Abstract
Background
Familial papillary thyroid microcarcinoma (FPTMC) appears to be more aggressive than sporadic papillary thyroid microcarcinoma (SPTMC). However, there are authors who indicate that unicentric FPTMC has a similar prognosis to SPTMC. The objective is to analyze whether unicentric FPTMC has a better prognosis than multicentric FPTMC.
Design and methods
Type of study: National multicenter longitudinal analytical observational study. Study population: Patients with FPTMC. Study groups: Two groups were compared: Group A (unicentric FPTMC) vs. Group B (multicentric FPTMC). Study variables: It is analyzed whether between the groups there are: a) differentiating characteristics; and b) prognostic differences. Statistical analysis: Cox regression analysis and survival analysis.
Results
Ninety-four patients were included, 44% (n = 41) with unicentric FPTMC and 56% (n = 53) with multicentric FPTMC. No differences were observed between the groups according to socio-familial, clinical or histological variables. In the group B a more aggressive treatment was performed, with higher frequency of total thyroidectomy (99 vs. 78%; p = 0.003), lymph node dissection (41 vs. 15%; p = 0.005) and therapy with radioactive iodine (96 vs. 73%; p = 0.002). Tumor stage was similar in both groups (p = 0.237), with a higher number of T3 cases in the group B (24 vs. 5%; p = 0.009). After a mean follow-up of 90 ± 68.95 months, the oncological results were similar, with a similar disease persistence rate (9 vs. 5%; p = 0.337), disease recurrence rate (21 vs. 8%; p = 0.159) and disease-free survival (p = 0.075).
Conclusions
Unicentric FPTMC should not be considered as a SPTMC due to its prognosis is similar to multicentric FPTMC.
Graphical Abstract
Similar content being viewed by others
Data availability
The data underlying this article will be shared on reasonable request to the corresponding author (Dr. Ríos).
References
A. Miyauchi, Clinical trials of active surveillance of papillary microcarcinoma of the thyroid. World J. Surg. 40, 516–22 (2016). https://doi.org/10.1007/s00268-015-3392-y
A. Ríos, J.M. Rodríguez, D. Navas, A. Cepero, N.M. Torregrosa, M.D. Balsalobre et al. Family screening in familial papillary carcinoma: the early detection of thyroid disease. Ann. Surg. Oncol. 23, 2564–70 (2016). https://doi.org/10.1245/s10434-016-5149-8
M. Capezzone, C. Secchi, N. Fralassi, S. Cantara, L. Brilli, C. Ciuoli et al. Should familial disease be considered as a negative prognostic factor in micropapillary thyroid carcinoma? J. Endocrinol. Invest 42, 1205–13 (2019). https://doi.org/10.1007/s40618-019-01039-0
E.L. Maxwell, F.T. Hall, J.L. Freeman, Familial non‐medullary thyroid cancer: a matched‐case control study. Laryngoscope 114, 2182–6 (2004). https://doi.org/10.1097/01.mlg.0000149454.91005.65
V. Nosé, Familial thyroid cancer: a review. Mod. Pathol. 24, S19–33 (2011). https://doi.org/10.1038/modpathol.2010.147
S. Uchino, S. Noguchi, H. Kawamoto, H. Yamashita, S. Watanabe, H. Yamashita et al. Familial nonmedullary thyroid carcinoma characterized by multifocality and a high recurrence rate in a large study population. World J. Surg. 26, 897–902 (2002). https://doi.org/10.1007/s00268-002-6615-y
R.S. Sippel, N.R. Caron, O.H. Clark, An evidence-based approach to familial nonmedullary thyroid cancer: screening, clinical management, and follow-up. World J. Surg. 31, 924–33 (2007). https://doi.org/10.1007/s00268-006-0847-1
X. Wu, B. Li, C. Zheng, X. He, Predicting factors of central lymph node metastases in patients with unilateral multifocal papillary thyroid microcarcinoma. Gland Surg. 9, 695–701 (2020). https://doi.org/10.21037/gs.2020.03.27
A.R. Shaha, V.V. Poorten, R.M. Tuttle, Multifocality in papillary thyroid carcinoma-An unresolved controversy. Eur. J. Surg. Oncol. 46, 1777–8 (2020). https://doi.org/10.1016/j.ejso.2020.06.026
J.W. Feng, Z. Qu, A.C. Qin, H. Pan, J. Ye, Y. Jiang, Significance of multifocality in papillary thyroid carcinoma. Eur. J. Surg. Oncol. 46, 1820–8 (2020). https://doi.org/10.1016/j.ejso.2020.06.015
C.R. Lee, S. Park, S.W. Kang, J. Lee, J.J. Jeong, K.H. Nam et al. Is familial papillary thyroid microcarcinoma more aggressive than sporadic form? Ann. Surg. Treat. Res 92, 129–35 (2017). https://doi.org/10.4174/astr.2017.92.3.129
G. Lupoli, G. Vitale, M. Caraglia, M.R. Fittipaldi, A. Abbruzzese, P. Tagliaferri et al. Familial papillary thyroid microcarcinoma: a new clinical entity. Lancet 353, 637–9 (1999). https://doi.org/10.1016/S0140-6736(98)08004-0
S. Lei, D. Wang, J. Ge, H. Liu, D. Zhao, G. Li, Z. Ding, Single-center study of familial papillary thyroid cancer in China: surgical considerations. World J. Surg. Oncol. 13, 115 (2015). https://doi.org/10.1186/s12957-015-0519-4
T.Y. Sung, Y.M. Lee, J.H. Yoon, K.W. Chung, S.J. Hong, Surgical management of familial papillary thyroid microcarcinoma: a single institution study of 94 cases. World J. Surg. 39, 1930–5 (2015). https://doi.org/10.1007/s00268-015-3064-y
J. Cao, C. Chen, C. Chen, Q.L. Wang, M.H. Ge, Clinicopathological features and prognosis of familial papillary thyroid carcinoma–a large-scale, matched, case-control study. Clin. Endocrinol. 84, 598–606 (2016). https://doi.org/10.1111/cen.12859
B.R. Haugen, E.K. Alexander, K.C. Bible, G.M. Doherty, S.J. Mandel, Y.E. Nikiforov et al. 2015 American thyroid association management guidelines for adult patients with thyroid nodules and differentiated thyroid cancer: the American thyroid association guidelines task force on thyroid nodules and differentiated thyroid cancer. Thyroid 26, 1–133 (2016). https://doi.org/10.1089/thy.2015.0020
K.R. Joseph, S. Edirimanne, G.D. Eslick, Multifocality as a prognostic factor in thyroid cancer: a meta-analysis. Int J. Surg. 550, 121–5 (2018). https://doi.org/10.1016/j.ijsu.2017.12.035
F. Wang, X. Yu, X. Shen, G. Zhu, Y. Huang, R. Liu, D. Viola, R. Elisei, E. Puxeddu, L. Fugazzola, C. Colombo, B. Jarzab, A. Czarniecka, A.K. Lam, C. Mian, F. Vianello, L. Yip, G. Riesco-Eizaguirre, P. Santisteban, C.J. O’Neill, M.S. Sywak, R. Clifton-Bligh, B. Bendlova, V. Sýkorová, Y. Wang, S. Liu, J. Zhao, S. Zhao, M. Xing, The prognostic value of tumor multifocality in clinical outcomes of papillary thyroid cancer. J. Clin. Endocrinol. Metab. 102, 3241–3250 (2017). https://doi.org/10.1210/jc.2017-00277
V. Harries, L.Y. Wang, M. McGill, B. Xu, R.M. Tuttle, R.J. Wong et al. Should multifocality be an indication for completion thyroidectomy in papillary thyroid carcinoma? Surgery 167, 10–17 (2020). https://doi.org/10.1016/j.surg.2019.03.031
A. La Greca, B. Xu, R. Ghossein, R.M. Tuttle, M.M. Sabra, Patients with multifocal macroscopic papillary thyroid carcinoma have a low risk of recurrence at early follow-up after total thyroidectomy and radioactive iodine treatment. Eur. Thyroid J. 6, 31–9 (2017). https://doi.org/10.1159/000448752
N. Qu, L. Zhang, Q.H. Ji, Y.X. Zhu, X.Y. Wang, Q. Shen et al. Number of tumor foci predicts prognosis in papillary thyroid cancer. BMC Cancer 14, 914–919 (2014). https://doi.org/10.1186/1471-2407-14-914
Z. Lu, J. Sheng, Y. Zhang, J. Deng, Y. Li, A. Lu et al. Clonality analysis of multifocal papillary thyroid carcinoma by using genetic profiles. J. Pathol. 239, 72–83 (2016). https://doi.org/10.1002/path.4696
A. Al Afif, B.A. Williams, M.H. Rigby, M.J. Bullock, S.M. Taylor, J. Trites et al. Multifocal papillary thyroid cancer increases the risk of central lymph node metastasis. Thyroid 25, 1008–12 (2015). https://doi.org/10.1089/thy.2015.0130
W.R. Choi, J.L. Roh, G. Gong, K.J. Cho, S.H. Choi, S.Y. Nam et al. Multifocality of papillary thyroid carcinoma as a risk factor for disease recurrence. Oral. Oncol. 94, 106–110 (2019). https://doi.org/10.1016/j.oraloncology.2019.05.023
Y. Ito, A. Miyauchi, H. Oda, Low-risk papillary microcarcinoma of the thyroid: a review of active surveillance trials. EJSO 44, 307–315 (2018). https://doi.org/10.1016/j.ejso.2017.03.004
Y.W. Jeon, H.G. Gwak, S.T. Lim, J. Schneider, Y.J. Suh, Long-term prognosis of unilateral and multifocal papillary thyroid microcarcinoma after unilateral lobectomy versus total thyroidectomy. Ann. Surg. Oncol. 26, 2952–2958 (2019). https://doi.org/10.1245/s10434-019-07482-w
J.F. Lin, P.K.C. Jonker, M. Cunich, S.B. Sidhu, L.W. Delbridge, A.R. Glover et al. Surgery alone for papillary thyroid microcarcinoma is less costly and more effective than long term active surveillance. Surgery 167, 110–116 (2020). https://doi.org/10.1016/j.surg.2019.05.078
B. Saravana-Bawan, A. Bajwa, J. Paterson, T. McMullen, Active surveillance of low-risk papillary thyroid cancer: a meta-analysis. Surgery 167, 46–55 (2020). https://doi.org/10.1016/j.surg.2019.03.040
D.I. Stefanova, A. Bose, T.M. Ullmann, J.N. Limberg, B.M. Finnerty, R. Zarnegar et al. Does the ATA risk stratification apply to patients with papillary thyroid microcarcinoma? World J. Surg. 44, 452–460 (2020). https://doi.org/10.1007/s00268-019-05215-4
X. Wang, W. Cheng, J. Li, A. Su, T. Wei, F. Liu et al. Endocrine tumours: familial nonmedullary thyroid carcinoma is a more aggressive disease: a systematic review and meta-analysis. Eur. J. Endocrinol. 172, R253–R262 (2015). https://doi.org/10.1530/EJE-14-0960
Acknowledgements
Thank the Spanish Association of Surgeons and the Endocrine Surgery Section for their support in carrying out this work. Thank the 30 Spanish Endocrine Surgery Units for having collaborated in the realization of this project and for having provided the data.
Author contributions
Authorship: Contribution to study of the Authors: 1) Conception and design: A.R. 2) Acquisition of a subtantial portion of data: A.R., J.R.-P., A.B.-R., J.A.P., P.M, E.M., E.F, M.A.M., J.M., M.D., J.M.B., D.C., M.P.S.E., N.M.T., A.E.R., G.M.F., A.M.C., O.V., F.H., G.R.-M., J.M.R. 3) Analysis and interpretation of data: A.R., J.R.-P., A.B.-R., J.A.P., P.M., E.M., E.F., M.A.M., J.M., M.D., J.M.B., D.C., M.P.S.-E., N.M.T., A.E.-R., G.M.-F., A.M.C., O.V., F.H., G.R.-M., J.M.R. 4) Drafting of the manuscript: A.R. 5) Critical revision of the manuscript for important intellectual content: A.R., J.R.-P., A.B.-R., J.A.P., P.M., E.M., E.F., M.A.M., J.M., M.D., J.M.B., D.C., M.P.S.-E., N.M.T., A.E.-R., G.M.-F., A.M.C., O.V., F.H., G.R.-M., J.M.R. 6) Statistical expertise: A.R., G.R.-M. 7) Obtaining funding for this project or study: A.R. 8) Supervision: A.R. 9) Final approval of the version to be published: A.R., J.R.-P., A.B.-R., J.A.P., P.M., E.M., E.F., M.A.M., J.M., M.D., J.M.B., D.C., M.P.S.-E., N.M.T., A.E.-R., G.M.-F., A.M.C., O.V., F.H., G.R.-M., J.M.R. 10) Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved: A.R., J.R.-P., A.B.-R., J.A.P., P.M., E.M., E.F., M.A.M., J.M., M.D., J.M.B., D.C., M.P.S.-E., N.M.T., A.E.-R., G.M.-F., A.M.C., O.V., FH., G.R.-M., J.M.R.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare no competing interests.
Ethical approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. The study protocol was approved by the committee of the Virgen de la Arrixaca University Hospital. Ethics Committee Code: 2021-2-13-HCUVA.
Additional information
Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
Ríos, A., Ruiz-Pardo, J., Balaguer-Román, A. et al. Is unicentric familial papillary thyroid microcarcinoma different from multicentric?. Endocrine 82, 613–621 (2023). https://doi.org/10.1007/s12020-023-03455-y
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12020-023-03455-y