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Surgical Morbidity and Short-Term Oncologic Status after Prophylactic-Intent Thyroidectomy in a Cohort of Children
Isaline Morard1, Frédéric Castinetti2, Isabelle Oliver Petit3
1Department of Pediatric Endocrinology, Diabetology, Gynecology, Obesity, Children's Hospital, University Hospital of Bordeaux, Bordeaux, France.
Background:
Children carrying germline RET mutations associated with multiple endocrine neoplasia type 2 (MEN2) are at high risk of developing medullary thyroid carcinoma (MTC). Prophylactic-intent thyroidectomy is recommended during childhood to prevent progression to advanced disease. Genotype-based recommendations combined with calcitonin measurements allow more individualized surgical timing. However, performing thyroidectomy at very young ages may expose children to long-term morbidity, particularly permanent hypoparathyroidism.
Methods:
We conducted a retrospective national multicenter cohort study within the French Groupe d'Étude des Tumeurs Endocrines, including children younger than 15 years who underwent prophylactic-intent total thyroidectomy between 2010 and 2020 in the absence of clinically apparent structural disease. Data collected included RET genotype, age at surgery, preoperative calcitonin values interpreted relative to each laboratory's upper limit of normal, surgical procedures performed, histopathologic findings, postoperative complications, and clinical status at last follow-up.
Results:
Sixty-four children (61 MEN2A, 3 MEN2B) underwent surgery at a median age of 4.6 years (interquartile range [IQR] = 3.2-8.3 years). Preoperative calcitonin was elevated in 44% of evaluable patients. Histopathology demonstrated C-cell hyperplasia in 52% and micro-MTC in 34%, while lymph node metastases were rare (3%). After a median follow-up of 6 years (IQR = 2.4-8.5 years), no patient had persistent structural disease. One patient had persistent moderate biochemical disease without structural evidence of MTC, although follow-up duration limits definitive long-term oncologic outcomes. Postoperative morbidity was notable: hypoparathyroidism occurred in 31% of patients and was permanent in 16%, predominantly among children operated before age 5 years.
Conclusions:
In this national contemporary cohort, prophylactic-intent thyroidectomy in pediatric MEN2 was associated with excellent short-term oncologic outcomes but also with a substantial rate of permanent hypoparathyroidism, particularly in very young children. These findings underscore the importance of multidisciplinary evaluation of both the timing and extent of surgery in expert centers.
Insights
Prophylactic thyroidectomy in children with multiple endocrine neoplasia type 2 (MEN2) shows excellent short-term results but carries a significant risk of permanent hypoparathyroidism, especially in younger children.
Area of Science:
- Endocrinology
- Pediatric Oncology
- Genetics
Background:
- Children with germline RET mutations for multiple endocrine neoplasia type 2 (MEN2) face a high risk of medullary thyroid carcinoma (MTC).
- Prophylactic thyroidectomy is recommended in childhood to prevent MTC progression.
- Early surgery may lead to long-term complications like permanent hypoparathyroidism.
Purpose of the Study:
- To evaluate the oncologic outcomes and postoperative morbidity of prophylactic thyroidectomy in pediatric MEN2 patients.
- To assess the impact of age at surgery on outcomes and complications.
Main Methods:
- Retrospective national multicenter cohort study of children under 15 undergoing prophylactic thyroidectomy (2010-2020).
- Data collected: RET genotype, age at surgery, calcitonin levels, surgical details, histopathology, complications, and follow-up status.
- Analysis of oncologic outcomes and incidence of hypoparathyroidism.
Main Results:
- 64 children (median age 4.6 years) underwent surgery; 44% had elevated preoperative calcitonin.
- Histopathology revealed C-cell hyperplasia (52%) or micro-MTC (34%); lymph node metastases were rare (3%).
- After median 6-year follow-up, no persistent structural disease; 16% experienced permanent hypoparathyroidism, higher in those operated before age 5.
Conclusions:
- Prophylactic thyroidectomy in pediatric MEN2 offers excellent short-term oncologic control.
- A substantial rate of permanent hypoparathyroidism necessitates careful consideration of surgical timing in very young children.
- Multidisciplinary evaluation is crucial for optimizing surgical timing and extent in expert centers.
