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
Corresponding author
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" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 11 | 18 |
| 2024 October | 61 | 50 | 111 |
| 2024 September | 89 | 29 | 118 |
| 2024 August | 110 | 59 | 169 |
| 2024 July | 81 | 37 | 118 |
| 2024 June | 92 | 43 | 135 |
| 2024 May | 71 | 34 | 105 |
| 2024 April | 72 | 21 | 93 |
| 2024 March | 91 | 30 | 121 |
| 2024 February | 69 | 35 | 104 |
| 2024 January | 68 | 38 | 106 |
| 2023 December | 66 | 15 | 81 |
| 2023 November | 76 | 22 | 98 |
| 2023 October | 54 | 25 | 79 |
| 2023 September | 58 | 23 | 81 |
| 2023 August | 55 | 11 | 66 |
| 2023 July | 60 | 43 | 103 |
| 2023 June | 63 | 20 | 83 |
| 2023 May | 63 | 27 | 90 |
| 2023 April | 49 | 28 | 77 |
| 2023 March | 48 | 20 | 68 |
| 2023 February | 84 | 27 | 111 |
| 2023 January | 45 | 33 | 78 |
| 2022 December | 57 | 52 | 109 |
| 2022 November | 37 | 28 | 65 |
| 2022 October | 33 | 22 | 55 |
| 2022 September | 22 | 42 | 64 |
| 2022 August | 26 | 40 | 66 |
| 2022 July | 31 | 38 | 69 |
| 2022 June | 30 | 25 | 55 |
| 2022 May | 50 | 46 | 96 |
| 2022 April | 64 | 44 | 108 |
| 2022 March | 87 | 61 | 148 |
| 2022 February | 51 | 43 | 94 |
| 2022 January | 63 | 52 | 115 |
| 2021 December | 67 | 41 | 108 |
| 2021 November | 55 | 50 | 105 |
| 2021 October | 67 | 43 | 110 |
| 2021 September | 32 | 37 | 69 |
| 2021 August | 27 | 23 | 50 |
| 2021 July | 66 | 28 | 94 |
| 2021 June | 51 | 24 | 75 |
| 2021 May | 37 | 23 | 60 |
| 2021 April | 67 | 84 | 151 |
| 2021 March | 68 | 29 | 97 |
| 2021 February | 60 | 27 | 87 |
| 2021 January | 33 | 21 | 54 |
| 2020 December | 37 | 19 | 56 |
| 2020 November | 36 | 26 | 62 |
| 2020 October | 23 | 14 | 37 |
| 2020 September | 43 | 16 | 59 |
| 2020 August | 26 | 15 | 41 |
| 2020 July | 28 | 16 | 44 |
| 2020 June | 24 | 29 | 53 |
| 2020 May | 19 | 21 | 40 |
| 2020 April | 22 | 20 | 42 |
| 2020 March | 24 | 21 | 45 |
| 2020 February | 3 | 3 | 6 |
| 2020 January | 4 | 2 | 6 |
| 2019 December | 4 | 3 | 7 |
| 2019 November | 4 | 1 | 5 |
| 2019 October | 0 | 1 | 1 |
| 2019 September | 0 | 1 | 1 |
| 2019 August | 4 | 6 | 10 |
| 2019 July | 4 | 4 | 8 |
| 2019 June | 4 | 28 | 32 |
| 2019 May | 5 | 58 | 63 |
| 2019 April | 3 | 33 | 36 |
| 2019 March | 2 | 8 | 10 |
| 2019 February | 0 | 6 | 6 |
| 2019 January | 3 | 0 | 3 |
| 2018 December | 1 | 0 | 1 |
| 2018 November | 1 | 0 | 1 |
| 2018 October | 1 | 0 | 1 |
| 2018 September | 3 | 1 | 4 |
| 2018 August | 0 | 1 | 1 |
| 2018 June | 0 | 2 | 2 |
| 2018 May | 0 | 6 | 6 |
| 2018 February | 24 | 7 | 31 |
| 2018 January | 33 | 5 | 38 |
| 2017 December | 25 | 9 | 34 |
| 2017 November | 28 | 10 | 38 |
| 2017 October | 24 | 9 | 33 |
| 2017 September | 17 | 7 | 24 |
| 2017 August | 25 | 8 | 33 |
| 2017 July | 24 | 10 | 34 |
| 2017 June | 28 | 36 | 64 |
| 2017 May | 29 | 18 | 47 |
| 2017 April | 16 | 26 | 42 |
| 2017 March | 15 | 27 | 42 |
| 2017 February | 11 | 9 | 20 |
| 2017 January | 17 | 6 | 23 |
| 2016 December | 13 | 15 | 28 |
| 2016 November | 23 | 20 | 43 |
| 2016 October | 18 | 20 | 38 |
| 2016 September | 0 | 5 | 5 |
| 2016 August | 0 | 1 | 1 |
| 2016 July | 0 | 2 | 2 |
| 2016 June | 1 | 3 | 4 |
| 2016 May | 2 | 1 | 3 |
| 2016 February | 0 | 1 | 1 |
| 2016 January | 0 | 7 | 7 |
| 2015 December | 0 | 1 | 1 |
| 2015 November | 0 | 1 | 1 |
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