The first case of papillary thyroid carcinoma in an adolescent with congenital dyshormonogenetic hypothyroidism in Serbia

Jelena Eremija; Tatjana Milenković; Katarina Mitrović; Sladjana Todorović; Rade Vuković; Ljiljana Plavšić

doi:10.2298/3860

Jelena Eremija Department of Endrocrinology, Institute for Mother and Child Health Care of Serbia „Dr Vukan Čupić“, Belgrade, Serbia
Tatjana Milenković Department of Endrocrinology, Institute for Mother and Child Health Care of Serbia „Dr Vukan Čupić“, Belgrade, Serbia
Katarina Mitrović Department of Endrocrinology, Institute for Mother and Child Health Care of Serbia „Dr Vukan Čupić“, Belgrade, Serbia
Sladjana Todorović Department of Endrocrinology, Institute for Mother and Child Health Care of Serbia „Dr Vukan Čupić“, Belgrade, Serbia
Rade Vuković Department of Endrocrinology, Institute for Mother and Child Health Care of Serbia „Dr Vukan Čupić“, Belgrade, Serbia
Ljiljana Plavšić Center for Family Planning, Institute for Mother and Child Health Care of Serbia „Dr Vukan Čupić“, Belgrade, Serbia

DOI: https://doi.org/10.2298/3860

Keywords: congenital hypothyroidism, thyroid neoplasms, carcinoma, papillary, thiroidectomy, child, treatment outcome, serbia,

Abstract

Introduction. Differentiated thyroid carcinoma (DTC) is a rare childhood malignancy, as it represents 0.3–0.4% of pediatric malignancies. Papillary carcinoma is the most common type of pediatric DTC and it represents about 90% of all DTC patients. Although rare, DTC arising from dyshormonogenetic goiter is the most serious complication of congenital hypothyroidism. Case report. We presented the development of thyroid papillary carcinoma in a 15-year-old girl diagnosed with congenital dyshormonogenetic hypothyroidism at neonatal age. Considering the early initiation and proper dosage of hormonal substitution, normal levels of thyreotropin and thyroid hormones were achieved quickly and maintained through a follow-up period. The girl remained euthyroid and asymptomatic until 13.8 years of age, when she presented with a large multinodular goiter. The patient underwent total thyroidectomy. Pathological examination revealed intrathyroid microcarcinoma in the right lobe. Conclusion. Although differentiated thyroid carcinoma is a rare pediatric malignancy, it is of great importance to have a certain degree of clinical caution and provide a multidisciplinary approach during the follow-up of patients with dyshormonogenetic hypothyroidism.

References

Brink JS, van Heerden JA, McIver B, Salomao DR, Farley DR, Grant CS, et al. Papillary thyroid cancer with pulmonary metastases in children: long-term prognosis. Surgery 2000; 128(6): 881−7.

Alkan S, Seven H, Sakalli E, Dadaş B. Papillary thyroid carci-noma in a 3-year-old child: case report. Int J Pediatr Otorhinolaryngol 2008; 72(2): 275−7.

Shapiro NL, Bhattacharyya N. Population-based outcomes for pediatric thyroid carcinoma. Laryngoscope 2005; 115(2): 337−40.

Srikumar S, Agada FO, Picton SV Squire R, Knight LC. Papillary carcinoma of the thyroid in a 2-year-old: case report with re-view of the literature. Int J Pediatr Otorhinolaryngol 2006; 1(4): 274−8.

Fagin JA. Familial nonmedullary thyroid carcinoma: The case for genetic susceptibility. J Clin Endocrinol Metab 1997; 82(2): 342−4.

Kikuchi S, Perrier ND, Ituarte P, Siperstein AE, Duh Q, Clark OH. Latency period of thyroid neoplasia after radiation exposure. Ann Surg 2004; 239(4): 536−43.

Millman B, Pellitteri PK. Thyroid carcinomain children and ado-lescents. Arch Otolaryngol Head Neck Sur 1995; 121(11): 1261−4.

Klopper JP, McDermott MT. Palpable pediatric thyroid abnormal-ities-diagnostic pitfalls necessitate a high index of clinical sus-picion: a case report. J Med Case Reports 2007; 1: 29.

Leboulleux S, Baudin E, Hartl DW, Travagli J, Schlumberger M. Follicular cell-derived thyroid cancer in children. Horm Res 2005; 63(3): 145−51.

Segal K, Shvero J, Stern Y, Mechlis S, Feinmesser R. Surgery of thy-roid cancer in children and adolescents. Head Neck 1998; 20(4): 293−7.

Grigsby PW, Galor A, Michalski JM, Doherty GM. Childhood and adolescent thyroid carcinoma. Cancer 2002; 95(4): 724−9.

Kumar A, Bal CS. Differentiated thyroid cancer. Indian J Pe-diatr 2003; 70(9): 707−13.

Poddar S, Basu S, Majumder A. Papillary carcinoma of thyroid in a 11 months old child. Indian J Med Paediatr Oncol 2008; 29(2): 33−5.

La Quaglia MP, Black T, Holcomb GW 3rd, Sklar C, Azizkhan RG, Haase GM, et al. Differentiated thyroid cancer: clinical characteristics, treatment, and outcome in patients under the 21 years of age who presented with distant metastases. A re-port from the surgical Committee of Children's Cancer Group. J Ped Surg 2000; 35(6): 955−9, discussion 960.

Spinelli C, Bertocchini A, Antonelli A, Miccoli P. Surgical therapy of the thyroid papillary carcinoma in children: experience with 56 patients < or =16 years old. J Pediatr Surg 2004; 39(10): 1500−5.

Kebebew E. Hereditary non-medullary thyroid cancer. World J Surg 2008; 32(5): 678−82.

Medeiros-Neto G, Stanbury JB. Thyroid malignancy and dyshor-monogenetic goiter. In: Medeiros-Neto G, Stanbury JB, editors. Inherited disorders of the thyroid system. Boca Raton, FL: CRC Press; 1994. p. 207−18.

Cooper DS, Axelrod L, DeGroot LJ, Vickery AL, Maloof F. Con-genital goiter and the development of metastatic follicular car-cinoma with evidence for a leak of nonhormonal iodide: clini-cal, pathological, kinetic, and biochemical studies and a review of the literature. J Clin Endocrinol Metab 1981; 52(2): 294−306.