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Revista de Senología y Patología Mamaria - Journal of Senology and Breast Disease
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Case report
Recurrent bilateral granulomatous mastitis due to Corynebacterium tuberculostearicum with mammary scrofula: A case report
Mastitis granulomatosa recurrente bilateral debida a Corynebacterium tuberculostearicum con escrófulas mamarias, a propósito de un caso
María Esther Sánchez Lópeza,
Corresponding author
marisanchezlopez@sescam.jccm.es

Corresponding author.
, Ana Sánchez Mozoa, Jessica Marcela Juez Barrerab
a Department of General Surgery, Complejo Hospitalario Universitario de Albacete, Albacete, Spain
b Department of Plastic Surgery, Complejo Hospitalario Universitario de Albacete, Albacete, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="s0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0025">Introduction</span><p id="p0005" class="elsevierStylePara elsevierViewall">Corynebacterium tuberculostearicum is part of the skin microbiota&#46; It is difficult to distinguish between infection&#44; colonization and contamination of this organism&#46; It is a pleomorphic Gram positive bacteria&#46; An association has been seen between <span class="elsevierStyleItalic">Corynebacterium</span> and infections after orthopedic surgery&#44; leprosy-like lesions or mastitis&#46;<a class="elsevierStyleCrossRef" href="#bb0005"><span class="elsevierStyleSup">1</span></a> Our goal is to review the treatments that we can offer to a patient with a breast infection due to Corynebacterium tuberculostearicum based on a clinical case&#46;</p></span><span id="s0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0030">Clinical case</span><p id="p0010" class="elsevierStylePara elsevierViewall">40-year-old patient with no relevant history&#44; a native of Colombia who has lived in Spain for 16&#8239;years&#46; She has two children&#44; her last pregnancy was 5&#8239;years ago&#46; She breastfed for 8&#8239;months&#46; She has been on contraceptive treatment in the last year&#46;</p><p id="p0015" class="elsevierStylePara elsevierViewall">She came to the consultation due to severe pain in the left breast and a mammary tumor with recurrent fistulas with pus draining on two occasions&#46;</p><p id="p0020" class="elsevierStylePara elsevierViewall">Mammography&#44; ultrasound and MRI revealed areas of increased density and collections with fistulous tracts to the skin &#40;<a class="elsevierStyleCrossRef" href="#f0005">Fig&#46; 1</a>&#41;&#46; In the biopsy&#44; there were no cellular alterations&#46; Histologically&#44; the biopsy shows lobular and interlobal inflammation with necrotic and hemorrhagic focus and granulomas &#40;<a class="elsevierStyleCrossRef" href="#f0010">Fig&#46; 2</a>&#41; without the presence of microorganisms&#46; Suggestive of granulomatous lobar mastitis&#46; The microbiological study was only positive for <span class="elsevierStyleItalic">Corynebacterium tuberculestearicum</span> in two of five samples&#46;</p><elsevierMultimedia ident="f0005"></elsevierMultimedia><elsevierMultimedia ident="f0010"></elsevierMultimedia><p id="p0025" class="elsevierStylePara elsevierViewall">In the first episode&#44; she was treated with amoxicillin 875&#8239;mg&#47;8&#8239;h for a month&#46; After showing no improvement&#44; she was admitted with intravenous corticosteroid treatment and drainage of mammary collections&#46; Once the acute process had been controlled&#44; she was maintained for three months with doxycycline 100&#8239;mg&#47;12&#8239;h&#44; followed by ciprofloxacin 500&#8239;mg&#47;12&#8239;h for one month and clindamycin 600&#8239;mg&#47;8&#8239;h until complete resolution of the condition was achieved&#46; In the second episode&#44; amoxicillin was started again for a month with clinical improvement&#46;</p><p id="p0030" class="elsevierStylePara elsevierViewall">One year after the first episode&#44; she presented the same infectious process in the contralateral breast with new fistulous tracts and intense pain&#44; being the microbiological study only positive for <span class="elsevierStyleItalic">Corynebacterium</span> sensitive to penicillin&#44; ciprofloxacin and linezolid&#46; Treatment was carried out with ciprofloxacin 750&#8239;mg&#47;12&#8239;h for 15&#8239;days&#46; After stopping antibiotics&#44; she developed new infections twice&#46; Maintenance antibiotic treatment was decided until CRP normalization and subsequent surgical treatment&#46; Surgical treatment was decided due to the patient&#39;s pain&#46; In this interval&#44; tests are done to rule out lupus&#44; sarcoidosis and tuberculosis&#46; As well as&#44; the patient stopped contraceptives&#46;</p><p id="p0035" class="elsevierStylePara elsevierViewall">Bilateral breast reduction&#44; Thorek-type pattern&#44; was performed&#44; maintaining antibiotics until the intervention&#46; The final anatomy shows exacerbated chronic inflammation without the presence of microorganisms&#46;</p><p id="p0040" class="elsevierStylePara elsevierViewall">One year after the intervention&#44; the patient has not presented new episodes of mastitis&#46;</p></span><span id="s0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0035">Discussion</span><p id="p0045" class="elsevierStylePara elsevierViewall">Corynebacteria are implicated in recurrent fistulous mastitis&#46; Cases associated with&#58; <span class="elsevierStyleItalic">Corynebacterium minutissimum</span>&#44; <span class="elsevierStyleItalic">amycolatum</span>&#44; <span class="elsevierStyleItalic">kroppenstedtii&#44; accolens</span> and tuberculostearicum&#44; among others&#44; have been described&#46; The initial approach is important since the delay entails disease progression and limiting physical and psychological discomfort&#46;</p><p id="p0050" class="elsevierStylePara elsevierViewall">Corynebacteria infections can appear to be tumors both clinically and radiologically&#44; so a differential diagnosis must be made&#46;<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a></p><p id="p0055" class="elsevierStylePara elsevierViewall">Imaging tests such as mammography&#44; ultrasound and MRI can help in the differential diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> For the diagnosis&#44; a biopsy with histological and microbiological study is necessary&#46;<a class="elsevierStyleCrossRef" href="#bb0020"><span class="elsevierStyleSup">4</span></a></p><p id="p0060" class="elsevierStylePara elsevierViewall">To date&#44; it is unknown whether <span class="elsevierStyleItalic">Corynebacterium</span> infection can be the cause of granulomatous mastitis or superinfection of granulomatous mastitis&#46;</p><p id="p0065" class="elsevierStylePara elsevierViewall">The etiology of granulomatous mastitis is unknown&#44; being considered in many cases ideopathic&#44; they have been related to autoimmune causes&#44;<a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a> contraceptives&#44; secretions&#44; trauma and infections&#46;<a class="elsevierStyleCrossRef" href="#bb0030"><span class="elsevierStyleSup">6</span></a></p><p id="p0070" class="elsevierStylePara elsevierViewall">The average age of appearance of granulomatous mastititis is usually 35&#8239;years&#44; patients who have had children and have breastfed&#46;<a class="elsevierStyleCrossRef" href="#bb0035"><span class="elsevierStyleSup">7</span></a></p><p id="p0075" class="elsevierStylePara elsevierViewall">Histologically&#44; the granulomas of granulomatous mastitis are lobule-centered&#44; can be suppurative&#44; and be associated with microabscesses&#46; They are also associated with cavities due to dissolution of lipids&#46; Granulomas have a specific appearance with an outer sleeve of histiocytes and giant cells surrounding a collection of polymorphonuclear cells with voids due to fatty solutions&#46; These granulomas are also called suppurative lipogranulomas&#46;</p><p id="p0080" class="elsevierStylePara elsevierViewall">Tuberculosis&#44; Wegener&#39;s granulomatosis&#44; giant cell arteritis&#44; polyarteritis nodosa&#44; syphilis&#44; sarcoidosis&#44; parasitic and fungal infections should be ruled out&#46;</p><p id="p0085" class="elsevierStylePara elsevierViewall">Wilson and Yau recommend for the management of superinfected granulomatous mastitis&#58;<a class="elsevierStyleCrossRef" href="#bb0040"><span class="elsevierStyleSup">8</span></a></p><p id="p0090" class="elsevierStylePara elsevierViewall">1&#46; Initial antibiotic treatment against the susceptibility of the pathogen&#44; the dose and duration being higher when dealing with soft tissues&#46; It may take months to control the disease&#46;</p><p id="p0095" class="elsevierStylePara elsevierViewall">2&#46; Corticosteroid treatment at a dose of 0&#46;6&#8239;mg&#47;kg&#47;day&#46;</p><p id="p0100" class="elsevierStylePara elsevierViewall">3&#46; Partial resections or mastectomy in recurrent cases&#46;</p><p id="p0105" class="elsevierStylePara elsevierViewall">Both medical and surgical treatment are usually effective&#44; but monitoring of granulomatous mastitis should be long-term due to the possibility of recurrence&#44; especially in cases with pain and fistulas&#46;<a class="elsevierStyleCrossRef" href="#bb0045"><span class="elsevierStyleSup">9</span></a> Corticosteroids can also be useful after relapse after surgical resection&#46;<a class="elsevierStyleCrossRef" href="#bb0050"><span class="elsevierStyleSup">10</span></a> Tuli proposes a dose of 0&#46;8&#8239;mg&#47;kg&#47;day&#46; Treatment with chronic methotrexate &#40;10&#8239;mg&#47;week&#41; or azatrioprine may be a maintenance option for non-superinfected recurrent mastitis&#46;<a class="elsevierStyleCrossRef" href="#bb0055"><span class="elsevierStyleSup">11</span></a></p></span><span id="s0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0040">Funding</span><p id="p0110" class="elsevierStylePara elsevierViewall">The authors declare that they did not receive any funding for the elaboration of this paper&#46;</p></span><span id="s0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0045">Confidentiality of data</span><p id="p0115" class="elsevierStylePara elsevierViewall">The authors declare that they have followedand the protocols of their Centre on the publication of patient data&#46;</p></span></span>"
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        "resumen" => "<span id="as0005" class="elsevierStyleSection elsevierViewall"><p id="sp0015" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Corynebacterium tuberculostearicum</span> is a Gram positive bacillus from the skin microbiota&#46; Sometimes it can produce recurrent fistulous mastitis associated with granulomas&#46; Its prevalence is very low&#46; The initial therapeutic approach is important to avoid progression&#46; The histopathological and microbiological study is essential for its diagnosis&#46; Differential diagnosis is made with tumor pathology&#46; There is no established therapeutic scheme to which we can adhere&#46; The initial treatment should be antibiotic therapy adjusted by antibiogram and corticosteroids&#46; The evolution can be torpid despite correct treatment&#46; In these cases&#44; surgical treatment with partial or total resections of the breast can be considered&#46;</p><p id="sp0020" class="elsevierStyleSimplePara elsevierViewall">We present the case of a 44-year-old woman who came to the consultation with pain and induration in the lower internal quadrants of the left breast with the formation of fistulous tracts of years of evolution despite antibiotic and corticosteroid treatment&#46;</p></span>"
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        "resumen" => "<span id="as0010" class="elsevierStyleSection elsevierViewall"><p id="sp0025" class="elsevierStyleSimplePara elsevierViewall">El C&#46; Tuberculostearicum es un Bacilo Gram positivo de la microbiota de la piel&#46; En ocasiones puede producir mastitis fistulosas de repetici&#243;n asociadas a granulomas&#46; Su prevalencia es muy baja&#46; El abordaje terape&#250;tico inicial es importante para evitar progresi&#243;n&#46; El estudio histopatol&#243;gico y microbiol&#243;gico es fundamental para su diagn&#243;stico&#46; El diagn&#243;stico diferencial se realiza con la patolog&#237;a tumoral&#46; No existe un esquema terap&#233;utico establecido al cual nos podamos ce&#241;ir&#46; El tratamiento inicial debe ser antibioterapia ajustada por antibiograma y corticoides&#46; La evoluci&#243;n puede ser t&#243;rpida a pesar de un tratamiento correcto&#46; En estos casos&#44; se puede plantear un tratamiento quir&#250;rgico con resecciones parciales&#44; o totales de la mama&#46;</p><p id="sp0030" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de una mujer de 44 a&#241;os que acude a la consulta con dolor e induraci&#243;n en cuadrantes inferointernos de mama izquierda es con formaci&#243;n de trayectos fistulosos de a&#241;os de evoluci&#243;n a pesar de tratamiento antibi&#243;tico y corticoideo&#46;</p></span>"
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos