Case and Research Letter
Osteonevus of Nanta: A Rare Skin Condition
Osteonevus de Nanta, un fenómeno cutáneo poco habitual
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Alvarado, J.J. Dávila-Rodríguez, B. Vélez, M. Montenegro-Zumárraga" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A.V." "apellidos" => "Alvarado" "email" => array:1 [ 0 => "avalvaradomd@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "J.J." "apellidos" => "Dávila-Rodríguez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "B." "apellidos" => "Vélez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "M." "apellidos" => "Montenegro-Zumárraga" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital San Francisco de Quito, Quito, Ecuador" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital San Francisco de Quito, Quito, Ecuador" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Osteonevus de Nanta, un fenómeno cutáneo poco habitual" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 599 "Ancho" => 900 "Tamanyo" => 123893 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Spicule with megakaryocytes and white and red blood cell precursors in the yellow bone marrow. Hematoxylin-eosin, original magnification ×10.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Osteonevus of Nanta is a rare condition characterized by osseus metaplasia in an intradermal nevus.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The lesion was first described by Heidesfield in 1908, and in 1911, it was reported in a publication by French dermatologist André Nanta.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Bone formation in the skin is uncommon and can be primary (when there is no evidence of a pre-existing lesion) or secondary to an inflammatory and/or neoplastic process.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Secondary bone formation has been reported in a range of lesions, including pilomatrixoma, basal cell carcinoma, acne, pyogenic granuloma, and dermatofibroma. Ossification of an intradermal nevus, however, is very rare.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,4</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">A 38-year-old man with no remarkable history presented with a hyperpigmented lesion on his left cheek that had grown and become progressively harder with time. Physical examination showed a hard, black nodule that measured 1.5<span class="elsevierStyleHsp" style=""></span>cm in diameter and was not painful on palpation (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The lesion was fully excised and a sample sent for evaluation. Histologic examination showed nests of nevus cells with appropriate maturation in the superficial dermis and, underneath, in the deep dermis, bone marrow trabeculae containing osteocytes (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Signs of intramedullary hematopoiesis and mature adipocytes were observed in the center (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The lesion was diagnosed as osteonevus of Nanta and the patient was scheduled for regular follow-up.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Primary cutaneous bone formation has been described in Albright hereditary osteodystrophy, progressive osseous heteroplasia, myositis ossificans progressiva, and osteoma cutis.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Secondary cases, in turn, have associated with scars, pyogenic granuloma, epidermal cyst, fibroxanthoma, and lipoma, and there have also been reports at the site of trauma or injection.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,4</span></a> Rarer associations include burns, dermabrasion, stasis dermatitis, and cutaneous metastases from breast, bladder, or bronchial cancer.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Benign tumors, and melanocytic nevi in particular, are the most common causes of secondary osteoma formation.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Clinically, osteonevus of Nanta resembles an intradermal nevus, is more common in women,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> and tends to be located in the upper part of the body, in particular the face, suggesting a potential pathogenic role for repeated hair follicle trauma and chronic inflammation.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,7</span></a> Lesions with necrosis, bleeding, and tissue regeneration could affect physical and chemical factors, such as calcium and phosphorous ion concentrations, pH, oxygen levels, and enzyme activity.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> These factors could induce a granulomatous reaction, triggering the transformation of mesenchymal cells into osteoblasts and resulting in bone formation.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> The most likely mechanism, however, is fibroblast differentiation (stimulated or not) in bone tissue.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The higher incidence observed in women has also raised the possibility of a pathogenic role for estrogen.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Estrogen would bind to osteoblast surface receptors, triggering the release of cytokines and inhibiting bone resorption and osteoclastic activity.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Certain cytokines, together with cell adhesion proteins and β-transforming factor in particular, would allow mesenchymal stem cells to differentiate into osteoblasts, initiating ossification.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Histologic examination of osteonevus of Nanta shows signs of ossification under nevus cells.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Characteristic findings include laminated or globular bone with a central cavity containing adipose tissue, osteoblasts, osteoclasts, blood vessels, and occasionally bone marrow elements; hair follicles are almost invariably observed in the lesions.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7,9</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">There is no established management for osteonevus of Nanta, and histologically, the lesion is benign. Culver and Burgdorf,<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> however, did report a case of malignant melanoma arising in an osteonevus of Nanta, leading some authors to advise monitoring of patients.</p><p id="par0045" class="elsevierStylePara elsevierViewall">We have presented the case of a patient with a nevus and histologic features consistent with osteonevus of Nanta. The lesion was fully excised and the patient is being monitored. No signs of recurrence have been observed.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:2 [ "identificador" => "xack405585" "titulo" => "Acknowledgments" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Alvarado AV, Dávila-Rodríguez JJ, Vélez B, Montenegro-Zumárraga M. Osteonevus de Nanta, un fenómeno cutáneo poco habitual. 2019;110:329–331.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1334 "Ancho" => 750 "Tamanyo" => 143757 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Black nodule with a diameter of 2 cm on the left cheek. Note the ill-defined borders and hair follicles.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 636 "Ancho" => 900 "Tamanyo" => 85054 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Benign nevus cells in the papillary dermis. Under these are bony spicules with osteocytes and osteoblasts. Hematoxylin-eosin, original magnification ×4.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 599 "Ancho" => 900 "Tamanyo" => 123893 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Spicule with megakaryocytes and white and red blood cell precursors in the yellow bone marrow. Hematoxylin-eosin, original magnification ×10.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Osteonevus of Nanta presenting as nodule over left eyebrow" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "G. Kamat" 1 => "A. Myageri" 2 => "R. Rao" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1155/2012/715672" "Revista" => array:5 [ "tituloSerie" => "Case Rep Dermatol Med." 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