Paralaryngeal solitary fibrous tumour treated with transoral ultrasonic surgery

Picher-Gómez, Diego J.; Martínez-Segura, Ana B.; Osete-Albaladejo, Jose M.; Diaz-Manzano, Jose A.
doi:10.1016/j.otoeng.2021.03.003
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1</a>&#41;&#46; In addition&#44; Magnetic Resonance Imaging &#40;MRI&#41; showed a tumour with an isointense signal in T1-weighted images &#40;T1WI&#41;&#44; hyperintense signal in T2-weighted images &#40;T2WI&#41; and highly enhanced by contrast &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; With these findings&#44; a solid&#44; non-vascular&#44; benign neoplastic lesion &#40;neurofibroma&#44; schwannoma or pleomorphic adenoma&#41; was suspected and surgical treatment was proposed&#46; Using the transoral ultrasonic surgery technique &#40;TOUSS&#41;&#44; with a 0&#176; endoscopic optic and ultrasonic scissors &#40;Harmonic Shears&#8482;&#44; Ethicon&#44; J&#38;J&#174;&#41;&#44; the tumour was completely removed with safe margins&#44; making a pyriform sinus mucosal flap &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; We had no intraoperative complications&#46; 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&#46; The anatomopathological examination revealed a non-capsulated mass composed by a proliferation of mesenchymal spindle cells without nucleolus&#44; moderate pleomorphism&#44; 2&#8211;3 mitosis per 10 high-power field&#44; sparse stroma and no necrosis areas&#46; Immunohistochemistry showed a high positivity to CD34&#44; CD99&#44; Bcl-2&#44; STAT6 and vimentin&#46; These findings were consistent with a solitary fibrous tumour &#40;SFT&#41;&#46; Due to the potential malignant transformation of these lesions&#44; we maintain periodical follow-up without signs of recurrence one year later&#46;</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&#46; Recent case reports have described SFT at several anatomic sites&#44; representing the head and neck area about 6&#37; of all of them&#44; being the larynx an uncommon location&#46;<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&#44; physical examination and imaging tests are very unspecific&#46; Most laryngeal cases complain of progressive dysphonia&#44; globus pharyngeus&#44; hoarseness&#44; dysphagia and odynophagia&#46;<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&#44; occasionally with calcifications and necrotic or cystic changes&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> SFT display variable attenuation on CT images&#46; On MRI&#44; SFT have low-to-intermediate signal intensity on T1WI and variable signal intensity on T2WI and are intensely contrast-enhanced&#44; reflecting their rich vascularity&#46; Imaging tests are not particularly useful for diagnosis but can help in choosing the appropriate surgical approach&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Histologically are well-circumscribed&#44; rounded masses&#44; with microcystic and sometimes haemorrhagic areas&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> The histologic distinction can be difficult in SFT containing highly cellular areas&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> In these cases&#44; immunohistochemistry is essential to make the diagnosis&#46; They are characteristically positive for CD34 &#40;90&#8211;95&#37;&#41;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;4</span></a> and frequently for CD99&#44; Bcl-2&#44; STAT6 and vimentin&#44; and negative for other soft tissue markers such as cytokeratin&#44; s100&#44; SMA and desmin&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;5</span></a> All these characteristics were consistent with our case&#46; There are some cases of malignant SFT defined by the presence of necrosis&#44; nuclear pleomorphism and high mitotic activity&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> There are also cases that show an infiltrative growing pattern&#44; which has not been described in the larynx&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The histological findings of our case were not suspicious of malignancy&#46;</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&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#8211;6</span></a> With the development of new transoral techniques&#44; such as TOUSS&#44; we have other treatment possibilities&#46; The TOUSS technique has been described as an alternative to transoral laser surgery &#40;TOLS&#41; for pharyngo-laryngeal lesions based on ultrasonic scalpels and resection tools&#44; with good surgical conditions and satisfactory functional outcomes&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;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&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> In addition&#44; the use of ultrasonic scalpels allows for bloodless surgery due to their coagulation properties&#44; which are more limited in laser surgery&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In our case&#44; we only had a small intraoperative haemorrhage that was easily controlled&#44; and we were able to remove the tumour without fragmentation&#46; There are few reports about the use of TOUSS for pharyngo-laryngeal lesions&#46; They suggest that the use of ultrasonic instruments helps to shorten the cutting time and bleeding when approaching preepiglottic and supraglottic structures&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> Besides&#44; endoscopically assisted approaches avoid the need for an external incision&#44; damage to surrounding vital structures and minimal blood loss&#44; short hospital stay&#44; and little postoperative pain&#46; Moreover&#44; this technique allows the surgery to be performed with a four-handed technique&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">SFT have a potential risk of recurrence&#44; malignancy and metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> Only 50&#37; of tumours classified histologically as malignant show this behaviour&#46; There are only two cases of recurrence described in the larynx&#44; one of them&#44; 22 months after excision&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Therefore&#44; long-term follow-up is considered necessary&#46;</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&#44; but we should consider it as a possible diagnosis when we have a laryngeal lesion with benign features on imaging tests&#46; Because of its potential risk of malignancy&#44; the treatment of choice is their resection with safe margins&#46; The development of the TOUSS technique represents a new way of treating these lesions&#44; with fewer complications than open or laser surgery and good outcomes&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Authors&#8217; contribution</span><p id="par0045" class="elsevierStylePara elsevierViewall">All authors have contributed intellectually to the development of the work&#44; qualify as authorship and have approved the final version&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">On their behalf&#44; I declare that the work is original and has not been previously published or is under review by any other journal&#46;</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&#46;</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&#46;</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&#46;</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&#46;</p></span></span>"
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        "titulo" => "Acknowledgements"
        "texto" => "<p id="par0075" class="elsevierStylePara elsevierViewall">We would like to express our gratitude to Mar&#237;a Isabel Oviedo-Ram&#237;rez&#44; MD &#40;Pathology Department&#44; Hospital Cl&#237;nico Universitario Virgen de la Arrixaca&#44; Murcia&#41; and to Francisca Vel&#225;zquez-Mar&#237;n&#44; MD &#40;Radiology Department&#44; Hospital Cl&#237;nico Universitario Virgen de la Arrixaca&#44; Murcia&#41; for providing the anatomopathological and radiological findings and figures of the case&#44; respectively&#46;</p>"
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