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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Malignant chondroid syringoma &#40;MCS&#41;&#44; which is also known as malignant cutaneous mixed tumor&#44; is an extremely rare malignant neoplasm that originates in the adnexa&#46; Approximately 50 cases have been reported in the literature to date&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> In contrast with its benign counterpart &#40;benign mixed tumor&#41;&#44; which shows a predilection for men and generally affects the head&#44; MCS is more common in women and mainly affects the limbs and trunk&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;3</span></a> Only 10 cases of MCS affecting the head have been reported to date&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;10</span></a> Below&#44; we report the case of giant facial MCS with distant metastasis&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 77-year-old man with myasthenia gravis and arterial hypertension under treatment presented with a painless tumor on the right cheek that had first appeared 2 years earlier&#46; The lesion had grown abruptly in the previous 3 months&#46; The patient reported difficulty chewing and reduced salivation&#46; Physical examination revealed a firm multilobulated skin-colored subcutaneous tumor nodule measuring 11<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>6&#46;7<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;5<span class="elsevierStyleHsp" style=""></span>cm with multiple telangiectasias adhering to deeper planes&#46; The nodule affected the malar and lower right maxillary areas of the face &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#41;&#46; Enlarged lymph nodes were palpable in the ipsilateral axilla&#46; Computed tomography of the head revealed a heterogeneous multilobulated lesion with hypodense areas measuring 10<span class="elsevierStyleHsp" style=""></span>cm at its widest diameter on the right side of the face&#46; The lesion was in contact with the masseter and affected the ipsilateral parotid gland&#46; Histology of an incisional biopsy specimen &#40;hematoxylin&#8211;eosin&#41; revealed tumor proliferation constituted by epithelial cells&#46; These formed solid nests&#44; cords&#44; and gland-like structures&#44; the main finding being foci of necrosis and other hypercellular foci with marked anisokaryosis&#44; evident nucleoli&#44; and several mitotic figures &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The surrounding stroma contained mainly sclerohyaline areas&#44; myxoid areas&#44; and other chondroid lesions&#46; Immunohistochemistry revealed positive findings for carcinoembryonic antigen &#40;CEA&#41;&#44; epithelial membrane antigen &#40;EMA&#41;&#44; protein S-100&#44; and cytokeratin and a KI-67 result of 25&#37;&#46; Based on these results&#44; we diagnosed the patient&#39;s condition as MCS&#46; Positron-emission tomography revealed hyperdense and hypermetabolic nodular lesions compatible with metastasis to the brain&#44; liver&#44; right axillary lymph nodes&#44; and soft tissue in the right arm&#46; The patient died of respiratory causes during admission&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">First described by Hirsch and Helwig in 1961&#44; chondroid syringoma is a benign tumor that originates in the eccrine or apocrine sweat glands&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> It is very uncommon&#44; accounting for only 0&#46;01&#37; of all primary skin neoplasms&#46; It presents as a solitary&#44; slow-growing&#44; painless intradermal or subcutaneous nodule that affects the head and neck&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> It more commonly affects middle-aged men&#46; The size of the tumor can range from 2<span class="elsevierStyleHsp" style=""></span>mm to more than 1<span class="elsevierStyleHsp" style=""></span>cm&#59; tumors measuring more than 3<span class="elsevierStyleHsp" style=""></span>cm are more likely to be malignant&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In contrast with its benign counterpart&#44; MCS mainly affects women&#44; has no predilection for age&#44; and is more commonly observed on the limbs or the trunk&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;3</span></a> The tumor progresses slowly and is characterized by a final burst of rapid growth that can result in local destruction&#44; metastasis&#44; and death&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> The most common site of distant metastasis is the lung&#44; followed by bone and the brain&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a> In the case reported here&#44; the tumor affected an elderly man&#44; was located on the head and neck&#44; and initially progressed slowly with a burst of growth later in its course&#46; Local invasion was detected at diagnosis&#44; as was metastasis to lymphatic tissue&#44; the brain&#44; liver&#44; and soft tissue&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">MCS can develop de novo or&#44; more rarely&#44; from an incompletely resected benign syringoma&#46; Recurrence of the lesion should alert to the possibility of malignancy&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> In the present case&#44; the tumor developed de novo&#44; and the patient reported previous removal of facial lesions&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">While some authors have pointed to local injury as playing a role in the etiology and pathogenesis of the tumor&#44; no clear risk factors for either benign or MCS have been identified to date&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Histologically&#44; chondroid syringoma is characterized by both epithelial and mesenchymal components&#46; The mesenchymal component may have myxoid&#44; chondroid&#44; osteoid&#44; adipose&#44; and fibrous characteristics and contain epithelial cell nests that differ depending on whether the eccrine or apocrine glands are affected&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The criteria for malignancy include cytological atypia&#44; increased mitotic activity&#44; tumor necrosis&#44; capsular invasion with compromise of deep structures&#44; and&#44; possibly&#44; satellite nodules&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;7</span></a> Immunohistochemistry reveals the simultaneous presence of luminal cells that were positive for cytokeratin&#44; CEA&#44; and EMA and myoepithelial cells that were positive for cytokeratin&#44; protein S-100&#44; and vimentin&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Given its uncharacteristic clinical presentation&#44; chondroid syringoma can only be diagnosed based on histopathology&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">The differential diagnosis includes epidermal cysts&#44; dermoid cysts&#44; pilar cysts&#44; pilomatrixoma&#44; trichoepithelioma&#44; schwannoma&#44; and neurofibroma&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Chondroid syringoma is treated by complete excision&#46; Control of the disease and prevention of recurrence are necessarily based on a wide free margin&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a> Local radiotherapy is often unsuccessful&#44; although bone metastases do respond to it&#46; Combination chemotherapy has not been reported to be beneficial in patients with metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Give the high rate of recurrence of MCS &#40;49&#37;&#41;&#44; the patient should be followed up closely&#46; More importantly&#44; follow-up should be long-term&#44; since there have been reports of recurrence with dissemination to the lymphatic system and distant metastasis up to 12&#8211;20 years after the initial removal of the tumor&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case and Research Letter
Malignant Chondroid Syringoma of the Face With Distant Metastasis
Siringoma condroide maligno de localización facial con metástasis a distancia
J.A. Mirandaa,
Autor para correspondencia
Ago_090@hotmail.com

Corresponding author.
, M.M. Mazzottab, É.V. Zalazara
a Cátedra de Dermatología, Hospital Nacional de Clínicas, Universidad Nacional de Córdoba, Córdoba, Argentina
b Cátedra de Anatomía Patológica, Hospital Nacional de Clínicas, Universidad Nacional de Córdoba, Córdoba, Argentina
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Firm multilobulated subcutaneous lesion adhering to deeper planes on the malar and lower maxillary area of the right half of the face&#46; Frontal view &#40;A&#41; and lateral view &#40;B&#41;&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Malignant chondroid syringoma &#40;MCS&#41;&#44; which is also known as malignant cutaneous mixed tumor&#44; is an extremely rare malignant neoplasm that originates in the adnexa&#46; Approximately 50 cases have been reported in the literature to date&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> In contrast with its benign counterpart &#40;benign mixed tumor&#41;&#44; which shows a predilection for men and generally affects the head&#44; MCS is more common in women and mainly affects the limbs and trunk&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;3</span></a> Only 10 cases of MCS affecting the head have been reported to date&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;10</span></a> Below&#44; we report the case of giant facial MCS with distant metastasis&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 77-year-old man with myasthenia gravis and arterial hypertension under treatment presented with a painless tumor on the right cheek that had first appeared 2 years earlier&#46; The lesion had grown abruptly in the previous 3 months&#46; The patient reported difficulty chewing and reduced salivation&#46; Physical examination revealed a firm multilobulated skin-colored subcutaneous tumor nodule measuring 11<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>6&#46;7<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;5<span class="elsevierStyleHsp" style=""></span>cm with multiple telangiectasias adhering to deeper planes&#46; The nodule affected the malar and lower right maxillary areas of the face &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#41;&#46; Enlarged lymph nodes were palpable in the ipsilateral axilla&#46; Computed tomography of the head revealed a heterogeneous multilobulated lesion with hypodense areas measuring 10<span class="elsevierStyleHsp" style=""></span>cm at its widest diameter on the right side of the face&#46; The lesion was in contact with the masseter and affected the ipsilateral parotid gland&#46; Histology of an incisional biopsy specimen &#40;hematoxylin&#8211;eosin&#41; revealed tumor proliferation constituted by epithelial cells&#46; These formed solid nests&#44; cords&#44; and gland-like structures&#44; the main finding being foci of necrosis and other hypercellular foci with marked anisokaryosis&#44; evident nucleoli&#44; and several mitotic figures &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The surrounding stroma contained mainly sclerohyaline areas&#44; myxoid areas&#44; and other chondroid lesions&#46; Immunohistochemistry revealed positive findings for carcinoembryonic antigen &#40;CEA&#41;&#44; epithelial membrane antigen &#40;EMA&#41;&#44; protein S-100&#44; and cytokeratin and a KI-67 result of 25&#37;&#46; Based on these results&#44; we diagnosed the patient&#39;s condition as MCS&#46; Positron-emission tomography revealed hyperdense and hypermetabolic nodular lesions compatible with metastasis to the brain&#44; liver&#44; right axillary lymph nodes&#44; and soft tissue in the right arm&#46; The patient died of respiratory causes during admission&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">First described by Hirsch and Helwig in 1961&#44; chondroid syringoma is a benign tumor that originates in the eccrine or apocrine sweat glands&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> It is very uncommon&#44; accounting for only 0&#46;01&#37; of all primary skin neoplasms&#46; It presents as a solitary&#44; slow-growing&#44; painless intradermal or subcutaneous nodule that affects the head and neck&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> It more commonly affects middle-aged men&#46; The size of the tumor can range from 2<span class="elsevierStyleHsp" style=""></span>mm to more than 1<span class="elsevierStyleHsp" style=""></span>cm&#59; tumors measuring more than 3<span class="elsevierStyleHsp" style=""></span>cm are more likely to be malignant&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In contrast with its benign counterpart&#44; MCS mainly affects women&#44; has no predilection for age&#44; and is more commonly observed on the limbs or the trunk&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;3</span></a> The tumor progresses slowly and is characterized by a final burst of rapid growth that can result in local destruction&#44; metastasis&#44; and death&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> The most common site of distant metastasis is the lung&#44; followed by bone and the brain&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a> In the case reported here&#44; the tumor affected an elderly man&#44; was located on the head and neck&#44; and initially progressed slowly with a burst of growth later in its course&#46; Local invasion was detected at diagnosis&#44; as was metastasis to lymphatic tissue&#44; the brain&#44; liver&#44; and soft tissue&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">MCS can develop de novo or&#44; more rarely&#44; from an incompletely resected benign syringoma&#46; Recurrence of the lesion should alert to the possibility of malignancy&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> In the present case&#44; the tumor developed de novo&#44; and the patient reported previous removal of facial lesions&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">While some authors have pointed to local injury as playing a role in the etiology and pathogenesis of the tumor&#44; no clear risk factors for either benign or MCS have been identified to date&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Histologically&#44; chondroid syringoma is characterized by both epithelial and mesenchymal components&#46; The mesenchymal component may have myxoid&#44; chondroid&#44; osteoid&#44; adipose&#44; and fibrous characteristics and contain epithelial cell nests that differ depending on whether the eccrine or apocrine glands are affected&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The criteria for malignancy include cytological atypia&#44; increased mitotic activity&#44; tumor necrosis&#44; capsular invasion with compromise of deep structures&#44; and&#44; possibly&#44; satellite nodules&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;7</span></a> Immunohistochemistry reveals the simultaneous presence of luminal cells that were positive for cytokeratin&#44; CEA&#44; and EMA and myoepithelial cells that were positive for cytokeratin&#44; protein S-100&#44; and vimentin&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Given its uncharacteristic clinical presentation&#44; chondroid syringoma can only be diagnosed based on histopathology&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">The differential diagnosis includes epidermal cysts&#44; dermoid cysts&#44; pilar cysts&#44; pilomatrixoma&#44; trichoepithelioma&#44; schwannoma&#44; and neurofibroma&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Chondroid syringoma is treated by complete excision&#46; Control of the disease and prevention of recurrence are necessarily based on a wide free margin&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a> Local radiotherapy is often unsuccessful&#44; although bone metastases do respond to it&#46; Combination chemotherapy has not been reported to be beneficial in patients with metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Give the high rate of recurrence of MCS &#40;49&#37;&#41;&#44; the patient should be followed up closely&#46; More importantly&#44; follow-up should be long-term&#44; since there have been reports of recurrence with dissemination to the lymphatic system and distant metastasis up to 12&#8211;20 years after the initial removal of the tumor&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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