Association between haemoglobin variants S and C and Mycobacterium ulcerans disease (Buruli ulcer): a case‐control study in Benin

• Published in: Tropical medicine & international health Vol. 12; no. 4; pp. 511 - 518
• Main Authors: Nackers, Fabienne, Tonglet, René, Slachmuylder, Véronique, Johnson, Roch C, Robert, Annie, Zinsou, Claude, Glynn, Judith R, Portaels, Françoise, Gala, Jean‐Luc
• Format: Journal Article
• Language: English
• Published: Oxford, UK Blackwell Publishing Ltd 01.04.2007; Blackwell Science
• Subjects: Adolescent; Adult; Age Distribution; Aged; Aged, 80 and over; Anemia, Sickle Cell - complications; Anemia, Sickle Cell - epidemiology; Anemia, Sickle Cell - genetics; Bacteria; Bacterial arthritis and osteitis; Bacterial diseases; Benin; Benin - epidemiology; Biological and medical sciences; Bénin; Case studies; Case-Control Studies; Child; Child, Preschool; Estudios caso‐control; Female; Gene Frequency; General aspects; Genetic testing; Genotype; Genotype & phenotype; Haemoglobin C; Haemoglobin S; Hemoglobin; Hemoglobin C - genetics; Hemoglobin C Disease - complications; Hemoglobin C Disease - epidemiology; Hemoglobin C Disease - genetics; Hemoglobin, Sickle - genetics; Hemoglobina C; Hemoglobina S; Hemoglobins - genetics; Human bacterial diseases; Humans; hémoglobine C; hémoglobine S; Infant; Infectious diseases; Male; Medical sciences; Middle Aged; Mycobacterium Infections, Nontuberculous - complications; Mycobacterium Infections, Nontuberculous - epidemiology; Mycobacterium Infections, Nontuberculous - genetics; Mycobacterium ulcerans; Mycobacterium ulcerans - genetics; Osteomielitis; Osteomyelitis; Osteomyelitis - complications; Osteomyelitis - epidemiology; Osteomyelitis - genetics; ostéomyélite; Risk Factors; Sex Distribution; Tropical diseases; Tuberculosis and atypical mycobacterial infections; Ulcers; Études cas‐témoins; Benin; Africa; Benin; Haemoglobin C; Skin disease; Diseases of the osteoarticular system; Mycobacterial infection; Case control study; Epidemiology; Case-Control Studies; Infection; Variant; Osteomyelitis; Haemoglobin S; Mycobacterium ulcerans; Association; Mycobacteriales; Buruli ulcer; Osteitis; Bacteriosis; Mycobacteriaceae; Hemoglobin; Bacteria; Tropical medicine; Actinomycetes; Public health
• ISSN: 1360-2276; 1365-3156
• DOI: 10.1111/j.1365-3156.2006.01808.x

Summary Risk factors for Buruli ulcer (BU) are poorly understood. We conducted a case‐control study in southern Benin to investigate the association between haemoglobin variants S or C and BU, and particularly the association between haemoglobinopathies HbSS/SC and BU osteomyelitis. We compared the haemoglobin genotype of 179 patients with BU and 44 with BU osteomyelitis to that of 242 community controls. We found no evidence of an increased risk of BU according to the presence of haemoglobin variants S and/or C [odds ratio adjusted for sex, age, region of residence and ethnicity: 1.24 (95%CI: 0.80–1.93), P = 0.34]. Haemoglobin variants S and C are unlikely to play a role in the BU burden. However, haemoglobinopathies HbSS/SC were more frequent among BU osteomyelitis patients than among controls (6.8%vs. 1.0%, Fisher's exact P‐value = 0.045), which may suggest that those disorders facilitate growth of Mycobacterium ulcerans in the bone matrix. Les facteurs de risque de l'ulcère de Buruli sont peu connus. Nous avons mené une étude cas‐témoins dans le sud du Bénin pour investiguer l'association entre les variantes S et C de l'hémoglobine et l'ulcère de Buruli et, en particulier, l'association entre les hémoglobinopathies HbSS/SC et les ostéomyélites àM.ulcerans. Nous avons comparé le génotype de l'hémoglobine de 179 patients atteints d'ulcère de Buruli et de 44 patients avec une ostéomyélite àM.ulceransà celui de 242 témoins de la communauté. Nous n'avons trouvé aucune évidence d'un plus grand risque d'ulcère de Buruli en présence des variantes S et/ou C de l'hémoglobine (OR ajusté pour le sexe, l’âge, la région de résidence et l'appartenance ethnique : 1,24 [IC95% : 0,80–1,93], P = 0,34). Il est peu probable que les variantes S et C de l'hémoglobine jouent un rôle dans le fardeau de l'ulcère de Buruli. Cependant, les hémoglobinopathies HbSS/SC étaient plus fréquentes chez les patients avec une ostéomyélite àM.ulcerans que chez les témoins (6,8% contre 1,0%, P‐valeur du test exact de Fisher = 0,045), ce qui permet de suggérer que ces troubles de l'hémoglobine pourraient faciliter la croissance de M.ulcerans dans le tissu osseux. Los factores de riesgo para la úlcera de Buruli no son bien conocidos. Hemos conducido un estudio de casos y controles en el sur de Benin para investigar la asociación entre las variantes de hemoglobina S o C y la úlcera de Buruli, y en particular la asociación entre hemoglobinopatías HbSS/SC y la ostomielitis por úlcera de Buruli. Hemos comparado el genotipo de hemoglobina de 179 pacientes con úlcera de Buruli y 44 con osteomielitis por úlcera de Buruli, frente a 242 controles de la comunidad. No encontramos evidencia de un riesgo aumentado de úlcera de Buruli según las variantes de hemoglobina S y/o (OR ajustado para sexo, edad, lugar de residencia y etnia 1.24 [95%CI: 0.80–1.93], P = 0.34). Las variantes de hemoglobina S y C no parecen jugar papel alguno en la carga por úlcera de Buruli. Sin embargo, las hemoglobinopatías HbSS/SC eran más frecuentes entre pacientes con osteomielitis por úlcera de Buruli que entre los controles (6.8% versus 1.0%, valor P exacto de Fisher = 0.045), lo cual podría sugerir que estos desórdenes facilitan el crecimiento de M. Ulcerans en la matriz ósea.