Papillary carcinoma in a lingual thyroid: an unusual presentation

A lingual thyroid is a mass of ectopic thyroid tissue located in the midline of the base of the tongue. Its estimated prevalence ranges from 1 in 3,000 to 1 in 10,000 population. We report the interesting case of a 28-year-old woman who presented with a primary papillary carcinoma in a lingual thyroid and a histologically normal thyroid gland. To the best of our knowledge, this case probably represents only the second reported case of a follicular variant of a papillary carcinoma arising in a lingual thyroid.     

Incidental metastatic papillary thyroid carcinoma in microvascular reconstruction

OBJECTIVES/HYPOTHESIS: Occult papillary thyroid carcinoma has a reported prevalence of 1% to 35% based on autopsy studies. Cervical lymphatic metastases from papillary thyroid carcinoma have been associated with a higher likelihood of recurrence with a questionable impact on survival. Without clinically evident disease in the thyroid or cervical nodes, management of these patients presents a treatment dilemma. We propose an individualized treatment plan for patients in whom metastatic papillary thyroid carcinoma is incidentally detected during neck exploration for other purposes. STUDY DESIGN: Retrospective review and discussion of the literature. METHODS: The clinical course of two patients with incidentally discovered metastatic papillary thyroid carcinoma to the cervical lymph nodes is described. Both patients had previously received head and neck irradiation in childhood and required free flap reconstruction of extensive skull base defects following extirpation of meningiomas. RESULTS: Neck dissection specimens from levels I and II obtained during exposure of recipient vessels for microvascular tissue transfer revealed papillary thyroid carcinoma in both cases. The patients subsequently underwent total thyroidectomy, neck dissection, and postoperative radioactive iodine ablation of residual thyroid tissue. After 1 year of follow-up, both patients were without evidence of recurrent disease. CONCLUSIONS: An individualized approach is justified to treat metastatic papillary thyroid carcinoma incidentally discovered during other procedures. The case reports underscore the importance of pathological analysis of surgical specimens obtained during head and neck reconstruction.     

Transoral videolaryngoscopic surgery for papillary carcinoma arising in lingual thyroid

Carcinoma arising in lingual thyroid is an extremely rare entity accounting for only 1% of all reported ectopic thyroids. Here, we report a case of carcinoma arising in lingual thyroid, which has been successfully managed by transoral resection and bilateral neck dissections.A lingual mass 4-cm in diameter with calcification was incidentally detected by computed tomography at medical check-up. No thyroid tissue was observed in normal position. Ultrasound examination showed bilateral multiple lymphadenopathies. Fine needle aspiration biopsy from lymph node in his right neck was diagnosed as Class III and thyroglobulin level of the specimen was 459 ng/ml. Due to the difficulty in performing FNA of the lingual masses, right neck dissection was performed in advance for diagnostic purpose. Pathological examination showed existence of large and small follicular thyroid tissues in several lymph nodes, suggesting lymph node metastasis from thyroid carcinoma. Two months after the initial surgery, video-assisted transoral resection of lingual thyroid with simultaneous left neck dissection was performed. Postoperative course was uneventful. Papillary carcinoma was found in the lingual thyroid and thyroid tissues were also found in left cervical lymph nodes.Video-assisted transoral resection was useful for the treatment of thyroid cancer arising in lingual thyroid.     

原发性异位甲状腺癌3例并文献复习

目的：探讨原发性异位甲状腺癌的临床特征及诊治原则。方法：回顾性分析我科1990年至今诊治的共3例原发性异位甲状腺癌患者的临床资料,并结合文献进行分析。结果：3例患者均为女性,年纪较轻,术前表现为颈部肿块,术后病理均证实为异位甲状腺乳头状癌,随访至今均无复发征象。结论：异位甲状腺是胚胎时期甲状腺始基迁移过程中发生异常所致,同样能发生癌变,其治疗原则在国内外颇有争议。本文报道的3例原发性异位甲状腺乳头状癌均予以手术切除,术后予以甲状腺素治疗,预后良好。     

Incidental metastases of well-differentiated thyroid carcinoma in lymph nodes of patients with squamous cell head and neck cancer: eight cases with a review of the literature

The examination of a large series of cervical lymph nodes in patients with head and neck cancer revealed the presence of incidental metastases of occult thyroid carcinoma in eight patients, of which six cases were squamous cell carcinoma of glottic and supraglottic sites of the larynx and two cases were pyriform sinus and tongue carcinomas. Three patients had two lymph nodes and the remaining patients had one lymph node each involved. The nodal chains affected were the jugular (n=5; level IV), Kuttner (level II), supraomohyoid (level III) and supraclavicular (level VI). In four cases, a subtotal thyroidectomy or unilateral lobectomy was performed during laryngectomy (for surgical reasons) or after histologic nodal examination; a minimal focus of thyroid papillary carcinoma was detected in one patient. Three of eight patients died from recurrence of the squamous cell carcinoma; no case presented clinical evidence of thyroid malignancy. The differential diagnosis from benign thyroid heterotopia was based on the presence of minimal nuclear atypia. The choice of treatment of patients with a coexisting neoplasm characterized by poor prognosis is difficult, and contrasting opinions exist regarding the use of radical thyroidectomy and the subsequent management. As reported in the literature (66 cases), the more aggressive squamous cell carcinoma will determine the prognosis of these patients; in fact, only one of the referred cases died of cerebellar metastases of the thyroid cancer. Our results emphasize the importance of an accurate re-evaluation and follow-up of patients with incidental occult metastases for detection of a primary thyroid tumor. In the general population, this incidental nodal involvement may be related to a minimal occult thyroid carcinoma.An erratum to this article can be found at     

Papillary thyroid cancer with an initial presentation of abdominal and flank pain

PURPOSE: Well-differentiated thyroid cancer typically presents as a thyroid mass. Common sites of metastases upon presentation include cervical lymph nodes, lung, and bone. Well-differentiated thyroid cancer with clinically apparent kidney metastases is rare, with fewer than 20 cases reported in the literature. In the vast majority of these cases, the patients had known thyroid neoplasms at the time the renal metastases were identified. We report a case of papillary thyroid carcinoma that presented with abdominal pain in a 25-year-old woman with no previous history of thyroid disease. STUDY DESIGN: This study is a case report. RESULTS: The patient underwent radical nephrectomy for a right renal mass, which was diagnosed as papillary thyroid carcinoma follicular variant. During subsequent evaluation, metastatic disease was also identified in the patient's lungs. The patient was treated with total thyroidectomy and iodine 131. CONCLUSIONS: Papillary cancer, which ordinarily behaves in an indolent manner, can have unusual presentation, including disseminated metastasis on presentation. Renal metastases are extremely rare.     

Uncommon metastatic site of renal adenocarcinoma: the oral tongue

Primary tumours metastasizing to the tongue are very unusual and only anecdotal cases have been reported. An exhaustive literature review covering the period from 1970 onwards disclosed only 22 cases of renal adenocarcinoma metastasizing to the tongue. We report the case of an 87-year-old female patient with oral tongue, lung, liver, thyroid gland, pancreas and renal adenocarcinoma metastases. She had undergone contralateral nephrectomy for clear cell carcinoma 10 years before diagnosis of the metastases. The tongue lesion was surgically removed under local anaesthesia. Tongue metastasis of renal adenocarcinoma is usually a manifestation of widespread disease. The prognosis for patients with lingual metastasis of renal adenocarcinoma is poor, the mean interval from diagnosis of tongue metastasis to death being 5.8 months. In our patient, metastatic involvement of the tongue was detected approximately 5 months before death. Treatment of renal adenocarcinoma metastasis to the tongue is usually palliative and aims to provide patient comfort by means of pain relief and prevention of bleeding and infection. Surgical excision is recommended as the primary treatment, with emphasis on preservation of tongue structure and function. Recent data regarding immunotherapy or immunochemotherapy for metastatic renal adenocarcinoma are encouraging.     

The first report of bilateral retropharyngeal lymph node metastasis from papillary thyroid carcinoma and review of the literature

The sites of lymph node metastasis of papillary thyroid carcinomas are typically the paratracheal and jugular lymph nodes. On the other hand, metastasis to the retropharyngeal or parapharyngeal nodes from papillary thyroid carcinomas is very rare. During the last two decades, limited to cases with a histologically definite diagnosis by surgery, only 39 cases have been reported. All reported cases were unilateral retropharyngeal or parapharyngeal node metastasis except one metachronous bilateral case, and there were no reports of simultaneous bilateral cases within our literature review. We report three cases of retropharyngeal node metastasis from thyroid papillary carcinoma, including a case of bilateral nodal metastasis. Retropharyngeal node metastasis was successfully resected in all three patients by the transcervical approach. As pointed out in past reports, this report also suggests that prior neck dissection and/or metastasis to cervical lymph nodes might alter the direction of lymphatic drainage to the retrograde fashion, resulting in the unusual metastasis to the retropharyngeal lymph nodes, and there is a possibility of a bilateral pattern. Also, it is necessary to consider the possibility of metastasis from a papillary thyroid carcinoma in the differential diagnosis of lymph node swelling in the parapharyngeal space.     

Papillary thyroid carcinoma in a thyroglossal duct cyst: primary tumor or metastasis?

Thyroglossal duct cysts are the most common anomaly in thyroid development. The occurrence of carcinoma in a cyst is reported to be about 1% of cases. Histopathological examinations reveal a papillary thyroid carcinoma in about 94% of these. We report the case of 38-year-old female having a massive, cervical cystic mass over a period of 18 months. An ectopic papillary thyroid carcinoma was diagnosed. A total thyoidectomy and bilateral neck dissection were performed. This revealed a small thyroid carcinoma and three positive lymphatic nodes. Whether the cystic carcinoma is the primary or a metastasis of the thyroid carcinomais discussed.     

Laryngeal carcinoma in a non-smoker female patient with thyroid carcinoma: report of a case

Laryngeal carcinoma is usually encountered in smoker men, and thyroid carcinoma is sometimes discovered incidentally during treatment for these patients. However, this coexistence of malignancies could occur in non-smoker female. We report an unusual case of multiple primary malignancies in the larynx and the thyroid gland. The laryngeal carcinoma was suspected to be related to the malignant transformation of the papillomas. The case suggests the importance of meticulous examination in the head and neck region for treatment of cervical metastatic lymph nodes with negative cytology in non-smoker female.     

Management considerations for differentiated thyroid carcinoma presenting as a metastasis to the skull base

OBJECTIVES/HYPOTHESIS: To characterize the salient features of skull base metastasis from differentiated thyroid carcinoma, discuss the diagnostic and treatment strategies, and propose rational management guidelines for such tumors. STUDY DESIGN: Case report. METHODS: Review of English literature from MEDLINE with the addition of our case. RESULTS: Skull base metastasis from differentiated thyroid carcinoma is rare, with only 20 cases reported to date, including our case report. On the basis of a review of all reported cases, both follicular and papillary thyroid cancers can metastasize to the skull base. Our case is unique because the lesion extends locally into the cavernous sinus and beyond. Histopathologic diagnosis is limited by the remote location of lesions. Most tumors are highly vascular, and there is potential for significant morbidity and mortality associated with surgical resection. The overall survival ranges from less than one year to 10+ years from the discovery of the metastasis and is similar in both tumor subtypes. There is no clear consensus on the management strategy for skull base metastasis from differentiated thyroid carcinoma. Interestingly, surgical resection of both the primary and metastatic lesions yields similar survival when compared with resection of the primary tumor alone. CONCLUSIONS: Distant metastasis from differentiated thyroid carcinoma needs to be considered in the differential diagnosis of destructive skull base lesions, regardless of the patient's age. Histopathologic tissue diagnosis should always be attempted, followed by total thyroidectomy, radioiodine, or external beam radiation, and chronic thyroid-stimulating hormone suppression. Surgical resection of the metastatic lesion should only be performed in carefully selected cases because it is associated with significant morbidity.     

Dual ectopic thyroid: report of a case

Dual ectopic thyroid is very rare. We report a case of dual ectopic thyroid in the lingual and infrahyoid areas in a 20-year-old female patient with no thyroid gland in its normal anatomical location. On physical examination, there was a 7 x 5 cm anterior midline neck swelling just below the hyoid bone and a 2 x 2 cm mass in the base of the tongue. Triiodothyronine (T(3)), thyroxine (T(4)), and thyroid-stimulating hormone (TSH) levels were normal. A thyroid scan with technetium-99m sodium pertechnate confirmed dual ectopic thyroid with no iodine uptake in the normal anatomical location of the thyroid gland. The infrahyoid ectopic thyroid was surgically removed for cosmetic reasons, and the lingual thyroid, which was symptomatic, was left untouched. The importance of thyroid scanning in the evaluation of anterior midline neck swellings and treatment options are discussed.     

A long-standing cystic lymph node metastasis from occult thyroid carcinoma--report of a case.

The authors describe a patient with a cervical cystic mass present for 14 years which proved to be a cystic metastasis from a papillary carcinoma of the thyroid gland. This is probably the first case report of a long-term lateral cervical cyst caused by an occult thyroid carcinoma.     

Incidence and significance of clinically unsuspected thyroid tissue in lymph nodes found during neck dissection in head and neck carcinoma patients

OBJECTIVES/HYPOTHESIS: The objectives were to quantify the incidence of clinically unsuspected thyroid tissue in cervical lymph nodes encountered during neck dissection in patients with head and neck carcinoma, to describe the location and histological aspect of these inclusions, and to assess their clinical significance. STUDY DESIGN: Retrospective study. METHODS: The histological records of 1123 neck dissections in 752 patients with head and neck carcinoma were reviewed. In cases with thyroid inclusions, the pathological diagnosis was reviewed and an immunohistochemical study against thyroglobulin and calcitonin was carried out. RESULTS: Clinically unsuspected thyroid tissue was found in lymph nodes in 11 of the 752 patients with head and neck carcinoma treated with neck dissection. In five cases, the thyroid inclusion was compatible with a metastases of an occult papillary thyroid carcinoma. In the other six cases, a collection of thyroid follicles without malignant characteristics was found beneath the lymph node capsule. These latter cases were considered benign thyroid inclusions. A thyroidectomy was performed in three of the patients with lymph node metastases of the papillary carcinoma. An occult papillary carcinoma was found in only one case. The other two patients had been treated previously with radiotherapy for an early-stage glottic carcinoma. Immunohistochemical study did not find calcitonin-positive cells within the benign thyroid inclusions. After a follow-up period ranging from 1.2 to 8.2 years, no patient had any kind of local, regional, or distant relapse related to the thyroid disease. CONCLUSION: The incidence of unsuspected thyroid tissue in lymph nodes of patients with head and neck carcinoma treated with neck dissection was 1.5%. Both lymph node metastases of a papillary carcinoma and benign thyroid inclusions were found. The study results suggest that the incidental finding of thyroid tissue in the lymph nodes during a neck dissection in patients with head and neck carcinoma does not necessarily indicate the need for aggressive therapy.     

Papillary thyroid carcinoma in a thyroglossal cyst

The incidence of Papillary Thyroid Carcinoma in a Thyroglossal Cyst is rare. Only about 160 cases have been reported in the last 85 years. We report a case of Thyroglossal Cyst who underwent Sistrunk ’s Operation. The Cyst was reported to contain a focus of papillary thyroid carcinoma. In the absence of metastases in thyroid gland and neck nodes, only thyroid suppression with Thyroxine was given. After I year of follow-up there are no metastases. The importance of Sistrunk’s operation lies not only in complete removal of Thyroglossal Cyst but also in management of small foci of Papillary thyroid Carcinoma.     

Simultaneous metastasized primary unknown signet ring cell carcinoma of the cervical lymph node and mucoepidermoid carcinoma of the parotid gland as double cancers

Lymph node metastasis from signet ring cellcarcinoma (SRCC) primary unknown is extremely rare. We here report a case of primary-unknown SRCC that metastasized to the cervical lymph nodes, co-existing with mucoepidermoid carcinoma (MEC) of the parotid gland as a simultaneous double cancer. A 68-year-old female patient with right swollen cervical lymph nodes consulted our medical center. A diagnosis of bilateral cervical lymph node metastasis and a right parotid tumor was made. After bilateral neck dissection and right parotidectomy, the pathological diagnosis was SRCC of primary unknown with metastasis to the cervical lymph node and MEC of the parotid gland. Examination of the CRTC1/3-MAML2 fusion gene showed no relation between SRCC of primary unknown with metastasis to the cervical lymph node and MEC of the parotid gland. Ten months after the first treatment, there was recurrence in the left neck lymph node, and left neck dissection was performed. Fourteen months after the first treatment, the patient is alive and cancer-free. This case is the fourth report of SRCC with lymph node metastasis, and highlights the value of fusion gene detection to determine relatedness between simultaneous cancers. Moreover, such cases should be closely monitored for the subsequent appearance of distant metastases.     

Medial lingual lymph node metastasis in carcinoma of the tongue

Lingual lymph node metastases are rarely seen in carcinoma of the tongue, and these nodes are not removed during neck dissection. Lingual lymph nodes are classified into medial and lateral groups, and metastasis to the former is extremely rare. A 55-year-old male with squamous cell carcinoma of the tongue, (stage T4aN0M0), underwent hemiglossectomy with neck dissection and free flap reconstruction. The lingual septum had a mass, 8 mm in size, which was diagnosed as medial lingual lymph node metastasis on histopathology. The patient developed multiple distant metastases and died of disease 18 months after the initial surgery. The presence of medial lymph node metastasis could result in contralateral neck metastases and worsen prognosis. Such cases may warrant more intensive therapy than recommended by current guidelines.     

双侧甲状腺癌1例

1病例报告 患者,女,48岁。因＂发现颈部肿物2个月余＂于2013年6月18日入院。患者2个月前无意中发现颈部有一肿物,无疼痛、发热、呼吸及吞咽困难,无食欲减退、盗汗、消瘦,无声嘶、情绪暴躁易怒。自发病来肿物无明显增大,体重未见下降,未予诊治。患者平素健康,无结核病及糖尿病病史。     

Squamous cell carcinoma and lingual thyroid

This report represents the first known case of squamous cell carcinoma associated with lingual thyroid tissue. Clinical examination of an exophytic mass at the base of tongue, CT imaging and a preoperative biopsy established a diagnosis of squamous cell carcinoma of the midline base of tongue. The therapeutic options and management of this carcinoma by surgical resection, irradiation or combined modalities were analyzed. The surgical option was chosen. This was accomplished via a midline labiomandibulo glossal split and primary repair of the wound. The final pathology report revealed a squamous cell carcinoma of the mucosa of the base of tongue directly over a benign lingual thyroid mass. Post-operative thyroid function studies were consistent with hypothyroidism and a thyroid scan confirmed the absence of thyroid tissue.     

"Collision" metastasis from unknown primary squamous cell carcinoma and papillary microcarcinoma of thyroid presenting as lateral cervical cystic mass

We report an unusual case of "collision metastasis". In a single lymph node, we found a metastatic mass composed of two immunohistochemically distinct components originating from two primary tumors: a papillary microfollicular thyroid cancer and an unknown primary squamous cell carcinoma. The clinical features and immunohistochemical profile are reported. Collision phenomena in oncology are extremely rare and pose diagnostic and management challenges which are discussed.