A clinicopathological review of 34 cases of inflammatory breast disease showing an association between corynebacteria infection and granulomatous mastitis. Pathology

Department of Anatomical Pathology, Labplus, Auckland Hospital, Park Road, PB 92024, Auckland 1, New Zealand.
Pathology (Impact Factor: 2.19). 05/2003; 35(2):109-19. DOI: 10.1080/0031302031000082197
Source: PubMed


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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    • "Its first isolation was described in 1998 from human sputum [1]. It has also been found in association with inflammatory breast diseases [2]. In 2003, Taylor et al. observed that the incidence of corynebacteria-associated inflammatory breast diseases was much higher in Maori women than women of European descent in whom this type of infection is rare [2]. "
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    ABSTRACT: Background. Corynebacterium kroppenstedtii (Ck) was first described in 1998 from human sputum. Contrary to what is observed in ethnic groups such as Maori, Ck is rarely isolated from breast abscesses and granulomatous mastitis in Caucasian women. Case Presentation. We herein report a case of recurrent breast abscesses in a 46-year-old Caucasian woman. Conclusion. In the case of recurrent breast abscesses, even in Caucasian women, the possible involvement of Ck should be investigated. The current lack of such investigations, probably due to the difficulty to detect Ck, may cause the underestimation of such an aetiology.
    Full-text · Article · Sep 2012
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    • "). The ability of C. kroppenstedtii to multiply in fat globules obviously links its lipophilic metabolism with pathogenicity by providing access to exogenous fatty acids for growth and thereby escaping the neutrophil response (Paviour et al., 2002; Taylor et al., 2003). The persistence of the bacterium in fat globules also limits the use of antimicrobials for the treatment of C. kroppenstedtii infections, since only the lipophilic antibiotic doxycycline turned out to be effective in vivo (Skinner et al., 2007). "
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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.
    Full-text · Article · Apr 2008 · Journal of Biotechnology
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    • "The women in our series were included because they had corynebacteria isolated from specimens; therefore, there is a bias in the way these patients were identified. However, when patients with the histological diagnosis of GLM were reviewed[10], corynebacteria accounted for 140% of the isolates recovered and far outnumbered any other single isolate. The next most common isolates were Enterobacteriaceae (14% of isolates), Staphylococcus aureus (13% of isolates), coagulasenegative staphylococci (14% of isolates), and P. acnes (6% of isolates). "
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    ABSTRACT: Corynebacteria were isolated from breast tissue, pus, or deep wound swabs of 24 women; the most common species isolated was the newly described Corynebacterium kroppenstedtii, followed by Corynebacterium amycolatum and Corynebacterium tuberculostearicum. Gram-positive bacilli were seen in samples sent for culture or in histological specimens for 12 women, and 9 of the 12 women from whom adequate histological specimens were obtained had conditions that met the criteria for granulomatous lobular mastitis, a chronic inflammatory disease of unknown etiology.
    Full-text · Article · Jan 2003 · Clinical Infectious Diseases
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