Multicentric reticulohistiocytosis in a patient with thymic carcinoma
Identifieur interne : 000310 ( Pmc/Corpus ); précédent : 000309; suivant : 000311Multicentric reticulohistiocytosis in a patient with thymic carcinoma
Auteurs : Samantha Shwe ; Aditi Sharma ; Ashley N. Elsensohn ; Linda Doan ; Janellen SmithSource :
- JAAD Case Reports [ 2352-5126 ] ; 2019.
Url:
DOI: 10.1016/j.jdcr.2019.06.012
PubMed: 31440567
PubMed Central: 6698724
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Multicentric reticulohistiocytosis in a patient with thymic carcinoma</title><author><name sortKey="Shwe, Samantha" sort="Shwe, Samantha" uniqKey="Shwe S" first="Samantha" last="Shwe">Samantha Shwe</name></author><author><name sortKey="Sharma, Aditi" sort="Sharma, Aditi" uniqKey="Sharma A" first="Aditi" last="Sharma">Aditi Sharma</name></author><author><name sortKey="Elsensohn, Ashley N" sort="Elsensohn, Ashley N" uniqKey="Elsensohn A" first="Ashley N." last="Elsensohn">Ashley N. Elsensohn</name></author><author><name sortKey="Doan, Linda" sort="Doan, Linda" uniqKey="Doan L" first="Linda" last="Doan">Linda Doan</name></author><author><name sortKey="Smith, Janellen" sort="Smith, Janellen" uniqKey="Smith J" first="Janellen" last="Smith">Janellen Smith</name></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">31440567</idno><idno type="pmc">6698724</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6698724</idno><idno type="RBID">PMC:6698724</idno><idno type="doi">10.1016/j.jdcr.2019.06.012</idno><date when="2019">2019</date><idno type="wicri:Area/Pmc/Corpus">000310</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000310</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Multicentric reticulohistiocytosis in a patient with thymic carcinoma</title><author><name sortKey="Shwe, Samantha" sort="Shwe, Samantha" uniqKey="Shwe S" first="Samantha" last="Shwe">Samantha Shwe</name></author><author><name sortKey="Sharma, Aditi" sort="Sharma, Aditi" uniqKey="Sharma A" first="Aditi" last="Sharma">Aditi Sharma</name></author><author><name sortKey="Elsensohn, Ashley N" sort="Elsensohn, Ashley N" uniqKey="Elsensohn A" first="Ashley N." last="Elsensohn">Ashley N. Elsensohn</name></author><author><name sortKey="Doan, Linda" sort="Doan, Linda" uniqKey="Doan L" first="Linda" last="Doan">Linda Doan</name></author><author><name sortKey="Smith, Janellen" sort="Smith, Janellen" uniqKey="Smith J" first="Janellen" last="Smith">Janellen Smith</name></author></analytic><series><title level="j">JAAD Case Reports</title><idno type="eISSN">2352-5126</idno><imprint><date when="2019">2019</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Kumar, B" uniqKey="Kumar B">B. Kumar</name></author><author><name sortKey="Singh, N" uniqKey="Singh N">N. Singh</name></author><author><name sortKey="Rahnama Moghadam, A" uniqKey="Rahnama Moghadam A">A. Rahnama-Moghadam</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Selmi, C" uniqKey="Selmi C">C. Selmi</name></author><author><name sortKey="Greenspan, A" uniqKey="Greenspan A">A. Greenspan</name></author><author><name sortKey="Huntley, A" uniqKey="Huntley A">A. Huntley</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Snow, J L" uniqKey="Snow J">J.L. Snow</name></author><author><name sortKey="Muller, S A" uniqKey="Muller S">S.A. Muller</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rudha, Y" uniqKey="Rudha Y">Y. Rudha</name></author><author><name sortKey="Starobinska, E" uniqKey="Starobinska E">E. Starobinska</name></author><author><name sortKey="Abdulqader, Y" uniqKey="Abdulqader Y">Y. Abdulqader</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">JAAD Case Rep</journal-id><journal-id journal-id-type="iso-abbrev">JAAD Case Rep</journal-id><journal-title-group><journal-title>JAAD Case Reports</journal-title></journal-title-group><issn pub-type="epub">2352-5126</issn><publisher><publisher-name>Elsevier</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">31440567</article-id><article-id pub-id-type="pmc">6698724</article-id><article-id pub-id-type="publisher-id">S2352-5126(19)30261-9</article-id><article-id pub-id-type="doi">10.1016/j.jdcr.2019.06.012</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Multicentric reticulohistiocytosis in a patient with thymic carcinoma</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Shwe</surname><given-names>Samantha</given-names></name><degrees>BS</degrees></contrib><contrib contrib-type="author"><name><surname>Sharma</surname><given-names>Aditi</given-names></name><degrees>MD</degrees></contrib><contrib contrib-type="author"><name><surname>Elsensohn</surname><given-names>Ashley N.</given-names></name><degrees>MD, MPH</degrees></contrib><contrib contrib-type="author"><name><surname>Doan</surname><given-names>Linda</given-names></name><degrees>MD, PhD</degrees></contrib><contrib contrib-type="author"><name><surname>Smith</surname><given-names>Janellen</given-names></name><degrees>MD</degrees><xref rid="cor1" ref-type="corresp">∗</xref></contrib></contrib-group><aff id="aff1">Department of Dermatology, University of California Irvine, Irvine, California</aff><author-notes><corresp id="cor1"><label>∗</label>Correspondence to: Janellen Smith, MD, Department of Dermatology, University of California Irvine, 118 Med Surg 1, Irvine, CA 92697-2400.</corresp></author-notes><pub-date pub-type="pmc-release"><day>05</day><month>8</month><year>2019</year></pub-date><pmc-comment> PMC Release delay is 0 months and 0 days and was based on .</pmc-comment>
<pub-date pub-type="collection"><month>8</month><year>2019</year></pub-date><pub-date pub-type="epub"><day>05</day><month>8</month><year>2019</year></pub-date><volume>5</volume><issue>8</issue><fpage>720</fpage><lpage>722</lpage><permissions><copyright-statement>© 2019 by the American Academy of Dermatology, Inc. Published by Elsevier, Inc.</copyright-statement><copyright-year>2019</copyright-year><copyright-holder>American Academy of Dermatology, Inc.</copyright-holder><license license-type="CC BY-NC-ND" xlink:href="http://creativecommons.org/licenses/by-nc-nd/4.0/"><license-p>This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).</license-p></license></permissions><kwd-group id="kwrds0010"><title>Key words</title><kwd>multicentric reticulohistiocytosis</kwd><kwd>paraneoplastic syndrome</kwd><kwd>thymic carcinoma</kwd></kwd-group><kwd-group id="kwrds0015"><title>Abbreviation used</title><kwd>MCR, multicentric reticulohistiocytosis</kwd></kwd-group></article-meta></front><body><sec id="sec1"><title>Introduction</title><p id="p0010">Multicentric reticulohistiocytosis (MCR) is a rare disease characterized by papulonodular skin lesions of the hands, forearms, face, and ears associated with erosive arthritis. A review of the literature shows an association with underlying malignancy in approximately 25% of cases but only 1 reported association with thymic carcinoma. Here, we describe a patient with an atypical presentation of MCR that previously had been diagnosed as thymic carcinoma. The rarity of this disease necessitates awareness of uncommon manifestations, and its potential to be a paraneoplastic syndrome warrants an evaluation for associated malignancy.</p></sec><sec id="sec2"><title>Case presentation</title><p id="p0015">A 61-year-old woman presented to the dermatology clinic with a 1-year history of increasing pruritus intermittently controlled with oral prednisone. After 1 year of these symptoms, she noted the onset of small papules over the posterior neck and along the left flank, which prompted a visit to the dermatologist.</p><p id="p0020">Shortly after the onset of pruritus a year earlier, she had developed arthralgias that were diagnosed as rheumatoid arthritis and treated with hydroxychloroquine and prednisone. Six months after the onset of arthritis, she experienced acute chest pain and was found on imaging to have a 7.5-cm mass in her anterior chest. Biopsy showed a thymic carcinoma with extensive local invasion and a metastasis to the right pelvic bone. She was treated with carboplatin/paclitaxel and radiation and presented to our dermatology clinic 5 months later.</p><p id="p0025">On examination, the patient had widespread areas of pruritus without primary lesions, as well as 2 nonpruritic areas with numerous 2- to 3-mm firm violaceous papules. These areas were located over the left flank and posterior neck (<xref rid="fig1" ref-type="fig">Fig 1</xref>). Of note, there were no lesions over her forearms, face, or ears. Although the patient was unaware of them, several slightly larger papules were noted over her fingers bilaterally (<xref rid="fig2" ref-type="fig">Fig 2</xref>). Ultrasonographic examination of her bilateral hands showed significantly active and symmetric inflammatory arthritis consistent with rheumatoid arthritis; no erosive changes were present. Laboratory workup for the pruritus was unremarkable.<fig id="fig1"><label>Fig 1</label><caption><p>Flesh-colored and violaceous grouped papules on (<bold>A</bold>) the posterior neck and <bold>(B)</bold> the left flank.</p></caption><graphic xlink:href="gr1"></graphic></fig><fig id="fig2"><label>Fig 2</label><caption><p>Papules on the dorsal hand.</p></caption><graphic xlink:href="gr2"></graphic></fig></p><p id="p0030">Biopsies of the posterior neck and left flank were performed, showing a well-defined dermal infiltrate composed of histiocytes and multinucleated giant cells with copious pink cytoplasm and a ground-glass appearance (<xref rid="fig3" ref-type="fig">Fig 3</xref>). A scattered lymphocytic infiltrate was present, but there were no eosinophils. Lesions had positive test results for CD68 and negative results for S100, p63, and CK5/6 markers, establishing a histologic diagnosis of MCR.<fig id="fig3"><label>Fig 3</label><caption><p>Histologic imaging of the posterior neck showing nodular aggregation of multinucleated histiocytes with ample pink cytoplasm.</p></caption><graphic xlink:href="gr3"></graphic></fig></p><p id="p0035">At the time of publication, the patient is contemplating alternative chemotherapeutic options for persistent disease and recovering from the acute effects of radiation. Her pruritus has resolved, and her skin lesions are almost clear. She denies pain, and her hands are unchanged from her initial visit.</p></sec><sec id="sec3"><title>Discussion</title><p id="p0040">MCR most commonly presents as flesh- to pink-colored papulonodules over the hands, forearms, face, and ears and is associated with an erosive arthritis. Roughly 300 cases have been reported worldwide, the majority of which involved middle-aged white women. Although the disease is rare, histopathologic findings are characteristic and include numerous histiocytes and multinucleated giant cells with ground-glass, eosinophilic cytoplasm. Immunostaining is characterized by monocyte-macrophage markers CD68 and CD45 and results are negative for S100, distinguishing this entity from Langerhans cell histiocytosis. Etiology of the disease has not been established but is postulated to involve stimulation of non-Langerhans cell histiocytes and osteoclast activation.<xref rid="bib1" ref-type="bibr"><sup>1</sup></xref></p><p id="p0045">Although the majority of MCR cases in the literature have no association with malignancy, an underlying cancer has been reported in approximately 25% of cases.<xref rid="bib2" ref-type="bibr"><sup>2</sup></xref> In most of these cases, cutaneous manifestations of MCR precede the diagnosis of cancer, prompting a screening for malignancy. The most common associations include breast cancer, leukemia, and gastrointestinal malignancies, but cases have also been reported of gynecologic, lung, and skin cancers.<xref rid="bib3" ref-type="bibr"><sup>3</sup></xref> In only 1 other case was there a coexistence of MCR with thymic carcinoma.<xref rid="bib4" ref-type="bibr"><sup>4</sup></xref> In this report, a 70-year-old man diagnosed with MCR was found to have thymic carcinoma 4 months later, after a bout of chest pain.</p><p id="p0050">Our patient presented with atypical features of MCR, including the location and timing. She also had pruritus, which may or may not have been a manifestation of the MCR and could have represented a separate paraneoplastic symptom or a reaction to treatment with chemotherapeutic agents. There was no significant inflammatory joint disease, often the sole presenting symptom of MCR, but it is worth noting that she was already receiving hydroxychloroquine. Finally, unlike other cases of malignancy associated with MCR, our patient had a diagnosis of thymic carcinoma before the discovery of MCR.</p><p id="p0055">We have reported this case because of its rarity and characteristic histopathologic findings. MCR will often remit after several years; however, in some cases it may progress to arthritis mutilans, causing significant morbidity. Improved prognosis may be afforded by early diagnosis and treatment and recognition of the need for further testing.</p></sec></body><back><ref-list id="cebib0010"><title>References</title><ref id="bib1"><label>1</label><element-citation publication-type="journal" id="sref1"><person-group person-group-type="author"><name><surname>Kumar</surname><given-names>B.</given-names></name><name><surname>Singh</surname><given-names>N.</given-names></name><name><surname>Rahnama-Moghadam</surname><given-names>A.</given-names></name></person-group><article-title>Multicentric reticulohistiocytosis: a multicenter case series and review of literature</article-title><source>J Clin Rheumatol</source><volume>24</volume><issue>1</issue><year>2018</year><fpage>45</fpage><lpage>49</lpage><pub-id pub-id-type="pmid">29059085</pub-id></element-citation></ref><ref id="bib2"><label>2</label><element-citation publication-type="journal" id="sref2"><person-group person-group-type="author"><name><surname>Selmi</surname><given-names>C.</given-names></name><name><surname>Greenspan</surname><given-names>A.</given-names></name><name><surname>Huntley</surname><given-names>A.</given-names></name></person-group><article-title>Multicentric reticulohistiocytosis: a critical review</article-title><source>Curr Rheumatol Rep</source><volume>17</volume><issue>6</issue><year>2015</year><fpage>511</fpage><pub-id pub-id-type="pmid">25900189</pub-id></element-citation></ref><ref id="bib3"><label>3</label><element-citation publication-type="journal" id="sref3"><person-group person-group-type="author"><name><surname>Snow</surname><given-names>J.L.</given-names></name><name><surname>Muller</surname><given-names>S.A.</given-names></name></person-group><article-title>Malignancy-associated multicentric reticulohistiocytosis: a clinical, histological and immunophenotypic study</article-title><source>Br J Dermatol</source><volume>133</volume><issue>1</issue><year>1995</year><fpage>71</fpage><lpage>76</lpage><pub-id pub-id-type="pmid">7669644</pub-id></element-citation></ref><ref id="bib4"><label>4</label><element-citation publication-type="journal" id="sref4"><person-group person-group-type="author"><name><surname>Rudha</surname><given-names>Y.</given-names></name><name><surname>Starobinska</surname><given-names>E.</given-names></name><name><surname>Abdulqader</surname><given-names>Y.</given-names></name></person-group><article-title>Multicentric reticulohistiocytosis associated with thymic carcinoma</article-title><source>Rheumatology (Oxford)</source><volume>56</volume><issue>10</issue><year>2017</year><fpage>1706</fpage><pub-id pub-id-type="pmid">28633354</pub-id></element-citation></ref></ref-list><fn-group><fn id="d31e42"><p id="ntpara0010">Funding sources: None.</p></fn><fn id="d31e45"><p id="ntpara0015">Conflicts of interest: None disclosed.</p></fn></fn-group></back></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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