Thyroid cancer in children: A multicenter international study highlighting clinical features and surgical outcomes of primary and secondary tumors

Cristina Martucci; Alessandro Crocoli; Maria Debora De Pasquale; Claudio Spinelli; Silvia Strambi; Paolo Brazzarola; Eleonora Morelli; Jessica Cassiani; Juliana Mancera; Juan Pablo Luengas; Pablo Lobos; Daniel Liberto; Estefanìa Astori; Sabine Sarnacki; Vincent Couloigner; François Simon; Cassandre Lambert; Simone de Campos Vieira Abib; Onivaldo Cervantes; Eliana Caran; Diana Delgado Lindman; Matthew O Jones; Rajeev Shukla; Paul D Losty; Alessandro Inserra

doi:10.3389/fped.2022.914942

Thyroid cancer in children: A multicenter international study highlighting clinical features and surgical outcomes of primary and secondary tumors

Front Pediatr. 2022 Jul 22:10:914942. doi: 10.3389/fped.2022.914942. eCollection 2022.

Authors

Cristina Martucci¹, Alessandro Crocoli¹, Maria Debora De Pasquale², Claudio Spinelli³, Silvia Strambi³, Paolo Brazzarola⁴, Eleonora Morelli⁴, Jessica Cassiani⁵, Juliana Mancera⁶, Juan Pablo Luengas⁷, Pablo Lobos⁸, Daniel Liberto⁸, Estefanìa Astori⁸, Sabine Sarnacki⁹, Vincent Couloigner¹⁰, François Simon¹⁰, Cassandre Lambert¹⁰, Simone de Campos Vieira Abib¹¹, Onivaldo Cervantes¹², Eliana Caran¹¹, Diana Delgado Lindman¹¹, Matthew O Jones¹³, Rajeev Shukla¹⁴, Paul D Losty¹⁵, Alessandro Inserra¹

Affiliations

¹ Department of Pediatric Surgery, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.
² Department of Pediatric Hematology/Oncology Cell and Gene Therapy, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.
³ Department of Pediatric Surgery, University of Pisa, Pisa, Italy.
⁴ Department of Surgery and Oncology, University and Hospital Trust of Verona, Verona, Italy.
⁵ Department of Hepatobiliary Surgery, University of Verona, Verona, Italy.
⁶ Department of Pediatric Surgery, Universidad Militar Nueva Granada, Bogotá, Colombia.
⁷ Department of Pediatric Oncology, National Cancer Institute, Liverpool, Colombia.
⁸ Department of Pediatric Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina.
⁹ Department of Pediatric Surgery, Necker-Enfants Malades Hospital, Université de Paris, Paris, France.
¹⁰ Department of Pediatric Otolaryngology, Necker-Enfants Malades Hospital, Université de Paris, Paris, France.
¹¹ Department of Pediatric Oncology Surgery and Pediatric Oncology, Pediatric Oncology Institute-GRACC, Federal University of São Paulo, São Paulo, Brazil.
¹² Department of Head and Neck Surgery, Federal University of São Paulo, São Paulo, Brazil.
¹³ Department of Pediatric Surgery, Alder Hey Children's Hospital NHS Foundation Trust, Liverpool, United Kingdom.
¹⁴ Department of Pathology, Alder Hey Children's Hospital NHS Foundation Trust, Liverpool, United Kingdom.
¹⁵ Department of Pediatric Surgery, Faculty of Health and Life Sciences, Alder Hey Children's Hospital NHS Foundation Trust, University of Liverpool, Liverpool, United Kingdom.

Abstract

Background: Thyroid gland malignancies are rare in pediatric patients (0.7% of tumors); only 1.8% are observed in patients aged <20 years, with a higher prevalence recorded in women and adolescents. Risk factors include genetic syndromes, MEN disorders, autoimmune diseases, and exposure to ionizing radiation. Radiotherapy is also associated with an increased risk of secondary thyroid cancer. This study describes the clinical features and surgical outcomes of primary and secondary thyroid tumors in pediatric patients.

Methods: Institutional data were collected from eight international surgical oncology centers for pediatric patients with thyroid cancer between 2000 and 2020. Statistical analyses were performed using the GraphPad Prism software.

Results: Among 255 total cases of thyroid cancer, only 13 (5.1%) were secondary tumors. Primary thyroid malignancies were more likely to be multifocal in origin (odds ratio [OR] 1.993, 95% confidence interval [CI].7466-5.132, p = 0.2323), have bilateral glandular location (OR 2.847, 95% CI.6835-12.68, p = 0.2648), and be metastatic at first diagnosis (OR 1.259, 95% CI.3267-5.696, p > 0.999). Secondary tumors showed a higher incidence of disease relapse (OR 1.556, 95% CI.4579-5.57, p = 0.4525) and surgical complications (OR 2.042, 95% CI 0.7917-5.221, p = 0.1614), including hypoparathyroidism and recurrent laryngeal nerve injury. The overall survival (OS) was 99% at 1 year and 97% after 10 years. No EFS differences were evident between the primary and secondary tumors (chi-square 0.7307, p = 0.39026).

Conclusions: This multicenter study demonstrated excellent survival in pediatric thyroid malignancies. Secondary tumors exhibited greater disease relapse (15.8 vs. 10.5%) and a higher incidence of surgical complications (36.8 vs. 22.2%).

Keywords: cancer; carcinoma; children; surgery; thyroid.

Copyright © 2022 Martucci, Crocoli, De Pasquale, Spinelli, Strambi, Brazzarola, Morelli, Cassiani, Mancera, Luengas, Lobos, Liberto, Astori, Sarnacki, Couloigner, Simon, Lambert, Abib, Cervantes, Caran, Delgado Lindman, Jones, Shukla, Losty and Inserra.