Virginie Pommelet1, Quentin B Vincent2, Marie-Françoise Ardant3, Ambroise Adeye3, Anca Tanase4, Laura Tondeur1, Adelaide Rega4, Jordi Landier1, Estelle Marion5, Alexandre Alcaïs6, Laurent Marsollier7, Arnaud Fontanet8, Annick Chauty3. 1. Emerging Diseases Epidemiology Unit, Institut Pasteur. 2. Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Recherche Médicale U980 (INSERM) Université Paris Descartes, Sorbonne Paris Cité, Imagine Institute. 3. Centre de Diagnostic et de Traitement de la Lèpre et de l'Ulcère de Buruli, Fondation Raoul Follereau, Pobè, Bénin. 4. Department of Pediatric Radiology, Robert Debré Children University Hospital, Assistance Publique-Hôpitaux de Paris. 5. Centre de Diagnostic et de Traitement de la Lèpre et de l'Ulcère de Buruli, Fondation Raoul Follereau, Pobè, Bénin ATOMycA, Inserm Avenir Team, CRCNA, Inserm U892, 6299 CNRS, Université et CHU LUNAM, Université d'Angers, France. 6. Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Recherche Médicale U980 (INSERM) Université Paris Descartes, Sorbonne Paris Cité, Imagine Institute CIC-0109 Cochin-Necker Inserm, Unité de Recherche Clinique, Paris Centre Descartes Necker Cochin, Assistance Publique-Hôpitaux de Paris et EA 3620, Université Paris Descartes Conservatoire National des Arts et Métiers, Paris. 7. ATOMycA, Inserm Avenir Team, CRCNA, Inserm U892, 6299 CNRS, Université et CHU LUNAM, Université d'Angers, France. 8. Emerging Diseases Epidemiology Unit, Institut Pasteur Conservatoire National des Arts et Métiers, Paris.
Abstract
BACKGROUND: Mycobacterium ulcerans is known to cause Buruli ulcer (BU), a necrotizing skin disease leading to extensive cutaneous and subcutaneous destruction and functional limitations. However, M. ulcerans infections are not limited to skin, and osteomyelitis, still poorly described in the literature, occurs in numerous young patients in Africa. METHODS: In a retrospective matched case-control study conducted in a highly endemic area in Benin, we analyzed demographic, clinical, biological, and radiological features in all patients with M. ulcerans infections with bone involvement, identified from a cohort of 1257 patients with polymerase chain reaction-proved M. ulcerans infections. RESULTS: The 81 patients studied had a median age of 11 years (interquartile range, 7-16 years) and were predominantly male (male-female ratio, 2:1). Osteomyelitis was observed beneath active BU lesions (60.5%) or at a distance from active or apparently healed BU lesions (14.8%) but also in patients without a history of BU skin lesions (24.7%). These lesions had an insidious course, with nonspecific clinical findings leading to delayed diagnosis. A comparison with findings in 243 age- and sex-matched patients with BU without osteomyelitis showed that case patients were less likely to have received BCG immunization than controls (33.3% vs 52.7%; P = .01). They were also at higher risk of longer hospital stay (118 vs 69 days; P = .001), surgery (92.6% vs 63.0%; P = .001), and long-term crippling sequelae (55.6% vs 15.2%; P < .001). CONCLUSIONS: This study highlighted the difficulties associated with diagnosis of M. ulcerans osteomyelitis, with one-fourth of patients having no apparent history of BU skin lesions, including during the current course of illness. Delays in treatment contributed to the high proportion (55.6%) of patients with crippling sequelae.
BACKGROUND:Mycobacterium ulcerans is known to cause Buruli ulcer (BU), a necrotizing skin disease leading to extensive cutaneous and subcutaneous destruction and functional limitations. However, M. ulcerans infections are not limited to skin, and osteomyelitis, still poorly described in the literature, occurs in numerous young patients in Africa. METHODS: In a retrospective matched case-control study conducted in a highly endemic area in Benin, we analyzed demographic, clinical, biological, and radiological features in all patients with M. ulcerans infections with bone involvement, identified from a cohort of 1257 patients with polymerase chain reaction-proved M. ulcerans infections. RESULTS: The 81 patients studied had a median age of 11 years (interquartile range, 7-16 years) and were predominantly male (male-female ratio, 2:1). Osteomyelitis was observed beneath active BU lesions (60.5%) or at a distance from active or apparently healed BU lesions (14.8%) but also in patients without a history of BU skin lesions (24.7%). These lesions had an insidious course, with nonspecific clinical findings leading to delayed diagnosis. A comparison with findings in 243 age- and sex-matched patients with BU without osteomyelitis showed that case patients were less likely to have received BCG immunization than controls (33.3% vs 52.7%; P = .01). They were also at higher risk of longer hospital stay (118 vs 69 days; P = .001), surgery (92.6% vs 63.0%; P = .001), and long-term crippling sequelae (55.6% vs 15.2%; P < .001). CONCLUSIONS: This study highlighted the difficulties associated with diagnosis of M. ulcerans osteomyelitis, with one-fourth of patients having no apparent history of BU skin lesions, including during the current course of illness. Delays in treatment contributed to the high proportion (55.6%) of patients with crippling sequelae.
Authors: N El Houmami; P Minodier; C Bouvier; H Seligmann; J-L Jouve; D Raoult; P-E Fournier Journal: Eur J Clin Microbiol Infect Dis Date: 2017-01-05 Impact factor: 3.267
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Authors: Carlos Capela; Ghislain E Sopoh; Jean G Houezo; René Fiodessihoué; Ange D Dossou; Patrício Costa; Alexandra G Fraga; João F Menino; Rita Silva-Gomes; Edgard M Ouendo; Fernando Rodrigues; Jorge Pedrosa Journal: PLoS Negl Trop Dis Date: 2015-09-10