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BRAFV600E mutation was been detected in skin tissue sample&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Computed tomography &#40;neck&#44; thorax&#44; abdomen&#44; pelvic&#41;&#44; also pituitary magnetic resonance scan failed to demonstrate systemic involvement&#46; The patient was diagnosed with LCH skin involvement&#46; He was referred to pulmonology&#44; medical oncology&#44; hematology and endocrinology departments and there was no extracutaneous involvement&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Treatment was started with topical corticosteroids and PUVA treatment with oral 8- Methoxypsoralen &#40;8-MOP&#41;&#46; The patient had photosensitivity reaction to PUVA after 46 sessions and treatment was changed to UVA with topical 0&#46;1&#37; 8-MOP gel&#46; Phototherapy resulted with healing of the erythematous papules&#44; erosions and thinning of the yellow-orange plaques &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The sixth month follow-up thoracic computed tomography image revealed parenchymal involvement consistent with LCH&#46; Bronchoalveolar lavage investigation showed a large number &#40;90&#37;&#41; of histiocytes and a small number of polymorphonuclear leukocytes&#46; The patient was diagnosed with multi-systemic LCH and started methotrexate treatment&#44; in the first week with a 5<span class="elsevierStyleHsp" style=""></span>mg test dose and then as 20<span class="elsevierStyleHsp" style=""></span>mg per weekly&#46; The patient was stable with methotrexate treatment for 6 months&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The diagnosis of LCH is based on clinical and histopathological evidence with microscopic examination and immunohistochemical features of langerhans cells&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a> Detection of LC markers CD1a and CD207 &#40;langerin&#41; is essential to confrim diagnosis and evaluation of Birberck granules on electronic microscopy has been using less frequently&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> &#304;n this case CD1a and CD207 were positive with immunohistochemical investigation of the cells&#44; with eosinophilic cytoplasm and grooved or lobular nucleus&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Skin lesions of LCH can be limited to a single anatomic region or generalized&#44; and consist of diffused papules&#44; nodules or plaques&#44; and can mimic seborrheic dermatitis in addition to prurigo nodularis&#44; eczema&#44; psoriasis&#44; candidiasis or intertrigo&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;5</span></a> Since skin involvement can be the initial symptom of systemic disease&#44; these patients should be evaluated in terms of multi-organ involvement&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> Our patient had atypical lesions&#44; xanthoma-like plaques and erythematous papules on his back and waist&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Clinical evidence of LCH vary based on the affected organ or system&#44; from self- healing disease to chronic recurrences and it may resulted in patient being underdiagnosed&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Podjasek et al reported a LCH case&#44; followed-up with a misdiagnosis of inflammatory intestinal disease&#44; was diagnosed with skin lesions and had gastrointestinal&#44; lung and liver involvement&#46; Skin biopsy may be valuable for diagnosis of LCH&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Our case developed lung involvement about 8 months after he was first diagnosed with LCH skin involvement&#46; Additionaly&#44; lung involvement of LCH is more frequently in adults and strongly associated with smoking&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">BRAFV600E mutation has been detected in approximately half of LCH samples&#46; Recently&#44; it is suggested to research BRAFV600E mutation in all samples with difficult cases&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> In our case&#44; we detected BRAFV600E mutation in skin tissue sample&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">LCH may affect any organs and revised classification contains 4 subtypes of LCH depending on the number of organs or systems involved&#44; lung involvement and involvement of risk organs &#40;liver&#44; spleen and bone marrow&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Treatment recommendations are determined according severity of disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The treatments used include topical and systemic steroid treatment&#44; phototherapy&#44; topical nitrogen mustard&#44; azathioprine and methotrexate&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;8</span></a> In our case erythematous papules&#44; perilesional erythema and eroded areas resolved with PUVA treatment but the xanthoma-like plaques showed thinning&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">LCH in adults is difficult to diagnose as it is rare and skin involvement can vary in appearance&#46; Skin biopsy is significant to diagnose skin involvement&#46; It can be the initial sign of multi-systemic LCH although the condition can rarely be limited to the skin&#46; Close follow-up of adult-onset LCH patients with skin involvement is therefore essential in terms of future potential multi-organ involvement&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest&#46;</p></span></span>"
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Case and Research Letters
Adult Onset Langerhans Cell Histiocytosis Diagnosed With Xanthoma-like Plaque
Placas similares a xantomas como forma de presentación de una histiocitosis de células de Langerhans en un paciente en edad adulta
E. Boyuka,
Autor para correspondencia
emineboyuk@gmail.com

Corresponding authors.
, I. Bulura, F. Canazb,
Autor para correspondencia
fcanaz@ogu.edu.tr

Corresponding authors.
, O. Cilingirc
a Servicio de Dermatología, Universidad de Eskişehir Osmangazi, Facultad de Medicina, Eskisehir, Turkey
b Servicio de Anatomía Patológica, Universidad de Eskişehir Osmangazi, Facultad de Medicina, Eskisehir, Turkey
c Servicio de Genética Médica, Universidad de Eskişehir Osmangazi, Facultad de Medicina, Eskisehir, Turkey
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he had stopped smoking 30 years prior to his diagnosis&#46; The dermatologic examination revealed xanthoma-like plaques on his back&#44; that had composed with the accumulation of yellow-orange papules on an erythematous background&#44; and erythematous papules &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Skin biopsy was taken for differential diagnosis of diffuse plane xanthoma&#46; Histopathological examination revealed dense clusters of Langerhans cells with an eosinophilic cytoplasm&#44; grooved or lobular nucleus&#59; histiocytes with abundant pale cytoplasm&#44; multinucleated giant cells and lymphocytes in the dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Langerhans cells stained the positive for CD1a&#44; CD207 &#40;langerin&#41; and S100 &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>a-3b&#41;&#46; The diagnosis of LCH skin involment was confirmed with histological and immunohistochemical analysis of skin biyopsy&#46; BRAFV600E mutation was been detected in skin tissue sample&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Computed tomography &#40;neck&#44; thorax&#44; abdomen&#44; pelvic&#41;&#44; also pituitary magnetic resonance scan failed to demonstrate systemic involvement&#46; The patient was diagnosed with LCH skin involvement&#46; He was referred to pulmonology&#44; medical oncology&#44; hematology and endocrinology departments and there was no extracutaneous involvement&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Treatment was started with topical corticosteroids and PUVA treatment with oral 8- Methoxypsoralen &#40;8-MOP&#41;&#46; The patient had photosensitivity reaction to PUVA after 46 sessions and treatment was changed to UVA with topical 0&#46;1&#37; 8-MOP gel&#46; Phototherapy resulted with healing of the erythematous papules&#44; erosions and thinning of the yellow-orange plaques &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The sixth month follow-up thoracic computed tomography image revealed parenchymal involvement consistent with LCH&#46; Bronchoalveolar lavage investigation showed a large number &#40;90&#37;&#41; of histiocytes and a small number of polymorphonuclear leukocytes&#46; The patient was diagnosed with multi-systemic LCH and started methotrexate treatment&#44; in the first week with a 5<span class="elsevierStyleHsp" style=""></span>mg test dose and then as 20<span class="elsevierStyleHsp" style=""></span>mg per weekly&#46; The patient was stable with methotrexate treatment for 6 months&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The diagnosis of LCH is based on clinical and histopathological evidence with microscopic examination and immunohistochemical features of langerhans cells&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a> Detection of LC markers CD1a and CD207 &#40;langerin&#41; is essential to confrim diagnosis and evaluation of Birberck granules on electronic microscopy has been using less frequently&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> &#304;n this case CD1a and CD207 were positive with immunohistochemical investigation of the cells&#44; with eosinophilic cytoplasm and grooved or lobular nucleus&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Skin lesions of LCH can be limited to a single anatomic region or generalized&#44; and consist of diffused papules&#44; nodules or plaques&#44; and can mimic seborrheic dermatitis in addition to prurigo nodularis&#44; eczema&#44; psoriasis&#44; candidiasis or intertrigo&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;5</span></a> Since skin involvement can be the initial symptom of systemic disease&#44; these patients should be evaluated in terms of multi-organ involvement&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> Our patient had atypical lesions&#44; xanthoma-like plaques and erythematous papules on his back and waist&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Clinical evidence of LCH vary based on the affected organ or system&#44; from self- healing disease to chronic recurrences and it may resulted in patient being underdiagnosed&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Podjasek et al reported a LCH case&#44; followed-up with a misdiagnosis of inflammatory intestinal disease&#44; was diagnosed with skin lesions and had gastrointestinal&#44; lung and liver involvement&#46; Skin biopsy may be valuable for diagnosis of LCH&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Our case developed lung involvement about 8 months after he was first diagnosed with LCH skin involvement&#46; Additionaly&#44; lung involvement of LCH is more frequently in adults and strongly associated with smoking&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">BRAFV600E mutation has been detected in approximately half of LCH samples&#46; Recently&#44; it is suggested to research BRAFV600E mutation in all samples with difficult cases&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> In our case&#44; we detected BRAFV600E mutation in skin tissue sample&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">LCH may affect any organs and revised classification contains 4 subtypes of LCH depending on the number of organs or systems involved&#44; lung involvement and involvement of risk organs &#40;liver&#44; spleen and bone marrow&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Treatment recommendations are determined according severity of disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The treatments used include topical and systemic steroid treatment&#44; phototherapy&#44; topical nitrogen mustard&#44; azathioprine and methotrexate&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;8</span></a> In our case erythematous papules&#44; 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