A 76-year-old man was referred to our hospital for investigation of a 20-day history of rapidly deteriorating dyspnea and severe cough. This was associated with dysphagia and cervical pain. The patient was an ex-marble craftsman and an active smoker. His medical history included chronic pulmonary obstructive disease (COPD). A chest X-ray (CXR) showed an imprint at the right side of the trachea. Thyroid ultrasound (US) revealed a large and heterogenous thyroid gland and the presence of a calcified mass, 4.76 × 4.01 cm with high blood perfusion in the right upper lobe of the gland. A contrast-enhanced computed tomography (CT) scan of the neck and thorax reported the corresponding lesion as heterogenous, with irregular borders, extending from the sternoclavicular joints to the upper borders of the thyroid gland. The mass was surrounding the trachea anterolaterally displacing it to the left and the right common artery to the right (). There were no enlarged lymph nodes. Bronchoscopy revealed a fracture of the first tracheal ring but there was no evidence of tumour invasion (). Cytology from bronchial lavage was negative for cancer cells. Laboratory tests were within normal limits with the exception of elevated D-dimers (2,994 μg/L) and elevated white blood cells (19.27 K/μL). Tumour markers were within normal range values.\nOn admission, the patient was intubated, due to acute respiratory failure, and he was admitted to the intensive care unit (ICU). Subsequently, he underwent an emergency procedure and a collar incision was carried out. At surgery, a suspicious looking tumour, which was in continuity with the superior pole of the right thyroid lobe, the sheath of right common carotid artery, and the anterolateral portion of the cartilaginous trachea, was identified. Macroscopically, the tumour looked whitish and scleroelastic with moderately regular borders and was firmly adhered to the aforementioned structures. After the right recurrent laryngeal nerve was identified, the tumour was carefully dissected off the right thyroid lobe and the adventitia of the right common carotid artery. A similar approach was carried out to dissect the tumour off the left lobe; hence, an en bloc tumour resection with total thyroidectomy and a regional lymph node dissection was performed. Rapid frozen section biopsy of the tumour in the right thyroid lobe proved to be malignant with features of squamous cell carcinoma and negative surgical margins.\nPostoperatively, the patient experienced swallowing difficulty and hoarseness and a tracheostomy was performed on day 2.\nFinal pathology examination confirmed frozen section's diagnosis of a squamous cell carcinoma invading the right lobe of the thyroid gland, as well as extracapsular structures of the left lobe. Microscopically, the tumour was described as nonencapsulated, irregularly lobulated, with broad fibrous septa, and lymphoplasmacytic, eosinophilic, histiocytic, and multinuclear infiltration. The neoplastic cells were largely polyhedral or oval with eosinophilic or amphophilic cytoplasm, arranged in islands or nests that showed focal keratinization. The nuclei were vesicular or hyperchromatic with nucleoli. A panel of immunohistochemical stains was performed; the tumour was positive for p63, CD117, CKH, CD5, and CD3 in the lymphocytic part (Figures , , and ). It was also negative for the expression of TTF1.\nThe patient subsequently improved and was discharged home on postoperative day 20. The patient did not receive any additional treatment.
Patient 1 was a 61-year-old female who presented with a 5-cm anterior neck mass with retrosternal extension. She was surgically treated with a right thyroid lobectomy at another institution. The lesion was interpreted to be a poorly differentiated papillary thyroid cancer. Postoperative treatment consisted of sequential and combined chemoradiotherapy. Local recurrence in the region of the right thyroid occurred 20 months later, and radiotherapy was performed to relieve symptoms. Six years later, the patient had local recurrence again and was referred to our institution for further treatment. After curative wide local excision and central neck dissection, postoperative radiotherapy was given to the thyroid bed and bilateral cervical lymph node areas (55 Gy/16 fractions). Histological re-evaluation of the recurrent tumor specimens revealed CASTLE (pT4bN1M0). A third local recurrence was diagnosed 1 year later, and concurrent chemotherapy (paclitaxel 210 mg/m2 and cisplatin 40 mg/m2) and radiotherapy (50.4 Gy /28 fractions) were applied. The patient died from acute respiratory distress 1 month after the initiation of treatment.