A clinicopathological review of 34 cases of inflammatory breast disease showing an association between corynebacteria infection and granulomatous mastitis
ABSTRACT Granulomatous mastitis is a rare condition of unknown aetiology. The great majority of cases has not been associated with bacterial pathogens if women with mammary tuberculosis are excluded. We noted that some women in Auckland with a histological diagnosis of granulomatous mastitis had both microbiological and histological evidence of corynebacteria infection and aimed to study this further.
Thirty-four women were reviewed who presented with inflammatory breast disease and had microbiological specimens from which corynebacteria were isolated and/or histological specimens containing coryneform bacteria. These 34 cases were compared with 28 controls with similar histology but no evidence of corynebacteria infection.
Twenty-seven (79%) of the cases and 21 (75%) of the controls had histological and/or cytological evidence of suppurative granulomas. Fourteen of the 34 cases also had Gram-positive bacilli (GPB), recognisable as coryneform bacteria, in histological sections. In all cases the bacilli were confined to empty spaces, consistent with dissolved lipid, and were surrounded by neutrophils and, frequently, suppurative granulomas. Corynebacterium species were isolated from 52 of 116 microbiological specimens taken from the 34 cases. Forty of these 52 cultures were pure. Twenty-four of the cultures were further classified biochemically and using 16S rRNA gene sequencing. Twenty of the 24 were lipophilic Corynebacterium species and 14 were identified as Corynebacterium kroppenstedtii. The cases were more likely to present with fever or neutrophilia and more often formed sinuses than the controls but other clinical features were similar. Maori and Pacific Islanders accounted for 77% of the women across both groups.
We suggest granulomatous mastitis can be associated with corynebacteria infection, particularly infection by C. kroppenstedtii. The significance of this finding, which has previously been described in only a single case report, is discussed.
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ABSTRACT: Corynebacterium kroppenstedtii is a lipophilic corynebacterial species that lacks in the cell envelope the characteristic alpha-alkyl-beta-hydroxy long-chain fatty acids, designated mycolic acids. We report here the bioinformatic analysis of genome data obtained by pyrosequencing of the type strain C. kroppenstedtii DSM44385 that was initially isolated from human sputum. A single run with the Genome Sequencer FLX system revealed 560,248 shotgun reads with 110,018,974 detected bases that were assembled into a contiguous genomic sequence with a total size of 2,446,804bp. Automatic annotation of the complete genome sequence resulted in the prediction of 2122 coding sequences, of which 29% were considered as specific for C. kroppenstedtii when compared with predicted proteins from hitherto sequenced pathogenic corynebacteria. This comparative content analysis of the genome data revealed a large repertoire of genes involved in sugar uptake and central carbohydrate metabolism and the presence of the mevalonate route for isoprenoid biosynthesis. The lack of mycolic acids and the lipophilic lifestyle of C. kroppenstedtii are apparently caused by gene loss, including a condensase gene cluster, a mycolate reductase gene, and a microbial type I fatty acid synthase gene. A complete beta-oxidation pathway involved in the degradation of fatty acids is present in the genome. Evaluation of the genomic data indicated that lipophilism is the dominant feature involved in pathogenicity of C. kroppenstedtii.Journal of Biotechnology 04/2008; 136(1-2):22-30. DOI:10.1016/j.jbiotec.2008.03.004 · 2.88 Impact Factor
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ABSTRACT: Introducción: La mastitis granulomatosa idiopática es una enfermedad inflamatoria benigna, infrecuente, de etiología desconocida. Puede simular carcinoma mamario, por lo cual su diagnóstico definitivo es histopatológico. No existe consenso respecto a su tratamiento. Realizamos una caracterización clínica de esta patología considerando su forma de presentación, métodos diagnósticos y tratamiento con especial énfasis en la efectividad de la corticoterapia. Material y método: Estudio retrospectivo realizado mediante protocolo tipo. Se revisaron los registros y fichas clínicas de los pacientes manejados con diagnóstico de mastitis granulomatosa idiopática en el Hospital Regional Valdivia entre los años 1995-2006. Resultados: La serie corresponde a 14 pacientes de sexo femenino. El promedio de edad fue de 31,6 años. La presentación clínica más frecuente fue absceso mamario en 9/14. Se realizó estudio imagenológico en 11/ 14 casos mediante mamografía en 7/14 y ecotomografía en 10/14. Las muestras para estudio histopatológico fueron obtenidas por biopsia percutánea en 8/14 y quirúrgica en 6/14. Se realizó tratamiento corticoidal con prednisona en 12/14 pacientes logrando buena respuesta en todos los casos. Se presentaron 2 recidivas durante el seguimiento que comprende un promedio de 28 meses. Conclusiones: La presentación clínica de las mastitis fue similar a la reportada en la literatura. La respuesta al tratamiento corticoesteroidal logró el control de la enfermedad en todos los casos. Las reacciones adversas al tratamiento fueron menores y las 2 recidivas fueron de menor magnitud y duración respecto al cuadro inicial.01/2007; 59(4). DOI:10.4067/S0718-40262007000400003
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ABSTRACT: Tuberculosis of the breast is an uncommon disease with non-specific clinical, radiological and histological findings. Misdiagnosis is common as biopsy specimens are pauci-bacillary and investigations such as microscopy and culture are frequently negative. We report a case of a breast abscess in a 34-year old Bangladeshi woman attributed to tuberculosis infection. Equivocal histology, negative Ziehl-Neelsen stain and culture for acid-fast bacilli resulted in the abscess initially being diagnosed as granulomatous mastitis and treated accordingly. However failure to respond to therapy raised suspicion of culture negative breast tuberculosis. Treatment with standard antituberculosis drugs was associated with complete resolution of the breast abscess. This case highlights the difficulty in differentiating culture negative tuberculosis from granulomatous mastitis and the importance of a high index of clinical suspicion.Cases Journal 11/2008; 1(1):273. DOI:10.1186/1757-1626-1-273