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    "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&#44; dyslipidemia&#44; and HLA-B27-positive seronegative spondyloarthropathy&#44; was referred from the hematology department for progressive asymptomatic thickening of the lip that had begun several months earlier&#46; The patient was undergoing tests for moderate iron deficiency anemia and a monoclonal immunoglobulin &#40;Ig&#41; G &#955; band&#46; Prior tests had revealed no other findings of interest&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient&#8217;s upper lip was thickened and hard to the touch&#44; without associated ulcers &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Neither locoregional lymphadenopathy nor hepatosplenomegaly were palpable&#46; Examination of the skin and mucosa revealed no other lesions&#46;</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 &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemistry was positive for CD20 and negative for IgG and IgG4&#44; and revealed monoclonal &#955; light chains but was negative for &#954; light chains &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</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&#46; Quantification of IgG &#955; light chain in serum continued to show elevated levels &#40;3&#46;20&#160;g&#47;dL&#41;&#46; The results of the autoimmunity study were normal&#46; The results of a bone marrow aspiration &#40;BMA&#41; biopsy were within the normal range&#46; The first positron emission tomography&#8211;computed tomography &#40;PET&#8211;CT&#41; scan showed foci of mild-to-moderate metabolic activity associated with paratracheal lymphadenopathy suggestive of benign inflammatory disease&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">What Is Your Diagnosis&#63;</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 &#955; plasmacytosis&#46;</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 &#40;2&#46;5&#37;&#41; and clonal B lymphocytes with weak cytoplasmic expression of IgG &#955; light chain&#46; A second PET&#8211;CT scan was performed 6 months later and compared with previous images&#46; The scan showed findings compatible with adenopathies suggestive of lymphomatous spread and a probable diagnosis of IgG &#955; lymphoplasmacytic lymphoma&#46; Treatment with cycles of rituximab&#44; cyclophosphamide&#44; and dexamethasone &#40;RCD&#41; resulted in a progressive decrease in the monoclonal component&#44; but had no effect on the patient&#8217;s lip condition&#46;</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 Sj&#246;gren syndrome&#44; in the early stages of mucosa-associated lymphoid tissue lymphoma&#44; and even in monoclonal gammopathy of uncertain significance&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Lip and minor salivary gland involvement&#44; as observed in the present case&#44; may be the first manifestation of lymphoma with systemic compromise&#46; Transition between lymphocytic infiltrate with apparently benign characteristics and lymphoma occurs relatively frequently&#44; in some cases separated by intermediate stages that are difficult to classify&#46; Therefore&#44; clonal proliferations in clinically accessible locations &#40;e&#46;g&#46; the lip&#44; which in our patient became progressively thickened&#41; should be evaluated with caution&#46; 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 Sj&#246;gren syndrome&#44; in which these clonal proliferations have also been described&#44; as well as IgG4-related disease&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> which was ruled out in our patient&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Histologically&#44; 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&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Ultimately&#44; our patient was diagnosed with IgG &#955; lymphoplasmacytic lymphoma&#46; 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&#44; lymphoplasmacytes&#44; and plasma cells&#46; It is frequently associated with a monoclonal IgM component &#40;Waldenstrom macroglobulinemia&#41; and less than 5&#37; of patients present a monoclonal IgG band&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> which was observed in our patient&#46;</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&#46;</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&#46;</p></span></span>"
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Case for Diagnosis
Progressive Asymptomatic Thickening of a Lip
Engrosamiento labial progresivo asintomático
R. Ruiz-Villaverde
Autor para correspondencia
ismenios@hotmail.com

Corresponding author.
, B. Rueda-Villafranca, M. Galvez-Moreno
Servicio de Dermatología, Hospital Universitario San Cecilio, Granada, Spain
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    "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&#44; dyslipidemia&#44; and HLA-B27-positive seronegative spondyloarthropathy&#44; was referred from the hematology department for progressive asymptomatic thickening of the lip that had begun several months earlier&#46; The patient was undergoing tests for moderate iron deficiency anemia and a monoclonal immunoglobulin &#40;Ig&#41; G &#955; band&#46; Prior tests had revealed no other findings of interest&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient&#8217;s upper lip was thickened and hard to the touch&#44; without associated ulcers &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Neither locoregional lymphadenopathy nor hepatosplenomegaly were palpable&#46; Examination of the skin and mucosa revealed no other lesions&#46;</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 &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemistry was positive for CD20 and negative for IgG and IgG4&#44; and revealed monoclonal &#955; light chains but was negative for &#954; light chains &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</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&#46; Quantification of IgG &#955; light chain in serum continued to show elevated levels &#40;3&#46;20&#160;g&#47;dL&#41;&#46; The results of the autoimmunity study were normal&#46; The results of a bone marrow aspiration &#40;BMA&#41; biopsy were within the normal range&#46; The first positron emission tomography&#8211;computed tomography &#40;PET&#8211;CT&#41; scan showed foci of mild-to-moderate metabolic activity associated with paratracheal lymphadenopathy suggestive of benign inflammatory disease&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">What Is Your Diagnosis&#63;</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 &#955; plasmacytosis&#46;</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 &#40;2&#46;5&#37;&#41; and clonal B lymphocytes with weak cytoplasmic expression of IgG &#955; light chain&#46; A second PET&#8211;CT scan was performed 6 months later and compared with previous images&#46; The scan showed findings compatible with adenopathies suggestive of lymphomatous spread and a probable diagnosis of IgG &#955; lymphoplasmacytic lymphoma&#46; Treatment with cycles of rituximab&#44; cyclophosphamide&#44; and dexamethasone &#40;RCD&#41; resulted in a progressive decrease in the monoclonal component&#44; but had no effect on the patient&#8217;s lip condition&#46;</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 Sj&#246;gren syndrome&#44; in the early stages of mucosa-associated lymphoid tissue lymphoma&#44; and even in monoclonal gammopathy of uncertain significance&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Lip and minor salivary gland involvement&#44; as observed in the present case&#44; may be the first manifestation of lymphoma with systemic compromise&#46; Transition between lymphocytic infiltrate with apparently benign characteristics and lymphoma occurs relatively frequently&#44; in some cases separated by intermediate stages that are difficult to classify&#46; Therefore&#44; clonal proliferations in clinically accessible locations &#40;e&#46;g&#46; the lip&#44; which in our patient became progressively thickened&#41; should be evaluated with caution&#46; 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 Sj&#246;gren syndrome&#44; in which these clonal proliferations have also been described&#44; as well as IgG4-related disease&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> which was ruled out in our patient&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Histologically&#44; 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&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Ultimately&#44; our patient was diagnosed with IgG &#955; lymphoplasmacytic lymphoma&#46; 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&#44; lymphoplasmacytes&#44; and plasma cells&#46; It is frequently associated with a monoclonal IgM component &#40;Waldenstrom macroglobulinemia&#41; and less than 5&#37; of patients present a monoclonal IgG band&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> which was observed in our patient&#46;</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&#46;</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&#46;</p></span></span>"
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