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and a large number of tortuous vessels in the stalk &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The tumor was excised and the defect reconstructed with a Dufourmentel flap&#46; Histopathology revealed the presence of lobules of basaloid cells showing maturation toward the surface&#44; the formation of cells with abundant eosinophilic cytoplasm&#44; sheets of eosinophilic keratinous debris&#44; and pilar keratinization&#46; The lobules were surrounded by a fibrous stroma with mononuclear&#44; histiocytic inflammatory infiltrate and foreign body-type multinucleated giant cells&#46; Numerous foci of dystrophic calcification were observed&#46; These findings were consistent with a diagnosis of giant pilomatrixoma with foreign-body giant-cell reaction &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; The patient progressed favorably and there was no recurrence after 1 year of follow-up&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Pilomatrixoma is a benign adnexal tumor with differentiation towards hair matrix cells&#44; and is most common in children and those over 60&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Although the etiology of this tumor is unknown&#44; in 75&#37; of cases a mutation has been identified in <span class="elsevierStyleItalic">CTNNB1</span>&#44; the gene encoding beta-catenin&#44; a signaling protein involved in hair follicle development&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Pilomatrixomas are slow-growing nodules measuring 1 to 1&#46;5<span class="elsevierStyleHsp" style=""></span>cm across&#59; they frequently calcify&#44; giving rise to bluish discoloration of the overlying skin&#44; which may extrude calcific material&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Nodules can be multiple or solitary&#46; Solitary tumors have been described in genetic syndromes such as Turner syndrome&#44; Sotos syndrome&#44; myotonic dystrophy&#44; Rubinstein-Taybi syndrome&#44; Gardner syndrome&#44; trisomy 9&#44; basal cell nevus syndrome&#44; and xeroderma pigmentosum&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Multiple pilomatrixomas have been reported in healthy patients&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Other clinicopathologic variants have also been described&#44; including blistering&#44; anetodermic&#44; perforating&#47;ulcerated&#44; lymphangiectasic&#44; and malignant forms&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Pilomatrixomas of over 5<span class="elsevierStyleHsp" style=""></span>cm in diameter are considered giant&#46; Only 4 cases of giant pilomatrixoma have been described&#44; all in adults<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a>&#58; 2 ulcerated and 2 with areas of erosion&#46; Two cases of giant pilomatrixoma associated with hypercalcemia have also been described&#44; both of which resolved after resection&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis includes dermatofibrosarcoma protuberans&#44; calcinosis cutis&#44; osteoma cutis&#44; cutaneous lymphomas&#44; sarcomas&#44; squamous carcinoma&#44; and cutaneous metastases&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The giant&#44; ulcerated clinical form must be differentiated from the malignant form&#44; and should be suspected in cases of sudden growth of a pre-existing lesion with ulceration and bleeding&#46; Indicators of malignancy include necrosis&#44; atypical mitotic figures&#44; perineural or perivascular invasion&#44; and the degree and extent of infiltration&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Ultrasound reveals an echogenic lesion with posterior acoustic shadowing&#46; 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Case and Research Letter
Ulcerated Giant Pilomatrixoma in a Child
Pilomatrixoma gigante y ulcerado en un niño
P. Stefanoa,
Corresponding author
paoladermatologia@yahoo.com.ar

Corresponding author.
, S. Apab, B. Balancinic
a Servicio de Dermatología, Hospital Nacional de Pediatría Dr. Prof. JP Garrahan, Buenos Aires, Argentina
b Servicio de Cirugía Plástica, Hospital Nacional de Pediatría Dr. Prof. JP Garrahan, Buenos Aires, Argentina
c Servicio de Diagnóstico por Imágenes, Hospital Nacional de Pediatría Dr. Prof. JP Garrahan, Buenos Aires, Argentina
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 7 year-old boy who presented with a lesion on the left arm that had developed 2 years earlier and had suddenly increased in size in the preceding month&#46; A Doppler ultrasound performed 1 month earlier revealed an echogenic mass of 1&#46;6 &#215; 1&#46;7 &#215; 1<span class="elsevierStyleHsp" style=""></span>cm with posterior acoustic shadowing&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">Examination revealed a stony-hard&#44; exophytic&#44; pedunculated tumor of 8<span class="elsevierStyleHsp" style=""></span>cm in diameter on the patient&#39;s left arm&#46; The overlying skin was absent&#44; exposing an eroded surface and a bleeding tumor bed with a crater-like appearance &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Nuclear magnetic resonance imaging &#40;MRI&#41; and magnetic resonance angiography showed an exophytic formation consisting of a heterogeneous nodule with a hypointense halo surrounded by hyperintense tissue&#44; and a large number of tortuous vessels in the stalk &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The tumor was excised and the defect reconstructed with a Dufourmentel flap&#46; Histopathology revealed the presence of lobules of basaloid cells showing maturation toward the surface&#44; the formation of cells with abundant eosinophilic cytoplasm&#44; sheets of eosinophilic keratinous debris&#44; and pilar keratinization&#46; The lobules were surrounded by a fibrous stroma with mononuclear&#44; histiocytic inflammatory infiltrate and foreign body-type multinucleated giant cells&#46; Numerous foci of dystrophic calcification were observed&#46; These findings were consistent with a diagnosis of giant pilomatrixoma with foreign-body giant-cell reaction &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; The patient progressed favorably and there was no recurrence after 1 year of follow-up&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Pilomatrixoma is a benign adnexal tumor with differentiation towards hair matrix cells&#44; and is most common in children and those over 60&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Although the etiology of this tumor is unknown&#44; in 75&#37; of cases a mutation has been identified in <span class="elsevierStyleItalic">CTNNB1</span>&#44; the gene encoding beta-catenin&#44; a signaling protein involved in hair follicle development&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Pilomatrixomas are slow-growing nodules measuring 1 to 1&#46;5<span class="elsevierStyleHsp" style=""></span>cm across&#59; they frequently calcify&#44; giving rise to bluish discoloration of the overlying skin&#44; which may extrude calcific material&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Nodules can be multiple or solitary&#46; Solitary tumors have been described in genetic syndromes such as Turner syndrome&#44; Sotos syndrome&#44; myotonic dystrophy&#44; Rubinstein-Taybi syndrome&#44; Gardner syndrome&#44; trisomy 9&#44; basal cell nevus syndrome&#44; and xeroderma pigmentosum&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Multiple pilomatrixomas have been reported in healthy patients&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Other clinicopathologic variants have also been described&#44; including blistering&#44; anetodermic&#44; perforating&#47;ulcerated&#44; lymphangiectasic&#44; and malignant forms&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Pilomatrixomas of over 5<span class="elsevierStyleHsp" style=""></span>cm in diameter are considered giant&#46; Only 4 cases of giant pilomatrixoma have been described&#44; all in adults<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a>&#58; 2 ulcerated and 2 with areas of erosion&#46; Two cases of giant pilomatrixoma associated with hypercalcemia have also been described&#44; both of which resolved after resection&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis includes dermatofibrosarcoma protuberans&#44; calcinosis cutis&#44; osteoma cutis&#44; cutaneous lymphomas&#44; sarcomas&#44; squamous carcinoma&#44; and cutaneous metastases&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The giant&#44; ulcerated clinical form must be differentiated from the malignant form&#44; and should be suspected in cases of sudden growth of a pre-existing lesion with ulceration and bleeding&#46; Indicators of malignancy include necrosis&#44; atypical mitotic figures&#44; perineural or perivascular invasion&#44; and the degree and extent of infiltration&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Ultrasound reveals an echogenic lesion with posterior acoustic shadowing&#46; In the present case the extension of the lesion was evaluated by MRI&#44; which revealed an exophytic formation with hypervascularization&#44; as described in a previous case of giant pilomatrixoma&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The pilomatrixoma case described here is of interest due to the sudden increase in the size of the lesion&#59; while this characteristic suggested a malignant tumor&#44; malignancy was subsequently ruled out by histopathology&#46;</p></span>"
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Article information
ISSN: 15782190
Original language: English
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Idiomas
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