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PCMCs are slow-growing tumors and have an indolent behavior&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> However&#44; late recurrence and metastases have been reported&#44; probably related to incomplete excision of the tumor&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Histologically&#44; it is indistinguishable from metastatic mucinous adenocarcinomas from other organs&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> particularly from the breast&#44; which can make diagnosis difficult&#46; We present a case of this rare entity and review the literature&#44; discussing the main recommendations and the immunohistochemical reactions useful to reach a correct diagnosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Description</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 68-year-old man with no past history of interest&#46; He was seen for a mobile nodular lesion that had been present for 2 years in the left axilla&#46; The lesion was not adherent to deeper planes and measured 1&#46;5<span class="elsevierStyleHsp" style=""></span>cm in diameter&#46; The clinical diagnosis was epidermal cyst&#46; Macroscopically&#44; the sample consisted of a number of reddish fragments of soft tissue&#44; including areas with a gelatinous appearance&#44; with an overall diameter of 3<span class="elsevierStyleHsp" style=""></span>cm&#46; Histologically&#44; there were fragments of skin with abundant eccrine and apocrine glands that were diffusely infiltrated by lakes of mucin &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; These lakes contained cords and solid nests of cuboidal and round epithelial cells that&#44; in some areas&#44; formed glandular structures&#44; with scattered cribriform lumina &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The cells presented cytologic atypia and occasional mitotic figures&#46; Periodic acid Schiff stain revealed an abundant quantity of mucin &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; Immunohistochemistry was positive for CK7 &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D&#41;&#44; CK19&#44; epithelial membrane antigen&#44; GATA3&#44; estrogen receptors &#40;ER&#41;&#44; progesterone receptors &#40;PR&#41;&#44; and p63 &#40;in isolated small cells&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>F&#41;&#59; the study was negative for CK20 &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E&#41;&#44; CK5&#47;6&#44; CD15&#44; and mammaglobin&#46; The pathologic diagnosis based on these findings was mucinous carcinoma&#46; Subsequently the patient underwent complete clinical evaluation&#44; including positron emission tomography &#40;PET&#41; in which no increased uptake was observed in other organs&#46; In cases such as ours&#44; it cannot be ruled out that the tumor may have arisen in a focus of ectopic breast tissue in the axilla&#59; however&#44; as no remnant of healthy breast tissue was observed in the surgical sample&#44; we made a definitive diagnosis of PCMC&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">PCMC is a rare entity that was first described in 1952 by Lenox et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> It usually shows an indolent clinical course and is typically present for several years before diagnosis&#59; it is often misdiagnosed clinically as an epidermal cyst&#44; sebaceous carcinoma&#44; cystic basal cell carcinoma&#44; squamous cell carcinoma&#44; neuroma&#44; or pilomatrixoma&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Late recurrence and metastases due to incomplete excision of the tumor<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> and lymphovascular invasion have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Apparently&#44; such recurrences have not responded well to treatment with radiotherapy or chemotherapy&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Morphologically&#44; it is not possible to differentiate PCMC from metastatic mucinous adenocarcinomas of noncutaneous organs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> Immunohistochemistry can exclude metastatic mucinous adenocarcinomas from the colon&#44; as these tumors are usually positive for CK20<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> and CDX2&#44; whereas PCMC is negative for these markers&#46; However&#44; immunohistochemistry is not particularly useful in distinguishing PCMC from mucinous adenocarcinoma of the breast&#44; as both tumors can express CK7&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> GATA3&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> mammaglobin&#44; REs&#44; and<span class="elsevierStyleHsp" style=""></span>PRs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">It has recently been suggested in the literature that detection of a myoepithelial component in the tumor &#40;confirmed by positivity for p63&#44; CK5&#47;6&#44; or calponin&#44; in isolated peripheral cells&#41; supports or suggests PCMC&#44; but unfortunately this is not a common finding&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1&#44;2</span></a> In our case&#44; we did detect the presence of this remnant of myoepithelial cells with immune stain for p63 &#40;Fig&#46;<span class="elsevierStyleHsp" style=""></span>1F&#41;&#59; however&#44; CK5&#47;6 was negative&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">In view of this situation&#44; it is strictly necessary to perform a metastatic workup &#40;computed tomography and&#47;or PET&#41;&#44; which must be negative in order to confirm that the lesion is not a metastasis from another primary site and to make a definitive diagnosis of PCMC&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion&#44; despite the low incidence of PCMC&#44; it must be included in the differential diagnosis of a mucosecretory adenocarcinoma in the skin&#46; Using immunohistochemistry techniques&#44; it is now possible to differentiate PCMC from metastatic mucinous adenocarcinomas from a primary in the colon&#44; as these colonic tumors are positive for CK20 and CDX2&#46; However&#44; no immunohistochemistry panel has yet been identified that will consistently differentiate PCMC from metastatic mucinous adenocarcinomas of the breast&#44; though detection of a residual myoepithelial component &#40;confirmed by positivity for p63&#44; CK5&#47;6&#44; or calponin in isolated peripheral cells&#41; suggests a primary cutaneous origin&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> Finally&#44; it is essential to establish an adequate clinical correlation and make the diagnosis of PCMC as a diagnosis of exclusion&#44; after ruling out the presence of a primary tumor in other organs&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1&#44;2</span></a></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of Interest</span><p id="par0040" 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
Primary Mucinous Carcinoma of the Skin: A Case Report
Carcinoma mucinoso primario cutáneo: presentación de un caso
C. Areán-Cunsa,
Autor para correspondencia
carolina.arean.cuns@navarra.es

Corresponding author.
, A. Córdoba-Iturriagagoitiaa, B. Aguiar-Losadaa, I. Yanguas-Bayonab
a Servicio de Anatomía Patológica, Complejo Hospitalario de Navarra, Pamplona, Navarra, Spain
b Servicio de Dermatología, Complejo Hospitalario de Navarra, Pamplona, Navarra, Spain
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A&#44; Skin sample after removal of the epidermis&#46; Normal skin adnexa may be observed in the mucinous tumor &#40;arrow&#41;&#46; Hematoxylin and eosin &#40;H&#38;E&#41;&#44; original magnification<span class="elsevierStyleHsp" style=""></span>&#215;2&#46; B&#44; Detail of the tumor with lakes of mucin that contain tumor nests&#44; with a cord-like and cribriform appearance in some areas&#46; H&#38;E&#44; original magnification<span class="elsevierStyleHsp" style=""></span>&#215;10&#46; C&#44; Positive staining with period acid Schiff &#40;PAS&#41; in the lakes of mucin&#46; PAS&#44; original magnification<span class="elsevierStyleHsp" style=""></span>&#215;10&#46; D&#44; Diffuse&#44; intense positivity for CK7 in the neoplastic cells&#46; Original magnification<span class="elsevierStyleHsp" style=""></span>&#215;10&#46; E&#44; Negative stain for CK20&#46; Original magnification<span class="elsevierStyleHsp" style=""></span>&#215;10&#46; F&#44; Two photomicrographs showing details of nuclear positivity for p63 in isolated cells within the tumor nests&#59; these cells correspond to a detectable myoepithelial component in the tumor&#44; supporting the cutaneous origin of the tumor&#46; Original magnification<span class="elsevierStyleHsp" style=""></span>&#215;20&#46;</p>"
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PCMCs are slow-growing tumors and have an indolent behavior&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> However&#44; late recurrence and metastases have been reported&#44; probably related to incomplete excision of the tumor&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Histologically&#44; it is indistinguishable from metastatic mucinous adenocarcinomas from other organs&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> particularly from the breast&#44; which can make diagnosis difficult&#46; We present a case of this rare entity and review the literature&#44; discussing the main recommendations and the immunohistochemical reactions useful to reach a correct diagnosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Description</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 68-year-old man with no past history of interest&#46; He was seen for a mobile nodular lesion that had been present for 2 years in the left axilla&#46; The lesion was not adherent to deeper planes and measured 1&#46;5<span class="elsevierStyleHsp" style=""></span>cm in diameter&#46; The clinical diagnosis was epidermal cyst&#46; Macroscopically&#44; the sample consisted of a number of reddish fragments of soft tissue&#44; including areas with a gelatinous appearance&#44; with an overall diameter of 3<span class="elsevierStyleHsp" style=""></span>cm&#46; Histologically&#44; there were fragments of skin with abundant eccrine and apocrine glands that were diffusely infiltrated by lakes of mucin &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; These lakes contained cords and solid nests of cuboidal and round epithelial cells that&#44; in some areas&#44; formed glandular structures&#44; with scattered cribriform lumina &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The cells presented cytologic atypia and occasional mitotic figures&#46; Periodic acid Schiff stain revealed an abundant quantity of mucin &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; Immunohistochemistry was positive for CK7 &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D&#41;&#44; CK19&#44; epithelial membrane antigen&#44; GATA3&#44; estrogen receptors &#40;ER&#41;&#44; progesterone receptors &#40;PR&#41;&#44; and p63 &#40;in isolated small cells&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>F&#41;&#59; the study was negative for CK20 &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E&#41;&#44; CK5&#47;6&#44; CD15&#44; and mammaglobin&#46; The pathologic diagnosis based on these findings was mucinous carcinoma&#46; Subsequently the patient underwent complete clinical evaluation&#44; including positron emission tomography &#40;PET&#41; in which no increased uptake was observed in other organs&#46; In cases such as ours&#44; it cannot be ruled out that the tumor may have arisen in a focus of ectopic breast tissue in the axilla&#59; however&#44; as no remnant of healthy breast tissue was observed in the surgical sample&#44; we made a definitive diagnosis of PCMC&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">PCMC is a rare entity that was first described in 1952 by Lenox et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> It usually shows an indolent clinical course and is typically present for several years before diagnosis&#59; it is often misdiagnosed clinically as an epidermal cyst&#44; sebaceous carcinoma&#44; cystic basal cell carcinoma&#44; squamous cell carcinoma&#44; neuroma&#44; or pilomatrixoma&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Late recurrence and metastases due to incomplete excision of the tumor<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> and lymphovascular invasion have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Apparently&#44; such recurrences have not responded well to treatment with radiotherapy or chemotherapy&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Morphologically&#44; it is not possible to differentiate PCMC from metastatic mucinous adenocarcinomas of noncutaneous organs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> Immunohistochemistry can exclude metastatic mucinous adenocarcinomas from the colon&#44; as these tumors are usually positive for CK20<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> and CDX2&#44; whereas PCMC is negative for these markers&#46; However&#44; immunohistochemistry is not particularly useful in distinguishing PCMC from mucinous adenocarcinoma of the breast&#44; as both tumors can express CK7&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> GATA3&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> mammaglobin&#44; REs&#44; and<span class="elsevierStyleHsp" style=""></span>PRs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">It has recently been suggested in the literature that detection of a myoepithelial component in the tumor &#40;confirmed by positivity for p63&#44; CK5&#47;6&#44; or calponin&#44; in isolated peripheral cells&#41; supports or suggests PCMC&#44; but unfortunately this is not a common finding&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1&#44;2</span></a> In our case&#44; we did detect the presence of this remnant of myoepithelial cells with immune stain for p63 &#40;Fig&#46;<span class="elsevierStyleHsp" style=""></span>1F&#41;&#59; however&#44; CK5&#47;6 was negative&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">In view of this situation&#44; it is strictly necessary to perform a metastatic workup &#40;computed tomography and&#47;or PET&#41;&#44; which must be negative in order to confirm that the lesion is not a metastasis from another primary site and to make a definitive diagnosis of PCMC&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion&#44; despite the low incidence of PCMC&#44; it must be included in the differential diagnosis of a mucosecretory adenocarcinoma in the skin&#46; Using immunohistochemistry techniques&#44; it is now possible to differentiate PCMC from metastatic mucinous adenocarcinomas from a primary in the colon&#44; as these colonic tumors are positive for CK20 and CDX2&#46; However&#44; no immunohistochemistry panel has yet been identified that will consistently differentiate PCMC from metastatic mucinous adenocarcinomas of the breast&#44; though detection of a residual myoepithelial component &#40;confirmed by positivity for p63&#44; CK5&#47;6&#44; or calponin in isolated peripheral cells&#41; suggests a primary cutaneous origin&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> Finally&#44; it is essential to establish an adequate clinical correlation and make the diagnosis of PCMC as a diagnosis of exclusion&#44; after ruling out the presence of a primary tumor in other organs&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1&#44;2</span></a></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of Interest</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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ISSN: 15782190
Idioma original: Inglés
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