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Objective: Current staging and risk-stratification systems for predicting survival or recurrence of patients with differentiated thyroid carcinoma may be ineffective at predicting outcomes in individual patients. In recent years, nomograms have been proposed as an alternative to conventional systems for predicting personalized clinical outcomes. We conducted a systematic review to evaluate the predictive performance of available nomograms for thyroid cancer patients. Design and methods: PROSPERO registration (CRD42022327028). A systematic search was conducted without time and language restrictions. PICOT questions: population, patients with papillary thyroid cancer; comparator prognostic factor, single-arm studies; outcomes, overall survival, disease-free survival, cancer-speci fic survival, recurrence, central lymph node metastases, or lateral lymph node metastases; timing, all periods; setting, hospital setting. Risk of bias was assessed through PROBAST tool. Results: Eighteen studies with a total of 20 prognostic models were included in the systematic review (90,969 papillary thyroid carcinoma patients). Fourteen models were at high risk of bias and four were at unclear risk of bias. The greatest concerns arose in the analysis domain. The accuracy of nomograms for overall survival was assessed in only one study and appeared limited (0.77, 95% CI: 0.75–0.79). The accuracy of nomograms for disease-free survival ranged from 0.65 (95% CI: 0.55–0.75) to 0.92 (95% CI: 0.91–0.95). The C-index for predicting lateral lymph node metastasis ranged from 0.72 to 0.92 (95% CI: 0.86–0.97). For central lymph node metastasis, the C-index of externally validated studies ranged from 0.706 (95% CI: 0.685–0.727) to 0.923 (95% CI: 0.893–0.946). Conclusions: Our work highlights the extremely high heterogeneity among nomograms and the critical lack of external validation studies that limit the applicability of nomograms in clinical practice. Further studies ideally using commonly adopted risk factors as the backbone to develop nomograms are required. Significance statement: Nomograms may be appropriate tools to plan treatments and predict personalized clinical outcomes in patients with papillary thyroid cancer. However, the nomograms developed to date are very heterogeneous, and their results seem to be closely related to the specific samples studied to generate the same nomograms. The lack of rigorous external validation procedures and the use of risk factors that sometimes appear to be far from those commonly used in clinical practice, as well as the great heterogeneity of the risk factors considered, limit the ability of nomograms to predict patient outcomes and thus their current introduction in clinical practice.

Autoimmune thyroid diseases are on the rise worldwide, and such a rapid increase is mainly driven by environmental factors related to changed lifestyles in “modern” societies. In this context, diet seems to play a crucial role. An unhealthy high-energy diet, rich in animal fat and proteins, salt and refined sugars (the so-called “Western diet”) negatively influences the risk of autoimmunity by altering the immune balance and the gut microbiota composition, enhancing oxidative stress and promoting inflammation. In contrast, the Mediterranean diet represents a unique model of healthy eating, characterized by a high intake of food from vegetable sources, a low consumption of saturated fats in favor of unsaturated fats (mainly, olive oil), a moderate consumption of fish (typically, the small oily fishes) and dairy products, as well as a moderate consumption of wine at meals, and a low intake of meat. Thanks to its nutritional components, the Mediterranean Diet positively influences immune system function, gut microbiota composition, and redox homeostasis, exerting anti-oxidants, anti-inflammatory, and immunomodulatory effects. The present review was aimed at exploring the existing knowledge on the correlations between dietary habits and thyroid autoimmunity, to evaluate the role of the Mediterranean diet as a protective model.

(1) Background: This paper aims to assess the existence of significant differences between two dietary regimes (omnivorous vs. semi-vegetarian) with reference to some oxidative stress markers (SOD, GPx, TRxR, GR, AGEs, and AOPPs) using non-parametric combination methodology based on a permutation test. (2) Methods: At the endocrinology unit of Messina University Hospital, two hundred subjects were asked to fill out a questionnaire about their dietary habits. None were under any pharmacological treatment. Using the NPC test, all comparisons were performed stratifying patients according to gender, age (≤40 or >40 years), BMI (normal weight vs. overweight), physical activity (sedentary vs. active lifestyle), TSH, FT4 levels in quartiles, and diagnosis of Hashimoto’s thyroiditis. We evaluated differences in oxidative stress parameters in relation to two examined dietary regimes (omnivorous vs. semi-vegetarian). (3) Results: The antioxidant parameters GPx and TRxR were significantly lower in subjects with an omnivorous diet than in semi-vegetarians, particularly in females, both age groups, subjects with normal weight, those not affected by Hashimoto’s thyroiditis, and both the sedentary and active lifestyle groups. Finally, the AGE and AOPP markers were significantly lower in semi-vegetarians. (4) Conclusion: Thanks to the NPC methodology, we can state that dietary patterns exert a significant influence on some oxidative stress parameters.

The use of radioiodine therapy (RIT) is debated in intermediate-risk differentiated thyroid cancer (DTC) patients. The understanding of the molecular mechanisms involved in the pathogenesis of DTC can be useful to refine patient selection for RIT. We analyzed the mutational status of BRAF, RAS, TERT, PIK3 and RET, and the expression of PD-L1 (as a CPS score), the NIS and AXL genes and the tumor-infiltrating lymphocytes (TIL, as the CD4/CD8 ratio), in the tumor tissue in a cohort of forty-six ATA intermediate-risk patients, homogeneously treated with surgery and RIT. We found a significant correlation between BRAF mutations and a less than excellent (LER, according to 2015 ATA classification) response to RIT treatment (p = 0.001), higher expression of the AXL gene (p = 0.007), lower expression of NIS (p = 0.045) and higher expression of PD-L1 (p = 0.004). Moreover, the LER patient group had a significantly higher level of AXL (p = 0.0003), a lower level of NIS (p = 0.0004) and a higher PD-L1 level (p = 0.0001) in comparison to patients having an excellent response to RIT. We also found a significant direct correlation between the AXL level and PD-L1 expression (p < 0.0001) and a significant inverse correlation between AXL and NIS expression and TILs (p = 0.0009 and p = 0.028, respectively). These data suggest that BRAF mutations and AXL expression are involved in LER among DTC patients and in the higher expression of PD-L1 and CD8, becoming new possible biomarkers to personalize RIT in the ATA intermediate-risk group, as well as the use of higher radioiodine activity or other possible therapies.

Familial hypocalciuric hypercalcemia (FHH) is an autosomal dominant inherited disorder due to inactivating mutations in the calcium-sensing receptor (CaSR), less commonly in the G-protein subunit α11 (GNA11) or the adaptor-related protein complex 2, sigma 1 subunit (AP2S1) genes. It is characterized by mild to moderate hypercalcemia, hypocalciuria, and inappropriately normal or high PTH levels. The disorder is usually asymptomatic, and bone or renal involvement is rare. Adequate differential diagnosis between primary hyperparathyroidism and FHH is important to avoid unnecessary surgery. We report the case of a male patient with FHH showing biochemical heterogeneity to highlight the difficulties of differential diagnosis

Secondary hyperparathyroidism (SHPT) is a common complication of chronic kidney disease (CKD). The progressive decline in renal function, in fact, leads to the alteration of the metabolism of calcium (Ca), phosphorus (P) and vitamin D. Difference of other forms of hyperparathyroidism, renal SHPT has many pathogenetic peculiarities, which have been only in part clarified. Furthermore, in the long course of CKD, SHPT sometimes transforms into a hypercalcemic condition resembling the autonomous form of hyperparathyroidism (tertiary hyperparathyroidism; THP). The clinical consequences of SHPT in CKD patients are manifold, encompassing not only bone and mineral disorders, but also other metabolic and organic changes which frequently burden these patients. Although the medical therapeutic tools have substantially increased in number and improved in their efficacy in recent decades, we have as yet no demonstration of a clear benefit regarding the major clinical outcomes. Furthermore, some of these patients still require a surgical approach. Herein we discussed the clinical management and surgical treatment for a case of a recurrent secondary hyperparathyroidism.

The case of a 46 years old man with single bone metastasis as the first sign of differentiated thyroid carcinoma is described and discussed along with advanced imaging.

Direct Monte Carlo (MC) simulation is considered the gold standard approach for internal dosimetry in nuclear medicine, and it is increasingly used in planning Trans-Arterial Radio-Embolization (TARE) of HepatoCellular Carcinoma (HCC) {and hepatic metastases}. However its computational times, longer with respect to other simplified approaches, constitute a limiting factor, especially when dealing with {large size and finely discretized voxelized volumes}. Aim of this work was the investigation of the influence of cuts on the production of secondary particles and of input CT images resamplings on dosimetric accuracy and computational time in patient-specific voxel-level MC simulations of 90Y-labelled glass microspheres TARE treatment, to find optimal combinations of settings for speeding up such simulations. GATE GEANT4 interface was used to perform simulations employing CT and 99mTc SPECT as input data, examining multiple CT resolutions (via CT resamplings characterized by voxel volume factors 2, 8, and 64 with respect to native one, and a {CT} resampling with SPECT resolution) and production cuts (0.01 mm, 0.05 mm, 0.1 mm, 0.5 mm and some more, specific for each resampling). Increasing cut length and reducing CT resolution produces an early rapid decrease followed by a late slow decrease of simulation time as a function of this two parameters. {A} reduction up to 30% with respect to reference simulation time, while preserving acceptable dosimetric accuracy, {was obtained}. The best combination of settings among the examined ones resulted the choice of CT resampling with 8 times the native voxel volume and of 0.1-0.5 mm cut, ensuring dosimetric agreement within 1% in liver-related VOIs, while reducing simulation time to 45%.

Background and Aim. Parathyroidectomy is the standard treatment for primary hyperparathyroidism (PHPT). Cinacalcet, an allosteric modulator of the calcium sensing receptor, has been shown to be effective in reducing serum calcium levels and it has gained prominence as a non-invasive treatment in PHTH. Aim of the study was to compare the efficacy and tolerability of medical therapy vs surgery in a cohort of PHPT patients. Patients and Methods. The study included 63 consecutive subjects (14 men and 49 women, median age 62.4 ± 12 yr) with PHPT: 50 subjects (40 F and 10 M, ratio F/M 4:1; median age 59.6 ± 16.6 years, range 37-82) underwent parathyroidectomy, while 13 (9 F and 4 M, ratio F/M 2.5:1, median age 76.5 ± 6.2 years, range 64-83), who were ineligible for surgery, were treated with cinacalcet. Cinacalcet was administered at increasing dosages until normal serum calcium was reached or side effects occurred. Serum calcium and PTH was measured and adverse events were monitored during a 12 month-duration follow-up. Results. All patients who had undergone parathyroidectomy normalized both serum calcium and PTH levels after surgery (P=0.001 vs baseline) and none of them experienced persistent and/or recurrent PHPT during follow-up. In the group under cinacalcet therapy, serum calcium significantly decreased in all patients within 4 weeks (P = 0.01 vs baseline) and normalized in all patients at prolonged follow-up (P=0.001 vs baseline), while PTH slightly decreased during follow-up (P=0.878 vs baseline) and never reached a normal value. Conclusion. Surgical treatment is curative and safe in most cases of PHPT. Cinacalcet represents an effective therapeutic option, from the perspective of hypercalcemia improvement, in PHPT patients who have contraindications to surgery or persistent PHPT after surgery.

99mTc-MIBI (MIBI) imaging is able to exclude malignancy of hypofunctioning thyroid nodules (TNs) with high probability but false positive results are frequent due to low specificity. Therefore, pre-test selection of appropriate TNs is crucial. For image evaluation visual and semiquantitative methods (Washout index, WOInd) are used. Aim of this study was to evaluate the diagnostic performance of MIBI imaging in hypofunctioning TNs with indeterminate fine-needle aspiration cytology results in a multicentric European setting. Patients with hypofunctioning TNs, EU-TIRADS 4 or 5, Bethesda III/IV and MIBI imaging were included. For visual evaluation the intensity of MIBI uptake in the TN was compared to normal thyroid tissue. 358 patients with 365 TNs (n = 68 malignant) were included. Planar imaging (SPECT) showed a sensitivity, specificity, positive predictive value, negative predictive value, and accuracy of 96% (94%), 21% (22%), 22% (15%), 96% (96%), and 35% (32%). The WOInd (38.9% of all cases, optimal cutoff: −19%) showed a sens 100% (spec 89%, PPV 82%, NPV 100%, ACC 93%). For hypofunctioning TNs at intermediate or high risk with indeterminate cytology, a MIBI negative result on visual evaluation is an effective tool to rule-out thyroid malignancy. The semi-quantitative method could considerably improve overall diagnostic performance of MIBI imaging.

Bone involvement in primary hyperparathyroidism (PHPT) is characterized by decreased bone mineral density, bone resorption at both trabecular and cortical sites and bone erosions, up to brown tumors (BT) and cysts, the so-called osteitis fibrosa cystica (OFC). Signs and symptoms of OFC include bone pain, muscle weakness, skeletal deformities and pathological fractures. In recent years, PHPT has greatly changed its clinical expression, especially in Western countries. For these reason BT, a typical expression of OFC, are always less observed and often mistaken for malignancy. An integrated diagnostic approach, considering first a complete biochemical panel and a confirmation by functional imaging, is crucial for a correct diagnosis, mostly considering that such skeletal manifestations may be reversible after surgical cure of PHPT.

Medullary thyroid cancer (MTC) is a rare disease, often displaying an aggressive behavior and a poor prognosis. Serum calcitonin represents the most sensitive marker of MTC and its routine measurement in patients with thyroid nodules allows early detection of occult MTC and appropriate surgical cure.Here we report on a 55-yr-old woman with a long-standing goiter, autonomously functioning thyroid nodules and subclinical thyrotoxicosis, who was diagnosed with MTC at an early stage by means of calcitoninscreening. This rare case highlights the importance of performing routine calcitonin measurementin thyroid nodules.

Rationale. Immune Checkpoint Inhibitors (ICIs) are approved for some advanced neoplasms, increasing survival. ICIs block inhibitor receptors cytotoxic T lymphocyte antigen 4 (CTLA4) and programmed death-1 (PD-1) and trigger T cell-mediated immunity against tumor. Their action is accompanied by several immunity-related adverse events (IRAEs), also involving the endocrine system (pituitary, thyroid, adrenals). We report two different cases of thyrotoxicosis following administration of the anti-PD-1 nivolumab. Patients. Patient 1, M, 75 years-old, treated for non-small cell lung carcinoma (NSCLC) since September 2016, with euthyroid multinodular goiter. In January 2017 (12 weeks from baseline), he developed frank hyperthyroidism, with positive TSH-receptor antibodies (TRAb) and thyroperoxidase antibodies (TPOAb). A Tc99m thyroid scintiscan showed diffuse uptake and “cold” areas. After nivolumab withdrawal, treatment with metimazole (MMI) 5 mg per day was started and euthyroidism was resumed, so to restart the drug in May 2017. Patient 2, M, 80 years-old, treated for a left-eye choroid melanoma since January 2017, with euthyroid nodular goiter. In April 2017 (6 weeks from baseline), thyrotoxicosis was detected, with positive thyroglobulin antibodies (Tg-Ab, 244 IU/ml, nv 4.5 µIU/ml (subclinical hypothyroidism). Patient was treated with replacement doses of levothyroxine (LT-4), and continued nivolumab infusions. Conclusions. Two forms of thyrotoxicosis were reported: the first with thyroid hyperfunction and positive TRAb, the latter as a destructive thyroiditis. In both cases (mean onset after 9 weeks), the moderate severity and the appropriate management of endocrine IRAEs allowed treatment continuation.

Abscess formation within a brain tumor is uncommon. Intrasellar or parasellar tumors are the most common neoplasms that develop such complications. Cerebral gliomas with abscesses are extremely rare. In this paper three rare cases of glioma associated with abscess formation are described. The diagnosis of brain tumor associated with abscess is particularly difficult by conventional neuroradiological studies. 99mTc-labeled sulesomab can be useful in the diagnosis of brain tumors with intratumoral abscesses. There are no precise guidelines for the diagnosis and treatment of cerebral gliomas associated with abscesses formation for the low number of cases reported to date. Appropriate treatment, aimed at radical surgery, and a suitable antibiotic-protocol, deferring adjuvant postoperative therapy, is associated with a more favorable outcome. A review of the pertinent literature is also performed.

The hepatocyte growth factor (HGF)/c-met axis plays a crucial role in cancer development by promoting cellular proliferation, motility, and morphogenesis, as well as angiogenesis. Different cellular distributions of both the ligand and the receptor in benign vs malignant lesions indicate this biological system as a candidate for a diagnostic biomarker of malignancy occurring in endocrine glands, such as the thyroid and pituitary. Furthermore, the HGF/c-met expression may help to identify a subset of patients eligible for potential targeted therapies with HGF/c-met inhibitors or antagonists in thyroid tumour, as well as in other malignancies. This may be relevant for iodine-refractory cancers, the treatment of which is still a major challenge. With this in mind, HGF/c-met expression in thyroid cancer tissue may be useful for prognostic and therapeutic stratification of patients.

A 63-year-old woman presented to the Orthopedic Unit of our hospital complaining of right hip pain of 6 months'duration associated with a worsening limp. Her past medical history included chronic renal insufficiency. Physical examination revealed deep pain in the iliac region and severe restriction of the right hip's articular function in the maximum degrees of range of motion. X-rays and CT scan detected an osteolytic and expansive lesion of the right supra-acetabular region with structural reabsorption of the right iliac wing. 99mTc-MDP whole-body bone scan showed an abnormal uptake in the right iliac region. Bone biopsy revealed an osteolytic lesion with multinucleated giant cells, indicating a brown tumor. Serum intact PTH was elevated (1020 pg/ml; normal values, 12 62 pg/ml), but her serum calcium was normal (total = 9.4 mg/dl, nv 8.5–10.5; ionized = 5.0 mg/dl, nv 4.2–5.4) due to the coexistence of chronic renal failure. 99mTc-MIBI scintigraphy revealed a single focus of sestamibi accumulation in the left retrosternal location, which turned out to be an intrathoracic parathyroid adenoma at surgical exploration. After surgical removal of the parathyroid adenoma, PTH levels decreased to 212 pg/ml. Three months after parathyroidectomy, the imaging studies showed complete recovery of the osteolytic lesion, thus avoiding any orthopedic surgery. This case is noteworthy because (1) primary hyperparathyroidism was not suspected due to the normocalcemia, likely attributable to the coexistence of chronic renal failure; and (2) it was associated with a brown tumor of unusual location (right supra-acetabular region).

Thyroid cancer harbours in about 5% of thyroid nodules. The majority of them are well-differentiated cancers originating from the follicular epithelium, and are subdivided into papillary and follicular carcinomas. Undifferentiated carcinomas and medullary thyroid carcinomas arising from C cells are less common. Although most thyroid nodules are benign, distinguishing thyroid cancer from benign lesions is crucial for an appropriate treatment and follow-up. The fi ne needle aspiration cytology (FNAC) allows the diagnosis of nature of thyroid nodules in the majority of cases. However, FNAC has some limitations, particularly in the presence of follicular lesions which can appear dubious in rare instances even at histology. In an effort to improve diagnostic accuracy and offer new prognostic criteria, several immunohistochemical and molecular markers have been proposed. However, most of them have to be validated on large series before being used in routine practice.

The Epidermal Growth Factor Receoptor (EGFR) family member human epidermal growth factor receptor 2 (HER2) is overexpressed in many human epithelial malignancies, representing a molecular target for specific anti-neoplastic drugs. Few data are available on HER2 status in differentiated thyroid cancer (DTC). The present study was aimed to investigate HER2 status in sporadic cancers of follicular cell origin to better clarify the role of this receptor in the stratification of thyroid cancer. By immunohistochemistry and fluorescence in-situ hybridization, HER2 expression was investigated in formalin-fixed paraffin-embedded surgical specimens from 90 DTC patients, 45 follicular (FTC) and 45 papillary (PTC) histotypes. No HER2 immunostaining was recorded in background thyroid tissue. By contrast, overall HER2 overexpression was found in 20/45 (44%) FTC and 8/45 (18%) PTC, with a significant difference between the two histotypes (p = 0.046). Five of the six patients who developed metastatic disease during a median nine-year follow-up had a HER2-positive tumor. Therefore, we suggest that HER2 expression may represent an additional aid to identify a subset of patients who are characterized by a worse prognosis and are potentially eligible for targeted therapy.

Purpose. To investigate the impact of the maximum standardized uptake value (SUVmax), size of primary lung lesion, and %ΔSUVmax on outcome (overall survival (OS) and 2-year disease-free survival (2-year DFS)) of patients with advanced nonsmall-cell lung cancer (NSCLC). Materials and Methods. 86 stage III-IV NSCLC patients underwent 18 F-FDGPET/CT, before and after chemotherapy, and were classified into subgroups according to the response criteria of the European Organization for Research and Treatment of Cancer. SUVmax values and tumor size with the best prognostic significance were searched. Correlation between the SUVmax value and the initial response to therapy (best response) and the relationship between %ΔSUVmax and OS were assessed. Results. In patients in PD (20/86), the average pretreatment SUVmax was 11.8±5.23, and the mean size of the primary lesion was 43.35 mm±16.63. In SD, PR, and CR patients (66/86), the average pretreatment SUVmax was 12.7±8.05, and the mean size of the primary lesion was 41.6 mm±21.15. Correlation was identified only for %ΔSUVmax; patients with PD (ΔSUVmax > +25%) showed a worse OS than patients with ΔSUVmax < +25% (CR, PR, and SD) (P=0.0235). Conclusions. In stage III-IV NSCLC, among the assessed factors, only %ΔSUVmax may be considered as a useful prognostic factor.

Thyroid cancer harbours in about 5% of thyroid nodules. The majority of them are well-differentiated cancers originating from the follicular epithelium, and are subdivided into papillary and follicular carcinomas. Undifferentiated carcinomas and medullary thyroid carcinomas arising from C cells are less common. Although most thyroid nodules are benign, distinguishing thyroid cancer from benign lesions is crucial for an appropriate treatment and follow-up. The fine needle aspiration cytology (FNAC) allows the diagnosis of nature of thyroid nodules in the majority of cases. However, FNAC has some limitations, particularly in the presence of follicular lesions which can appear dubious in rare instances even at histology. In an effort to improve diagnostic accuracy and offer new prognostic criteria, several immunohistochemical and molecular markers have been proposed. However, most of them have to be validated on large series before being used in routine practice.

In patients affected by differentiated thyroid cancer, the whole-body scan (WBS) with 131-radioiodine, especially when performed after a therapeutic activity of131I, represents a sensitive procedure for detecting thyroid remnant and/or metastatic disease. Nevertheless, a wide spectrum of potentially pitfalls has been reported. Herein we describe a 63-year-old woman affected by follicular thyroid cancer, who was accidentally found to have an abdominal mass at post-dose WBS (pWBS). pWBS showed abnormal radioiodine uptake in the upper mediastinum, consistent with lymph-node metastases, and a slight radioiodine uptake in an abdominal focal area. Computed tomography revealed an inhomogeneous mass in the pelvis, previously unrecognized. The lesion, surgically removed, was found to be a typical dermoid cyst of the ovary, without any evidence of thyroid tissue. By immunohistochemistry, a moderate expression of the sodium-iodine symporter (NIS) was demonstrated in the epithelial cells, suggesting a NIS-dependent uptake of radioiodine by the cyst.

SUMMARY Papillary thyroid carcinoma (PTC) is the most common type of thyroid cancer. On the contrary, primary thyroid lymphoma (PTL) is a rare disease, accounting for 2% to 5% of all thyroid malignancies. Despite several cases in which both PTC and PTL arise in the setting of Hashimoto's thyroiditis (HT), the coexistence of both tumors in HT patients is very rare. Herein we report the case of a 66-year-old woman with long-standing nodular HT under replacement therapy, who presented with a fast, painless enlargement in the right anterior side of the neck. Thyroid ultrasonography demonstrated increased growth of a hypoechoic nodule in the right lobe measuring 32 × 20 mm. A total thyroidectomy was performed, and histology revealed a diffuse large B-cell lymphoma (DLBCL) on a background of florid HT. Moreover, a unifocal papillary microcarcinoma, classical variant (7 mm, pT1aNxMx), was discovered. The patient was then treated with chemotherapy for the PTL, but she did not undergo radioactive iodine ablation treatment for the microPTC as per guidelines. Two years after surgery, the patient had no evidence of recurrence of either malignancy. This rare case highlights the importance of monitoring HT patients with nodular lesions, especially if they have long-standing disease. In addition, PTL should be considered for differential diagnosis in elder HT patients who present with sudden thyroid enlargement.

Thyroid nodules in children are less common than in adults but they are approximately two- to three-fold more likely to be malignant in children. Among thyroid nodular diseases, Plummer's adenoma occurs very rarely in pediatrics, and currently, there is no literature providing evidence of this diagnosis in patients with Prader–Willi syndrome (PWS). We report the case of a 9-year-old Caucasian boy affected by PWS presenting with a rapidly growing palpable mass in the thyroid lodge associated with subclinical hyperthyroidism. Laboratory and other examinations (thyroid ultrasound, fine-needle aspiration of the nodule, and scintigraphy) were strongly suggestive for Plummer's adenoma; therefore, the patient underwent left hemithyroidectomy surgery, and anatomo-pathological examination confirmed the diagnosis. Our case describes the first evidence of an isolated follicular adenoma in children with PWS. Surgery is the only therapeutic option in younger children. Further evidence is needed to assess the possible correlation between these two conditions and the existence of potential risk factors.