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However&#44; there have been very few reports of oral metastases&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> which mainly affect the gingiva&#44; tongue&#44; tonsils&#44; and mandible&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe the case of an 86-year-old white man admitted with a black lesion on the tongue that had persisted for about 2 months&#46; During the consultation&#44; the patient rejected the idea that he had a disease and complained about the appearance&#44; concurrently with the lingual lesions&#44; of dark nodules on his neck and upper limbs associated with intense and persistent itching&#44; which had on occasion spread to the trunk&#46; These skin lesions had been treated with antihistamines and topical corticosteroids prescribed initially by his family physician and subsequently by a dermatologist&#44; 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3</a>A&#41;&#59; <span class="elsevierStyleItalic">b</span>&#41; 3 pigmented blue-brown lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#44; C&#44; second arrow&#41; situated 14<span class="elsevierStyleHsp" style=""></span>mm from the first lesion&#59; and <span class="elsevierStyleItalic">c</span>&#41; an irregularly shaped variegated brown lesion on the right parascapular region&#44; of 14<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>15<span class="elsevierStyleHsp" style=""></span>mm &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#44; first arrow&#41; characterized by a fragmented thickened network and areas of regression&#44; consistent with the diagnosis of melanoma &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>B and C&#41;&#46; Histopathology examination of an incisional biopsy of the parascapular lesion showed a dermis colonized by atypical epithelioid cells indicating a melanoma &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; Unexpectedly&#44; after the diagnosis of cutaneous melanoma was confirmed&#44; the patient refused to undergo oral biopsy&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">A diagnosis of cutaneous melanoma with oral metastases was confirmed on the basis of the clinical&#44; dermoscopic&#44; and histological data&#46; A chest radiograph demonstrated no active parenchymal lesions&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">A computed tomography scan of the maxillofacial region&#44; neck&#44; brain&#44; thorax and abdomen revealed no further metastases&#44; but generalized lymphadenopathy&#46; Due to the presence of in-transit metastases&#44; sentinel lymph-node biopsy was not performed&#44; as recently reported&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was referred to an oncology unit&#44; but no therapeutic regimen was undertaken given his age&#44; his refusal to consent to treatment&#44; and the number and extent of his lesions&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Melanoma is a tumor caused by the malignant transformation of melanocytes&#44; a cell line derived from the neuroectoderm&#46; Although the skin continues to be the most frequent site of primary disease &#40;95&#37; of cases&#41;&#44; the embryologic origin of melanocytes explains why melanoma is not exclusively a skin cancer&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In fact&#44; melanomas may also arise in extracutaneous sites&#44; including the mucosal surfaces of the respiratory&#44; gastrointestinal&#44; and genitourinary tracts and other sites where neural crest cells migrate&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The appearance of a primary melanoma located in the oral cavity is very rare&#44; accounting for only 1&#8211;2&#37; of all mucosal melanomas and 0&#46;5&#37; of all oral malignancies&#59; secondary or metastatic forms are even more rare&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;8</span></a> When the maxillofacial region is involved&#44; metastases of cutaneous melanoma are mainly reported in the tongue&#44; tonsils&#44; mandible&#44; gingiva&#44; and parotid glands&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">When a patient presents with a pigmented oral lesion&#44; an extraoral clinical examination should be performed&#59; in fact&#44; according to Greene and coworkers&#44;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;9</span></a> in order to consider an oral melanoma as primary&#44; the following 3 criteria must be met&#58; <span class="elsevierStyleItalic">a</span>&#41; demonstration of melanoma only in the oral cavity&#59; <span class="elsevierStyleItalic">b</span>&#41; presence of junctional activity&#59; and <span class="elsevierStyleItalic">c</span>&#41; inability to demonstrate extra-oral primary melanoma&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In our case&#44; the presence of a cutaneous lesion with a histopathological diagnosis of melanoma allowed us to consider the oral lesions as metastatic&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">When analyzing a suspicious skin lesion&#44; it is important to bear in mind the ABCD rule in which A refers to asymmetry&#44; B to border irregularities&#44; C to color heterogeneity&#44; and D to dynamics &#40;in color&#44; elevation&#44; or size&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Furthermore&#44; it has been noted&#44; that dermoscopy always enhances the diagnostic accuracy and identifies lesions that must be biopsied&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> as was the case in this patient&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion&#44; the appearance of a metastatic lingual melanoma is a very rare event&#44; hence close collaboration among different specialists is very important in case of suspicious pigmented lesions to ensure their early detection and a prompt treatment of such aggressive neoplasm&#46;</p></span>"
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Case and Research Letter
Metastatic Melanoma of the Tongue: A Rare Case
Melanoma metastásico de la lengua: un caso raro
D. Compilatoa,
Corresponding author
compilato@odonto.unipa.it

Corresponding author.
, S. Amatob, G. Campisia
a Department of Surgical and Oncological Disciplines, V. Margiotta Oral Medicine Unit, Palermo University Hospital, Palermo, Italy
b Dermatology Unit, ARNAS, Ospedale Civico e Fatebenefratelli G. Di Cristina e Maurizio Ascoli, Palermo, Italy
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However&#44; there have been very few reports of oral metastases&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> which mainly affect the gingiva&#44; tongue&#44; tonsils&#44; and mandible&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe the case of an 86-year-old white man admitted with a black lesion on the tongue that had persisted for about 2 months&#46; During the consultation&#44; the patient rejected the idea that he had a disease and complained about the appearance&#44; concurrently with the lingual lesions&#44; of dark nodules on his neck and upper limbs associated with intense and persistent itching&#44; which had on occasion spread to the trunk&#46; These skin lesions had been treated with antihistamines and topical corticosteroids prescribed initially by his family physician and subsequently by a dermatologist&#44; without clinical improvement&#46; Intraoral examination revealed the presence of a wide&#44; irregularly shaped&#44; sepia-black macular lesion that was firm to the touch and asymptomatic&#44; extending from the dorsal to the ventral surface of the tongue&#46; This was associated with small stiff nodules with an ulcerated surface on the left margin and the midline of the anterior third of the tongue &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Extraoral examination &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A and B&#41; revealed the presence of blackish oval nodular lesions&#44; palpable and painless&#44; localized to the neck and upper limbs&#46; A diffuse cervical lymphadenopathy was also detected&#46; These clinical findings gave rise to a suspected diagnosis of oral melanoma&#46; While considering it important to establish whether the oral lesions were primary or metastatic&#44; we preferred to start the diagnostic work-up of the suspected melanoma with an examination of the skin lesions&#46; The results of routine blood tests were within normal limits&#44; with the exception of an increased erythrocyte sedimentation rate&#46; Tests for neoplastic markers revealed the carcino-embryogenic antigen&#46; A more detailed clinical and dermoscopic examination of the skin &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#41; revealed the following findings&#58; <span class="elsevierStyleItalic">a</span>&#41; a brown-black variegated pigmented nevus on the spinal column between the shoulders&#44; irregular in shape and color measuring 21<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>16<span class="elsevierStyleHsp" style=""></span>mm &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#44; third arrow&#41; and characterized by a blue&#8211;gray veil and irregularly shaped globules&#59; the presumptive diagnosis was melanoma with in-transit metastases &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>A&#41;&#59; <span class="elsevierStyleItalic">b</span>&#41; 3 pigmented blue-brown lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#44; C&#44; second arrow&#41; situated 14<span class="elsevierStyleHsp" style=""></span>mm from the first lesion&#59; and <span class="elsevierStyleItalic">c</span>&#41; an irregularly shaped variegated brown lesion on the right parascapular region&#44; of 14<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>15<span class="elsevierStyleHsp" style=""></span>mm &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#44; first arrow&#41; characterized by a fragmented thickened network and areas of regression&#44; consistent with the diagnosis of melanoma &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>B and C&#41;&#46; Histopathology examination of an incisional biopsy of the parascapular lesion showed a dermis colonized by atypical epithelioid cells indicating a melanoma &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; Unexpectedly&#44; after the diagnosis of cutaneous melanoma was confirmed&#44; the patient refused to undergo oral biopsy&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">A diagnosis of cutaneous melanoma with oral metastases was confirmed on the basis of the clinical&#44; dermoscopic&#44; and histological data&#46; A chest radiograph demonstrated no active parenchymal lesions&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">A computed tomography scan of the maxillofacial region&#44; neck&#44; brain&#44; thorax and abdomen revealed no further metastases&#44; but generalized lymphadenopathy&#46; Due to the presence of in-transit metastases&#44; sentinel lymph-node biopsy was not performed&#44; as recently reported&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was referred to an oncology unit&#44; but no therapeutic regimen was undertaken given his age&#44; his refusal to consent to treatment&#44; and the number and extent of his lesions&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Melanoma is a tumor caused by the malignant transformation of melanocytes&#44; a cell line derived from the neuroectoderm&#46; Although the skin continues to be the most frequent site of primary disease &#40;95&#37; of cases&#41;&#44; the embryologic origin of melanocytes explains why melanoma is not exclusively a skin cancer&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In fact&#44; melanomas may also arise in extracutaneous sites&#44; including the mucosal surfaces of the respiratory&#44; gastrointestinal&#44; and genitourinary tracts and other sites where neural crest cells migrate&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The appearance of a primary melanoma located in the oral cavity is very rare&#44; accounting for only 1&#8211;2&#37; of all mucosal melanomas and 0&#46;5&#37; of all oral malignancies&#59; secondary or metastatic forms are even more rare&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;8</span></a> When the maxillofacial region is involved&#44; metastases of cutaneous melanoma are mainly reported in the tongue&#44; tonsils&#44; mandible&#44; gingiva&#44; and parotid glands&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">When a patient presents with a pigmented oral lesion&#44; an extraoral clinical examination should be performed&#59; in fact&#44; according to Greene and coworkers&#44;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;9</span></a> in order to consider an oral melanoma as primary&#44; the following 3 criteria must be met&#58; <span class="elsevierStyleItalic">a</span>&#41; demonstration of melanoma only in the oral cavity&#59; <span class="elsevierStyleItalic">b</span>&#41; presence of junctional activity&#59; and <span class="elsevierStyleItalic">c</span>&#41; inability to demonstrate extra-oral primary melanoma&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In our case&#44; the presence of a cutaneous lesion with a histopathological diagnosis of melanoma allowed us to consider the oral lesions as metastatic&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">When analyzing a suspicious skin lesion&#44; it is important to bear in mind the ABCD rule in which A refers to asymmetry&#44; B to border irregularities&#44; C to color heterogeneity&#44; and D to dynamics &#40;in color&#44; elevation&#44; or size&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Furthermore&#44; it has been noted&#44; that dermoscopy always enhances the diagnostic accuracy and identifies lesions that must be biopsied&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> as was the case in this patient&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion&#44; 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ISSN: 15782190
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