Squamous cell carcinoma in a thyroglossal duct cyst: A case report with review of the literature




Abstract


Carcinoma arising within a thyroglossal duct cyst (TGDC) is exceedingly uncommon, occurring in about 1% of TGDC (Chrisoulidou et al., 2013 [ ]). Since the first description of TGDC carcinoma in 1911 by Brentano, approximately 200 cases have been reported in the world literature, mostly as single case reports making its true incidence difficult to determine (Hanna, 1996 ). There are numerous reports in the literature of papillary and follicular TGDC carcinoma, but only limited reports of squamous cell carcinoma. In order for a lesion to qualify as squamous cell carcinoma of a TGDC, the lesion must arise from the epithelial lining. Recognition and understanding of this entity are important in staging and planning of treatment. Exclusion of primary squamous cell carcinoma arising from an adjacent structure or metastatic central compartment nodal disease is important in the evaluation for diagnosis to be confirmed. A thorough work up should be undertaken to exclude other primary malignancies that may have metastasized. We present a unique case of squamous cell carcinoma, along with supporting pathology and radiology imaging. We then review this rare entity and provide some insight into the diagnosis and management.



Case presentation


A 47-year-old male smoker presented with a 6-month history of a right-sided neck mass with associated pain, hoarseness, and productive cough. Significant past medical history included an unknown thyroid disorder, Chronic Obstructive Pulmonary Disorder (COPD), and Obstructive Sleep Apnea (OSA). On examination, he had a mildly tender, soft, right neck mass that elevated with swallowing. The thyroid gland was midline and non-enlarged, and there was no evidence of cervical lymphadenopathy. Computed tomography (CT) of the neck with contrast revealed an 8 × 8 × 5 cm cystic lesion with thick septations and solid areas of enhancement arising from the thyroid notch and extending to the midline tongue base. This lesion was deep to the strap muscles. These findings are shown below in Figs. 1 and 2 . Cytological sampling of the neck mass revealed benign and enucleated squamous cells in a background of proteinaceous debris. Based on these findings, thyroglossal duct cyst was the suspected diagnosis and the decision to perform a Sistrunk procedure was made.




Fig. 1


Axial CT of the soft tissue neck with intravenous contrast demonstrates multiseptated cystic type lesion centered at the thyroid notch. Thick nodular septation of this lesion is noted. Image provided courtesy of Dr. Azita Khorsandi MD, Department of neuroradiology at New York Eye and Ear Infirmary.



Fig. 2


Sagittal CT of the soft tissue neck with intravenous contrast demonstrates multiseptated cystic type lesion centered at the thyroid notch. Thick nodular septation of this lesion is noted. Image provided courtesy of Dr. Azita Khorsandi MD, Department of neuroradiology at New York Eye and Ear Infirmary.


During the resection, the mass was punctured and thick yellow fluid was noted to extrude from it. At the lower extent of the resection, it was noted that the mass was abutting the thyroid gland. The mass was carefully dissected from the thyroid gland and the thyroid gland was left intact. The thyroid gland appeared normal, and no suspicious lymph nodes were seen. The cyst and cyst contents were sent for pathological review. The patient tolerated the procedure well with no post-operative complications.


Histologic analysis of the lesion showed numerous malignant squamous cells in the specimen with frequent mitotic figures as shown in Figs. 3 and 4 .




Fig. 3


40 × magnification of H&E stain of tumor specimen showing numerous mitotic figures in tumor. Original image provided courtesy of Austin Anderson MD and Iacob Codrin MD, Department of Pathology New York Eye and Ear Infirmary.



Fig. 4


20 × magnification of H&E stain of transition between columnar epithelial to malignant squamous cells. Original image provided courtesy of Austin Anderson MD and Iacob Codrin MD, Department of Pathology New York Eye and Ear Infirmary.


The diagnosis of squamous cell carcinoma was confirmed, and the patient was sent for PET/CT scan, which did not reveal regional or distant metastatic disease. The patient was post-operatively sent for evaluation by radiation oncology and medical oncology for chemotherapy and radiation. The patient received weekly cisplatin, as well as 6600 Gy of external beam radiation divided into 33 fractions. Given the likely descent from the tongue base, this region was included in the radiation field. Currently, the patient is due for a three month post-treatment follow-up during which repeat imaging will be ordered as well.

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Aug 23, 2017 | Posted by in OTOLARYNGOLOGY | Comments Off on Squamous cell carcinoma in a thyroglossal duct cyst: A case report with review of the literature

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