Case for Diagnosis
Progressive Asymptomatic Thickening of a Lip
Engrosamiento labial progresivo asintomático
R. Ruiz-Villaverde
, B. Rueda-Villafranca, M. Galvez-Moreno
Corresponding author
Servicio de Dermatología, Hospital Universitario San Cecilio, Granada, Spain
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The reddish-violaceous homogeneous lesion is surrounded by a biphasic halo. The first halo has a yellowish-orange tinge (*) and the second halo on the periphery is reddish. The hair follicles remain intact.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "F.J. Navarro-Triviño" "autores" => array:1 [ 0 => array:2 [ "nombre" => "F.J." 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A Cross-Sectional Study at a Field Hospital" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "640" "paginaFinal" => "644" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "¿Son las alteraciones en la mucosa oral un signo de COVID-19? Estudio transversal en un Hospital de Campaña" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 926 "Ancho" => 1804 "Tamanyo" => 270054 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Characteristic tongue swelling or macroglossia (6.6%) associated with transient U-shaped lingual papillitis (11.5%) in patients with COVID-19. A, Illustration showing tongue changes with indentation on both sides of the tongue and inflammation of anterior papillae, probably caused by friction. B–E, Tongue swelling and transient U-shaped lingual papillitis during COVID-19. COVID-19 indicates coronavirus disease 2019.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Nuño González, K. Magaletskyy, P. Martín Carrillo, B. Lozano Masdemont, A. Mayor Ibarguren, M. Feito Rodríguez, P. Herranz Pinto" "autores" => array:7 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Nuño González" ] 1 => array:2 [ "nombre" => "K." "apellidos" => "Magaletskyy" ] 2 => array:2 [ "nombre" => "P." "apellidos" => "Martín Carrillo" ] 3 => array:2 [ "nombre" => "B." "apellidos" => "Lozano Masdemont" ] 4 => array:2 [ "nombre" => "A." "apellidos" => "Mayor Ibarguren" ] 5 => array:2 [ "nombre" => "M." "apellidos" => "Feito Rodríguez" ] 6 => array:2 [ "nombre" => "P." "apellidos" => "Herranz Pinto" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219021001700?idApp=UINPBA000044" "url" => "/15782190/0000011200000007/v1_202107020822/S1578219021001700/v1_202107020822/en/main.assets" ] "en" => array:17 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case for Diagnosis</span>" "titulo" => "Progressive Asymptomatic Thickening of a Lip" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "645" "paginaFinal" => "646" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "R. Ruiz-Villaverde, B. Rueda-Villafranca, M. Galvez-Moreno" "autores" => array:3 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Ruiz-Villaverde" "email" => array:1 [ 0 => "ismenios@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "B." "apellidos" => "Rueda-Villafranca" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Galvez-Moreno" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Universitario San Cecilio, Granada, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Engrosamiento labial progresivo asintomático" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1253 "Ancho" => 2174 "Tamanyo" => 333805 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Immunohistochemistry, original magnification ×10. Left, negative staining for immunoglobulin (Ig) G κ light chain. Right, positive staining for IgG λ light chain.</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 65-year-old woman with no drug allergies and a medical history of type II diabetes mellitus, dyslipidemia, and HLA-B27-positive seronegative spondyloarthropathy, was referred from the hematology department for progressive asymptomatic thickening of the lip that had begun several months earlier. The patient was undergoing tests for moderate iron deficiency anemia and a monoclonal immunoglobulin (Ig) G λ band. Prior tests had revealed no other findings of interest.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient’s upper lip was thickened and hard to the touch, without associated ulcers (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Neither locoregional lymphadenopathy nor hepatosplenomegaly were palpable. Examination of the skin and mucosa revealed no other lesions.</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">Histopathology showed lymphoid clusters consisting mainly of plasma cells with a periglandular distribution (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Immunohistochemistry was positive for CD20 and negative for IgG and IgG4, and revealed monoclonal λ light chains but was negative for κ light chains (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Additional Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Additional tests revealed a normal complete blood count and a normal biochemical profile. Quantification of IgG λ light chain in serum continued to show elevated levels (3.20 g/dL). The results of the autoimmunity study were normal. The results of a bone marrow aspiration (BMA) biopsy were within the normal range. The first positron emission tomography–computed tomography (PET–CT) scan showed foci of mild-to-moderate metabolic activity associated with paratracheal lymphadenopathy suggestive of benign inflammatory disease.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">What Is Your Diagnosis?</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Diagnosis</span><p id="par0025" class="elsevierStylePara elsevierViewall">Lymphocytic hyperplasia with monoclonal IgG λ plasmacytosis.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Clinical Course and Treatment</span><p id="par0030" class="elsevierStylePara elsevierViewall">BMA biopsy was repeated 3 months later and revealed an increase in plasma cells (2.5%) and clonal B lymphocytes with weak cytoplasmic expression of IgG λ light chain. A second PET–CT scan was performed 6 months later and compared with previous images. The scan showed findings compatible with adenopathies suggestive of lymphomatous spread and a probable diagnosis of IgG λ lymphoplasmacytic lymphoma. Treatment with cycles of rituximab, cyclophosphamide, and dexamethasone (RCD) resulted in a progressive decrease in the monoclonal component, but had no effect on the patient’s lip condition.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Comment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Monoclonal plasmacytosis in minor salivary gland biopsies is observed in autoimmune diseases such as Sjögren syndrome, in the early stages of mucosa-associated lymphoid tissue lymphoma, and even in monoclonal gammopathy of uncertain significance.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Lip and minor salivary gland involvement, as observed in the present case, may be the first manifestation of lymphoma with systemic compromise. Transition between lymphocytic infiltrate with apparently benign characteristics and lymphoma occurs relatively frequently, in some cases separated by intermediate stages that are difficult to classify. Therefore, clonal proliferations in clinically accessible locations (e.g. the lip, which in our patient became progressively thickened) should be evaluated with caution. It is necessary to clinically rule out processes such as granulomatous cheilitis in Melkersson-Roshental syndrome<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and Sjögren syndrome, in which these clonal proliferations have also been described, as well as IgG4-related disease,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> which was ruled out in our patient.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Histologically, features that suggest benignity include preserved acinar architecture and the presence of small lymphocytes and plasma cells in the interfollicular regions with a nondiffuse pattern distinct from that of lymphoproliferative infiltrate.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Ultimately, our patient was diagnosed with IgG λ lymphoplasmacytic lymphoma. In 2008<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> the World Health Organization defined this condition as a B-cell neoplasm consisting of coexisting clonal populations of small B cells, lymphoplasmacytes, and plasma cells. It is frequently associated with a monoclonal IgM component (Waldenstrom macroglobulinemia) and less than 5% of patients present a monoclonal IgG band,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> which was observed in our patient.</p></span></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Funding</span><p id="par0050" class="elsevierStylePara elsevierViewall">No funding was received for this study.</p></span><span id="sec0050" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conflicts of interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 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" => "Additional Tests" ] 4 => array:3 [ "identificador" => "sec0025" "titulo" => "What Is Your Diagnosis?" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0030" "titulo" => "Diagnosis" ] 1 => array:2 [ "identificador" => "sec0035" "titulo" => "Clinical Course and Treatment" ] 2 => array:2 [ "identificador" => "sec0040" "titulo" => "Comment" ] ] ] 5 => array:2 [ "identificador" => "sec0045" "titulo" => "Funding" ] 6 => array:2 [ "identificador" => "sec0050" "titulo" => "Conflicts of interest" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-04-02" "fechaAceptado" => "2019-07-14" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Ruiz-Villaverde R, Rueda-Villafranca B, Galvez-Moreno M. Engrosamiento labial progresivo asintomático. Actas Dermosifiliogr. 2021;112:645–646.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1674 "Ancho" => 1257 "Tamanyo" => 148696 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Symmetrical thickening of the lower lip.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1249 "Ancho" => 2174 "Tamanyo" => 364491 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Hematoxylin-eosin, original magnification ×10.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1253 "Ancho" => 2174 "Tamanyo" => 333805 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Immunohistochemistry, original magnification ×10. Left, negative staining for immunoglobulin (Ig) G κ light chain. Right, positive staining for IgG λ light chain.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clonally expanded lymphocytes in the minor salivary glands of Sjögren’s syndrome patients without lymphoproliferative disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "J.L. Pablos" 1 => "P.E. Carreira" 2 => "L. Morillas" 3 => "G. Montalvo" 4 => "C. Ballestin" 5 => "J.J. Gomez-Reino" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1002/art.1780371006" "Revista" => array:6 [ "tituloSerie" => "Arthritis Rheum." 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 22 | 12 | 34 |
| 2024 October | 75 | 42 | 117 |
| 2024 September | 84 | 18 | 102 |
| 2024 August | 113 | 49 | 162 |
| 2024 July | 79 | 21 | 100 |
| 2024 June | 98 | 30 | 128 |
| 2024 May | 87 | 34 | 121 |
| 2024 April | 70 | 31 | 101 |
| 2024 March | 69 | 25 | 94 |
| 2024 February | 85 | 37 | 122 |
| 2024 January | 71 | 39 | 110 |
| 2023 December | 60 | 18 | 78 |
| 2023 November | 92 | 37 | 129 |
| 2023 October | 56 | 26 | 82 |
| 2023 September | 62 | 35 | 97 |
| 2023 August | 66 | 19 | 85 |
| 2023 July | 61 | 40 | 101 |
| 2023 June | 53 | 20 | 73 |
| 2023 May | 59 | 29 | 88 |
| 2023 April | 52 | 37 | 89 |
| 2023 March | 71 | 29 | 100 |
| 2023 February | 27 | 20 | 47 |
| 2023 January | 41 | 41 | 82 |
| 2022 December | 45 | 57 | 102 |
| 2022 November | 20 | 27 | 47 |
| 2022 October | 32 | 15 | 47 |
| 2022 September | 43 | 43 | 86 |
| 2022 August | 32 | 32 | 64 |
| 2022 July | 28 | 35 | 63 |
| 2022 June | 21 | 26 | 47 |
| 2022 May | 45 | 46 | 91 |
| 2022 April | 49 | 28 | 77 |
| 2022 March | 60 | 71 | 131 |
| 2022 February | 47 | 42 | 89 |
| 2022 January | 39 | 36 | 75 |
| 2021 December | 40 | 41 | 81 |
| 2021 November | 57 | 44 | 101 |
| 2021 October | 54 | 71 | 125 |
| 2021 September | 39 | 51 | 90 |
| 2021 August | 60 | 53 | 113 |
| 2021 July | 83 | 37 | 120 |
| 2021 June | 46 | 28 | 74 |
| 2021 May | 41 | 32 | 73 |
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