Cystic neutrophilic granulomatous mastitis: The Cleveland Clinic experience with diagnosis and management

Gautham, Ila; Radford, Diane M.; Kovacs, Christopher S.; Calhoun, Benjamin C.; Procop, Gary W.; Shepardson, Laura B.; Dawson, Andrea E.; Downs‐Kelly, Erinn; Zhang, Gloria; Al‐Hilli, Zahraa; Fanning, Alicia; Wilson, Deborah A.; Sturgis, Charles D.

doi:10.1111/tbj.13160

Cited by 42 publications

(63 citation statements)

References 20 publications

(60 reference statements)

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“…Regarding the demographics of patients with CNGM, our cohort has similarities compared with other case series (parous females of reproductive age). [1][2][3][4][5][6][7][8][9][10] All our patients were female with a mean age of 39.8 years. The majority were parous.…”

Section: Discussionmentioning

confidence: 99%

“…As emphasised by many authors, it can be extremely difficult to identify the causative pathogens in surgical pathology slides from cases of CNGM. [1][2][3][4][5][6][7][8][9][10] The organisms are typically lipophilic Gram-positive members of the Corynebacterium spp., which are found most often within the cyst-like lipid droplets. [1][2][3][4][5][6][7][8][9][10] The organisms may be scarce or clustered within the lipid vacuoles.…”

Section: Discussionmentioning

confidence: 99%

“…3 CNGM has a distinct histopathological pattern characterised by neutrophilic and granulomatous inflammation surrounding clear lipid-filled cyst-like spaces. [1][2][3][4][5][6][7][8][9][10] Sparse clusters of bacteria, typically Gram-positive Corynebacterium spp. (usually C. kroppenstedtii), may be seen localised within the lipid-filled spaces with Gram stain.…”

Section: Introductionmentioning

confidence: 99%

“…(usually C. kroppenstedtii), may be seen localised within the lipid-filled spaces with Gram stain. [1][2][3][4][5][6][7][8][9][10] Corynebacteria are Gram-positive bacilli and a component of normal skin flora, some of which are lipophylic. 1,4 It has been postulated that the bacterium may gain entry to breast tissue via lactiferous ducts during lactation and proliferate within static secretory material and fatty stromal tissue.…”

Section: Introductionmentioning

confidence: 99%

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Cystic neutrophilic granulomatous mastitis – a review of 12 consecutive cases

et al. 2020

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Cystic neutrophilic granulomatous mastitisa review of 12 consecutive cases Aims: Cystic neutrophilic granulomatous mastitis (CNGM) is an uncommon but increasingly recognised cause of mastitis, often associated with Corynebacterium ssp. infection. We studied the histopathological and clinical features of CNGM in a Canadian setting, and the work-up required to identify pathogenic microorganisms. Methods and results: A retrospective search for breast specimens with abscess, acute, chronic and/or granulomatous inflammation from 1998 to 2018 was performed. Haematoxylin and eosin slides were reviewed for typical histological features of CNGM. Histochemically stained slides for microorganisms were also reviewed. Repeat Gram stains were performed if initially negative. Electronic medical records were abstracted for microbiology results and relevant clinical data. Twelve cases were identified. All were female, aged 25-57 years, mainly Caucasian, with one Venezuelan and two of Chinese ethnicity. Most were parous (10 of 12); five of 12 had an endocrinopathy. Bacteria were identified in one or more specimens from eight of 12 patients; additional Gram stains revealed organisms in four of 12 cases. Of four bacterial cultures, one grew Corynebacterium kroppenstedtii. 16S polymerase chain reaction for three samples was negative. Two patients had multiple breast biopsies, showing early palisaded granulomas followed by classic features of CNGM. The patients had various management approaches, including surgery and antimicrobials. Conclusions: CNGM may present as palisaded granulomatous inflammation, without the expected 'cystic' pattern, suggesting that there is an evolution of histomorphology with this infection. Most patients with CNGM are parous, and there may be an association with endocrinopathies. Application of multiple Gram stains increases the yield of microorganism identification. Recognition of CNGM in breast biopsies and collaborative communications are essential to direct appropriate therapy.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Cystic neutrophilic granulomatous mastitis – a review of 12 consecutive cases

et al. 2020

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“…Two main groups of breast diseases may share granulomatous patterns: breast cancer and infectious diseases. Among granulomatous breast infections, tuberculosis mastitis is the most common [71] ( Figure 3C), followed by various infections (cat-scratch disease, Proprionibacterium, or Corynebacterium) [72,73]. A third etiologic group is represented by autoimmune diseases and includes well-defined clinicopathological granulomatous diseases such as Crohn's disease, granulomatosis with polyangiitis (Wegener's disease), or sarcoidosis, as well as more recently described conditions such as IgG4-related disease, systemic erythematosus lupus, or SjS [74][75][76].…”

Section: Granulomatous Mastitismentioning

confidence: 99%

Mastitis in Autoimmune Diseases: Review of the Literature, Diagnostic Pathway, and Pathophysiological Key Players

Goulabchand

Hafidi

Perre

et al. 2020

JCM

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Mastitis frequently affects women of childbearing age. Of all the pathological breast conditions requiring specific management, autoimmune mastitis is in the third position after infection and breast cancer. The aim of this literature review was to make a comprehensive description of autoimmune diseases targeting the mammary gland. Four main histological patterns of autoimmune mastitis are described: (i) lymphocytic infiltrates; (ii) ductal ectasia; (iii) granulomatous mastitis; and (iv) vasculitis. Our literature search found that all types of autoimmune disease may target the mammary gland: organ-specific diseases (diabetes, thyroiditis); connective tissue diseases (such as systemic erythematosus lupus or Sjögren’s syndrome); vasculitides (granulomatosis with polyangiitis, eosinophilic granulomatosis with polyangiitis, giant cell arteritis, polyarteritis nodosa, Behçet’s disease); granulomatous diseases (sarcoidosis, Crohn’s disease); and IgG4-related disease. Cases of breast-specific autoimmune diseases have also been reported, including idiopathic granulomatous mastitis. These breast-limited inflammatory diseases are sometimes the first symptom of a systemic autoimmune disease. Although autoimmune mastitis is rare, it is probably underdiagnosed or misdiagnosed. Early diagnosis may allow us to detect systemic diseases at an earlier stage, which could help to initiate a prompt, appropriate therapeutic strategy. In case of suspected autoimmune mastitis, we hereby propose a diagnostic pathway and discuss the potential pathophysiological pathways leading to autoimmune breast damage.

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