Case and Research Letters
Histologic diagnosis of ossifying fibromyxoid tumor: 2 cases in the past 20 Years
Diagnóstico histológico del tumor fibromixoide osificante: 2 casos en los últimos 20 años
J. Hernández-Godoy
, C. Casado-Sánchez, L. Landín, A.A. Rosell
Autor para correspondencia
Servicio de Cirugía Plástica, Hospital Universitario La Paz, Madrid, Spain
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Tinción con H-E con aumento ×<span class="elsevierStyleHsp" style=""></span>2 donde se aprecia TFMO con la característica cápsula incompleta de hueso lamelar (A). Tinción H-E con aumento ×<span class="elsevierStyleHsp" style=""></span>40 mostrando células epitelioides monomorfas con pequeños nucléolos y bordes nítidos (B). Inmunohistoquímica con aumento ×<span class="elsevierStyleHsp" style=""></span>20, positividad difusa para S-100 (C).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J. Hernández-Godoy, C. Casado-Sánchez, L. Landín, A.A. Rosell" "autores" => array:4 [ 0 => array:2 [ "nombre" => "J." "apellidos" => "Hernández-Godoy" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Casado-Sánchez" ] 2 => array:2 [ "nombre" => "L." "apellidos" => "Landín" ] 3 => array:2 [ "nombre" => "A.A." 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Hernández-Godoy, C. Casado-Sánchez, L. Landín, A.A. Rosell" "autores" => array:4 [ 0 => array:4 [ "nombre" => "J." "apellidos" => "Hernández-Godoy" "email" => array:1 [ 0 => "jhdezgodoy@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Casado-Sánchez" ] 2 => array:2 [ "nombre" => "L." "apellidos" => "Landín" ] 3 => array:2 [ "nombre" => "A.A." "apellidos" => "Rosell" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Cirugía Plástica, Hospital Universitario La Paz, Madrid, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Diagnóstico histológico del tumor fibromixoide osificante: 2 casos en los últimos 20 años" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 399 "Ancho" => 1600 "Tamanyo" => 308404 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histology images. A, The ossifying fibromyxoid tumor with its characteristic, incomplete capsule of lamellar bone; hematoxylin and eosin (H&E), original magnification ×<span class="elsevierStyleHsp" style=""></span>2. B, Monomorphic epithelioid cells with small nucleoli and clearly distinct borders; H&E, original magnification ×<span class="elsevierStyleHsp" style=""></span>40.<span class="elsevierStyleHsp" style=""></span>C, Immunohistochemistry showing diffuse positivity for protein S-100; original magnification<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>20.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The ossifying fibromyxoid tumor (OFMT) was originally described as a small benign tumor of the subcutaneous tissue. It is formed of small cells arranged in cords and nests in a fibromyxoid stroma, covered by a bony capsule.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> However, recent publications have reported histologic findings of malignancy associated with metastatic disease.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> There is controversy regarding the histologic origin of the tumor. Despite initially being considered to be distinct from schwannian or cartilaginous tumors,<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> based on ultrastructural and immunohistochemical characteristics (positivity for protein S-100), more recent proteomic and genetic analyses support a neuronal or myoepithelial origin.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">3,4</span></a> In the last decade, the idea that malignant OFMTs do not exist has been proposed, as they do not satisfy the traditional histological description, and could correspond to other malignant soft tissue tumors.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">In the last 20 years, 2 cases of OFMT have been identified in our hospital, one on the scalp of a 55-year-old man the other on the hand of a 46-year-old man (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The tumors were painless. Histology of the excisional biopsies were consistent with the classic description of OFMT: well-defined capsule; areas of fibrosis formed of laminae of uniform, ovoid cells with round nuclei in a hyaline stroma; other areas of myxoid appearance with lower cellularity; and moderate diffuse positivity for protein S-100 (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The surgical margins were not extended in either case. No signs of local recurrence or metastases have been detected after follow-up of 18 years and 21 months, respectively.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">This tumor typically affects men, and the mean age at presentation is 50 years. It usually arises in the proximal regions of the limbs, most commonly the lower limbs.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1,3–6</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Immunohistochemistry closely reflects the controversy regarding the histogenesis of the tumor. The origins that have been postulated with greatest emphasis are Schwann cells (because of the existence of well-developed, duplicated basal lamina and positivity for protein S-100<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a>) and cartilage (due to the presence of irregular cell borders with small projections and intracellular microfillaments with positivity for S-100<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a>). Other authors have suggested a myoepithelial lineage based on ultrastructural findings,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">4</span></a> or neuronal lineage due to positivity for CD56, a neural adhesion molecule, and for EAAT4, a neuronal glutamic acid transporter.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Cytogenetic analysis of this type of tumor focuses attention on 2 genes, <span class="elsevierStyleItalic">INI-1</span>,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> a tumor suppressor gene, and <span class="elsevierStyleItalic">PHF1</span>,<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> which codes for a protein that, among other functions, regulates the activity of the polycomb-repressive complex 2, which silences genes responsible for development. Dysregulation of this complex, secondary to changes in these or other adjacent genes, has been suggested as a mechanism of development of OFMTs.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In 1995, Kilpatrick<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a> was the first to describe the existence of OFMTs with a malignant behavior, associated with atypical morphological findings. Folpe and Weiss<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> identified criteria predictive of an aggressive or malignant behavior. Subsequently, Miettinen<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> rejected the existence of malignant OFMTs, stating that the majority of those tumors did not satisfy the classic description of OFMTs and could be better classified as other types of sarcoma. Graham,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> continuing the work initiated by Folpe, validated their classification on finding that the malignant subtype was associated with a more aggressive behavior during follow-up.</p><p id="par0035" class="elsevierStylePara elsevierViewall">A biologically aggressive behavior was also investigated in the largest series of OFMTs. Folpe<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> detected local recurrence in 18% and metastases in 16%. Miettinen,<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> including only typical OFMTs, reported local recurrence in 22% and no cases of metastasis. Finally, Graham<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> reported local recurrence rates of 4.3% and metastasis in 6.5%, considering only malignant OFMTs. Grouping the data from those series, the metastatic risk of the typical variant is less than 5%; this indicates that, even if benign histological characteristics are observed, OFMT can give rise to metastases.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The scarcity of OFMTs recruited in the past 20 years in our hospital, given that the catchment population is close to 1 million, would appear to be related to a diagnostic vision closer to that of Miettinen than to Folpe and Graham. Thus, tumors that do not fit the typical description of OFMTs, and that showed characteristics of malignancy, were classified as low-grade sarcomas rather than as malignant OFMTs. This could explain the absence of recurrence in either of our 2 patients, despite having performed excisions with narrow margins.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion, OFMT is a rare disease, whose epidemiology may be biased by the diagnostic considerations of each pathology departament regarding atypical and malignant variants. Despite showing benign characteristics, these tumors can present aggressive behavior, and they should not therefore be classified as benign, but rather considered as tumors of intermediate malignancy.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Hernández-Godoy J, Casado-Sánchez C, Landín L, Rosell AA. Diagnóstico histológico del tumor fibromixoide osificante: 2 casos en los últimos 20 años. Actas Dermosifiliogr. 2015;106:772–774.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 450 "Ancho" => 1800 "Tamanyo" => 187413 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Preoperative Image of the tumor in the first commissure. B, Intraoperative view of the resection. C, The excised ossifying fibromyxoid tumor.</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" => 399 "Ancho" => 1600 "Tamanyo" => 308404 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histology images. A, The ossifying fibromyxoid tumor with its characteristic, incomplete capsule of lamellar bone; hematoxylin and eosin (H&E), original magnification ×<span class="elsevierStyleHsp" style=""></span>2. B, Monomorphic epithelioid cells with small nucleoli and clearly distinct borders; H&E, original magnification ×<span class="elsevierStyleHsp" style=""></span>40.<span class="elsevierStyleHsp" style=""></span>C, Immunohistochemistry showing diffuse positivity for protein S-100; original magnification<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>20.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Ossifying fibromyxoid tumor of soft parts. 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José Juan Pozo-Kreilinger, Adrien Yvon, and Estefania Alonso-Rodríguez for their close collaboration, which made this study possible.</p>" "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000010600000009/v1_201511040157/S1578219015002474/v1_201511040157/en/main.assets" "Apartado" => array:4 [ "identificador" => "6157" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case and Research Letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000010600000009/v1_201511040157/S1578219015002474/v1_201511040157/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219015002474?idApp=UINPBA000044" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 8 | 11 | 19 |
| 2024 Octubre | 61 | 50 | 111 |
| 2024 Septiembre | 89 | 29 | 118 |
| 2024 Agosto | 110 | 59 | 169 |
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| 2024 Junio | 92 | 43 | 135 |
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| 2024 Marzo | 91 | 30 | 121 |
| 2024 Febrero | 69 | 35 | 104 |
| 2024 Enero | 68 | 38 | 106 |
| 2023 Diciembre | 66 | 15 | 81 |
| 2023 Noviembre | 76 | 22 | 98 |
| 2023 Octubre | 54 | 25 | 79 |
| 2023 Septiembre | 58 | 23 | 81 |
| 2023 Agosto | 55 | 11 | 66 |
| 2023 Julio | 60 | 43 | 103 |
| 2023 Junio | 63 | 20 | 83 |
| 2023 Mayo | 63 | 27 | 90 |
| 2023 Abril | 49 | 28 | 77 |
| 2023 Marzo | 48 | 20 | 68 |
| 2023 Febrero | 84 | 27 | 111 |
| 2023 Enero | 45 | 33 | 78 |
| 2022 Diciembre | 57 | 52 | 109 |
| 2022 Noviembre | 37 | 28 | 65 |
| 2022 Octubre | 33 | 22 | 55 |
| 2022 Septiembre | 22 | 42 | 64 |
| 2022 Agosto | 26 | 40 | 66 |
| 2022 Julio | 31 | 38 | 69 |
| 2022 Junio | 30 | 25 | 55 |
| 2022 Mayo | 50 | 46 | 96 |
| 2022 Abril | 64 | 44 | 108 |
| 2022 Marzo | 87 | 61 | 148 |
| 2022 Febrero | 51 | 43 | 94 |
| 2022 Enero | 63 | 52 | 115 |
| 2021 Diciembre | 67 | 41 | 108 |
| 2021 Noviembre | 55 | 50 | 105 |
| 2021 Octubre | 67 | 43 | 110 |
| 2021 Septiembre | 32 | 37 | 69 |
| 2021 Agosto | 27 | 23 | 50 |
| 2021 Julio | 66 | 28 | 94 |
| 2021 Junio | 51 | 24 | 75 |
| 2021 Mayo | 37 | 23 | 60 |
| 2021 Abril | 67 | 84 | 151 |
| 2021 Marzo | 68 | 29 | 97 |
| 2021 Febrero | 60 | 27 | 87 |
| 2021 Enero | 33 | 21 | 54 |
| 2020 Diciembre | 37 | 19 | 56 |
| 2020 Noviembre | 36 | 26 | 62 |
| 2020 Octubre | 23 | 14 | 37 |
| 2020 Septiembre | 43 | 16 | 59 |
| 2020 Agosto | 26 | 15 | 41 |
| 2020 Julio | 28 | 16 | 44 |
| 2020 Junio | 24 | 29 | 53 |
| 2020 Mayo | 19 | 21 | 40 |
| 2020 Abril | 22 | 20 | 42 |
| 2020 Marzo | 24 | 21 | 45 |
| 2020 Febrero | 3 | 3 | 6 |
| 2020 Enero | 4 | 2 | 6 |
| 2019 Diciembre | 4 | 3 | 7 |
| 2019 Noviembre | 4 | 1 | 5 |
| 2019 Octubre | 0 | 1 | 1 |
| 2019 Septiembre | 0 | 1 | 1 |
| 2019 Agosto | 4 | 6 | 10 |
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| 2019 Febrero | 0 | 6 | 6 |
| 2019 Enero | 3 | 0 | 3 |
| 2018 Diciembre | 1 | 0 | 1 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 3 | 1 | 4 |
| 2018 Agosto | 0 | 1 | 1 |
| 2018 Junio | 0 | 2 | 2 |
| 2018 Mayo | 0 | 6 | 6 |
| 2018 Febrero | 24 | 7 | 31 |
| 2018 Enero | 33 | 5 | 38 |
| 2017 Diciembre | 25 | 9 | 34 |
| 2017 Noviembre | 28 | 10 | 38 |
| 2017 Octubre | 24 | 9 | 33 |
| 2017 Septiembre | 17 | 7 | 24 |
| 2017 Agosto | 25 | 8 | 33 |
| 2017 Julio | 24 | 10 | 34 |
| 2017 Junio | 28 | 36 | 64 |
| 2017 Mayo | 29 | 18 | 47 |
| 2017 Abril | 16 | 26 | 42 |
| 2017 Marzo | 15 | 27 | 42 |
| 2017 Febrero | 11 | 9 | 20 |
| 2017 Enero | 17 | 6 | 23 |
| 2016 Diciembre | 13 | 15 | 28 |
| 2016 Noviembre | 23 | 20 | 43 |
| 2016 Octubre | 18 | 20 | 38 |
| 2016 Septiembre | 0 | 5 | 5 |
| 2016 Agosto | 0 | 1 | 1 |
| 2016 Julio | 0 | 2 | 2 |
| 2016 Junio | 1 | 3 | 4 |
| 2016 Mayo | 2 | 1 | 3 |
| 2016 Febrero | 0 | 1 | 1 |
| 2016 Enero | 0 | 7 | 7 |
| 2015 Diciembre | 0 | 1 | 1 |
| 2015 Noviembre | 0 | 1 | 1 |
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