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      Thyroid metastasis presenting as backache and lower limb weakness without any primary tumor - a case report

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            Abstract

            Background:

            Differentiated thyroid cancer is the commonest endocrine malignancy. Mortality in the presence of distant metastasis can increase dramatically. Bony metastasis often leads to increased morbidity and mortality. We report a case of a 60-year-old male who had metastatic spread of follicular cancer of the thyroid, without any identifiable primary.

            Case Presentation:

            A 60-year-old male presented with backache and weakness of lower limbs due to the collapse of the 11th thoracic vertebra. He underwent laminectomy and internal fixation. The histopathology revealed that it was the metastatic spread of follicular cancer of the thyroid. The histopathology revealed no malignancy in the thyroidectomy specimen. Subsequent administration of radioactive iodine showed a fall in thyroglobulin from 6,000 to 203 ng/ml.

            Conclusion:

            In cases of metastasis from an unknown primary, follicular thyroid cancer should be included in the differential diagnosis. Treatment after thyroidectomy can lead to good results.

            Main article text

            Background

            Although a rare type of cancer, differentiated thyroid cancer is the commonest endocrine malignancy [1-3]. It usually has a rather indolent course; however, the patients can present with complications such as the distant spread of the disease [4]. Distant spread of thyroid cancer has been reported in the literature to be anywhere between 2% and 20% [5,6]. Mortality in the presence of distant metastasis can increase dramatically with a 10-year survival rate as low as 40%, as compared to 80%-95% in patients with non-metastasized disease [5,7,8]. Common sites of metastasis include the lungs and bones [9,10]. Bony metastasis often leads to increased morbidity and mortality [9,11]. We report an unusual case of a 60-year-old male who presented with backache and weakness of lower limbs due to the collapse of the 11th thoracic vertebra, which proved to be due to metastatic spread of follicular cancer of the thyroid, without any primary tumor in the thyroid.

            Case Report

            A 60-year-old male with back pain and inability to walk presented to a neurologist. An magnetic resonance imaging (MRI) was advised that revealed a partial collapse of the 11th thoracic vertebra with cord compression and severe right and moderate left foramen stenosis (Figure 1). The adjacent intervertebral heights were intact. The differential diagnosis included metastasis, myeloma, and lymphoma. He underwent laminectomy and internal fixation. The histopathology revealed glandular architecture lined by stratified columnar cells. There was moderate nuclear pleomorphism and hyperchromasia. Immunohistochemistry was positive for thyroglobulin (Tg), TTF1, and CK-7, thus confirming that it was metastatic spread of follicular cancer of the thyroid (Figure 2). To relieve the patient’s symptoms 20 Gy radiation was delivered in 5 fractions to 10th-12th thoracic vertebrae. An ultrasound and computed tomography (CT) of the neck were performed, which failed to reveal anything unusual in the thyroid. A CT chest was performed which revealed no pulmonary metastasis, while the bone scan revealed uptake only in the 11th thoracic vertebra (Figure 3). Subsequently, the patient underwent a total thyroidectomy. The histopathology revealed no malignancy in the thyroidectomy specimen. Tg level was in excess of 6,000 ng/ml (normal < 50 ng/ml), while the anti-Tg antibody (Anti-Tg) level was 10 IU/ml (normal 29 IU/ml). After discussion in the multidisciplinary clinic, it was decided that a dose of 7.4 GBq (200 mCi) of radioactive iodine (RAI) (I-131) be given to the patient. A post-therapy scan was performed which revealed uptake in the thyroid bed, with metastasis in the region of the lower thoracic vertebral region (Figure 4). Post-RAI Tg levels came down to 408 ng/ml. Six months later repeat blood test revealed Tg levels of 203 ng/ml (Anti-Tg = 0.02 IU/ml). A repeat of Tg levels at 1 year revealed an increase to 250 ng/ml (Anti-Tg = 2.8 IU/ml). It was decided that another dose of 7.4 MBq of RAI be given to the patient. The whole-body scan revealed uptake in the surgical site, with no other focus of abnormal tracer uptake noted (Figure 5). The post-second dose Tg decreased slightly to 236 ng/ml (Anti-Tg = 3.07 IU/ml) (Table 1).

            Table 1.
            A brief overview of the case report.
            The patient presented to a neurologist Back pain and inability to walk
            MRI Partial collapse of 11th thoracic vertebra with cord compression
            Referred to neurosurgeonLaminectomy plus internal fixation
            HistopathologyMetastatic follicular ca thyroid
            Radiotherapy20 Gy given to 10-12th thoracic vertebrae
            Ultrasound and CT neckNo lesion detected in thyroid
            Bone scanUptake in 11th thoracic vertebra
            Patient underwent total thyroidectomyNo malignancy found on histopathology
            Tg levels measured>6,000 ng/ml
            RAI administered7.4 GBq
            Whole body scanUptake in thyroid bed and the lower thoracic vertebral region
            Tg levels measured408 ng/ml
            Repeat dose of I-131 after 1 year7.4 GBq
            Whole body scanUptake in the region of the lower thoracic vertebra only
            Tg levels measured236 ng/ml
            Figure 1.

            MRI of the patient revealing collapse of 11th thoracic vertebra (arrow).

            Figure 2.

            Immunohistochemistry staining showing Tg positivity (left panel) and TTF-1 staining (right panel) in the laminectomy sample.

            Figure 3.

            99mTc-MDP whole body bone scan showing intensely increased tracer uptake in the 11th thoracic vertebra.

            Discussion

            Nearly a third of patients with cancer have a distant spread of the disease [12]. As many as 3%-4% of patients with bone metastasis have no identifiable primary tumor [13]. We report a case of metastasis of follicular cancer of the thyroid to the spine. However, when the thyroid gland was removed and histopathology performed, no primary tumor was discovered. In the review of the literature, one case series of seven cases of metastatic papillary thyroid cancer was found where no primary tumor could be found [14]. Boz et al. [15] have also reported a case of metastatic follicular cancer of the thyroid without an identifiable primary. Akdemir et al. [10] have reported a case where there were skeletal metastases in a patient without any primary being detected in the thyroid. In a 2012 study published in the Australia and New Zealand Journal of Surgery, regarding papillary thyroid cancer patients, three patients were reported to have distant metastasis without any discernible primary lesion [8]. Meanwhile, Anastasilakis et al. have reported a patient in whom they found lymph node metastasis of papillary cancer without any discernible primary [16].

            Figure 4.

            Post-ablation scan after administration of the first dose of 7.4 GBq of I-131 showing uptake in the thyroid bed (arrowhead) and the region of the lower thoracic vertebrae (outline arrow).

            Figure 5.

            Post-ablation scan after the second dose of 7.4 GBq of I-131 still revealing uptake in the operation site.

            It is rare to have a metastatic spread of the disease in follicular thyroid cancer without a primary. The lack of the detectable primary may be because of the spontaneous regression of the primary lesion.

            Conclusion

            This case report highlights that in cases of metastasis from an unknown primary, follicular thyroid cancer should be included in the differential diagnosis. Also, even if a primary is not found in the thyroid, total thyroidectomy with post-surgical administration of RAI can lead to a good prognosis as measured by the serum Tg levels.

            What is new?

            Follicular thyroid cancer must be a differential in the case of metastasis of unknown origin. There may be instances where a primary may not be found in the thyroid upon total thyroidectomy. Administration of RAI in such cases can lead to a good prognosis.

            List of Abbreviations

            Anti-Tg

            Anti-thyroglobulin antibodies

            CT

            Computed tomography

            GBq

            Gigabecquerels

            MBq

            Megabecquerels

            MRI

            Magnetic resonance imaging

            RAI

            Radioactive iodine

            Tg

            Thyroglobulin

            Conflict of interest

            The authors declare that there is no conflict of interest regarding the publication of this article.

            Funding

            This research did not receive any funding or grant.

            Consent for publication

            A written informed consent to publish/present this case was obtained from the patient.

            Ethical approval

            Ethical approval is not required at our institutions to publish an anonymous case report.

            References

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            2. Kilfoy BA, Zheng T, Holford TR, Han X, Ward MH, Sjodin A, et al.. International patterns and trends in thyroid cancer incidence, 1973-2002. Cancer Causes Control. 2009. Vol. 20(5):525–31. [Cross Ref]

            3. Rubab N, Afzal M, Shahbaz M, Imran M. Complete remission after primary single dose of radioactive iodine in metastatic papillary thyroid carcinoma: a case report. Eur J Med Case Rep. 2021. 345–9. [Cross Ref]

            4. Choksi P, Papaleontiou M, Guo C, Worden F, Banerjee M, Haymart M. Skeletal complications and mortality in thyroid cancer: A population-based study. J Clin Endocrinol Metab. 2017. Vol. 102(4):1254–1260. [Cross Ref]

            5. Benbassat CA, Mechlis-Frish S, Hirsch D. Clinicopathological characteristics and long-term outcome in patients with distant metastases from differentiated thyroid cancer. World J Surg. 2006. Vol. 30(6):1088–95. [Cross Ref]

            6. Chen D, Huang L, Chen S, Huang Y, Hu D, Zeng W, et al.. Innovative analysis of distant metastasis in differentiated thyroid cancer. Oncol lett. 2020. Vol. 19(3):1985–92. [Cross Ref]

            7. Ramadan S, Ugas MA, Berwick RJ, Notay RJ, Cho H, Jerjes W, et al.. Spinal metastasis in thyroid cancer. Head Neck Oncol. 2012. Vol. 4(1):1–19. [Cross Ref]

            8. Ban EJ, Andrabi A, Grodski S, Yeung M, McLean C, Serpell J. Follicular thyroid cancer: minimally invasive tumours can give rise to metastases. ANZ J Surg. 2012. Vol. 82(3):136–9. [Cross Ref]

            9. O’Sullivan GJ, Carty FL, Cronin CG. Imaging of bone metastasis: an update. World J Radiol. 2015. Vol. 7(8):202–11. [Cross Ref]

            10. Akdemir I, Erol FS, Akpolat N, Ozveren MF, Akfirat M, Yahsi S. Skull metastasis from thyroid follicular carcinoma with difficult diagnosis of the primary lesion. Neurol Med Chir (Tokyo). 2005. Vol. 45(4):205–8. [Cross Ref]

            11. Kallel F, Hamza F, Charfeddine S, Amouri W, Jardak I, Ghorbel A, et al.. Clinical features of bone metastasis for differentiated thyroid carcinoma: a study of 21 patients from a Tunisian center. Indian J Endocrinol Metab. 2014. Vol. 18(2):185–90. [Cross Ref]

            12. Tomuleasa C, Zaharie F, Muresan M-S, Pop L, Fekete Z, Dima D, et al.. How to diagnose and treat a cancer of unknown primary site. J Gastrointestin Liver Dis. 2017. Vol. 26(1):69–79. [Cross Ref]

            13. Hage WD, Aboulafia AJ, Aboulafia DM. Incidence, location, and diagnostic evaluation of metastatic bone disease. Orthope Clin North Am. 2000. Vol. 31(4):515–28. [Cross Ref]

            14. Xu B, Scognamiglio T, Cohen PR, Prasad ML, Hasanovic A, Tuttle RM, et al.. Metastatic thyroid carcinoma without identifiable primary tumor within the thyroid gland: a retrospective study of a rare phenomenon. Hum Pathol. 2017. Vol. 65:133–9. [Cross Ref]

            15. Boz A, Tazegul G, Bozoglan H, Dogan O, Sari R, Altunbas HA, et al.. Bone metastases without primary tumor: A well-differentiated follicular thyroid carcinoma case. J Cancer Res Thera. 2018. Vol. 14(2):447–50. [Cross Ref]

            16. Anastasilakis AD, Polyzos SA, Makras P, Kampas L, Valeri RM, Dimitrios K, et al.. Papillary thyroid microcarcinoma presenting as lymph node metastasis--a diagnostic challenge: case report and systematic review of literature. Hormone (Athens). 2012. Vol. 11(4):419–27. [Cross Ref]

            Summary of the case

            1 Patient (gender, age) Male, 60 years
            2 Final diagnosis Metastatic papillary ca thyroid
            3 Symptoms Backache and inability to walk
            4 Medications RAI
            5 Clinical procedure Surgery
            6 Specialty Nuclear Medicine

            Author and article information

            Journal
            European Journal of Medical Case Reports
            EJMCR
            Discover STM Publishing Ltd.
            2520-4998
            30 June 2023
            : 7
            : 6
            : 125-129
            Affiliations
            [1 ]Department of Nuclear Medicine, KIRAN, Karachi, Pakistan
            [2 ]Department of Nuclear Medicine, GINUM, Gujranwala, Pakistan
            Author notes
            [* ] Correspondence to: Javaid Iqbal Department of Nuclear Medicine, KIRAN, Karachi, Pakistan. drjiqbal@ 123456hotmail.com
            Article
            ejmcr-7-125
            10.24911/ejmcr/173-1673679828
            5c882183-5788-413d-a501-4df9e3806291
            © Javaid Iqbal, Basit Iqbal, Talal A. Rahman, Salman Habib, Hasnain Dilawar, Imran Hadi, Akhtar Ahmad

            This is an open access article distributed in accordance with the Creative Commons Attribution (CC BY 4.0) license: https://creativecommons.org/licenses/by/4.0/) which permits any use, Share — copy and redistribute the material in any medium or format, Adapt — remix, transform, and build upon the material for any purpose, as long as the authors and the original source are properly cited.

            History
            : 14 January 2023
            : 20 July 2023
            Categories
            CASE REPORT

            case report,metastasis of unknown origin,unknown primary,follicular cancer,Differentiated thyroid cancer

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