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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A full-term male newborn born by vaginal delivery&#44; after an uncomplicated gestation&#44; was observed at our department on the first day of life for a congenital cutaneous lesion&#46; At physical examination&#44; we observed on the left scapular region an 8&#160;mm ulcerated nodule&#44; with a central black crust&#44; a bright elevated pink border&#44; and a peripheral erythema &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Dermoscopy revealed a central reddish-black crust&#44; with a rim of sparse red globules&#44; and a pink border with fine white scale &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; No other lesions were evident&#44; and the remaining physical examination had no abnormalities&#46; Eye red reflex and otoacoustic emissions screenings were normal&#46; His family history was unremarkable&#46; A punch biopsy was made&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Histopathology showed an ulcerated lesion with an infiltrate composed of large epithelioid cells with a kidney-shaped nucleus&#44; with epidermotropism &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Mitotic figures were observed&#46; The background infiltrate was composed of lymphocytes&#44; plasma cells and a significant number of eosinophils &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; Immunostaining was positive for CD1a &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>A&#41;&#44; langerin &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>B&#41;&#44; S100 protein and CD45&#44; and negative for CD68 and CD34&#46; A presumptive diagnosis of solitary congenital self-healing Langerhans cell histiocytoma was made&#46; Complete Blood Count &#40;CBC&#41;&#44; Erythrocyte Sedimentation Rate &#40;ESR&#41;&#44; coagulation times&#44; kidney and liver function tests and lactate dehydrogenase were within the normal range for the age&#46; The newborn underwent chest radiography and abdominal ultrasound&#44; which revealed no abnormalities&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The infant maintains regular follow-ups&#46; At 6 months appointment&#44; no systemic involvement was noticed and there was a complete involution of the skin lesion&#46; The overall well-appearing state of the neonate&#44; lack of systemic signs&#44; and spontaneous involution of the lesion&#44; associated with the histopathology and immunohistochemistry findings&#44; were compatible with congenital self-healing histiocytosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">Langerhans cell histiocytosis &#40;LCH&#41; is a rare neoplasm&#44; characterized by a pathological Langerhans cell proliferation&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> LCH can present as a single organ or multi-systemic involvement&#44; with a wide spectrum of manifestations&#44; ranging from self-resolving skin lesions to disseminated forms&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The prognosis depends on the extent of systemic involvement&#44; with the single-system disease having a good prognosis&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Congenital self-healing Langerhans cell histiocytosis &#40;CSHLCH&#41; is a rare variant of LCH&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It usually presents as multiple papules or nodules &#40;from the &#8220;blueberry muffin baby&#8221; spectrum&#41;&#44; but uncommonly a solitary lesion may occur&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a> LC histiocytoma &#40;or solitary CSHLCH&#41; is considered a unimodular or paucinodular CSHLCH variant&#46; It presents as a single reddish nodule at birth or within the first weeks of life&#44; that progresses to crusting and ulceration&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Cutaneous lesions typically regress within a few months&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Neonates are usually healthy&#44; with no systemic involvement&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> A recent retrospective review of 82 neonates showed complete involution of the lesion in all cases who have not been submitted to excision&#46; Furthermore&#44; the authors observed no progression&#44; recurrence&#44; or multisystemic involvement&#44; with a median follow-up of 15 months&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Histopathology is characterized by a dense dermal infiltrate composed of large cells with abundant eosinophilic cytoplasm and round-oval or kidney-shaped nucleus&#44; accompanied by an infiltrate comprising lymphocytes and eosinophils&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;6</span></a> Histopathological differences have been between solitary CSHLCH and multiple papular or nodular forms&#46; In the former&#44; there is a deeper infiltrate of the whole dermis and often extends to superficial subcutis&#44; superficial ulceration or crusting&#44; frequent parakeratosis and necrosis&#44; and little or no epidermotropism&#46; In solitary CSHLCH&#44; there are large-sized cells&#44; sometimes with &#8220;ground-glass&#8221; cytoplasm and numerous mitoses&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Diagnosis is confirmed by positive immunostaining for CD1a&#44; S100&#44; and langerin&#44; markers of Langerhans cells&#46; Electron microscopy reveals the characteristic Birbeck granules&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> but usually is not needed&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">There is some variability in dermoscopy reports of LCH&#44; due to the clinical spectrum and different lesion stages&#46; Reddish-lilac color of nodular lesions&#44; with peripheral telangiectasias&#44; violaceous lacunae and clods&#44; and whitish areas may be present&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7&#44;8</span></a> However&#44; dermoscopy descriptions are sparse in the literature&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">There are no specific guidelines for the management and follow-up of solitary CSHLCH&#46; Initial work-up should include a complete physical examination &#40;exclude other skin or mucosal lesions&#44; lymphadenopathy&#44; or hepatosplenomegaly&#41;&#44; laboratory analysis &#40;CBC&#44; ESR&#44; C-reactive protein&#44; coagulation studies&#44; hepatic and renal function tests&#44; blood and urine osmolarities&#41; and imaging exams &#40;abdominal ultrasound&#44; chest X-Ray&#44; complete skeletal radiographs&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In summary&#44; solitary CSHLCH diagnosis should be raised in the presence of a single ulcerated congenital lesion and usually portends a good prognosis&#46; CSHLCH diagnosis is made in retrospect after work-up and follow-up of these patients&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> showing a spontaneous resolution and absence of systemic involvement&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Financial support</span><p id="par0050" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Authors&#39; contributions</span><p id="par0055" class="elsevierStylePara elsevierViewall">Tiago Fernandes Gomes&#58; Data collection&#44; analysis and interpretation&#59; critical literature review&#59; preparation and writing of the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Jos&#233; Carlos Cardoso&#58; Approval of the final version of the manuscript&#59; data collection&#44; analysis and interpretation&#59; manuscript critical review&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Victoria Guiote&#58; Approval of the final version of the manuscript&#59; data collection&#44; analysis&#44; and interpretation&#59; manuscript critical review&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Felicidade Santiago&#58; Approval of the final version of the manuscript&#59; data collection&#44; analysis&#44; and interpretation&#59; intellectual participation in propaedeutic and&#47;or therapeutic management of studied cases&#59; manuscript critical review&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflicts of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Letter - Clinical
Congenital ulcerated nodule: self-healing Langerhans cell histiocytosis
Tiago Fernandes Gomesa,
Corresponding author
tiagofgomesd@gmail.com

Corresponding author.
, José Carlos Cardosob, Victoria Guiotea, Felicidade Santiagoa
a Department of Dermatology, Centro Hospitalar de Leiria, Portugal
b Department of Dermatology, Centro Hospitalar e Universitário de Coimbra, Portugal
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A full-term male newborn born by vaginal delivery&#44; after an uncomplicated gestation&#44; was observed at our department on the first day of life for a congenital cutaneous lesion&#46; At physical examination&#44; we observed on the left scapular region an 8&#160;mm ulcerated nodule&#44; with a central black crust&#44; a bright elevated pink border&#44; and a peripheral erythema &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Dermoscopy revealed a central reddish-black crust&#44; with a rim of sparse red globules&#44; and a pink border with fine white scale &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; No other lesions were evident&#44; and the remaining physical examination had no abnormalities&#46; Eye red reflex and otoacoustic emissions screenings were normal&#46; His family history was unremarkable&#46; A punch biopsy was made&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Histopathology showed an ulcerated lesion with an infiltrate composed of large epithelioid cells with a kidney-shaped nucleus&#44; with epidermotropism &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Mitotic figures were observed&#46; The background infiltrate was composed of lymphocytes&#44; plasma cells and a significant number of eosinophils &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; Immunostaining was positive for CD1a &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>A&#41;&#44; langerin &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>B&#41;&#44; S100 protein and CD45&#44; and negative for CD68 and CD34&#46; A presumptive diagnosis of solitary congenital self-healing Langerhans cell histiocytoma was made&#46; Complete Blood Count &#40;CBC&#41;&#44; Erythrocyte Sedimentation Rate &#40;ESR&#41;&#44; coagulation times&#44; kidney and liver function tests and lactate dehydrogenase were within the normal range for the age&#46; The newborn underwent chest radiography and abdominal ultrasound&#44; which revealed no abnormalities&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The infant maintains regular follow-ups&#46; At 6 months appointment&#44; no systemic involvement was noticed and there was a complete involution of the skin lesion&#46; The overall well-appearing state of the neonate&#44; lack of systemic signs&#44; and spontaneous involution of the lesion&#44; associated with the histopathology and immunohistochemistry findings&#44; were compatible with congenital self-healing histiocytosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">Langerhans cell histiocytosis &#40;LCH&#41; is a rare neoplasm&#44; characterized by a pathological Langerhans cell proliferation&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> LCH can present as a single organ or multi-systemic involvement&#44; with a wide spectrum of manifestations&#44; ranging from self-resolving skin lesions to disseminated forms&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The prognosis depends on the extent of systemic involvement&#44; with the single-system disease having a good prognosis&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Congenital self-healing Langerhans cell histiocytosis &#40;CSHLCH&#41; is a rare variant of LCH&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It usually presents as multiple papules or nodules &#40;from the &#8220;blueberry muffin baby&#8221; spectrum&#41;&#44; but uncommonly a solitary lesion may occur&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a> LC histiocytoma &#40;or solitary CSHLCH&#41; is considered a unimodular or paucinodular CSHLCH variant&#46; It presents as a single reddish nodule at birth or within the first weeks of life&#44; that progresses to crusting and ulceration&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Cutaneous lesions typically regress within a few months&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Neonates are usually healthy&#44; with no systemic involvement&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> A recent retrospective review of 82 neonates showed complete involution of the lesion in all cases who have not been submitted to excision&#46; Furthermore&#44; the authors observed no progression&#44; recurrence&#44; or multisystemic involvement&#44; with a median follow-up of 15 months&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Histopathology is characterized by a dense dermal infiltrate composed of large cells with abundant eosinophilic cytoplasm and round-oval or kidney-shaped nucleus&#44; accompanied by an infiltrate comprising lymphocytes and eosinophils&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;6</span></a> Histopathological differences have been between solitary CSHLCH and multiple papular or nodular forms&#46; In the former&#44; there is a deeper infiltrate of the whole dermis and often extends to superficial subcutis&#44; superficial ulceration or crusting&#44; frequent parakeratosis and necrosis&#44; and little or no epidermotropism&#46; In solitary CSHLCH&#44; there are large-sized cells&#44; sometimes with &#8220;ground-glass&#8221; cytoplasm and numerous mitoses&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Diagnosis is confirmed by positive immunostaining for CD1a&#44; S100&#44; and langerin&#44; markers of Langerhans cells&#46; Electron microscopy reveals the characteristic Birbeck granules&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> but usually is not needed&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">There is some variability in dermoscopy reports of LCH&#44; due to the clinical spectrum and different lesion stages&#46; Reddish-lilac color of nodular lesions&#44; with peripheral telangiectasias&#44; violaceous lacunae and clods&#44; and whitish areas may be present&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7&#44;8</span></a> However&#44; dermoscopy descriptions are sparse in the literature&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">There are no specific guidelines for the management and follow-up of solitary CSHLCH&#46; Initial work-up should include a complete physical examination &#40;exclude other skin or mucosal lesions&#44; lymphadenopathy&#44; or hepatosplenomegaly&#41;&#44; laboratory analysis &#40;CBC&#44; ESR&#44; C-reactive protein&#44; coagulation studies&#44; hepatic and renal function tests&#44; blood and urine osmolarities&#41; and imaging exams &#40;abdominal ultrasound&#44; chest X-Ray&#44; complete skeletal radiographs&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In summary&#44; solitary CSHLCH diagnosis should be raised in the presence of a single ulcerated congenital lesion and usually portends a good prognosis&#46; CSHLCH diagnosis is made in retrospect after work-up and follow-up of these patients&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> showing a spontaneous resolution and absence of systemic involvement&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Financial support</span><p id="par0050" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Authors&#39; contributions</span><p id="par0055" class="elsevierStylePara elsevierViewall">Tiago Fernandes Gomes&#58; Data collection&#44; analysis and interpretation&#59; critical literature review&#59; preparation and writing of the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Jos&#233; Carlos Cardoso&#58; Approval of the final version of the manuscript&#59; data collection&#44; analysis and interpretation&#59; manuscript critical review&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Victoria Guiote&#58; Approval of the final version of the manuscript&#59; data collection&#44; analysis&#44; and interpretation&#59; manuscript critical review&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Felicidade Santiago&#58; Approval of the final version of the manuscript&#59; data collection&#44; analysis&#44; and interpretation&#59; intellectual participation in propaedeutic and&#47;or therapeutic management of studied cases&#59; manuscript critical review&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflicts of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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