title: Findings in patients from Benin with osteomyelitis and polymerase chain reaction-confirmed Mycobacterium ulcerans infection.
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.
authors.affiliation: Emerging Diseases Epidemiology Unit, Institut Pasteur.
authors.forename: Virginie
authors.initials: V
authors.lastname: Pommelet
authors.affiliation: 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.
authors.forename: Quentin B
authors.initials: QB
authors.lastname: Vincent
authors.affiliation: Centre de Diagnostic et de Traitement de la Lèpre et de l'Ulcère de Buruli, Fondation Raoul Follereau, Pobè, Bénin.
authors.forename: Marie-Françoise
authors.initials: MF
authors.lastname: Ardant
authors.affiliation: Centre de Diagnostic et de Traitement de la Lèpre et de l'Ulcère de Buruli, Fondation Raoul Follereau, Pobè, Bénin.
authors.forename: Ambroise
authors.initials: A
authors.lastname: Adeye
authors.affiliation: Department of Pediatric Radiology, Robert Debré Children University Hospital, Assistance Publique-Hôpitaux de Paris.
authors.forename: Anca
authors.initials: A
authors.lastname: Tanase
authors.affiliation: Emerging Diseases Epidemiology Unit, Institut Pasteur.
authors.forename: Laura
authors.initials: L
authors.lastname: Tondeur
authors.affiliation: Department of Pediatric Radiology, Robert Debré Children University Hospital, Assistance Publique-Hôpitaux de Paris.
authors.forename: Adelaide
authors.initials: A
authors.lastname: Rega
authors.affiliation: Emerging Diseases Epidemiology Unit, Institut Pasteur.
authors.forename: Jordi
authors.initials: J
authors.lastname: Landier
authors.affiliation: 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.
authors.forename: Estelle
authors.initials: E
authors.lastname: Marion
authors.affiliation: 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.
authors.forename: Alexandre
authors.initials: A
authors.lastname: Alcaïs
authors.affiliation: ATOMycA, Inserm Avenir Team, CRCNA, Inserm U892, 6299 CNRS, Université et CHU LUNAM, Université d'Angers, France.
authors.forename: Laurent
authors.initials: L
authors.lastname: Marsollier
authors.affiliation: Emerging Diseases Epidemiology Unit, Institut Pasteur Conservatoire National des Arts et Métiers, Paris.
authors.forename: Arnaud
authors.initials: A
authors.lastname: Fontanet
authors.affiliation: Centre de Diagnostic et de Traitement de la Lèpre et de l'Ulcère de Buruli, Fondation Raoul Follereau, Pobè, Bénin.
authors.forename: Annick
authors.initials: A
authors.lastname: Chauty
mesh_terms: D000293:Adolescent; D001541:Benin / Q000453:epidemiology; D054312:Buruli Ulcer / Q000453:epidemiology* / Q000382:microbiology / Q000473:pathology; D016022:Case-Control Studies; D002648:Child; D005260:Female; D006801:Humans; D008297:Male; D019911:Mycobacterium ulcerans / Q000235:genetics* / Q000302:isolation & purification; D010019:Osteomyelitis / Q000453:epidemiology* / Q000382:microbiology / Q000473:pathology; D016133:Polymerase Chain Reaction; D012189:Retrospective Studies
keywords: Buruli ulcer; Mycobacterium ulcerans; osteomyelitis