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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Oral Carcinoma Cuniculatum &#40;OCC&#41; corresponds to a rare&#44; well-differentiated variant of Oral Squamous Cell Carcinoma &#40;OSCC&#41; first included in the World Health Organization &#40;WHO&#41; Classification of Head and Neck tumors in 2005&#46; According to the WHO&#44; OCC is defined as a malignant neoplastic proliferation of stratified squamous epithelium in broad processes with keratin cores and keratin-filled crypts which seem to burrow into the bone&#44; without obvious cytological features of malignancy&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is estimated that OCC accounts for up to 2&#46;7&#37; of all OSCC&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Up to date&#44; less than 60 cases have been reported&#46; Here we report an unsual case of an oral carcinoma cuniculatum affecting the gingiva of a child&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">An 11-year-old boy was consulted because of local pain and paresthesia of the left lower lip during an unknown amount of time&#46; He had a history of grade II ependymoma&#44; which was appropriately treated with surgery and radiotherapy and controlled routinely&#46; He had no systemic symptoms&#44; known allergies or medical history of any other illness&#46; Extra-oral examination revealed no anomalies&#46; Intraorally&#44; a mass involving both the buccal and lingual gingiva between the lower left second molar and lower left first premolar was observed&#46; An extensive verrucous white patch was covering the buccal side of the mass&#44; while cystic-like white nodules with superficial blood vessels were also present on both the lingual and buccal sides of the tumor &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The teeth involved in the mass were mobile&#46; Cone-beam computed tomography revealed a well-defined large radiolucency involving the whole thickness of the posterior left mandible perforating the buccal bone plate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; An incisional biopsy was taken which showed a tumoral mass composed of multiple branching keratin-filled crypts sheets lined by epidermoid epithelial cells&#46; The endophytic epithelial growth pattern burrowed deep inside into the alveolar bone&#44; forming a complex network of connected canaliculi&#44; resembling rabbit burrows&#46; The epithelial cells were well differentiated&#44; and no atypia&#44; mitosis&#44; perineural nor lymphovascular invasion was observed&#46; A chronic inflammatory infiltrates consisting mainly of lymphocytes with local areas of neutrophil accumulation was observed in some areas of the stroma &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; The lesion was diagnosed as a carcinoma cuniculatum of the oral cavity&#46; Differential diagnosis with a solid variant of an odontogenic keratocyst was done due to its histopathological similarity&#46; The CT scan examination revealed no lymph node involvement and no metastasis&#44; and the patient was referred for surgical treatment&#46; The patient was controlled with regularity and no recurrences have been observed after a 5-years-follow up period&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Carcinoma Cuniculatum &#40;CC&#41; is a rare&#44; low-grade variant of Squamous Cell Carcinoma &#40;SCC&#41; most commonly located on the plant of the foot&#44; but any anatomic site can be affected&#44; including the oral cavity&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> According to a recent systemic review that analyzed 43 cases of OCC&#44; this tumor has a slight predilection for females&#44; being more frequent between the sixth and seventh decade of life&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Our case is the second report of an OCC in children&#46; The first case was reported by Hutton el al&#46; in 2010&#44; which corresponded to an OCC affecting the anterior maxillary gingiva of a 7-year-old girl&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> When affecting the oral cavity&#44; the most common location is the mandibular gingiva followed by the maxillary gingiva&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Cases affecting the alveolar ridge&#44; tongue and palate have also been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> When affecting the gingiva&#44; bone involvement is common&#46; Imaging usually shows a radiolucency with ill-defined margins reabsorbing the adjacent cortical bone&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The most common clinical symptom is pain&#44; followed by ulceration&#44; swelling&#44; and induration&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5&#44;6</span></a> Our patient apart from the aforementioned symptoms also reported paresthesia&#44; which to our knowledge&#44; has not been reported before&#46; The main differential diagnosis of CC is Verrucous Carcinoma &#40;VC&#41;&#44; another well-differentiated uncommon variant of SCC&#46; But other keratinizing lesions&#44; such as keratinizing odontogenic cysts have to be ruled out&#44; especially with dealing with small incisional biopsies&#46; In the early days&#44; CC was thought to be a VC&#44; but is now considered a distinct entity of SCC&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Clinically&#44; both CC and VC can have a verrucous appearance&#44; although CC might exhibit a sessile pink-reddish papillary surface&#44; which is not observed in VC&#46; Histologically&#44; CC shows a tortuous invasive &#40;endophytic&#41; component with keratin plugging&#44; which contrasts with the more exophytic verrucous growth and &#8220;pushing borders&#8221; observed in VC&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Microabscesses filled with neutrophils are also commonly reported in CC&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The main treatment of choice is surgical excision&#46; Although OCC can be locally aggressive&#44; lymph node and distant metastases are uncommon&#44;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;6</span></a> and the 5-year survival rate has been reported above 90&#37;&#46; <a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Local recurrences have been reported&#44; but are not very common&#46; In respect to our case&#44; the patient remained disease-free after a follow-up period of 5 years&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0030" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#39; contributions</span><p id="par0035" class="elsevierStylePara elsevierViewall">Sven Niklander&#58; Preparation and writing of the manuscript&#59; approval of the final version of the manuscript&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Luz Mar&#237;a Sernuda&#58; Approval of the final version of the manuscript&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ren&#233; Martinez&#58; Preparation and writing of the manuscript&#59; approval of the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">None declared&#46;</p><p id="par0055" 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; The study was done in Vi&#241;a del Mar&#44; 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Case Letter
Unusual case of carcinoma cuniculatum affecting the oral cavity of an 11-year-old boy
Sven Niklandera,
Corresponding author
sven.niklander@unab.cl

Corresponding author.
, Luz María Sernudab, René Martineza
a Department of Surgery and Oral Pathology, Faculty of Dentistry, Universidad Andres Bello, Santiago, Chile
b Maxillofacial Surgery, Quilpué Hospital, Viña del Mar-Quillota Health Service, Quilpué, Chile
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    "titulo" => "Unusual case of carcinoma cuniculatum affecting the oral cavity of an 11-year-old boy"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Oral Carcinoma Cuniculatum &#40;OCC&#41; corresponds to a rare&#44; well-differentiated variant of Oral Squamous Cell Carcinoma &#40;OSCC&#41; first included in the World Health Organization &#40;WHO&#41; Classification of Head and Neck tumors in 2005&#46; According to the WHO&#44; OCC is defined as a malignant neoplastic proliferation of stratified squamous epithelium in broad processes with keratin cores and keratin-filled crypts which seem to burrow into the bone&#44; without obvious cytological features of malignancy&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is estimated that OCC accounts for up to 2&#46;7&#37; of all OSCC&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Up to date&#44; less than 60 cases have been reported&#46; Here we report an unsual case of an oral carcinoma cuniculatum affecting the gingiva of a child&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">An 11-year-old boy was consulted because of local pain and paresthesia of the left lower lip during an unknown amount of time&#46; He had a history of grade II ependymoma&#44; which was appropriately treated with surgery and radiotherapy and controlled routinely&#46; He had no systemic symptoms&#44; known allergies or medical history of any other illness&#46; Extra-oral examination revealed no anomalies&#46; Intraorally&#44; a mass involving both the buccal and lingual gingiva between the lower left second molar and lower left first premolar was observed&#46; An extensive verrucous white patch was covering the buccal side of the mass&#44; while cystic-like white nodules with superficial blood vessels were also present on both the lingual and buccal sides of the tumor &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The teeth involved in the mass were mobile&#46; Cone-beam computed tomography revealed a well-defined large radiolucency involving the whole thickness of the posterior left mandible perforating the buccal bone plate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; An incisional biopsy was taken which showed a tumoral mass composed of multiple branching keratin-filled crypts sheets lined by epidermoid epithelial cells&#46; The endophytic epithelial growth pattern burrowed deep inside into the alveolar bone&#44; forming a complex network of connected canaliculi&#44; resembling rabbit burrows&#46; The epithelial cells were well differentiated&#44; and no atypia&#44; mitosis&#44; perineural nor lymphovascular invasion was observed&#46; A chronic inflammatory infiltrates consisting mainly of lymphocytes with local areas of neutrophil accumulation was observed in some areas of the stroma &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; The lesion was diagnosed as a carcinoma cuniculatum of the oral cavity&#46; Differential diagnosis with a solid variant of an odontogenic keratocyst was done due to its histopathological similarity&#46; The CT scan examination revealed no lymph node involvement and no metastasis&#44; and the patient was referred for surgical treatment&#46; The patient was controlled with regularity and no recurrences have been observed after a 5-years-follow up period&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Carcinoma Cuniculatum &#40;CC&#41; is a rare&#44; low-grade variant of Squamous Cell Carcinoma &#40;SCC&#41; most commonly located on the plant of the foot&#44; but any anatomic site can be affected&#44; including the oral cavity&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> According to a recent systemic review that analyzed 43 cases of OCC&#44; this tumor has a slight predilection for females&#44; being more frequent between the sixth and seventh decade of life&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Our case is the second report of an OCC in children&#46; The first case was reported by Hutton el al&#46; in 2010&#44; which corresponded to an OCC affecting the anterior maxillary gingiva of a 7-year-old girl&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> When affecting the oral cavity&#44; the most common location is the mandibular gingiva followed by the maxillary gingiva&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Cases affecting the alveolar ridge&#44; tongue and palate have also been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> When affecting the gingiva&#44; bone involvement is common&#46; Imaging usually shows a radiolucency with ill-defined margins reabsorbing the adjacent cortical bone&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The most common clinical symptom is pain&#44; followed by ulceration&#44; swelling&#44; and induration&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5&#44;6</span></a> Our patient apart from the aforementioned symptoms also reported paresthesia&#44; which to our knowledge&#44; has not been reported before&#46; The main differential diagnosis of CC is Verrucous Carcinoma &#40;VC&#41;&#44; another well-differentiated uncommon variant of SCC&#46; But other keratinizing lesions&#44; such as keratinizing odontogenic cysts have to be ruled out&#44; especially with dealing with small incisional biopsies&#46; In the early days&#44; CC was thought to be a VC&#44; but is now considered a distinct entity of SCC&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Clinically&#44; both CC and VC can have a verrucous appearance&#44; although CC might exhibit a sessile pink-reddish papillary surface&#44; which is not observed in VC&#46; Histologically&#44; CC shows a tortuous invasive &#40;endophytic&#41; component with keratin plugging&#44; which contrasts with the more exophytic verrucous growth and &#8220;pushing borders&#8221; observed in VC&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Microabscesses filled with neutrophils are also commonly reported in CC&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The main treatment of choice is surgical excision&#46; Although OCC can be locally aggressive&#44; lymph node and distant metastases are uncommon&#44;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;6</span></a> and the 5-year survival rate has been reported above 90&#37;&#46; <a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Local recurrences have been reported&#44; but are not very common&#46; In respect to our case&#44; the patient remained disease-free after a follow-up period of 5 years&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0030" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#39; contributions</span><p id="par0035" class="elsevierStylePara elsevierViewall">Sven Niklander&#58; Preparation and writing of the manuscript&#59; approval of the final version of the manuscript&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Luz Mar&#237;a Sernuda&#58; Approval of the final version of the manuscript&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ren&#233; Martinez&#58; Preparation and writing of the manuscript&#59; approval of the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">None declared&#46;</p><p id="par0055" 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; The study was done in Vi&#241;a del Mar&#44; 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                      "titulo" => "Mandibular cuniculatum carcinoma&#58; apropos of 3 cases and literature review"
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                            5 => "F&#46; Gruffaz"
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                      "doi" => "10.1002/hed.21493"
                      "Revista" => array:6 [
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                        "volumen" => "34"
                        "paginaInicial" => "291"
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        "identificador" => "xack568755"
        "titulo" => "Acknowledgments"
        "texto" => "<p id="par0060" class="elsevierStylePara elsevierViewall">We would like to thank Dr&#46; Javiera Fuentes for her help with the interpretation of the cone-beam computed tomography images&#46;</p>"
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Article information
ISSN: 03650596
Original language: English
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