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Granulomatous Mastitis Due to Non-Tuberculous Mycobacteria: a Diagnostic and Therapeutic Dilemma

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Granulomatous Mastitis Due to Non-Tuberculous Mycobacteria: a Diagnostic and Therapeutic Dilemma Case Report Granulomatous Mastitis Due to Non-Tuberculous Mycobacteria: A Diagnostic and Therapeutic Dilemma Owais Ahmed Patel * , Girish D. Bakhshi, Amogh R. Nadkarni and Zarin S. Rangwala Grant Government Medical College and Sir JJ Group of Hospitals, Mumbai 400008, India; [email protected] (G.D.B.); [email protected] (A.R.N.); [email protected] (Z.S.R.) * Correspondence: [email protected]; Tel.: +91-6362256141 Abstract: Non-tuberculous mycobacterial (NTM) infections of the breast are rare. These infections present as cellulitis of the breast or breast abscess. Their diagnosis poses a challenge as they manifest signs of acute inflammation, unlike tuberculous mycobacterial infections which present in a chronic pattern. However, on aspiration of pus from the site of infection, primary smear may show acid fast bacilli. This poses a diagnostic dilemma. The present case is that of a 34-year-old woman who presented with recurrent mastitis. She had history of right breast swelling, for which surgical excision had been performed three months prior at another facility. Her histopathology had showed cystic granulomatous neutrophilic mastitis (CNGM). The patient again presented with right breast abscess which was confirmed on ultrasonography. Incision and drainage along with removal of necrotic tissue was done. Primary smear of pus showed acid fast bacilli on Ziehl–Neelson staining. Bacterial culture and line probe speciation revealed non-tuberculous mycobacterium M. abscessus, which responded well to prolonged anti-microbial therapy. These rapidly growing NTM require prolonged Citation: Patel, O.A.; Bakhshi, G.D.; Nadkarni, A.R.; Rangwala, Z.S. treatment and are quite often recurrent. M. abscessus is a rare cause of CNGM, with this being only Granulomatous Mastitis due to the third reported case in literature. A brief case report with a review of literature is presented. Non-Tuberculous Mycobacteria: A Diagnostic and Therapeutic Dilemma. Keywords: breast; mastitis; non-tuberculous mycobacteria; breast abscess Clin. Pract. 2021, 11, 228–234. https:// doi.org/10.3390/ clinpract11020034 1. Introduction Academic Editors: Cystic neutrophilic granulomatous mastitis (CNGM) remains a little-known benign Vishwanath Venketaraman and Vipul breast entity with obscure aetiology first reported in 1972 as a differential mimicking D. Yagnik breast carcinoma [1]. Less than 1% of all breast specimens show CNGM. It is commonly observed in women of childbearing age, often with a history of breast feeding. It is typically Received: 7 December 2020 associated with the gram-positive bacillus Corynebacterium kroppenstedtii. A mainstay Accepted: 25 March 2021 Published: 14 April 2021 of therapy in CNGM remains focused on lipophilic antibiotics and surgical excision in refractory cases. CNGM is rarely associated with non-tuberculous mycobacteria (NTM) [2]. NTM may cause infections of breast tissue after cosmetic surgery and in immunosuppressed Publisher’s Note: MDPI stays neutral with regard to jurisdictional claims in individuals. These mycobacterial species show resistance to conventional antibiotic therapy. published maps and institutional affil- We hereby present a case of Mycobacterium abscessus associated with CNGM in India. iations. 2. Case Presentation A 34-year-old woman from Maharashtra, India presented with right breast swelling, generalized malaise, and decreased appetite. She gave prior history of bilateral breast abscesses three months back, for which she underwent incision and drainage at another Copyright: © 2021 by the authors. Licensee MDPI, Basel, Switzerland. facility. Histopathology reports had demonstrated acute on chronic granulomatous mastitis This article is an open access article with the presence of Langhans’ type of giant cells with acid-fast bacilli seen on Ziehl– distributed under the terms and Neelson staining. She was started on a course of anti-tubercular drugs at the previous conditions of the Creative Commons facility. The patient denied any history of previous trauma to the breast or diabetes mellitus. Attribution (CC BY) license (https:// She reported no history of breastfeeding during the period when she developed the abscess. creativecommons.org/licenses/by/ At presentation, she reported a swelling on her right breast associated with throbbing 4.0/). pain and low-grade fever. The left breast showed a healthy scar of previous surgery. Vital Clin. Pract. 2021, 11, 228–234. https://doi.org/10.3390/clinpract11020034 https://www.mdpi.com/journal/clinpract Clin. Pract. 2021, 12, FOR PEER REVIEW 2 facility. The patient denied any history of previous trauma to the breast or diabetes melli- tus. She reported no history of breastfeeding during the period when she developed the Clin. Pract. 2021, 11 abscess. 229 At presentation, she reported a swelling on her right breast associated with throbbing pain and low-grade fever. The left breast showed a healthy scar of previous surgery. Vital parametersparameters werewere unremarkableunremarkable andand locallocal examinationexamination demonstrateddemonstrated aa tendertender swellingswelling overover the right breast breast in in the the upper upper and and outer outer aspect. aspect. It Itwas was about about 10 10cm cm × 7× cm7 in cm size in sizeand andassociated associated with withlocal localwarmth, warmth, induration, induration, and fluctuation. and fluctuation. Blood tests Blood revealed tests revealed a leuco- acyte leucocyte count of count 18,000/mm of 18,000/mm3 with neutrophilia.3 with neutrophilia. A diagnosis A of diagnosis recurrent of breast recurrent abscess breast was abscessmade. She was underwent made. She incision underwent and drainage incision of andthe breast drainage under of general the breast anaesthesia. under general Intra- anaesthesia.operatively an Intra-operatively indurated mass an with indurated multiple mass small with abscesses multiple containing small abscesses greyish containing green pus greyishwas seen green (Figure pus 1A was,B). seen About (Figure 150 cc1A,B). of pus About was 150evacuated cc of pus and was all evacuatednecrotic tissue and was all necroticdebrided. tissue This waspus debrided.cavity was This in conti pusnuity cavity with was previously in continuity drained with abscess previously. Post drained opera- abscess.tively she Post was operativelystarted on a shecourse was ofstarted intravenous on a coursethird generation of intravenous cephalosporin. third generation Pus was cephalosporin.sent for Gram staining, Pus was Ziehl sent for–Neelson Gram staining,(ZN) staining Ziehl–Neelson and culture. (ZN) Gram staining staining and was culture. neg- Gramative for staining bacteria, was but negative primary for ZN bacteria, staining but demonstrated primary ZN staining acid-fast demonstrated bacilli in fair acid-fastnumber, bacillisuggestive in fair of number,mycobacterial suggestive infection. of mycobacterial Histopathology infection. report was Histopathology suggestive of report periductal was suggestiveand perilobular of periductal inflammati andon perilobular comprising inflammation lymphocytes, comprising plasma cells, lymphocytes, polymorphs, plasma foamy cells,macrophages, polymorphs, histiocytes foamy, and macrophages, occasional histiocytes,Langhans’ type and of occasional giant cells, Langhans’ along with type areas of giantof micro cells,-abscesses along with and areas frank of oedema. micro-abscesses Further, andcystic frank spaces oedema. surrounded Further, by cystic neutrophilic spaces surroundedaggregates were by neutrophilic noted corresponding aggregates to were CNGM. noted No corresponding micro-calcifications to CNGM. or granulomas No micro- calcificationswere reported or (Figure granulomas 2). As wereper hospital reported protocol (Figure Cartridge2). As per- hospitalbased Nucleic protocol Acid Cartridge- Amplifi- ® basedcation NucleicTest for AcidMycobacterium Amplification tuberculosis Test for (GeneXpertMycobacterium® ) was tuberculosis ordered to(GeneXpert rule out tubercu-) was orderedlosis. The to test rule was out negative. tuberculosis. The test was negative. FigureFigure 1.1. ((A):): AbscessAbscess containingcontaining pus (B): Communicating abscesses (C): Actively draining sites of previousprevious incisionincision (black(black arrows).arrows). NewNew onsetonset indurationinduration (white(white arrow).arrow). Clin. Pract. 2021, 11 230 Clin. Pract. 2021, 12, FOR PEER REVIEW 3 FigureFigure 2. 2.Histopathological Histopathological photograph, photograph, HaematoxylinHaematoxylin && EosinEosin stain, 40×; ;white white arrow arrow showing showing epithelioid cells around a granuloma; black arrow showing neutrophilic infiltrate. epithelioid cells around a granuloma; black arrow showing neutrophilic infiltrate. AerobicAerobic culture culture obtained obtained after after two two weeks weeks revealed revealed rapidly rapidly growing growing non-tuberculous non-tuberculous mycobacteria.mycobacteria. LineLine probeprobe speciation speciation further further demonstrated demonstratedMycobacterium Mycobacterium abscessus abscessussub. sub. abscessusabscessusas as the the causative causative organism. organism. Based Based on on histological histological and and microbiological microbiological reporting, reporting, diagnosisdiagnosis of of cystic cystic neutrophilic neutrophilic granulomatous granulomatous mastitis mastitis secondary secondary to Mycobacterium to Mycobacterium absces- ab- susscessuwass made.was made. Patient Patient by this by timethis time had developedhad developed a new a new abscess
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