Pediatric thyroid disease is relatively rare compared to adult thyroid disease, contributing to low annual numbers of operative thyroid cases in children, even in high volume surgery centers. These low numbers lead to a paucity of large sample populations from which we can draw conclusions regarding updated practices in pediatric thyroid disease. This study was undertaken to examine the changing trends in practice for pediatric thyroid disease at a single institution after the establishment of an Endocrine Surgery Center. This retrospective review of 78 pediatric thyroid operations between 1994 and 2009 demonstrated an increase in the volume of pediatric thyroid cases, an increase in operations for benign thyroid disease, and an increase in total thyroidectomies over partial removal for benign disease since the formation of the Endocrine Surgery Center. Moreover, we demonstrate a trend of increased utilization of FNA in diagnosis of pediatric thyroid nodules, and a distribution of thyroid cancer cases that mirrors the percentages found in larger nationwide studies (
14), although the number of operations for medullary thyroid cancer decreased significantly in the later time group. Interestingly, this study revealed an increase in the number of surgeons performing pediatric thyroid operations and in the number of cases performed by pediatric and otolaryngology surgeons since the establishment of the Endocrine Surgery Center. However, this trend was not associated with a change in complication rates, which were uniformly low (9% of all cases), on the same level as published adult series.
Both national database studies and single institution studies advocate that pediatric endocrine surgical care be performed by surgeons with high-volume practices at high-volume surgical centers due to lower complication rates and shorter lengths of stay (
9–
11,
15,
16). Interestingly, this relationship holds true despite the fact that both high-volume surgeons and pediatric surgeons in the Healthcare Cost and Utilization Project Nationwide Inpatient Sample (HCUP-NIS) performed an average of only 2 pediatric cases per year (
9). This suggests that there are qualities outside of pediatric-specific endocrine surgical volume that are reducing length of stay and complications for pediatric patients. Our findings support this idea; there was no significant change in complications after the endocrine surgery center was created at our institution, even though there was an increase in the number of cases performed by either pediatric or otolaryngology specialists (4% each prior to the endocrine center vs. 12% and 15%, respectively). Despite not having an endocrine surgery center prior to 2001, our institution has been a high-volume adult and pediatric surgical institution since well before our prospectively collected database began. This suggests that complication rates and other negative outcomes are limited at least in part by the experience of our other healthcare staff with pediatric patients. In addition, the increase in the proportion of cases performed by an otolaryngologist or pediatric surgeon with the formation of the endocrine surgery center demonstrates an increase in interdisciplinary collaboration advocated by a recent single-institution study by Wood et al. (
10), thereby minimizing specialist-specific limitations and optimizing patient care. In our institution, this collaboration is facilitated by a regular case conference during which challenging cases are addressed by multidisciplinary faculty.
One of the benefits of an interdisciplinary endocrine surgery center is a more ready adoption of technology. Fine needle aspiration has been recommended by the American Thyroid Association (ATA) as a first-line diagnostic tool in adults (
7), but it has been less uniformly encouraged in children, even though the risk of malignancy in children who present with nodules is higher than in adults (
17,
18). A recent meta-analysis demonstrates a 98.2% negative predictive value of FNA in children, while a retrospective study of 110 patients shows a positive predictive value of 95% in FNAs that are read as malignant. These findings suggest FNA should play a central role in the diagnostic algorithm of pediatric patients with thyroid nodules to exclude malignancy (
18) and confirm the need to remove nodules that are atypical on FNA just as in adults (
19). Our findings of increased use of FNA for pediatric thyroid nodules likely corresponds with an increased appreciation for the accuracy in pediatrics patients, as well as the coordination within our multidisciplinary practice with the adult endocrine practioners who ability to perform the procedure with minimal anxiety on the part of the patient.
This concern for higher risk of complications in children carries over to thyroidectomies as well. A previous single-institution study found higher complication rates in children than adults, and thus recommend partial thyroidectomy for all single malignant nodules (
20). However, our study shows complication rates similar to adult series, with no significant increase in complications associated with the increase in total thyroidectomies. Moreover, it has been shown that children with even small tumors at presentation have high rates of recurrence (20–30%) requiring reoperation when not initially treated with total thyroidectomy and radioactive iodine treatment (
8,
21). Still, thyroid malignancy in children is a universally recognized indication for surgical removal, as is concern for multiple endocrine neoplasia 2 (MEN2) hereditary syndromes. This is supported by the ATA Guidelines for management of medullary thyroid cancer (
22,
23), with recommendations for thyroidectomy based on malignant potential in genetic testing. While we showed no change in the number of prophylactic operations, likely due to the steady detection of MEN2 syndromes with genetic testing protocols, the number of operations for medullary thyroid cancer decreased significantly. This may be a reflection of the efficacy of increased awareness of the prophylactic guidelines and early detection of genetic mutations.
In concordance with population-based studies over recent years (
6), our study shows an increase over time in the percentage of total thyroidectomy versus subtotal thyroidectomy as the surgical treatment of choice for thyroid disease (increasing from 58% to 85%), with a significant increase in patients with benign indications undergoing total thyroidectomy after the establishment of an Endocrine Surgery center. While there are concerns for higher rates of complications in children compared to adults, studies of total thyroidectomy for benign disease in both adults (
5) and children (
4) show low complications rates and no disease recurrence at a median of 9 years and 12.9 years of follow up, respectively (
4,
5).
With definitive surgical treatment for benign disease, we allow pediatric patients to avoid the difficulty of compliance and potential toxicity, as well as high relapse rates, associated with antithyroid medications like propylthiouracil and methimazole (
24,
25). We also avoid the risks of radioactive iodine (RAI) treatment in pediatric patients for treatment of benign disease. While there was no change in RAI treatment between patient groups in our study, no patients with benign disease were subjected to RAI in Group 2, while 2 patients with benign disease underwent RAI in Group 1 (8%). Further studies have addressed this issue particularly in Graves’ Disease, which was the most common benign indication for surgery in our study. These studies proposed total or near total thyroidectomy as the most appropriate operative treatment of Graves’ Disease, because it eliminates the risk of recurrent disease and the resulting hypothyroidism can be predictably controlled with thyroxine replacement (
2,
3,
5).
The reticence to recommend following the adult guidelines completely in children is likely due to a concern for complications in pediatric patients. While some population database studies suggest that children have higher complication rates than adults after thyroid surgery, these differences were only in post-operative hypocalcemia. Non-endocrine-related complications were comparable between adults and children, at a rate of 10.5–11.5%, as are recurrent laryngeal nerve related complications (
14). The single-institution study from Boston Children’s Hospital demonstrates overall complication rates similar to ours (
1), which correspond with rates from adult series (
5,
6,
11). We conclude that, with fellowship-trained surgeons at a high-volume endocrine center, complication rates can be kept low, especially with collaboration between surgical specialties in very young patients.
This study possesses the inherent limitations of retrospective reviews, in that there is no design for targeted results, and the data are subject to the limitations of observations. However, this study benefits compared to other retrospective reviews in that the data were collected prospectively at the time of operation, and follow up data have been tracked. Additionally, pediatric thyroid disease is relatively rare, meaning that patient samples are small. Many comparisons in this study approach significance, and it is unclear if significance is not reached due to the lack of power with the small sample sizes. Regarding comparisons based on disease classification, the benign versus malignant determination is made by pathologic results, and not on an intent-to-treat diagnosis. This information is not reliably recorded in our database and so was not used. This study may also provide a skewed representation of thyroid disease rates in the population because this sample is comprised only of patients who underwent an operation, although the frequency of the cancer diagnoses shown here generally follows the pattern in the population at large (
14). However, contrary to other single-institution studies that reported a predominance of patients with thyroid nodules as the indication for operation (
2,
20), we demonstrated an increasing number of patients undergoing an operation for benign disease, which may indicate that this sample is a closer representation of thyroid disease in the general pediatric population.
Despite these limitations, we are able to show convincing aspects of how the surgical treatment of pediatric thyroid disease has evolved at our institution with the development of our Endocrine Surgery Center. We advocate for the use of ultrasound in the preoperative evaluation of pediatric patients, as well as increased use of FNA as an accurate diagnostic tool with minimal risk. Based on our results, we find the practice trending toward total thyroidectomy for both malignant and benign indications. These operations can have a low associated risk of complication in high-volume centers with experienced surgeons, even in young children. Specifically, the benefits of an Endocrine Surgery Center include the easy access to interdisciplinary collaboration in challenging cases, as well as a specialized understanding of pediatric endocrine disease by healthcare workers involved in every facet of patient care. With such a care system in place, we are able to provide children with safe, definitive surgical treatment for thyroid disease.