Case study
Osteochondroma of hyoid
Osteocondroma de hioides
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identificación de la lesión (A) con exéresis de la pieza en bloque con el cuerpo del hioides (B) y visión microscópica de la lesión bien delimitada constituida en periferia por rodetes cartilaginosos con condrocitos maduros sin atipias, dispuestos en remolinos y en cuyo centro presenta osificación endocondral de disposición trabecular, quedando sus espacios ocupados por elementos representativos de las tres series hematopoyéticas, sin signos de malignidad histológica (C).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Francisco Javier García-Callejo, Pablo Tiziano Guastella-Almeida, John Deiver Cardona-Henao, Miguel Juantegui-Azpilicueta" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Francisco Javier" "apellidos" => "García-Callejo" ] 1 => array:2 [ "nombre" => "Pablo Tiziano" "apellidos" => "Guastella-Almeida" ] 2 => array:2 [ "nombre" => "John Deiver" "apellidos" => "Cardona-Henao" ] 3 => array:2 [ "nombre" => "Miguel" "apellidos" => "Juantegui-Azpilicueta" 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The mass did not cause dyspnoea or dysphagia and she reported having had it since at least the age of 10 years, with gradual, slow growth. She had not consulted earlier for sociocultural reasons.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Nasofibroscopy did not detect any anomalies in the upper aerodigestive tract. The CT imaging study reported a solid tumour with well-defined lobulated contours not infiltrative, of 51 × 22 × 21 mm (axial, anteroposterior, and craniocaudal diameters), with homogeneous density and multiple gross calcifications inside, without a fatty cleavage plane with the hyoid bone (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><p id="par0015" class="elsevierStylePara elsevierViewall">It was decided to excise it using a median horizontal cervicotomy, detecting an expansive, hard, pearly and papilliform mass under the platysma. It was overlapping the prelaryngeal muscular plane along the midline and depended on the inferior face of the hyoid body, and both were removed en bloc (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), without intra or postoperative complications. The histopathological report of the piece was large nodules of mature cartilage without atypia and with central ossification of endochondral nature arranged in trabeculae (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), all of which consistent with osteochondroma of the hyoid bone.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">One year after the operation, the cervical and pharyngolaryngeal examination was normal.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0025" class="elsevierStylePara elsevierViewall">Osteochondromas or cartilaginous exostoses are the most common bone tumours, accounting for approximately 35%–50% of all benign bone neoplasms and 8%–15% of all primary tumours.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In 85% of cases they are single lesions, predominantly in the metaphyseal zone of the long bones, although they have been described in any segment of the skeleton developing due to endochondral ossification, especially in children and young adults.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3</span></a> The systemic form encompasses 15% of cases, and is a multiple osteochondromatosis (MO) with exostosis of symmetrical distribution throughout almost the entire skeleton and an average age of presentation of 3 years, congenital and of autosomal dominant inheritance associated with mutations in the EXT 1 and EXT 2 genes involved in the biosynthesis of heparan sulphate proteoglycans, involved in epiphyseal growth.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Osteochondromas of the head and neck are extremely rare, since most maxillofacial bones develop by intramembranous ossification.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> In this area, the most common location is the mandible, where they represent .6% of all osteochondromas, very typically in the condylar segment,<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> embryonic derivative of the first branchial arch or Meckel’s cartilage. Our case would have originated in the third arch.</p><p id="par0040" class="elsevierStylePara elsevierViewall">To date, only 3 cases of hyoid osteochondroma have been reported. Torous et al. reported in 2015 the case of a 31-year-old woman with a left cervical mass of 16 mm maximum diameter, without symptoms, slow-growing over 2 years. Excision of the tumour confirmed its dependence on the lesser horn.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Raggio et al. reported in 2018 a 17-year-old male and MO with several previous exereses of other similar lesions in extremities, with a cervical midline mass of 15 mm maximum diameter and one month since onset, causing pain and dysphagia. It was removed along with a segment of the body of the hyoid.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> In 2020 Shameen et al. presented a 62-year-old male with a right cervical tumour of about 5 years’ evolution and 53 mm maximum diameter, asymptomatic, but with obliteration of the homolateral vallecula on fibroscopy. Cervicotomy revealed its origin in the greater horn of the hyoid bone, and the bone was removed in its entirety.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">As they are typically asymptomatic, the exact prevalence of osteochondroma is unknown, and some authors do not consider them to be an authentic lesion, although it is possible that 3% of the population may have them, and up to 4% of patients with osteochondroma may present some complication.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,10</span></a> When they grow subcutaneously, they are characteristically hard and painless. Symptoms are caused by of their intraosseous expansion due to fracture or mechanical irritation in other bones, compression of nerves, vessels or soft tissues, or due to malignant transformation, which occurs in 1% of cases.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,10</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The WHO defines osteochondroma as an osteocartilaginous exostosis with continuity of cortical and medullary bone covered by a layer of hyaline cartilage.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> They are thought to be lesions resulting from the separation of a fragment of epiphyseal growth plate, herniating through the normal bone surrounding the growth plate with intense chondrogenic potential, possibly increased by repeated microtrauma and even radiotherapy.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5,10</span></a> This is why CT is an excellent diagnostic tool as it easily detects the lesion’s corticomedullary continuity from the bone from which it originates—even in scenarios of complex anatomy—or coarse dot-and-comma calcifications on tumour areas of hyaline cartilage, highly specific radiological features of the tumour.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,10</span></a> MRI offers optimal complementary measurement of cartilage layer thickness, an important criterion for chondrosarcoma differentiation. Needle aspiration tests are often insensitive due to the hardness and inaccessibility of the lesions.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The only effective treatment is surgical excision including all the perichondrium surrounding the base of the tumour, otherwise it may recur. This occurs in 2% of osteochondromas,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,6</span></a> more frequently in childhood due to the potential for growth. Recurrence consists of a new osteochondroma or a low-grade chondrosarcoma.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The case presented is very unusual because of its location and size, due to the patient’s reluctance to seek advice, but its continued growth after bone maturity required its removal to prevent upper airway problems. The striking hard consistency of the tumour and the imaging tests are diagnostic, but only by complete excision of the lesion with a large segment of the original bone can it be confirmed and cured.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interests</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interests" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-09-02" "fechaAceptado" => "2021-10-23" "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" => 520 "Ancho" => 1500 "Tamanyo" => 94704 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cervical protrusion of the mass (A) and axial (B) and sagittal (C) CT projections.</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" => 1081 "Ancho" => 1500 "Tamanyo" => 281284 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cervicotomy and identification of the lesion (A) with exeresis of the piece en bloc with the body of the hyoid (B) and microscopic view of the well-delimited lesion with cartilaginous rods constituting the periphery with mature chondrocytes without atypia, arranged in whorls presenting endochondral ossification of trabecular arrangement in the centre, its spaces being occupied by representative elements of the three haematopoietic series, without signs of histological malignancy (C).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Osteocondroma: diagnóstico radiológico, complicaciones y variantes" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "P. 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