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Massive bilateral mandibular metastasis from papillary thyroid carcinoma
Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx
Contents lists available at ScienceDirect
Egyptian Journal of Ear, Nose, Throat and Allied Sciences journal homepage: www.ejentas.com
Case report
Massive bilateral mandibular metastasis from papillary thyroid carcinoma Siti Asmat Md Arepen ⇑, Irfan Mohamad Department of Otorhinolaryngology-Head & Neck Surgery, School Of Medical Sciences, Universiti Sains Malaysia Health Campus, 16150 Kota Bharu, Kelantan, Malaysia
a r t i c l e
i n f o
Article history: Received 13 June 2016 Accepted 8 December 2016 Available online xxxx Keywords: Papillary thyroid carcinoma Metastasis Mandible
a b s t r a c t Papillary thyroid carcinoma (PTC) comprises almost eighty percent of differentiated thyroid malignancy. It affects female more than male in the age range between 40–50 years old. It usually metastasizes to ipsilateral regional lymph nodes of neck and rarely have distant metastasis. Common route of dissemination of tumour is through lymphatic system. We reported a rare case of bilateral mandibular metastasis from incomplete resection of follicular variant of PTC. Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/).
1. Introduction Thyroid carcinoma is a common endocrine carcinoma. It is variably typed depend on either types of cells or pattern of the growth. Most common type is papillary thyroid carcinoma (PTC) followed by follicular types carcinoma (FTC). PTC has 3 subtypes depend on the growth pattern such as pure papillary, follicular and mixed. Distant metastasis of malignant tumor to the mandible is a rare condition and account about 1 per cent of all malignant tumor.1 The reported incidence of mandibular metastasis from thyroid carcinomas is rare and estimated approximately also about 1 per cent.1 Because of their bloodstream dissemination, most of them are a consequence of the FTC compare to PTC which metastasizes via lymphatic route.2 2. Case report A 68-year-old female presented with history of right thyroid swelling which was painless and progressively increasing in size for six month. Fine needle aspiration for cytology (FNAC) was suggestive of follicular thyroid neoplasm (FTN). Computed tomography (CT) scan showed aggressive right thyroid mass measuring 9 cm 4 cm 4 cm with superior extension seen till the right angle of mandible and thrombosis of right internal jugular vein. Right hemithyroidectomy was performed and histopathological examination (HPE) reported as follicular variant of PTC. Patient Peer review under responsibility of Egyptian Society of Ear, Nose, Throat and Allied Sciences. ⇑ Corresponding author. E-mail address: [email protected] (S.A.M. Arepen).
then was planned for completion of thyroidectomy but refused and defaulted follow up. She presented again 4 years later with history of left mandibular swelling for one year which rapidly increasing in size within three-month duration (Fig. 1). It was also associated with on and off gum bleeding. Examination showed that patient having hard left mandibular swelling about 6 cm 4 cm and right mandibular swelling 3 cm 2 cm, with no skin changes and non tender on palpation. CT of the neck and thorax showed recurrent thyroid carcinoma with right and left mandibular metastasis (Fig. 2). Thorax and brain parenchyma showed no evidence of distant metastasis. Biopsy taken from the oral cavity showed metastasis from papillary thyroid carcinoma. Then patient underwent completion of thyroidectomy and was referred to our center for continuation of treatment for mandibular metastases. However there are no surgical intervention done because patient refused and patient was started on palliative radiotherapy after was evaluated by oncology and oromaxillofacial team. After completed three cycles radiotherapy, the swelling had slightly reduced and she was planned for another radiotherapy. However she again defaulted follow up. 3. Discussion Thyroid carcinoma is common and making up around one per cent of all cancers.3 About 75 per cent of thyroid cancer in Malaysia occur in female and commonly occur at age of 40–70 years old. Our own local data of thyroid cancers made up 4.9 percent of all cancers and the most common malignancy was PTC which comprises about 76.6%.4 PTC is frequently multifocal and small in size. Primary lesion can present as multinodular appearance or diffused mass with
http://dx.doi.org/10.1016/j.ejenta.2016.12.004 2090-0740/Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Please cite this article in press as: Arepen S.A.M., Mohamad I. Massive bilateral mandibular metastasis from papillary thyroid carcinoma. Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.004
2
S.A.M. Arepen, I. Mohamad / Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx
Fig. 1. (Anterior view) Left mandible swelling extending from left ear lobe up to the right chin measuring about 9 6 cm.
Fig. 2. CT scan axial view showed swelling from both sides of the mandible.
alteration in texture. There are three growth patterns of PTC which are pure papillary, follicular and mixed. Pathologically, PTC cell is oval in shape due to nuclear changes and shows chromatin margination along the nuclear membrane giving a clear nuclear appearance known as ‘‘Orphan Annie” nuclei. PTC usually metastasizes via lymphatic system compare to follicular carcinoma which spread via blood stream. Metastases are usually to ipsilateral regional lymph nodes with rarely having distant metastasis. The reported incidence of mandibular metastasis from thyroid carcinomas is estimated at approximately one percent.1 The most common site involved are ramus and angle regions by producing a poorly defined osteolytic lesion with ragged borders.5 Both of them commonly involved due to high vascularity. Patient may present with swelling over the mandible, loosening and displacement of teeth; rarely, pathologic fracture; minimal of lymph node metastasis; and evidence of hypervascularity often characterized by pulsations and/or an audible bruit on auscultation. FTN has been used to described cytological finding when present of ‘gray-zone’ either benign or malignant cells with undetermined nature. Although FNAC give a 83% of sensitivity, 92% of specificity and a accuracy of 95%, there is still present of
weakness of FNAC in differentiate nodular goiter, follicular adenoma (FA) as well as in well differentiated carcinomas [follicular carcinoma (FC) and follicular variant of papillary thyroid carcinoma (FVPTC)].6 In this case, histological evaluation will help for proper diagnosis. In our case, initial FNAC result came back as follicular neoplasm and HPE after hemithyroidectomy came back as FVPTC. When FNAC reveals an FTN, final diagnosis of FC depends on histological demonstration of capsular and/or vascular invasion; definitive diagnosis of FVPTC depends on a cytological evaluation with a better sample and/or histological diagnosis.7,8 Appropriate surgical management for FTN is still controversial. Some institutions practice hemithyroidectomy and some proceed with total thyroidectomy. The main criteria to decide for hemithyroidectomy were a single nodule, age of less than 45 years old, the absence of thyroiditis or clinical/intraoperative suspicion of malignancy. The increased number of FTN diagnoses has led to more thyroid surgical procedures, demonstrating that malignancy rate associated with FTN is low (10–30%).9,10 Nonetheless, neither preoperative ultrasound (US) features nor molecular markers, nor intra-operative consultation are so accurate to predict malignancy, and, an increased number of diagnostic thyroidectomies have been reported. On the other hand, in several patients who underwent lobectomy, a completion thyroidectomy was required. For the low-risk follicular neoplasm solitary lesions, hemithyroidectomy is still the safest standard of care with lower hospitalization and costs while for multiglandular disease or thyroiditis, that might be associated with a higher risk of cancer, total thyroidectomy should be recommended. Further investigation is warranted to achieve a better preoperative follicular neoplasm diagnostic accuracy in order to reduce the amount of unnecessary surgical operations with a diagnostic aim. In most of the reported cases, for resectable mass over the mandible, the best treatment is the surgical operation with plating and reconstruction and total thyroidectomy at same setting. For this patient, due to large mass over the mandible which needs extensive removal of mandible and patient factor, only palliative treatment with radiotherapy was given to her. In conclusion, thyroid carcinoma with mandibular metastasis is a very rare occurrence. Although FNAC is a main modality used for the diagnosis of a thyroid mass, there still present of limitation for certain cases to determine whether patient having benign or malignant lesion. So that proper evaluation and other supportive investigation can help to decide proper surgical operation for the patient to avoid inadequate treatment.
References 1. Meyer I, Shklar G. Malignant tumors metastatic to mouth and jaws. J Oral Surg. 1965;20:350–362. 2. Ostrosky A, Mareso EA, Klurfan FJ, et al.. Mandibular metastasis of follicular thyroid carcinoma. Case report. Med Oral. 2003;8:224–227. 3. Murray D. The thyroid gland. In: Kovacs L, Asa SL, eds. Functional endocrine pathology. Oxford: Blackwell Science; 1998:295–369. 4. Othman NH, Omar E, Naing NN. Spectrum of thyroid lesions in Hospital Universiti Sains Malaysia over 11 years and a review of thyroid cancers in Malaysia. Asian Pac J Cancer. 2009;10(1):87–90. 5. Kashore Kumar RV, Chakravathy C, Rajasekhar G, et al.. Metastatic thyroid carcinoma presenting as hypervascular lesion of the mandible: A Case report and review of literature. J Oral Maxillofac Surg. 2010;68:2613–2616. 6. Antunes CM, Taveira-Gomes A. Lobectomy in follicular thyroid neoplasms treatment. Int J Surg. 2013;11(9):919–922. 7. Wang TS, Roman SA, Sosa JA. Management of follicular tumors of the thyroid. Minerva Chirurgica. 2007;62:373–382. 8. Sobrinho-Simoes M, Eloy C, Magalhaes J, Lobo C, Amaro T. Follicular thyroid carcinoma. Mod Pathol. 2011;24(suppl. 2):S10–8. 9. Smith J, Cheifetz RE, Schneidereit N, et al.. Can cytology accurately predict benign follicular nodules? Am J Surg. 2005;189(5):592–595. 10. Lee KH, Shin JH, Ko ES, et al.. Predictive factors of malignancy in patients with cytologically suspicious for Hurthle cell neoplasm of thyroid nodules. Int J Surg. 2013;11(9):898–902.
Please cite this article in press as: Arepen S.A.M., Mohamad I. Massive bilateral mandibular metastasis from papillary thyroid carcinoma. Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.004
Contents lists available at ScienceDirect
Egyptian Journal of Ear, Nose, Throat and Allied Sciences journal homepage: www.ejentas.com
Case report
Massive bilateral mandibular metastasis from papillary thyroid carcinoma Siti Asmat Md Arepen ⇑, Irfan Mohamad Department of Otorhinolaryngology-Head & Neck Surgery, School Of Medical Sciences, Universiti Sains Malaysia Health Campus, 16150 Kota Bharu, Kelantan, Malaysia
a r t i c l e
i n f o
Article history: Received 13 June 2016 Accepted 8 December 2016 Available online xxxx Keywords: Papillary thyroid carcinoma Metastasis Mandible
a b s t r a c t Papillary thyroid carcinoma (PTC) comprises almost eighty percent of differentiated thyroid malignancy. It affects female more than male in the age range between 40–50 years old. It usually metastasizes to ipsilateral regional lymph nodes of neck and rarely have distant metastasis. Common route of dissemination of tumour is through lymphatic system. We reported a rare case of bilateral mandibular metastasis from incomplete resection of follicular variant of PTC. Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/).
1. Introduction Thyroid carcinoma is a common endocrine carcinoma. It is variably typed depend on either types of cells or pattern of the growth. Most common type is papillary thyroid carcinoma (PTC) followed by follicular types carcinoma (FTC). PTC has 3 subtypes depend on the growth pattern such as pure papillary, follicular and mixed. Distant metastasis of malignant tumor to the mandible is a rare condition and account about 1 per cent of all malignant tumor.1 The reported incidence of mandibular metastasis from thyroid carcinomas is rare and estimated approximately also about 1 per cent.1 Because of their bloodstream dissemination, most of them are a consequence of the FTC compare to PTC which metastasizes via lymphatic route.2 2. Case report A 68-year-old female presented with history of right thyroid swelling which was painless and progressively increasing in size for six month. Fine needle aspiration for cytology (FNAC) was suggestive of follicular thyroid neoplasm (FTN). Computed tomography (CT) scan showed aggressive right thyroid mass measuring 9 cm 4 cm 4 cm with superior extension seen till the right angle of mandible and thrombosis of right internal jugular vein. Right hemithyroidectomy was performed and histopathological examination (HPE) reported as follicular variant of PTC. Patient Peer review under responsibility of Egyptian Society of Ear, Nose, Throat and Allied Sciences. ⇑ Corresponding author. E-mail address: [email protected] (S.A.M. Arepen).
then was planned for completion of thyroidectomy but refused and defaulted follow up. She presented again 4 years later with history of left mandibular swelling for one year which rapidly increasing in size within three-month duration (Fig. 1). It was also associated with on and off gum bleeding. Examination showed that patient having hard left mandibular swelling about 6 cm 4 cm and right mandibular swelling 3 cm 2 cm, with no skin changes and non tender on palpation. CT of the neck and thorax showed recurrent thyroid carcinoma with right and left mandibular metastasis (Fig. 2). Thorax and brain parenchyma showed no evidence of distant metastasis. Biopsy taken from the oral cavity showed metastasis from papillary thyroid carcinoma. Then patient underwent completion of thyroidectomy and was referred to our center for continuation of treatment for mandibular metastases. However there are no surgical intervention done because patient refused and patient was started on palliative radiotherapy after was evaluated by oncology and oromaxillofacial team. After completed three cycles radiotherapy, the swelling had slightly reduced and she was planned for another radiotherapy. However she again defaulted follow up. 3. Discussion Thyroid carcinoma is common and making up around one per cent of all cancers.3 About 75 per cent of thyroid cancer in Malaysia occur in female and commonly occur at age of 40–70 years old. Our own local data of thyroid cancers made up 4.9 percent of all cancers and the most common malignancy was PTC which comprises about 76.6%.4 PTC is frequently multifocal and small in size. Primary lesion can present as multinodular appearance or diffused mass with
http://dx.doi.org/10.1016/j.ejenta.2016.12.004 2090-0740/Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Please cite this article in press as: Arepen S.A.M., Mohamad I. Massive bilateral mandibular metastasis from papillary thyroid carcinoma. Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.004
2
S.A.M. Arepen, I. Mohamad / Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx
Fig. 1. (Anterior view) Left mandible swelling extending from left ear lobe up to the right chin measuring about 9 6 cm.
Fig. 2. CT scan axial view showed swelling from both sides of the mandible.
alteration in texture. There are three growth patterns of PTC which are pure papillary, follicular and mixed. Pathologically, PTC cell is oval in shape due to nuclear changes and shows chromatin margination along the nuclear membrane giving a clear nuclear appearance known as ‘‘Orphan Annie” nuclei. PTC usually metastasizes via lymphatic system compare to follicular carcinoma which spread via blood stream. Metastases are usually to ipsilateral regional lymph nodes with rarely having distant metastasis. The reported incidence of mandibular metastasis from thyroid carcinomas is estimated at approximately one percent.1 The most common site involved are ramus and angle regions by producing a poorly defined osteolytic lesion with ragged borders.5 Both of them commonly involved due to high vascularity. Patient may present with swelling over the mandible, loosening and displacement of teeth; rarely, pathologic fracture; minimal of lymph node metastasis; and evidence of hypervascularity often characterized by pulsations and/or an audible bruit on auscultation. FTN has been used to described cytological finding when present of ‘gray-zone’ either benign or malignant cells with undetermined nature. Although FNAC give a 83% of sensitivity, 92% of specificity and a accuracy of 95%, there is still present of
weakness of FNAC in differentiate nodular goiter, follicular adenoma (FA) as well as in well differentiated carcinomas [follicular carcinoma (FC) and follicular variant of papillary thyroid carcinoma (FVPTC)].6 In this case, histological evaluation will help for proper diagnosis. In our case, initial FNAC result came back as follicular neoplasm and HPE after hemithyroidectomy came back as FVPTC. When FNAC reveals an FTN, final diagnosis of FC depends on histological demonstration of capsular and/or vascular invasion; definitive diagnosis of FVPTC depends on a cytological evaluation with a better sample and/or histological diagnosis.7,8 Appropriate surgical management for FTN is still controversial. Some institutions practice hemithyroidectomy and some proceed with total thyroidectomy. The main criteria to decide for hemithyroidectomy were a single nodule, age of less than 45 years old, the absence of thyroiditis or clinical/intraoperative suspicion of malignancy. The increased number of FTN diagnoses has led to more thyroid surgical procedures, demonstrating that malignancy rate associated with FTN is low (10–30%).9,10 Nonetheless, neither preoperative ultrasound (US) features nor molecular markers, nor intra-operative consultation are so accurate to predict malignancy, and, an increased number of diagnostic thyroidectomies have been reported. On the other hand, in several patients who underwent lobectomy, a completion thyroidectomy was required. For the low-risk follicular neoplasm solitary lesions, hemithyroidectomy is still the safest standard of care with lower hospitalization and costs while for multiglandular disease or thyroiditis, that might be associated with a higher risk of cancer, total thyroidectomy should be recommended. Further investigation is warranted to achieve a better preoperative follicular neoplasm diagnostic accuracy in order to reduce the amount of unnecessary surgical operations with a diagnostic aim. In most of the reported cases, for resectable mass over the mandible, the best treatment is the surgical operation with plating and reconstruction and total thyroidectomy at same setting. For this patient, due to large mass over the mandible which needs extensive removal of mandible and patient factor, only palliative treatment with radiotherapy was given to her. In conclusion, thyroid carcinoma with mandibular metastasis is a very rare occurrence. Although FNAC is a main modality used for the diagnosis of a thyroid mass, there still present of limitation for certain cases to determine whether patient having benign or malignant lesion. So that proper evaluation and other supportive investigation can help to decide proper surgical operation for the patient to avoid inadequate treatment.
References 1. Meyer I, Shklar G. Malignant tumors metastatic to mouth and jaws. J Oral Surg. 1965;20:350–362. 2. Ostrosky A, Mareso EA, Klurfan FJ, et al.. Mandibular metastasis of follicular thyroid carcinoma. Case report. Med Oral. 2003;8:224–227. 3. Murray D. The thyroid gland. In: Kovacs L, Asa SL, eds. Functional endocrine pathology. Oxford: Blackwell Science; 1998:295–369. 4. Othman NH, Omar E, Naing NN. Spectrum of thyroid lesions in Hospital Universiti Sains Malaysia over 11 years and a review of thyroid cancers in Malaysia. Asian Pac J Cancer. 2009;10(1):87–90. 5. Kashore Kumar RV, Chakravathy C, Rajasekhar G, et al.. Metastatic thyroid carcinoma presenting as hypervascular lesion of the mandible: A Case report and review of literature. J Oral Maxillofac Surg. 2010;68:2613–2616. 6. Antunes CM, Taveira-Gomes A. Lobectomy in follicular thyroid neoplasms treatment. Int J Surg. 2013;11(9):919–922. 7. Wang TS, Roman SA, Sosa JA. Management of follicular tumors of the thyroid. Minerva Chirurgica. 2007;62:373–382. 8. Sobrinho-Simoes M, Eloy C, Magalhaes J, Lobo C, Amaro T. Follicular thyroid carcinoma. Mod Pathol. 2011;24(suppl. 2):S10–8. 9. Smith J, Cheifetz RE, Schneidereit N, et al.. Can cytology accurately predict benign follicular nodules? Am J Surg. 2005;189(5):592–595. 10. Lee KH, Shin JH, Ko ES, et al.. Predictive factors of malignancy in patients with cytologically suspicious for Hurthle cell neoplasm of thyroid nodules. Int J Surg. 2013;11(9):898–902.
Please cite this article in press as: Arepen S.A.M., Mohamad I. Massive bilateral mandibular metastasis from papillary thyroid carcinoma. Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.004