Bilateral Cystic Neutrophilic Granulomatous Mastitis Due to Corynebacterium tuberculostearicum

Abstract

Introduction Cystic neutrophilic granulomatous mastitis (CNGM) is a form of breast lobulocentric granulomatous inflammation characterized by cystic spaces of dissolved lipid, surrounded by neutrophils. Corynebacterium is frequently the etiological agent, specially C kroppenstedtii. It is often painful and can simulate malignancy by clinical and imaging features. Clinical Case A 36-year-old woman, three pregnancies with puerperal mastitis in all. Consults by increase in left breast volume, painful, 1-month evolution, with erythema and not quantified fever. With antibiotherapy, erythema resolved, but the mass persisted. Ultrasound showed a 28-mm right nodule and extensive left architecture distortion, hypoechogenic, poorly delimited, which compromised superior quadrants, both BIRADS 4. Bilateral core biopsies had typical findings of CNGM, with bacteria inside cysts, identified with HE, which were Gram positive. The cultures gave Corynebacterium, typified as tuberculostearicum by MALDI-TOF methodology, with antibiotic multisensitivity. After 6 weeks of evolution, the last two with ciprofloxacin, on the left the mass had decreased, with 3-cm periareolar collection. Right lesion fistulized on the biopsy path. Conclusions We present a case of bilateral cystic neutrophilic granulomatous mastitis with clinical and imaging features suspected of malignancy. After 6 weeks of evolution, the last two with ciprofloxacin therapy, she had reduction of volume of the larger lesion and fistulization contralateral. She would probably require in its management, in addition to surgery to resolve the fistula, use of lipophilic antibiotics and corticosteroids.

Cystic Neutrophilic Granulomatous Mastitis: A Clinicopathological Study With 16s rRNA Sequencing for the Detection of Corynebacteria in Formalin-Fixed Paraffin-Embedded Tissue

“…The term “cystic neutrophilic granulomatous mastitis” was used by Renshaw et al 3 in 2011 for a lesion that had been described in 2003 by Taylor et al 1 Subsequent to the 2011 article, 3 there have been case series 4,12-20 and sporadic case reports. 6,21-44 Interestingly, however, a report of breast abscess due to C renale had been published as early as 1981, 45 and the association of corynebacteria with granulomatous mastitis had been made by Binelli et al in 1996. 22 Paviour et al 2 and Taylor et al 1 demonstrated Gram-positive bacilli confined to cystic spaces on sections and isolated Corynebacterium species in culture, in women with inflammatory breast disease.…”

“…CNGM appears to be more common in certain ethnic groups like Maoris, Pacific Islanders, Hispanics, Chileans, and Asians compared with Caucasians 1,2,12-20 and usually affects parous women in the postlactational period. While C kroppenstedtii has emerged as the predominant causative species, 1,2,4-6,16 other species associated with CNGM include C tuberculostearicum , 1,2,35,40,43 C accolens , 1,28 C amycolatum , 1,2,15,24,30 C minutissimum , 1,21 C freneyi , 46 C striatum , 15,23 C bovis , 16,41 C jeikeium , 44 and C glucuronolyticum . 46 Of these, C amycolatum, C minutissimum, C glucuronolyticum , and C freneyi are non-lipophilic, 46 with all others being lipophilic.…”