An 84-year-old man was referred to our institution for dysphonia present for 2 months. Fiberoptic laryngoscopy demonstrated a polypoid lesion of the right true vocal fold with normal motility. Excisional biopsy of a submucosal thickening of the right vocal fold with vocal ligament calcification was performed. Histology revealed squamous cell epithelium without malignancy. Postoperative endoscopic follow-up was regular. Six months after surgery, dysphonia and dyspnea occurred. Flexible laryngoscopy demonstrated right hemilaryngeal paralysis, edema, and partial airway obstruction. Necrotic tissue and purulent secretions were visible at the anterior commissure, right vocal fold, and ipsilateral vestibule. A second microlaryngoscopic surgery included multiple biopsies and debridement of the necrotic tissue. Histological report was negative for malignancy, and purulent chondritis was diagnosed. Intravenous antibiotic therapy was administered with improvement of infection, inflammation, dysphonia, and dyspnea. The patient was discharged

De Rossi, S

Fabbris, C

Gazzini, L

Marchioni, D

Molteni, G

Soloperto, D

Valotto, G

English

Molteni G.

Soloperto D.

Fabbris C.

Gazzini L.

De Rossi S.

Valotto G.

Marchioni D.

Archivio istituzionale della ricerca - Università di Modena e Reggio Emilia

Laryngoscopic ClinicLeiomyosarcoma of the Larynx:A Complex DiagnosisGabriele Molteni, MD1, Davide Soloperto, MD, PhD1,Cristoforo Fabbris, MD1, Luca Gazzini, MD1, Stefano De Rossi, MD1,Giovanni Valotto, MD2, and Daniele Marchioni, MD1Case ReportAn 84-year-old man was referred to our institution for dyspho-nia present for 2 months. Fiberoptic laryngoscopy demon-strated a polypoid lesion of the right true vocal fold withnormal motility.Excisional biopsy of a submucosal thickening of the rightvocal fold with vocal ligament calcification was performed.Histology revealed squamous cell epithelium without malig-nancy. Postoperative endoscopic follow-up was regular.Six months after surgery, dysphonia and dyspnea occurred.Flexible laryngoscopy demonstrated right hemilaryngealparalysis, edema, and partial airway obstruction. Necrotic tis-sue and purulent secretions were visible at the anterior com-missure, right vocal fold, and ipsilateral vestibule. A secondmicrolaryngoscopic surgery included multiple biopsies anddebridement of the necrotic tissue. Histological report wasnegative for malignancy, and purulent chondritis was diag-nosed. Intravenous antibiotic therapy was administered withimprovement of infection, inflammation, dysphonia, and dys-pnea. The patient was discharged.Two months later, a new sudden and severe dyspneaoccurred. He was admitted to the emergency department, andoropharyngeal intubation was necessary because of respiratoryfailure.Computed tomography (CT) scan showed thickening of theright laryngeal wall with wide fluid collection (48-43 mm dia-meter) that almost completely obstructed the airway, with ero-sion of the right thyroid and cricoid cartilage (Figure 1).Debridement of the necrotic tissue and drainage of the puru-lent collection was performed with direct laryngoscopy. Cul-ture was positive for Staphylococcus epidermidis and Candidaalbicans. Intravenous antibiotic, antimicotic, and corticosteroidtherapy was administered, and other 3 surgical debridementsand drainage were performed, but without clinical or radiolo-gical improvements. Moreover, patient developed signs of sep-sis. Since no improvement had been achieved, totallaryngectomy was planned even without a positive histologicbiopsy. Intraoperative findings showed transglottic extensionof the ulcerated material with full-thickness infiltration of thelaryngeal wall and external spread through the thyroidcartilage, with invasion of prelaryngeal tissues (Figure 2). Thepatient’s clinical condition improved, and he started oral feed-ing without pharyngocutaneous fistula. Morphological andimmunohistochemical features were consistent with leiomyo-sarcoma (Fédération Nationale des Centres de Lutte Contre LeCancer [FNCLCC] grade 3; Figure 3).Multidisciplinary consultation suggested adjuvant radia-tion therapy, but preirradiation staging CT scan showed rapidlocal recurrence and lung metastasis. The poor clinical con-dition of the patient contraindicated first-line chemotherapy.Figure 1. Computed tomography image showing an almost completeerosion of the laryngeal framework cartilages due to LLMS. Residualairway is minimal therefore an emergency oropharyngeal intubation isneeded.1 Otolaryngology Department, University Hospital of Verona, Verona, Italy2 Pathology Department, University Hospital of Verona, Verona, ItalyReceived: August 19, 2018; revised: August 27, 2018; accepted: August 28,2018Corresponding Author:Cristoforo Fabbris, MD, Otolaryngology Department, University Hospital ofVerona, Piazzale Aristide Stefani 1, Verona 37126, Italy.Email: cristoforo.fabbris@gmail.comEar, Nose & Throat Journal2020, Vol. 99(5) 300–302ª The Author(s) 2019Article reuse guidelines:sagepub.com/journals-permissionsDOI: 10.1177/0145561319840204journals.sagepub.com/home/earAt 3-month follow-up, the patient was alive and receiving bestsupportive care.DiscussionThe first case of laryngeal leiomyosarcoma (LLMS) wasreported by Jackson et al in 1939.1 After that, fewer than 50cases of LLMS have been reported in international literature,2including an 87-year-old man with laryngopyocele and LLMSwho was treated with total laryngectomy.3Simultaneous presence of laryngeal carcinoma, laryngopyo-cele, and laryngomucocele was described by Cassano et al,4and Mitro et al,5 but no leiomyosarcoma was found in thesepatients or in cases reviewed in similar articles.Purulent chondritis of laryngeal cartilages (PCLC) is a veryuncommon abscess that grows between inner and outer peri-chondria of the laryngeal framework. To date, only 5 caseshave been described in the literature,6-8 and none of them wasassociated with neoplasm.In our case, radiological suspicion of laryngeal abscess andno clear radiological signs of malignant tumor were detectable.Laryngeal biopsies were negative for malignancy, and cultureswere consistent with PCLC, but combined antibiotic therapy,surgical drainage, and debridement did not improve the clinicalcondition, and the patient developed sepsis. Fortunately, aftertotal laryngectomy, his clinical condition improved quicklywith complete sepsis resolution.Etiology of LLMS is still unclear. A possible factor is anabnormal postoperative healing process.9 In our case, thepatient developed worsening dysphonia and dyspnea, 6 monthsafter his first vocal fold biopsy with negative findings.Treatment guidelines for LLMS are lacking due to its rarity.First-line treatment should be tumor resection with wide mar-gins, reserving radiotherapy as adjuvant treatment or in case ofrecurrence or persistent tumor.10,11 Chemotherapy usually issuggested for distant metastases.12Even though if LLMS is a very rare laryngeal malignancy, itshould be considered when dealing with laryngeal abscess,especially with cartilage involvement.Authors’ NoteThe human participant involved in this research signed an informedconsent before any further treatment. All procedures performed werein accordance with the ethical standards of the institutional and/ornational research committee and with the 1964 Helsinki declarationand its later amendments or comparable ethical standards.Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect tothe research, authorship, and/or publication of this article.FundingThe author(s) received no financial support for the research, author-ship, and/or publication of this article.References1. Jackson C, Jackson CL. Sarcoma of the larynx. In: Jackson C. (ed).Cancer of the larynx. Philadelphia: W. B. Saunders; 1939:164-168.2. Yadav J, Bakshi J, Chouhan M, Modi R. Head and Neck Leio-myosarcoma. Indian J Otolaryngol Head Neck Surg. 2013;65(Suppl 1):S1-S5.3. Helmberger RC, Croker BP, Mancuso AA. Leiomyosarcoma ofthe Larynx Presenting as a Laryngopyocele. AJNR Am J Neuror-adiol. 1996;17(6):1112-1114.4. Cassano L, Lombardo P, Marchese-Ragona R, Pastore A. Laryn-gopyocele: three new clinical cases and review of the literature.Eur Arch Otorhinolaryngol. 2000;257(9):507-511.Figure 2. Intraoperative picture showing the almost complete ero-sion of laryngeal cartilages with necrotic material substituting normallaryngeal tissue.Figure 3. Histopathology of the lesion: SMA (smooth muscle specificactin) positivity demonstrates the smooth muscle nature of the neo-plastic population. Written histological report shows a malignantneoplasm with spindle cells, high mitotic index (10-14 mitosis 10HPF), and large areas of tissue necrosis (>50%).Molteni et al 3015. Mitroi M, Căpitănescu A, Popescu FC, et al. Laryngocele associ-ated with laryngeal carcinoma. Rom J Morphol Embryol. 2011;52(1):183-185.6. Eliashar R, Gross M, Goldfarb A, Sichel JY. Purulent chondritisof the laryngeal framework cartilages. Ann Otol Rhinal Laryngol.2005;114(3):219-222.7. Lee JK, Cho HH, Kim HD, Lim SC. Purulent chondritis of thethyroid cartilage. Am J Otolaryngol. 2007;28(1):64-66.8. Leiva-Salinas C, Mukherjee S. Purulent chondritis of the cricoid.Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133(2):137-139.9. Khadivi E, Taziky MH, Jafarian AH, Nasseri Sadr M. Laryngealleiomyosarcoma, a case report and review of articles. Iran J Otor-hinolaryngol. 2013;25(73):253-258.10. Abbas A, Ikram M, Yagoob N. Leiomyosarcoma of the larynx:A case report. Ear Nose Throat J. 2005;84(7):435-436.11. Kleinsasser O., Glanz H. Myogenic tumors of the larynx. ArchOtorhinolaryngol. 1979;225(2):107-119.12. Ismi O, Arpaci RB, Puturgeli T, Ozcan C, Gorur K. Leiomyosar-coma of Larynx as a Metachronous Tumor of Squamous CellCarcinoma: An Unusual Case. J Voice. 2016;30(2):242-245.302 Ear, Nose & Throat Journal 99(5)

Leiomyosarcoma of the Larynx: A Complex Diagnosis

Molteni G

Soloperto D

Fabbris C

Gazzini L

De Rossi S

Valotto G

Marchioni D

Catalogo dei prodotti della ricerca

Laryngoscopic ClinicLeiomyosarcoma of the Larynx:A Complex DiagnosisGabriele Molteni, MD1, Davide Soloperto, MD, PhD1,Cristoforo Fabbris, MD1, Luca Gazzini, MD1, Stefano De Rossi, MD1,Giovanni Valotto, MD2, and Daniele Marchioni, MD1Case ReportAn 84-year-old man was referred to our institution for dyspho-nia present for 2 months. Fiberoptic laryngoscopy demon-strated a polypoid lesion of the right true vocal fold withnormal motility.Excisional biopsy of a submucosal thickening of the rightvocal fold with vocal ligament calcification was performed.Histology revealed squamous cell epithelium without malig-nancy. Postoperative endoscopic follow-up was regular.Six months after surgery, dysphonia and dyspnea occurred.Flexible laryngoscopy demonstrated right hemilaryngealparalysis, edema, and partial airway obstruction. Necrotic tis-sue and purulent secretions were visible at the anterior com-missure, right vocal fold, and ipsilateral vestibule. A secondmicrolaryngoscopic surgery included multiple biopsies anddebridement of the necrotic tissue. Histological report wasnegative for malignancy, and purulent chondritis was diag-nosed. Intravenous antibiotic therapy was administered withimprovement of infection, inflammation, dysphonia, and dys-pnea. The patient was discharged.Two months later, a new sudden and severe dyspneaoccurred. He was admitted to the emergency department, andoropharyngeal intubation was necessary because of respiratoryfailure.Computed tomography (CT) scan showed thickening of theright laryngeal wall with wide fluid collection (48-43 mm dia-meter) that almost completely obstructed the airway, with ero-sion of the right thyroid and cricoid cartilage (Figure 1).Debridement of the necrotic tissue and drainage of the puru-lent collection was performed with direct laryngoscopy. Cul-ture was positive for Staphylococcus epidermidis and Candidaalbicans. Intravenous antibiotic, antimicotic, and corticosteroidtherapy was administered, and other 3 surgical debridementsand drainage were performed, but without clinical or radiolo-gical improvements. Moreover, patient developed signs of sep-sis. Since no improvement had been achieved, totallaryngectomy was planned even without a positive histologicbiopsy. Intraoperative findings showed transglottic extensionof the ulcerated material with full-thickness infiltration of thelaryngeal wall and external spread through the thyroidcartilage, with invasion of prelaryngeal tissues (Figure 2). Thepatient’s clinical condition improved, and he started oral feed-ing without pharyngocutaneous fistula. Morphological andimmunohistochemical features were consistent with leiomyo-sarcoma (Fédération Nationale des Centres de Lutte Contre LeCancer [FNCLCC] grade 3; Figure 3).Multidisciplinary consultation suggested adjuvant radia-tion therapy, but preirradiation staging CT scan showed rapidlocal recurrence and lung metastasis. The poor clinical con-dition of the patient contraindicated first-line chemotherapy.Figure 1. Computed tomography image showing an almost completeerosion of the laryngeal framework cartilages due to LLMS. Residualairway is minimal therefore an emergency oropharyngeal intubation isneeded.1 Otolaryngology Department, University Hospital of Verona, Verona, Italy2 Pathology Department, University Hospital of Verona, Verona, ItalyReceived: August 19, 2018; revised: August 27, 2018; accepted: August 28,2018Corresponding Author:Cristoforo Fabbris, MD, Otolaryngology Department, University Hospital ofVerona, Piazzale Aristide Stefani 1, Verona 37126, Italy.Email: cristoforo.fabbris@gmail.comEar, Nose & Throat Journal1–3ª The Author(s) 2019Article reuse guidelines:sagepub.com/journals-permissionsDOI: 10.1177/0145561319840204journals.sagepub.com/home/earAt 3-month follow-up, the patient was alive and receiving bestsupportive care.DiscussionThe first case of laryngeal leiomyosarcoma (LLMS) wasreported by Jackson et al in 1939.1 After that, fewer than 50cases of LLMS have been reported in international literature,2including an 87-year-old man with laryngopyocele and LLMSwho was treated with total laryngectomy.3Simultaneous presence of laryngeal carcinoma, laryngopyo-cele, and laryngomucocele was described by Cassano et al,4and Mitro et al,5 but no leiomyosarcoma was found in thesepatients or in cases reviewed in similar articles.Purulent chondritis of laryngeal cartilages (PCLC) is a veryuncommon abscess that grows between inner and outer peri-chondria of the laryngeal framework. To date, only 5 caseshave been described in the literature,6-8 and none of them wasassociated with neoplasm.In our case, radiological suspicion of laryngeal abscess andno clear radiological signs of malignant tumor were detectable.Laryngeal biopsies were negative for malignancy, and cultureswere consistent with PCLC, but combined antibiotic therapy,surgical drainage, and debridement did not improve the clinicalcondition, and the patient developed sepsis. Fortunately, aftertotal laryngectomy, his clinical condition improved quicklywith complete sepsis resolution.Etiology of LLMS is still unclear. A possible factor is anabnormal postoperative healing process.9 In our case, thepatient developed worsening dysphonia and dyspnea, 6 monthsafter his first vocal fold biopsy with negative findings.Treatment guidelines for LLMS are lacking due to its rarity.First-line treatment should be tumor resection with wide mar-gins, reserving radiotherapy as adjuvant treatment or in case ofrecurrence or persistent tumor.10,11 Chemotherapy usually issuggested for distant metastases.12Even though if LLMS is a very rare laryngeal malignancy, itshould be considered when dealing with laryngeal abscess,especially with cartilage involvement.Authors’ NoteThe human participant involved in this research signed an informedconsent before any further treatment. All procedures performed werein accordance with the ethical standards of the institutional and/ornational research committee and with the 1964 Helsinki declarationand its later amendments or comparable ethical standards.Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect tothe research, authorship, and/or publication of this article.FundingThe author(s) received no financial support for the research, author-ship, and/or publication of this article.References1. Jackson C, Jackson CL. Sarcoma of the larynx. In: Jackson C. (ed).Cancer of the larynx. Philadelphia: W. B. Saunders; 1939:164-168.2. Yadav J, Bakshi J, Chouhan M, Modi R. Head and Neck Leio-myosarcoma. Indian J Otolaryngol Head Neck Surg. 2013;65(Suppl 1):S1-S5.3. Helmberger RC, Croker BP, Mancuso AA. Leiomyosarcoma ofthe Larynx Presenting as a Laryngopyocele. AJNR Am J Neuror-adiol. 1996;17(6):1112-1114.4. Cassano L, Lombardo P, Marchese-Ragona R, Pastore A. Laryn-gopyocele: three new clinical cases and review of the literature.Eur Arch Otorhinolaryngol. 2000;257(9):507-511.Figure 2. Intraoperative picture showing the almost complete ero-sion of laryngeal cartilages with necrotic material substituting normallaryngeal tissue.Figure 3. Histopathology of the lesion: SMA (smooth muscle specificactin) positivity demonstrates the smooth muscle nature of the neo-plastic population. Written histological report shows a malignantneoplasm with spindle cells, high mitotic index (10-14 mitosis 10HPF), and large areas of tissue necrosis (>50%).2 Ear, Nose & Throat Journal XX(X)5. Mitroi M, Căpitănescu A, Popescu FC, et al. Laryngocele associ-ated with laryngeal carcinoma. Rom J Morphol Embryol. 2011;52(1):183-185.6. Eliashar R, Gross M, Goldfarb A, Sichel JY. Purulent chondritisof the laryngeal framework cartilages. Ann Otol Rhinal Laryngol.2005;114(3):219-222.7. Lee JK, Cho HH, Kim HD, Lim SC. Purulent chondritis of thethyroid cartilage. Am J Otolaryngol. 2007;28(1):64-66.8. Leiva-Salinas C, Mukherjee S. Purulent chondritis of the cricoid.Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133(2):137-139.9. Khadivi E, Taziky MH, Jafarian AH, Nasseri Sadr M. Laryngealleiomyosarcoma, a case report and review of articles. Iran J Otor-hinolaryngol. 2013;25(73):253-258.10. Abbas A, Ikram M, Yagoob N. Leiomyosarcoma of the larynx:A case report. Ear Nose Throat J. 2005;84(7):435-436.11. Kleinsasser O., Glanz H. Myogenic tumors of the larynx. ArchOtorhinolaryngol. 1979;225(2):107-119.12. Ismi O, Arpaci RB, Puturgeli T, Ozcan C, Gorur K. Leiomyosar-coma of Larynx as a Metachronous Tumor of Squamous CellCarcinoma: An Unusual Case. J Voice. 2016;30(2):242-245.Molteni et al 3

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Leiomyosarcoma of the Larynx: A Complex Diagnosis

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