CASE REPORT

Amy Chen, MD, Section Editor

Laryngeal osteosarcoma: Case report and literature review Kiyomi Kuba, MD,* Hitoshi Inoue, MD, Takahiro Hayashi, MD, Satoko Matsumura, MD, Kazuhiko Minami, MD, Humihiko Takajo, MD, Kei Morita, MD, Mitsuhiko Nakahira, MD, PhD, Masashi Sugasawa, MD, PhD Department of Head and Neck Surgery and Otolaryngology, Saitama Medical University International Medical Center, Hidaka, Saitama, Japan.

Accepted 10 May 2014 Published online 24 July 2014 in Wiley Online Library (wileyonlinelibrary.com). DOI 10.1002/hed.23767

ABSTRACT: Background. Sarcomas of the larynx are uncommon tumors, and osteosarcoma is the rarest. Only 24 cases of osteosarcoma of the larynx have been reported. The only case reported in Asia was from Thailand in 1981. Methods. A 55-year-old male patient was referred to our hospital because of an 8-month history of odynophagia and a gradually enlarging left neck mass. A neck CT scan was performed. A literature review was also performed for this report. Results. The CT scan revealed extensive calcification with enlargement of the thyroid cartilage. The patient was diagnosed with osteosarcoma and was successfully treated by surgery alone.

Conclusion. Adequate surgical resection is required for successful treatment of laryngeal sarcomas. Adjuvant therapy is not contraindicated, but chemotherapy may improve the prognosis in patients with high-grade C 2014 Wiley Periodicals, Inc. Head Neck tumors or metastatic disease. V 37: E26–E29, 2015

INTRODUCTION

Figure 2). Fluorodeoxyglucose positron emission tomography showed focally intense fluorodeoxyglucose uptake in the tumor, but no evidence of metastasis. Based on its clinical and radiologic characteristics, the tumor was thought to be a chondroma or chondrosarcoma. Performance of fine-needle aspiration cytology seemed difficult because of the high degree of calcification. Therefore, we planned surgical resection for diagnosis and treatment with preservation of laryngeal function. With the patient under general anesthesia, a thyrotomy was performed to resect the mass with the left thyroid cartilage. The left upper pole of the thyroid gland was also resected because of the possibility of invasion. The recurrent laryngeal nerve and supralaryngeal nerve were preserved. Finally, a tracheostomy was performed. Macroscopically, the surgical specimen was composed of well-circumscribed, white, hard tissue. Histologically, the tumor comprised spindle cells with an osteoid matrix, and mitoses were numerous (see Figure 3). The tumor was diagnosed as a high-grade osteoblastic osteosarcoma, and the surgical margin was negative. Although left vocal cord paralysis occurred, the patient’s postoperative course was uneventful, and he was discharged after the tracheal stoma had closed. He declined adjuvant chemotherapy, but is still alive 20 months after surgery with no recurrence or metastasis.

Sarcomas of the larynx are uncommon tumors. They comprise about 1% of all malignant laryngeal tumors, and osteosarcomas are extremely rare.1 Only 24 cases of osteosarcoma of the larynx have been reported. The only case reported in Asia was from Thailand in 1981.2 We herein present another case of osteosarcoma of the larynx and discuss the current literature. This is the second case report from Asia and the first from Japan.

CASE REPORT A 55-year-old male patient was referred to our hospital because of an 8-month history of odynophagia and a gradually enlarging left-sided neck mass. He had no history of smoking or alcohol intake. Physical examination revealed a 20 3 30 mm hard, elastic mass in the left side of the neck just anterior to the thyroid cartilage. Although laryngoscopy showed no abnormalities, neck CT revealed extensive calcification with enlargement of the thyroid cartilage. No lymph node swelling was present, and the tumor did not infiltrate the surrounding tissue. The other cartilage was intact (see Figure 1). MRI of the neck also showed an enlarged mass in the thyroid cartilage. The mass was black on T1 and T2weighted images and was enhanced with gadolinium (see

KEY WORDS: laryngeal osteosarcoma, hoarseness, odynophagia, thyroid cartilage, head and neck

DISCUSSION *Corresponding author: K. Kuba, Department of Head and Neck Surgery and Otolaryngology, Saitama Medical University International Medical Center, 13971, Yamane, Hidaka, Saitama, 350-1298, Japan. E-mail: [email protected]

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Head and neck osteosarcomas are rare and account for 1% of all head and neck cancers. They are most frequently located in the mandible (45% to 56%) and

A

CASE OF LARYNGEAL OSTEOSARCOMA

FIGURE 1. (A) Axial contrastenhanced CT image and (B) coronal bone-window CT image revealed extensive calcification with enlargement of the left thyroid cartilage (white arrows).

maxilla (32% to 40%).3,4 According to the United States National Cancer Data Base report on head and neck osteosarcoma (n 5 496), the median age at the time of diagnosis was 38.0 years, the sex distribution was equal, and the 5-year disease-specific survival rate was 59.7%.5 Histologic subtypes of head and neck osteosarcoma include osteoblastic, chondroblastic, fibroblastic, telangiectatic, and dedifferentiated. Tumors are graded on the basis of the predominant cell type and degree of anaplasia of the neoplastic cells. Generally, osteoblastic, telangiectatic, and dedifferentiated types are high-grade neoplasias, and chondroblastic and fibroblastic types are low-grade or intermediate-grade neoplasias. The majority (63%) of head and neck osteosarcomas are high-grade tumors, followed by low-grade (22%) and intermediate-grade (15%) tumors.6 Osteosarcoma of the larynx is extremely rare and demonstrates many differences from osteosarcoma of other head and neck sites. Only 24 cases of laryngeal osteosarcoma were reported from 1942 to 2009 (Table 1),1,2,7–26

excluding 2 patients who were not described in detail and were lost to follow-up shortly after surgery. Among these 24 cases, the median age at diagnosis was 64 years (range, 47–80 years), and most patients were men (92%). The most common presenting complaints among these previously reported cases were hoarseness and dyspnea. Dysphasia, odynophagia, and acute airway obstruction were also seen in some patients. There was no evidence of a correlation with alcohol intake or smoking in this small number of reported cases. Two patients presented with a history of radiotherapy and likely developed radiation-induced tumors. The most common primary site was the true vocal cord, followed by the cricoid cartilage and thyroid cartilage. When the tumor occurred in the thyroid cartilage, hoarseness, dysphagia, and odynophagia were likely to develop, whereas dyspnea was rare. CT usually showed an infiltrating lesion with calcifications, but such findings are also seen in other tumors,

FIGURE 2. MRI showed a mass on the left thyroid cartilage, which was black on (A) T1-weighted and (B) T2-weighted images and (C) enhanced with gadolinium (white arrow). [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]

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TABLE 2. Treatment and clinical course of 24 patients with laryngeal osteosarcoma.

Treatment

TL/TPL TL 1 CRT TL 1 chemo PL 1 RT Local resection RT

No. of patients

Recurrence/ residual

Distant metastasis

Free of disease

15 3 1 1 2 2

11 3 1 0 1 2

10 3 1 0 -

4 1 0 1 1 0

Abbreviations: TL, total laryngectomy; TPL, total pharyngolaryngectomy; CRT, chemoradiotherapy; chemo, chemotherapy; PL, partial laryngectomy; RT, radiation therapy.

FIGURE 3. Hematoxylin-eosin staining of the surgical specimen at 3400 magnification showed a tumor comprising neoplastic spindle cells with an osteoid matrix. There was no chondrocyte proliferation.

such as chondrosarcomas. MRI is used to define the intramedullary and extramedullary extent of such tumors. Diagnosis of this tumor is difficult because of submucosal involvement, and several microlaryngoscopic biopsies or external incision is needed. The mainstay of treatment is surgery. Adequate resection is extremely important for head and neck osteosarcoma. Smeele et al27 demonstrated the importance of the margin status, with a 40% survival advantage noted in patients with craniofacial osteosarcoma with clear margins of resection. Among previous cases, 22 of 24 patients underwent surgical treatment (Table 2). Nineteen patients underwent total laryngectomy (including 1 case of total pharyngolarTABLE 1. Clinical characteristics of 24 patients with laryngeal osteosarcoma. Characteristic

No. of patients Sex male:female ratio Median age, y Symptoms Hoarseness Dyspnea Dysphasia Acute airway obstruction Odynophagia Pain radiation to ear Globus sensation Anatomic site, number True vocal cord Cricoid cartilage Thyroid cartilage Transglottic Entire of larynx Anterior fissure Supraglottic Not seated

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Value

24 22:2 64.4 (range, 49–84 y) 66% 58% 16% 8% 8% 4% 4% 9 4 3 2 2 2 1 1

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yngectomy). Three patients underwent tumor resection with preservation of laryngeal function, and 1 patient developed local recurrence. Two patients underwent radiation treatment as the initial therapy, but both tumors were residual or recurred. In the present case, the tumor had not extended to form other lesions, and we thus planned local resection with preservation of laryngeal function. Although the survival rate of laryngeal osteosarcoma is uncertain because of the small number of reported cases, only a few patients were free of recurrence and metastasis. Local recurrence occurred after an average of 9.8 months, and distant spread occurred after an average of 11.0 months. Lung metastasis occurred in 9 patients (33%); 2 had bone metastasis, 1 had mediastinal metastasis, and 2 had cervical skin metastasis. More than 60% of patients died 3 to 20 months after surgery. This mortality rate is obviously worse than that in patients with head and neck osteosarcomas at other sites. However, patients with skin metastasis who underwent salvage resection remained alive for more than 60 months.11,18 Thus, surgery should be carried out without hesitation if metastases are resectable. The benefits of adjuvant chemotherapy and radiotherapy are unknown. Osteosarcomas are relatively radioresistant.6 A few patients who underwent primary radiation therapy demonstrated poor survival. The other patients underwent postoperative radiation therapy because of close surgical margins, lymph node metastasis, or local recurrence, but this treatment seemed to confer no benefit over surgery alone. Adjuvant chemotherapy is widely performed in patients with non-head and neck osteosarcoma, and it markedly improves survival.28 Methotrexate, adriamycin, and cisplatin are the most frequently used drugs. However, the impact of such treatment on survival in patients with head and neck osteosarcoma is controversial. Although some authors have described a benefit of adjuvant chemotherapy in patients with head and neck osteosarcoma, there was no clear effectiveness in the 24 previously reported cases. Smith et al5 reported that equivalent survival rate in the surgery-only group with adjuvant chemotherapy group in the United States National Data Base on Head and Neck Osteosarcoma. He reports that patients with high-grade tumors or with metastatic disease were more likely to receive adjuvant chemotherapy, whereas patients with smaller tumors were more frequently treated with surgery alone. In our case,

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surgical margin was negative but relatively close, and it was a histologically high-grade tumor. Our patient refused any of the adjuvant chemotherapy but completed the disease-free period of 20 months by surgical treatment alone.

CONCLUSION Laryngeal osteosarcoma is extremely rare, with poor prognosis. Adequate surgical resection is a must for successful treatment. Adjuvant therapy was not unestablished, but chemotherapy may improve the prognosis in patients with high-grade tumors or with metastatic disease.

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CASE OF LARYNGEAL OSTEOSARCOMA

10. Rydzewski B, Matusiak M. Osteosarcoma of the larynx ‘in Polish’. Otolaryngol Pol 2005;59:285–288. 11. Madrigal FM, Godoy LM, Daboin KP, Casiraghi O, Garcia AM, Luna MA. Laryngeal osteosarcoma: a clinicopathologic analysis of four cases and comparison with a carcinosarcoma. Ann Diagn Pathol 2002;6:1–9. 12. Athre RS, Vories A, Mudrovich S, Ducic Y. Osteosarcomas of the larynx. Laryngoscope 2005;115:74–77. 13. Topaloglu I, Is¸iksac¸an V, Ulusoy S, Sis¸man S. Osteosarcoma of the larynx. Otolaryngol Head Neck Surg 2004;131:789–790. 14. Myssiorek D, Patel M, Wasserman P, Rofeim O. Osteosarcoma of the larynx. Ann Otol Rhinol Laryngol 1998;107:70–74. 15. Rossi RM, Landas SK, Kelly DR, Marsh WL. Osteosarcoma of the larynx. Otolaryngol Head Neck Surg 1998;118(3 Pt 1):385–388. 16. Berge JK, Kapadia SB, Myers EN. Osteosarcoma of the larynx. Arch Otolaryngol Head Neck Surg 1998;124:207–210. 17. Sheen TS, Wu CT, Hsieh T, Hsu MM. Postirradiation laryngeal osteosarcoma: case report and literature review. Head Neck 1997;19:57–62. 18. Pinsolle J, LeCluse I, Demeaux H, Laur P, Rivel J, Siberchicot F. Osteosarcoma of the soft tissue of the larynx: report of a case with electron microscopic studies. Otolaryngol Head Neck Surg 1990;102:276–280. 19. van Laer CG, Helliwell TR, Atkinson MW, Stell PM. Osteosarcoma of the larynx. Ann Otol Rhinol Laryngol 1989;98(12 Pt 1):971–974. 20. Levine HL, Tubbs R. Nonsquamous neoplasms of the larynx. Otolaryngol Clin North Am 1986;19:475–488. 21. Remagen W, L€ ohr J, von Westernhagen B. Osteosarcoma of the larynx ‘in German’. HNO 1983;31:366–368. 22. Gorenstein A, Neel HB III, Weiland LH, Devine KD. Sarcomas of the larynx. Arch Otolaryngol 1980;106:8–12. 23. Haar JG, Chaudhry AP, Karanjia MD, Milley PS. Chondroblastic osteosarcoma of the larynx. Arch Otolaryngol 1978;104:477–481. 24. Morley AR, Cameron DS, Watson AJ. Osteosarcoma of the larynx. J Laryngol Otol 1973;87:997–1005. 25. Sprinkle PM, Allen MS, Brookshire PF. Osteosarcoma of the larynx. (A true primary sarcoma of the larynx). Laryngoscope 1966;76:325–333. 26. Clerf LN. Sarcoma of the larynx: report of eight cases. Arch Otolaryngol 1946;44:517–524. 27. Smeele LE, Kostense PJ, van der Waal I, Snow GB. Effect of chemotherapy on survival of craniofacial osteosarcoma: a systematic review of 201 patients. J Clin Oncol 1997;15:363–367. 28. Ferrari S, Mercuri M, Picci P, et al. Nonmetastatic osteosarcoma of the extremity: results of a neoadjuvant chemotherapy protocol (IOR/OS-3) with high-dose methotrexate, intraarterial or intravenous cisplatin, doxorubicin, and salvage chemotherapy based on histologic tumor response. Tumori 1999;85:458–464.

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Laryngeal osteosarcoma: case report and literature review.

Sarcomas of the larynx are uncommon tumors, and osteosarcoma is the rarest. Only 24 cases of osteosarcoma of the larynx have been reported. The only c...
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