, Pedrosa J, Portaels F, , 2008. Follow-up of the first case of Mycobacterium ulcerans infection documented by PCR, genotyping and culture in the Republic of Congo-Brazzaville]. Méd Trop 68: 137143. [Google Scholar]

Data & Media loading...

  • Received : 13 Oct 2009
  • Accepted : 27 Apr 2010
  • Published online : 05 Aug 2010

Most Cited This Month

This is a required field
Please enter a valid email address
Approval was a Success
Invalid data
An Error Occurred
Approval was partially successful, following selected items could not be processed due to error