Cases for Diagnosis
Diffuse Firm Swelling of the Right Areola
Infiltración difusa unilateral de la aréola mamaria
H. Escolà
, S. Segura Tigell, R.M. Pujol
Corresponding author
Departamento de Dermatología, Hospital del Mar, Parc de Salut Mar, Universitat Autònoma de Barcelona, Barcelona, Spain
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"apellidos" => "Pujol" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Dermatología, Hospital del Mar, Parc de Salut Mar, Universitat Autònoma de Barcelona, Barcelona, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Infiltración difusa unilateral de la aréola mamaria" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 807 "Ancho" => 1207 "Tamanyo" => 94053 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous-violaceous, thickened, and infiltrated right mammary areola.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Medical history</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 40-year-old man, with no relevant past personal history, with a 5-month history of erythema, itching, and diffuse infiltration of the right mammary areola, without any triggering factor or associated systemic symptoms.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed the presence of an erythematous-violaceous, thickened, infiltrated right mammary areola, palpable on touch (<a class="elsevierStyleCrossRef" href="#fig0005">fig. 1</a>), with no locoregional lymphadenopathy or other lesions elsewhere on the skin integumentary system.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">Skin biopsy showed a dense dermal lymphoid infiltrate composed of intermediate-sized cells with occasional large cells, and a few accompanying eosinophils (<a class="elsevierStyleCrossRef" href="#fig0010">fig. 2</a>). Presence of non-confluent germinal centers in deep dermis. Immunophenotypic study: mixed infiltrate of T cells (CD3+, CD4+, CD7+) CD30− with occasional PD1+ cells, and B cells (CD20+, CD79a+) bcl2− with focal positivity for CD10 and CD21. No restriction of light chains of immunoglobulins by in situ hybridization was observed. Rearrangement of T-cell receptor genes and immunoglobulin heavy chains: polyclonal pattern.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Other additional tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Complete blood count, standard count, formula, and biochemistry showed no changes. Breast ultrasound revealed thickening of the cutaneous complex of the areola-nipple, with no identification of fibroglandular tissue or underlying focal lesions.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">What is your diagnosis?</span><p id="par9025" class="elsevierStylePara elsevierViewall">.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">Reactive cutaneous lymphoid hyperplasia.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Course of the disease and treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Upon further questioning, the patient reported having traveled to Sweden 6 months prior to the consultation, admitting to having suffered several tick bites. Serologies for <span class="elsevierStyleItalic">Borrelia burgdorferi</span> were requested, showing negative IgM antibodies, weakly positive IgG antibodies, and positive PCR for <span class="elsevierStyleItalic">B. burgdorferi</span> (OspA gene) in the skin biopsy, leading to the diagnosis of lymphocytoma cutis due to <span class="elsevierStyleItalic">Borrelia</span>. A 2-week regimen of doxycycline 100 mg every 12 hours was initiated with complete resolution of the clinical picture.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Comment</span><p id="par0040" class="elsevierStylePara elsevierViewall">Reactive cutaneous lymphoid hyperplasias (RCLH) or cutaneous pseudolymphomas constitute a heterogeneous group of benign hyperplastic lymphoproliferative reactions that clinically and/or histologically mimic a malignant lymphoproliferative process.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Lymphocytoma cutis (LC) is considered the prototype of nodular cutaneous B-cell pseudolymphoma with follicular pattern. LC can be induced by various antigenic stimuli, including arthropod bites, drugs, and vaccines. In endemic regions, <span class="elsevierStyleItalic">B. burgdorferi</span> is the main causative agent, being rare in our environment. <span class="elsevierStyleItalic">Borrelia</span>-induced LC is a rare cutaneous sign of Lyme disease that occurs weeks or months after the bite of an <span class="elsevierStyleItalic">Ixodes</span> tick. It is usually observed in children and young adults with a slight predominance in women. There are several descriptions in the literature of cases similar to the one presented here.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">2-4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Clinically, it presents as a painless erythematous-violaceous nodule or plaque that mainly affects the earlobe, the areola-nipple complex, or the scrotum.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">5</span></a> It is usually accompanied by regional lymphadenopathy.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Histopathological examination shows a nodular lymphoid infiltrate, without cellular atypia, affecting the entire dermis, with formation of large and confluent germinal centers with a reduced or absent mantle zone. The infiltrate consists of plasma cells, eosinophils, reactive T lymphocytes, and macrophages with apoptotic cells phagocytized inside (tingible bodies). Immunophenotyping demonstrates lymphoid proliferation with predominance of B-cell germinal center cells, positive for CD20, CD10, and Bcl-6 and negative for Bcl-2, with polytypic light chains expression of immunoglobulins.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The diagnosis of <span class="elsevierStyleItalic">Borrelia</span>-induced LC is clinical and requires a high level of suspicion.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">7</span></a> The presence of diffuse infiltration of the mammary areola, with histopathological findings of nodular dermal lymphoid infiltration with germinal center formation, should raise suspicion of the diagnosis of a B-cell RCLH with nodular pattern and, within the appropriate epidemiological context, due to B. <span class="elsevierStyleItalic">burgdorferi</span>. Diagnosis is confirmed by histopathological examination, serologies, and/or detection of <span class="elsevierStyleItalic">B. burgdorferi</span> DNA in tissue by PCR.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The clinical differential diagnosis is established with primary or secondary cutaneous lymphomas, sarcoidosis, nodular gynecomastia, urticarial follicular mucinosis, lupus mastitis, and other etiologies of RCLH.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Treatment includes the administration of doxycycline 100 mg twice a day, or amoxicillin 500 mg every 8 hours for 2-3 weeks.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a></p></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Funding</span><p id="par0075" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0050" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conflicts of interest</span><p id="par0080" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:11 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Medical history" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Histopathology" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Other additional tests" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "What is your diagnosis?" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Diagnosis" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Course of the disease and treatment" ] 7 => array:2 [ "identificador" => "sec0040" "titulo" => "Comment" ] 8 => array:2 [ "identificador" => "sec0045" "titulo" => "Funding" ] 9 => array:2 [ "identificador" => "sec0050" "titulo" => "Conflicts of interest" ] 10 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 807 "Ancho" => 1207 "Tamanyo" => 94053 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous-violaceous, thickened, and infiltrated right mammary areola.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 874 "Ancho" => 1207 "Tamanyo" => 417884 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Skin biopsy of the right areola. Hematoxylin and eosin, ×100. Dense dermal lymphoid infiltrate composed of intermediate-sized cells with occasional large cells. Presence of germinal centers and non-confluent in deep dermis.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0045" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cutaneous pseudolymphoma — A review on the spectrum and a proposal for a new classification" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "C. Mitteldorf" 1 => "W. Kempf" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/cup.13532" "Revista" => array:6 [ "tituloSerie" => "J Cutan Pathol." 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 19 | 12 | 31 |
| 2024 October | 152 | 60 | 212 |
| 2024 September | 142 | 41 | 183 |
| 2024 August | 139 | 62 | 201 |
| 2024 July | 320 | 68 | 388 |
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| 2024 May | 5 | 18 | 23 |
