Case study
Paralaryngeal solitary fibrous tumour treated with transoral ultrasonic surgery
Tumor fibroso solitario tratado mediante cirugía transoral ultrasónica
Diego J. Picher-Gómeza,b,
, Ana B. Martínez-Segurac, Jose M. Osete-Albaladejoa,b, Jose A. Diaz-Manzanoa,b
Corresponding author
a ENT Department, Hospital Clínico Universitario Virgen de la Arrixaca, Ctra. Madrid-Cartagena, s/n, 30120 El Palmar, Murcia, Spain
b Instituto Murciano de Investigación Biosanitaria (IMIB), ENT Area, University of Murcia, Calle Campo, s/n, 30120 El Palmar, Murcia, Spain
c Radiology Department, Hospital Clínico Universitario Virgen de la Arrixaca, Ctra. Madrid-Cartagena, s/n, 30120 El Palmar, Murcia, Spain
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Picher-Gómez, Ana B. Martínez-Segura, Jose M. Osete-Albaladejo, Jose A. Diaz-Manzano" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Diego J." "apellidos" => "Picher-Gómez" "email" => array:1 [ 0 => "diegovichp@gmail.com" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Ana B." "apellidos" => "Martínez-Segura" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 2 => array:3 [ "nombre" => "Jose M." "apellidos" => "Osete-Albaladejo" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Jose A." "apellidos" => "Diaz-Manzano" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "ENT Department, Hospital Clínico Universitario Virgen de la Arrixaca, Ctra. Madrid-Cartagena, s/n, 30120 El Palmar, Murcia, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Instituto Murciano de Investigación Biosanitaria (IMIB), ENT Area, University of Murcia, Calle Campo, s/n, 30120 El Palmar, Murcia, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Radiology Department, Hospital Clínico Universitario Virgen de la Arrixaca, Ctra. Madrid-Cartagena, s/n, 30120 El Palmar, Murcia, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tumor fibroso solitario tratado mediante cirugía transoral ultrasónica" ] ] "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" => 1280 "Ancho" => 1500 "Tamanyo" => 228777 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">TOUSS images. (A) Preoperative transoral endoscopy showing the submucous left paralaryngeal mass. (B) Paralaryngeal approach performing a pyriform sinus mucosal flap with ultrasonic scissors. (C) Dissection of the mass (*). (D) Closure of the mucosal flap.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case presentation</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 44-year-old woman presented with dysphonia, dysphagia and pharyngeal foreign body sensation lasting 6 months. A rounded submucosal mass was observed by nasofiberlaryngoscopy in the left paralaryngeal space. Computed tomography (CT) revealed a solid left paralaryngeal lesion of 3<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2.2<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>1.8<span class="elsevierStyleHsp" style=""></span>cm with a marked contrast enhancement, compressing the airway (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). In addition, Magnetic Resonance Imaging (MRI) showed a tumour with an isointense signal in T1-weighted images (T1WI), hyperintense signal in T2-weighted images (T2WI) and highly enhanced by contrast (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). With these findings, a solid, non-vascular, benign neoplastic lesion (neurofibroma, schwannoma or pleomorphic adenoma) was suspected and surgical treatment was proposed. Using the transoral ultrasonic surgery technique (TOUSS), with a 0° endoscopic optic and ultrasonic scissors (Harmonic Shears™, Ethicon, J&J®), the tumour was completely removed with safe margins, making a pyriform sinus mucosal flap (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). We had no intraoperative complications. The patient started oral feeding after 48<span class="elsevierStyleHsp" style=""></span>h of the intervention with only mild dysphagia to liquids complaint within the first week and was discharged after 4 days. The anatomopathological examination revealed a non-capsulated mass composed by a proliferation of mesenchymal spindle cells without nucleolus, moderate pleomorphism, 2–3 mitosis per 10 high-power field, sparse stroma and no necrosis areas. Immunohistochemistry showed a high positivity to CD34, CD99, Bcl-2, STAT6 and vimentin. These findings were consistent with a solitary fibrous tumour (SFT). Due to the potential malignant transformation of these lesions, we maintain periodical follow-up without signs of recurrence one year later.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0010" class="elsevierStylePara elsevierViewall">SFT are infrequent mesenchymal spindle cell neoplasms originally described in the pleura. Recent case reports have described SFT at several anatomic sites, representing the head and neck area about 6% of all of them, being the larynx an uncommon location.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Early diagnosis is difficult because the clinical symptoms, physical examination and imaging tests are very unspecific. Most laryngeal cases complain of progressive dysphonia, globus pharyngeus, hoarseness, dysphagia and odynophagia.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">CT and MRI studies usually show well-defined contoured masses, occasionally with calcifications and necrotic or cystic changes.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> SFT display variable attenuation on CT images. On MRI, SFT have low-to-intermediate signal intensity on T1WI and variable signal intensity on T2WI and are intensely contrast-enhanced, reflecting their rich vascularity. Imaging tests are not particularly useful for diagnosis but can help in choosing the appropriate surgical approach.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Histologically are well-circumscribed, rounded masses, with microcystic and sometimes haemorrhagic areas.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> The histologic distinction can be difficult in SFT containing highly cellular areas.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> In these cases, immunohistochemistry is essential to make the diagnosis. They are characteristically positive for CD34 (90–95%)<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,4</span></a> and frequently for CD99, Bcl-2, STAT6 and vimentin, and negative for other soft tissue markers such as cytokeratin, s100, SMA and desmin.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,5</span></a> All these characteristics were consistent with our case. There are some cases of malignant SFT defined by the presence of necrosis, nuclear pleomorphism and high mitotic activity.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> There are also cases that show an infiltrative growing pattern, which has not been described in the larynx.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The histological findings of our case were not suspicious of malignancy.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Most of the described cases of laryngeal SFT have been treated with open transcervical approaches or transoral CO<span class="elsevierStyleInf">2</span> laser excisions.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2–6</span></a> With the development of new transoral techniques, such as TOUSS, we have other treatment possibilities. The TOUSS technique has been described as an alternative to transoral laser surgery (TOLS) for pharyngo-laryngeal lesions based on ultrasonic scalpels and resection tools, with good surgical conditions and satisfactory functional outcomes.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7,8</span></a> It avoids some of the disadvantages of TOLS such as the need to often cut the tumour into multiple pieces and the limited intraoperative field of vision.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> In addition, the use of ultrasonic scalpels allows for bloodless surgery due to their coagulation properties, which are more limited in laser surgery.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In our case, we only had a small intraoperative haemorrhage that was easily controlled, and we were able to remove the tumour without fragmentation. There are few reports about the use of TOUSS for pharyngo-laryngeal lesions. They suggest that the use of ultrasonic instruments helps to shorten the cutting time and bleeding when approaching preepiglottic and supraglottic structures.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> Besides, endoscopically assisted approaches avoid the need for an external incision, damage to surrounding vital structures and minimal blood loss, short hospital stay, and little postoperative pain. Moreover, this technique allows the surgery to be performed with a four-handed technique.</p><p id="par0035" class="elsevierStylePara elsevierViewall">SFT have a potential risk of recurrence, malignancy and metastasis.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> Only 50% of tumours classified histologically as malignant show this behaviour. There are only two cases of recurrence described in the larynx, one of them, 22 months after excision.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Therefore, long-term follow-up is considered necessary.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conclusion</span><p id="par0040" class="elsevierStylePara elsevierViewall">SFT are very uncommon in the laryngeal region, but we should consider it as a possible diagnosis when we have a laryngeal lesion with benign features on imaging tests. Because of its potential risk of malignancy, the treatment of choice is their resection with safe margins. The development of the TOUSS technique represents a new way of treating these lesions, with fewer complications than open or laser surgery and good outcomes.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Authors’ contribution</span><p id="par0045" class="elsevierStylePara elsevierViewall">All authors have contributed intellectually to the development of the work, qualify as authorship and have approved the final version.</p><p id="par0050" class="elsevierStylePara elsevierViewall">On their behalf, I declare that the work is original and has not been previously published or is under review by any other journal.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Informed consent</span><p id="par0055" class="elsevierStylePara elsevierViewall">Informed consent was obtained from the patient included in the study.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Copyright transfer</span><p id="par0060" class="elsevierStylePara elsevierViewall">Authors transfer copyright to the publisher as part of a journal publishing agreement.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Financial disclosure</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that this study has received no financial support.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case presentation" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conclusion" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Authors’ contribution" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Informed consent" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Copyright transfer" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Financial disclosure" ] 7 => array:2 [ "identificador" => "sec0040" "titulo" => "Conflict of interest" ] 8 => array:2 [ "identificador" => "xack651730" "titulo" => "Acknowledgements" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-01-02" "fechaAceptado" => "2021-03-12" "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" => 1562 "Ancho" => 1500 "Tamanyo" => 197999 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">High resolution computed tomography (A: axial; B: coronal) and magnetic resonance (C: axial; D: coronal) images show a rounded, well defined, contrast enhanced mass, located in the left paralaryngeal space.</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" => 1280 "Ancho" => 1500 "Tamanyo" => 228777 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">TOUSS images. (A) Preoperative transoral endoscopy showing the submucous left paralaryngeal mass. (B) Paralaryngeal approach performing a pyriform sinus mucosal flap with ultrasonic scissors. (C) Dissection of the mass (*). (D) Closure of the mucosal flap.</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" => "Solitary fibrous tumors of the head and neck: a multi-institutional clinicopathologic study" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "S.C. Smith" 1 => "W.E. Gooding" 2 => "M. Elkins" 3 => "R.M. Patel" 4 => "P.W. Harms" 5 => "A.S. 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Silvestrini" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s00405-015-3570-4" "Revista" => array:6 [ "tituloSerie" => "Eur Arch Otorhinolaryngol" "fecha" => "2015" "volumen" => "272" "paginaInicial" => "2551" "paginaFinal" => "2555" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25721199" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack651730" "titulo" => "Acknowledgements" "texto" => "<p id="par0075" class="elsevierStylePara elsevierViewall">We would like to express our gratitude to María Isabel Oviedo-Ramírez, MD (Pathology Department, Hospital Clínico Universitario Virgen de la Arrixaca, Murcia) and to Francisca Velázquez-Marín, MD (Radiology Department, Hospital Clínico Universitario Virgen de la Arrixaca, Murcia) for providing the anatomopathological and radiological findings and figures of the case, respectively.</p>" "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/21735735/0000007300000006/v2_202302131157/S2173573522000606/v2_202302131157/en/main.assets" "Apartado" => array:4 [ "identificador" => "5881" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case Studies" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735735/0000007300000006/v2_202302131157/S2173573522000606/v2_202302131157/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573522000606?idApp=UINPBA00004N" ]