Case study
Chondroid Tenosynovial Giant Cell Tumour of the Temporomandibular Joint: A Case Report
Tumor de células gigantes tenosinovial condroide de articulación temporomandibular. Presentación de un caso
Mercedes Álvarez-Buylla Blanco
, Manuel López Amado, Anselmo Padin Seara, Ana Reguera Arias
Corresponding author
Servicio de Otorrinolaringología, Hospital Universitario A Coruña, La Coruña, Spain
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Presentación de un caso" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "128" "paginaFinal" => "130" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Chondroid tenosynovial giant cell tumour of the temporomandibular joint: A case report" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 641 "Ancho" => 1800 "Tamanyo" => 108744 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Imagen de TC en la que se visualiza una lesión isodensa expansiva con destrucción de la pared anterior del CAE, articulación temporomandibular y fosa craneal media izquierda. 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Presentación de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 530 "Ancho" => 1500 "Tamanyo" => 244613 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histological image of the chondroid tenosynovial giant cell tumour with 10 and 20 enlargements, respectively.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Report</span><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a 42-year old patient who sought medical consultation due to a sensation of obstruction and deafness in the left ear of 2-year onset. During examination, the otoscopy of the left ear showed exostosis of the external auditory canal (EAC), with a light reduction of approximately 50%; a normal integral tympanic membrane was observed. Audiometry showed a mean threshold of airway in the left ear of 30<span class="elsevierStyleHsp" style=""></span>dB with a drop in high-pitched sounds of 50<span class="elsevierStyleHsp" style=""></span>dB. CT was requested of the middle ear and the petrous part of the temporal bone, which identified an irregularly edged mass that was destroying the EAC, the anterosuperior wall of the mastoids and invading the middle cranial fossa and left temporomandibular articulation, with irregularity of the mandibular condyle, subchrondral cysts and bone spurs that suggested degenerative-associated changes; it displaced the tympanic membrane and was in contact with the ossicular chain. A diagnosis of possible left cholesteatoma was diagnosed, without ruling out carcinoma.</p><p id="par0010" class="elsevierStylePara elsevierViewall">NMR of the temporal bone showed a heterogenous mass with chondroid matrix in the anteroinferior part of the left temporal bone, with invasion of the middle left cranial fossa which discreetly displaced the left temporal lobe and the pterygoid musculature; it was destroying the anterior part of the mastoids and invading the left EAC and left temporomandibular joint.</p><p id="par0015" class="elsevierStylePara elsevierViewall">No restriction for diffusion was determined (characteristic trait of cholesteatoma). Contrast injection showed up a small enhanced focus 13×5<span class="elsevierStyleHsp" style=""></span>mm. The combination of findings described suggested as first possibility a chondral type tumour.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Given the situation, the tumour was removed using a combined transmastoid approach through the middle fossa. The pathological anatomical analysis revealed a chondroid tenosynovial giant cell tumour of the temporomandibular joint of the left ear. Treatment was subsequently initiated with complementary radiotherapy due to the persistence of the tumour, with favourable results up to the present time.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0025" class="elsevierStylePara elsevierViewall">Giant cell tumour is a benign tumour with a proliferative pattern which develops in the synovial membranes of joints, particularly in the knee, shoulder and hand.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> Although it represents one of the main tumours in these locations, its development in the temporomandibular joint is infrequent.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">It is mainly manifested through pain, palpable mass and limitation of temporomandibular joint movmement<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2,4</span></a> with a mean period of time between the onset of symptoms and diagnosis at 15 months.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Giant cell tumour located at the base of the cranium has a tendency towards chondromatous metaplasia, imitating other chondroid tumours, such as chondroblastoma.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In this, its symptoms of presentation is a mass in the EAC accompanied by otological symptoms: deafness, otaligia, otorrhea, tinnitus, vertigo and sensation of obstruction in the ear.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Our patient began with symptoms of deafness and a sensation of obstruction in the ear.</p><p id="par0040" class="elsevierStylePara elsevierViewall">This tumour is divided into 4 subtypes depending on its growth pattern (localised or diffuse) and anatomical site (intra-or extraarticular). The localised type is the most frequent in the body and is asymptomatic in most cases.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> However, at the base of the cranium the diffuse type is the most common and usually affects adults with a mean age of 45 years<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> without any gender differences. It presents as a mass of poorly defined soft tissues, with an aggressive local growth and high rate of recurrence (up to 55%) after surgery.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Aetiology is unknown: inflammatory and neoplastic origin is mostly described.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Trauma, changes of lipids in the metabolism or haemorrhages’ are also reported.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">When there is pain or a palpable mass in the temporomandibular joint, differential diagnosis is established, from radiological patterns and later from anatomopathological patterns, between pseudotumores, such as osteochondroma, chondrosarcoma, osteiod osteoma, plasmacytoma, synovial chondromatosis, eosinophilic granuloma, pigmented villonodular synovitis, cholesteatoma, intraosseous meningioma, rabdomiosarcoma, granuloma or metastasis.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Imaging studies are able to outline the spread of the tumour and show whether it affects the surrounding solid structures and serve as a guide for surgical treatment.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The CT radiographic study shows lytic juxta-articular dislocation in the giant cell tumour with central areas of calcification and irregular peripheral bone erosion (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). In the NMR there is marked enhancement in T2 which is highly characteristic of these tumours, with variable contrast uptake<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Specifically, the haemosiderin deposit suggests a hypointense focal or diffuse signal in T1 and T2.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,6</span></a></p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0065" class="elsevierStylePara elsevierViewall">The overlapping of the histological patterns between the chondroblastoma and the giant cell tumour impedes its diagnosis, both in radiological studies through NMR and in pathological anatomy studies.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a>Thus, the chondroblastoma in enhanced sequences in T1 is hypo- or isointense; in the enhanced sequences in T2, it is hypointense if the immature chondroid matrix, haemosiderin, hypercelluarity of chondroblasts and calcifications predominate, or hypointense if there are intralesional aneurysmal cysts. In the giant cell tumour a marked hypointensity in T2 is characteristic.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,5,6</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In the anatomopathological analysis, the chondroblastoma is formed by rounded or poligonal chondroblastic cells and by multinucleated giant cells of osteoclastic type with small amounts of eossinophil intercellular controid, with necrotic areas and focal calcifications (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>.) The presence of aneurysmal bone cysts is also typical.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The giant cell tumour is composed of synovial mononuclear cells and of osteoclast type multinucleated giant cells with abundant eosinophilic and intracytoplasmatic haemosiderin.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0075" class="elsevierStylePara elsevierViewall">The treatment of choice is surgical, with an extensive local resection being considered whenever possible, due to the high rate of relapse. When the tumour has spread to the EAC, middle ear and mastoids, as it had in this patient, a combined transmastoid and middle fossa approach is required.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">The risk of recurrence essentially depends on the spread of the primary tumour and on the complete resection of the tumour.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The basic follow-up method established is by NMR 3 months after surgery. If resection has been complete, the NMR is repeated after 2 years, and review periods are progressively increased. When resection is incomplete follow-up must be more frequent. Salvage surgery and radiotherapy are used in recurrences, depending on location, tumour size and specific patient traits.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Financing</span><p id="par0085" class="elsevierStylePara elsevierViewall">No financing was received for this study.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of Interest</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:5 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case Report" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Financing" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of Interest" ] 4 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-07-28" "fechaAceptado" => "2019-11-08" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Álvarez-Buylla Blanco M, López Amado M, Padin Seara A, Reguera Arias A. Tumor de células gigantes tenosinovial condroide de articulación temporomandibular. Presentación de un caso. Acta Otorrinolaringol Esp. 2021;72:128–130.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 641 "Ancho" => 1800 "Tamanyo" => 108744 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) CT imaging where an isodense expansive tumour is observed with destruction of the anterior wall of the EAC, temporomandibular joint and middle left cranial fossa. (B) NMR imagine of enhanced sequence in T1 which shows a heterogenous mass of chondroid matrix with invasion of the EAC, temporomandibular joint and middle left cranial fossa.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 530 "Ancho" => 1500 "Tamanyo" => 244613 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histological image of the chondroid tenosynovial giant cell tumour with 10 and 20 enlargements, respectively.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tenosynovial giant cell tumor of the temporomandibular joint and lateral skull base: review of 11 cases" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "M. 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