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Background Parathyroid carcinoma is extremely rare. Correct preoperative and even histopathological diagnosis may be difficult owing to the deceptively bland cytoarchitectural features, especially when presenting with localized disease. Recurrence/metastases developing years later then make the malignant nature obvious. Methods and Results We present here an unusual case of a 32-year-old patient with carcinoma of the left upper parathyroid gland, initially diagnosed as parathyroid adenoma, treated with endoscopic left parathyroidectomy, and later developing subcutaneous metastatic nodules over the medial end of the right clavicle and right anterior chest wall, followed by a right breast deposit. The recurrences, especially subcutaneous ones, were probably secondary to tumor seeding along the track of insertion of the endoscope. Conclusion Involvement of subcutaneous tissue and the breast in parathyroid carcinoma is extremely rare. The case is being reported for its uniqueness along with a discussion of possible appropriate course of management, which may have averted the aggressive clinical course of the disease. © 2015 Wiley Periodicals, Inc. Head Neck, 2015

Purpose Parathyroid carcinoma (PC) is remarkable for its rare occurrence and challenging diagnostics. PC accounts for 0.1–5% cases of primary hyperparathyroidism (PHPT). The differentiation from benign tumours is difficult even by morphological criteria. To address these issues, we assessed the PC frequency in two separate European PHPT cohorts and evaluated the demographic, clinical, morphological and molecular background. Methods A retrospective study was carried out, using continuously maintained database (2005–2014) of PHPT patients from two tertiary referral university hospitals in Europe. The demographic, clinical data and frequency of PC among surgically treated PHPT was detected. Immunohistochemistry (IHC) was performed to detect parafibromin, representing protein product ofHRPT2gene and proliferation marker Ki-67. Results Both PHPT cohorts were characterised by close mean age values (58.6 and 58.0years) and female predominance. The frequency of PC differed significantly between the cohorts: 2.1 vs. 0.3%;p = 0.004. PC was characterised by invariable complete loss of parafibromin contrasting with parathyroid adenomas. The proliferation fraction was similar in both PC cohorts (10.6 and 11.0%). PC showed significantly higher proliferation fraction than typical parathyroid adenomas (1.6%), atypical adenomas (1.6%) or adenomas featuring focal loss of parafibromin (2.2%). Conclusions PC frequency can range significantly between the two European cohorts. The differences can be attributable to selection bias of patients referred for surgery and are not caused by discordant definition of malignant parathyroid histology. Diffuse loss of parafibromin and increased proliferation fraction by Ki-67 are valuable adjuncts in PC diagnostics due to significant differences with various clinical and morphological subtypes of adenoma. Keywords Parathyroid carcinomaPrimary hyperparathyroidismImmunohistochemistryKi-67Parafibromin&nbsp

Background Primary hyperparathyroidism is primarily caused by parathyroid adenoma, followed by hyperplasia and parathyroid carcinoma. In the era of minimally invasive, targeted parathyroidectomy, the main challenge remains that of distinguishing intraoperatively pathological parathyroid from normal glands and peri-thyroid fat tissue. The aim of this study is to evaluate the surgical outcomes of a novel minimally invasive technique called ultrasound-guided dye-assisted parathyroidectomy (USDAP). Methods We perform a retrospective analysis of patients affected by parathyroid adenoma, treated with USDAP at our institution between 2014 and 2019. Data were collected on patient age and sex, tumor location and size, preoperative investigations, histopathology, perioperative complications and surgical outcomes. Results Between January 2014 and June 2019, 43 patients underwent parathyroidectomy in our Institute. Each case was discussed by the Institutional Multidisciplinary Board. All patients undergoing thyroidectomy together with USDAP or patients undergoing USDAP under endoscopic control were excluded from the present study. The final cohort, the largest to our knowledge, consisted of 29 patients. All patients were successfully treated with USDAP and remained disease-free during follow up. In all cases, pathological parathyroid was correctly identified and removed. There was no postoperative allergic reaction, nor were there neurotoxicity complications. USDAP permitted a shortening of operative and hospitalization time. Conclusions USDAP is an effective and safe procedure both as first line treatment and as a re-operative procedure after previous surgical failures in selected cases.

Primary hyperparathyroidism (PHPT) is relatively common among adults but rarely encountered in children and adolescents. According to the western literature, young PHPT is different from adult PHPT and is associated with more severe hypercalcemia. PHPT in the adult Indian population is different from its western counterpart. Here we present the clinical, biochemical, and surgical characteristics of young patients with PHPT treated at our tertiary care center.PHPT patients were divided into adult (≥25 years) and young (25 years) groups. The clinical, biochemical, hormonal, and histopathologic characteristics and treatment outcomes in the groups were compared.Out of 358 patients, 47 patients were young and 311 patients were adults. The mean ages of the groups were 19 ± 4 and 45 ± 12 years, respectively. The corresponding female-to-male ratios were 1.24:1 and 3.38:1 ( P.05). The nature and frequency of presenting symptoms were comparable between the 2 groups. The most common symptom in young patients with PHPT was bone pain and was not significantly different from adults (57% vs. 61%, respectively). The most common symptom in adult PHPT was fatigue, which was also not significantly different from young patients (63% vs. 53%, respectively), The serum calcium, phosphate, 25-hydroxyvitamin D levels; alkaline phosphatase Z-score; and parathyroid hormone levels were comparable between the 2 groups. Parathyroid adenoma was the most common histopathologic finding, while hyperplasia was rare in both groups.We observed that young PHPT is not markedly different from its adult counterpart in an Indian population.ALP = alkaline phosphatase; Ca = calcium; Cr = creatinine; iPTH = intact parathyroid hormone; 25(OH)D = 25-hydroxyvitamin D; P = phosphate; PHPT = primary hyperparathyroidism; PTH = parathyroid hormone; RR = reference range

Parathyroid cancer is a rare neoplasm accounting for 0.1% of all Malignancies. There is no validated indication and little experience to guide us in the diagnosis and treatment of this condition that requires a high level of clinical suspicion. The present report concerns a case of parathyroid cancer that primarily manifested through the appearance of a brown tumor of the mandible, a circumstance that up to date has only been described exceptionally. This unusual presentation of an already uncommon medical condition in an otherwise healthy and asymptomatic Woman lead to a delay in diagnosis and to futile investigations. We treated the patient performing radical en-bloc excision of the mass and of the homolateral thyroid lobe. In the absence of clear guidelines and having found no pathological nodes at surgical exploration we decided to refrain from performing a cervical lymphoadenectomy and to put the patient under strict follow-up. Further research, more information and the definition of common rules for management of parathyroid carcinoma are needed to help recognize and treat this rare neoplasm promptly and correctly.

La rupture spontanée du tendon quadricipital est rare, ainsi qu'une atteinte bilatérale et simultanée. Nous rapportons un patient de 53 ans suivi pour adénome parathyroïdien compliqué d'insuffisance rénale, admis pour rupture bilatérale et spontanée des deux tendons quadricipitaux. Il a bénéficié d'une réparation chirurgicale qui lui a permis une autonomie.

Objective: Present a case of an atypical parathyroid adenoma in the mediastinum as a part of multiple endocrine neoplasia type 1 (MEN1).Methods: Clinical, laboratory, imaging, and histolopathogic analyses were performed.Results: A 50-year-old man who was diagnosed with chronic kidney disease secondary to bilateral nephrolithiasis was referred for evaluation of incidentally detected hypercalcemia. He had an acromegaloid facies and lipomas over the forehead. Laboratory evaluation revealed hypercalcemia (3.33 mmol/L), hyperphosphatemia, and elevated intact parathyroid hormone level (215 pmol/L). Serum insulin-like growth factor 1 was elevated and growth hormone was non-suppressible on oral glucose load. Contrast-enhanced magnetic resonance imaging of the sella showed a pituitary microadenoma. A scan using 99mTc-Sestamibi and combined positron emission and computed tomography using fluorocholine F-18 revealed a mediastinal ectopic parathyroid adenoma, thereby attributing hypercalcemia to primary hyperparathyroidism. A clinical diagnosis of MEN1 was made. Genetic analysis revealed an A>T substitution at cDNA 253 in exon 2 of the MEN1 gene. He underwent median sternotomy and a 3 × 3-cm mass was excised, weighing 14 g. Histopathology showed parathyroid tissue with moderate pleomorphism, pushing margins, fibrous trabeculae, atypical mitoses, and low Ki-67 index, suggestive of atypical parathyroid neoplasm. After surgery the patient became normocalcemic. On follow-up, he is asymptomatic without any evidence of recurrence of primary hyperparathyroidism.Conclusion: Parathyroid involvement in MEN1 is almost exclusively in the form of benign hyperplasia. Atypical parathyroid adenomas and parathyroid carcinomas in MEN1 are very rare, limited to only a few anecdotal case reports to which ours contributes a new case.Abbreviations: CKD = chronic kidney disease;iPTH = intact parathyroid hormone;MEN1 = multiple endocrine neoplasia type 1

Parathyroid or hypercalcaemic crisis is a rare presenting manifestation of primary hyperparathyroidism and usually seen in relation to parathyroid carcinoma and multiple myeloma. We present a middle-aged woman with extreme hypercalcaemia due to parathyroid adenoma presenting as acute kidney injury. This case highlights the need to consider primary hyperparathyroidism in differentials of a patient presenting with severe hypercalcaemia and renal dysfunction. Also this is the second reported case in literature of a patient surviving such extreme hypercalcaemia due to parathyroid adenoma.

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The aim of this article is to explore the value of ultrasonic diagnosis and localization of intrathyroid parathyroid diseases.The medical records of 15 patients with surgically confirmed intrathyroid parathyroid diseases were reviewed. We analyzed the diagnosis process and recorded sonographic features of these intrathyroid parathyroid lesions.The patients included 11 females (73%, 11/15) and 4 males (27%, 4/15) with a mean age of 46.2 ± 10.2 years and a mean lesion size of 2.1 ± 1.1 cm. A total of 11 intrathyroid parathyroid lesions in this study presented as hypoechoic (73.3%, 11/15). Nine lesions were located in the right lobe of the thyroid (60%, 9/15), and most of the lesions were located in the middle and inferior thyroid (80%, 12/15). All of the 15 intrathyroid parathyroid lesions were variable in shape and well defined. Only 1 lesion showed microcalcification, which was confirmed as parathyroid adenocarcinoma by the postoperative pathological diagnosis. The blood supply of 13 lesions was plentiful (86.7%, 13/15). A hyperechoic line on the parathyroid lesion was detected in 13 lesions (86.7%, 13/15). Based on the degree to which the parathyroid gland was embedded in the thyroid gland, 12 cases were classified as the complete type (80%, 12/15), and 3 cases were classified as the incomplete type (20%, 3/15). Ultrasound was used to diagnose 10 cases, and sestamibi-SPECT was used to diagnose 11 cases preoperatively; the 2 imaging methods were complementary.Ultrasonic preoperative localization could be helpful in the diagnosis and management of intrathyroid parathyroid diseases.

Parathyroid hormone (PTH) has a short half-life and is cleared by the liver and kidneys. This study examined whether declining estimated glomerular filtration rate (eGFR) affects application of the Miami criterion for intraoperative PTH (ioPTH) decline during parathyroidectomy for primary hyperparathyroidism (pHPT). A retrospective review of consecutive patients undergoes parathyroidectomy for pHPT. Patients with multi-gland disease, without ioPTH, failure-to-cure and those

Rationale: Primary hyperparathyroidism (PHPT) during pregnancy is rare. Nevertheless, hypercalcemia secondary to gestational PHPT may be masked by physiological changes in calcium homeostasis during pregnancy. Gestational PHPT constitutes a serious danger to mother and fetus. Surgery is the only curative treatment when conservative treatment could not control the condition. Due to the lack of guidelines concerning PHPT during pregnancy, the optimal anesthetic management of PHPT during pregnancy needs to be individualized. Patient concerns: We report a case of PHPT with successful surgical treatment under combined cervical plexus block and general anesthesia. Diagnosis: She was diagnosed with hypercalcemia, PHPT, a possible parathyroid adenoma, and a 19-week intrauterine pregnancy. Interventions: The patient underwent heparin-free hemodialysis before the surgery in the nephrology department in the presence of a cardiologist. She then received a successful parathyroidectomy under combined bilateral superficial cervical plexus block and general anesthesia in her 19th week of pregnancy. Outcomes: She was released from the hospital with no maternal or fetal complications on postoperative day 9. A healthy baby boy was uneventfully born at 37 weeks of gestation. Lessons: We suggest that surgical removal of the lesion after lowering the blood calcium concentration in mid-pregnancy is currently the optimal treatment option for pregnant patients with PHPT. Furthermore, multidisciplinary perioperative management is particularly important.

WOS: 000422772500022, PubMed ID: 29303141, Simultaneous existence of parathyroid adenoma and thyroid nonmedullary carcinoma is rarely observed. A 52-year-old female was diagnosed approximately 4 years ago with primary hyperparathyroidism (PHPT) on the basis of hypercalcemia and elevated serum parathyroid hormone (PTH) level. Clinically, PHPT diagnosed patient was examined with Tc-99m MIBI scintigraphy to investigate parathyroid adenoma. Early Tc-99m MIBI images showed focal focused enhanced activity retention on both thyroid left lobe upper pole and thyroid lower pole inferior neighborhood. However, late images indicated thyroid activity washout and persistence of activity on left lobe lower pole. The observed lesion on left lobe lower pole neighborhood was identified as parathyroid adenoma. One-day thyroid scintigraphy was also conducted to correlate enhanced activity retention on the left lobe upper pole. Results on thyroid scintigraphy showed hypoactivity, and thyroid fine-needle aspiration cytology process was suggested from the nodule. Simultaneous existence of parathyroid adenoma and thyroid nonmedullary carcinoma is rarely observed. Diagnosis and treatment of hyperparathyroidism is crucial during preoperative period. Even observed rarely, parathyrotoxicosis diagnosed patients can develop thyroid cancer along with parathyroid adenoma.

Tumors involving the salivary, thyroid, and parathyroid glands in the pediatric age group range from benign tumors, to indeterminant malignant potential tumors to frankly malignant tumors. In addition, certain tumors may be associated with hereditary syndromes. During the past several decades, cytogenetics and molecular genetics have provided certain tumor-defining features. With the advent of rapid sequencing techniques, further definition of these tumors and identification of molecular targets for diagnosis, prognosis, and treatment have been identified. This chapter reviews the molecular features of salivary, thyroid, and parathyroid gland tumors.

Background: In the era of immunotherapy as a therapeutic option for solid tumors, the immuno-tumor characterization of parathyroid neoplasms is timely and necessary. Since conventional therapeutic options are limited for controlling the progression of parathyroid carcinoma, new modalities of treatment are desperately needed. Four distinct tumor microenvironments have been proposed based on the existence of tumor-infiltrating lymphocytes (TILs) and programmed death-ligand 1 (PD-L1); type I (TILs+/PD-L1+), type II (TILs-/PD-L-), type III (TILs-/PD-L1+), and type IV (TILs+/PD-L1-). These immunogenic subtypes may predict responses to these therapies. Aim: The aim of this study was characterized programmed death-ligand 1 (PD-L1) expression and tumoralinfiltrating lymphocytes (TILs) in parathyroid carcinoma (PC) and atypical parathyroid neoplasms (ANs). Methods: Our tissue samples constitute 30 parathyroid tumors (17 PC and 13 ANs) which were available from a prospectively collected database at the University of Texas M.D Anderson Cancer Center from 1996 to 2016. All samples were meticulously reviewed utilizing the current World Health Organization histopathological criteria. Each of the samples was immunohistochemically analysed for PD-L1, CD3, CD8 and CD68 with an automated staining system (BOND-MAX, Leica Biosystems, Buffalo Grove, IL). The Novocastra Bond Polymer Refine Detection Kit (Leica Biosystems) was used to detect PD-L1, CD3, CD8, and CD68 expression in tumor cells. Digital image analysis was performed using Aperio software (Leica Biosystems); PD-L1 expression was analysed with a tumor membrane-specific algorithm, CD3 and CD8 expression with a tumor nuclear staining algorithm, and CD68 expression was evaluated with a tumor cytoplasmic staining algorithm. The PD-L1 H score was calculated using the formula (1 × (% cells 1+))+(2 × (% cells 2+))+(3 × (% cells 3+)) and the other immunomarkers were calculated in accordance with the number of TILs positive by mm2 of the parathyroid tumor. For the immuno-classification of the PC in this study, a PD-L1 ≥ 1 was used as a cut-off for positivity. High expression of TILs was defined as cases with CD3+ and CD8+ density higher than that in the median PC group. Results: The biochemical and immunogenic characteristics of the PCs compared with ANs. The analysis of the immuno-data showed no difference in the median of PD-L1 H score between PC and ANs. All locoregional recurrences (n=6), distant metastases (n=5), and deaths due to disease (n=3) occurred in the PC group. We found four cases with a PD-L1 H score ≥ 1 associated with high expression of CD3+ (median 59.9 (27.4-986.8) cells/mm2), CD8+ (median=50.6 (4.5-1107.1) cells/mm2), and CD68+ (median 221.7 (53.0-741.1) cells/mm2) tumor cell density, and two had distant metastases (lung and liver). We identified 9 PC with a median CD68 tumor cell density>194 cells/mm2, and four in this PC group developed locoregional recurrences with or without distant metastases. We immuno-classify PC based on the four immunotypes. 14 PC cases were in the immunotype with PD-L1 negative and 4 PC cases had PD-L1 H score ≥ 1 with or without TILs positive. Conclusions: PCs tend to display immune tolerance tumor microenvironment (type IV). Additionally, 22% of PCs had patterns of PD-L1 and TILs expression in their microenvironment (type I) suggesting a potential benefit from immunotherapy. Thus, tumor microenvironment profiling could be useful to identify PC cases that could benefit from immunotherapy, and additional investigation is warranted.

Background: Multiple endocrine neoplasia type 1 (MEN1) is a rare, autosomal dominant inherited syndrome caused by mutations in the MEN1 tumor suppressor gene. The diagnosis is defined clinically by the presence of 2 or more primary MEN1 tumors (parathyroid, anterior pituitary, and pancreatic islet). We describe the case of a patient who presented with classic history and imaging findings for MEN1. Case Report: A male in his early thirties with a history of hyperparathyroidism and a transsphenoidal prolactinoma resection presented years later with abdominal symptoms concerning for Zollinger-Ellison syndrome: worsening epigastric abdominal pain, nausea, vomiting, and diarrhea. Contrast-enhanced computed tomography (CT) of the abdomen revealed hyperenhancing pancreatic lesions and duodenal inflammation, suggesting pancreatic neuroendocrine tumor (gastrinoma) with secondary duodenitis. Bilateral indeterminate hypoattenuating adrenal nodules were also seen on contrast-enhanced CT, and follow-up magnetic resonance imaging confirmed benign adrenal adenomas. Furthermore, thyroid ultrasound and sestamibi scintigraphy revealed a parathyroid adenoma. With confirmatory imaging findings, history, and presenting symptoms, the patient was clinically diagnosed with MEN1 syndrome and underwent surgical and medical management. Conclusion: This case exhibits the classic history with corresponding imaging findings of MEN1 syndrome, including pancreatic neuroendocrine tumors, parathyroid adenoma, and adrenal adenomas. High clinical suspicion for MEN1 should lead to endocrinology evaluation with appropriate laboratory workup and targeted imaging evaluation of the typical endocrine organs as described for this patient.

Ectopic parathyroids can often present a diagnostic and therapeutic conundrum for clinicians. Mediastinal parathyroid adenomas are usually small and located anteriorly within thymic tissue. To our knowledge, this is the first reported case of a large cystic parathyroid adenoma presenting as a 6-cm posterior mediastinal mass. After a successful thoracoscopic resection, parathyroid hormone levels normalized and the patient was discharged home on postoperative day 2.

To identify the importance of the ectopic, overly descended superior parathyroid adenoma variant and its prevalence in primary and reoperative parathyroid surgery and the implications for successful initial parathyroidectomy.Case series with planned data collection.Tertiary endocrine surgery practice in an academic medical center.An analysis was undertaken of 561 consecutive patients undergoing parathyroid surgery in a tertiary endocrine surgery practice from March 2004 to April 2013. There were 270 patients who had curative primary or reoperative surgery for single-gland parathyroid adenomas during this time. Clinical records, imaging studies, operative reports, and pathology findings were evaluated, and cases from a subset of patients who had an ectopic, overly descended superior parathyroid adenoma were further analyzed. The prevalence of this entity in primary and revision surgeries was calculated.Among the 270 patients with single-gland parathyroid adenomas, there were 251 primary operations and 19 reoperative procedures referred from outside institutions. An ectopic, overly descended superior parathyroid adenoma was present in 23 (9.2%) primary cases and 4 (21.1%) reoperative cases.An overly descended superior parathyroid adenoma is frequently encountered during primary parathyroid surgery. It is more than twice as common in reoperative parathyroidectomy, reflecting the propensity to be missed at the first exploration. Recognition and proper treatment of this entity at the initial operation will reduce the need for revision surgery.

The authors present clinicopathological features of parathyroid lipoadenoma in a 48-year-old woman who presented with symptomatic primary hyperparathyroidism manifesting with pathological fractures and osteoporosis. Preoperative sestamibi scan failed to localize the source of her disease. Exploratory surgery identified an enlarged parathyroid gland with abundant fat tissue. The significant drop of intraoperative serum parathyroid hormone after the removal of this gland and postoperative biochemical cure justified the presence of a single gland disease presenting as parathyroid lipoadenoma. From an educational perspective, the presented case emphasizes why the historical approach to parathyroid proliferations by assessing alone the ratio of parenchymal cells to adipocytes is not a reliable method in the diagnostic evaluation of parathyroid disease. While the accurate size and weight of a parathyroid gland are defining parameters of an abnormal gland, intraoperative and postoperative biochemical workup distinguishes uniglandular disease (adenoma) from multiglandular disease (hyperplasia). The authors also provide a brief review of the previously published cases of parathyroid lipoadenomas to highlight their clinicopathological characteristics of relevance to surgical pathologists.

Objectives: Parathyroid carcinoma (PC) is a rare parathyroid tumor compared to parathyroid adenoma (PA) and atypical parathyroid adenoma (APA). Recent studies have suggested parafibromin has a role in the differential diagnosis of parathyroid tumors. We sought to determine the role of parafibromin as well as galectin-3, Ki-67, and HBME-1 as diagnostic markers in the differential diagnosis of parathyroid tumors. Methods: A total of 92 cases diagnosed with PA, APA, or PC at Sifa University and Private Ege Pathology Laboratory between 2006–2012 were included in the study. Parafibromin (microarray), galectin-3, Ki-67, and HBME-1 were evaluated using immunohistochemistry in all parathyroid tumors. Results: Eighty-four cases were diagnosed with PA, six with APA, and two with PC. The study group consisted of 82 females and 10 males. Their mean age was 50.9 years, and the mean tumor diameter was 1.97 cm. Parafibromin was negative in the two PC cases but positive in all APA and PA cases. Positivity was observed with galectin-3 in 17 adenoma cases, three atypical adenomas, and two carcinoma cases. Positivity with HBME-1 was found in 26 PA cases and one PC case. Parafibromin and galectin-3 expression was significant between the three tumor groups but not for HBME-1 expression. Parafibromin expression increased in PA whereas galectin-3 expression decreased. A statistical significance was found between the three tumor groups according to the Ki-67 score (p=0.010). Additionally, the Ki-67 proliferation index was under 1% in PAs. Conclusion: The number of PCs in our series was small so our data mostly reflects the immunohistochemical characteristics of PAs. Parafibromin expression, galectin-3 negativity, and a Ki-67 proliferation index under 1% were estimated as beneficial in the differential diagnosis of difficult parathyroid tumors. © 2015, Oman Medical Specialty Board. All rights reserved.

BackgroundProper localization is crucial in performing minimally invasive parathyroidectomy for primary hyperparathyroidism. Ultrasonography (US) and Tc-99m sestamibi (MIBI) scintigraphy are common methods used for localization. As the appearance and activity of the thyroid gland may impact parathyroid localization, the purpose of this study was to determine how exogenous use of the thyroid hormone, levothyroxine (LT), affects parathyroid localization.MethodsAdult patients with non-familial primary hyperparathyroidism who underwent initial parathyroidectomy from 2000-2014 were retrospectively identified. LT (+LT) and non-LT (-LT) patients were matched 1:3 based on age, gender, goiter status, and preoperative parathyroid hormone levels. Subgroup analysis was performed on patients previously treated with radioactive iodine and patients undergoing single adenoma resection.ResultsOf the 1737 patients that met inclusion criteria, 286 were on LT at the time of their parathyroid localization scan. Use of LT did not impact the percentage of correct MIBI localization scans when compared with -LT patients (P=0.83). Interestingly, use of LT significantly hindered localization by US in comparison with the -LT group (48.4 versus 62.2%, P&nbsp

Non-functional parathyroid carcinoma is an exceedingly rare disease with 31 reported cases since 1909. Because of the scarce number of cases of non-functional parathyroid carcinoma, there are no evidence-based recommendations for its optimal treatment. Surgery, including en bloc resection of the carcinoma, ipsilateral thyroid lobe and isthmus together with a neck dissection only in case of lymph node involvement, is the main treatment for non-functioning parathyroid carcinoma. The patient usually has a poorer prognosis because of detection at advanced stages, the relative ineffectiveness of adjuvant treatment modalities and the lack of adequate parameters for clinical follow-up. In this report, we present a case of non-functional parathyroid carcinoma at our institution, and we review the previous literature to discuss the latest advances in the diagnosis and treatment of this rare disease.

BACKGROUND AND PURPOSE: Minimally invasive parathyroidectomy requires accurate preoperative localization of suspected adenomas, and multiphase CT allows adenoma characterization while providing detailed anatomic information. The purpose of this study was to assess the feasibility of a protocol using only arterial and venous phases to localize pathologic glands in patients with primary hyperparathyroidism. MATERIALS AND METHODS: We identified 278 patients with primary hyperparathyroidism who had undergone 2-phase CT with surgical cure. All scans were read prospectively by board-certified neuroradiologists. A neuroradiology fellow retrospectively reviewed images and reports and classified suspected adenomas on the basis of anatomic location. Accuracy was determined by comparing imaging results with surgical findings. The ability of 2-phase CT to localize adenomas to 1 of 4 neck quadrants and lateralize them to the correct side was assessed. Accuracy of identifying multigland disease was also evaluated. RESULTS: In patients with single-gland disease, the sensitivity and specificity of 2-phase CT to correctly localize the quadrant were 55.4% and 85.9%, respectively. The sensitivity and specificity of correct lateralization were 78.8% and 67.8%, respectively. The sensitivity and specificity to identify multigland disease were 22.9% and 79.5%, respectively. CONCLUSIONS: While the 2-phase CT protocol in this study demonstrates lower accuracy compared with reports of other techniques, its lower radiation compared with 3- and 4-phase techniques may make it a feasible alternative for preoperative parathyroid localization. Further prospective studies are needed to identify patients for whom this technique is most suitable.

Background Intraneural parathyroid adenomas are rare, with only 9 cases of intravagal adenomas reported. All but one of the reported cases was found after multiple neck explorations. To the best of our knowledge, we report the first case of nonsupernumerary ectopic intravagal parathyroid identified at primary exploration. Methods and Results A 17-year-old girl with primary hyperparathyroidism and nephrolithiasis was referred with a sestamibi scan reporting a left lower parathyroid adenoma. No eutopic parathyroid tissue was identified during full exploration of the left side of the neck. Exploration of the carotid sheath revealed a fusiform swelling of the vagus nerve at the level of the carotid bifurcation. Longitudinal incision of the vagal perineurium revealed a 7-mm parathyroid adenoma, which was enucleated. The patient recovered uneventfully, with normalization of serum calcium, parathyroid hormone (PTH), and normal vocal cord function. Conclusion We believe that this is the first reported case of nonsupernumerary intravagal parathyroid adenoma resected at initial exploration. The vagus nerve is a rare location for a parathyroid adenoma, but one that should be considered, even during primary exploration. © 2015 Wiley Periodicals, Inc. Head Neck 37: E200–E204, 2015

There is uncertainty about accuracies of dual-phase (DP) and dual-tracer (DT) parathyroid scintigraphy with the newly added SPECT/CT. Although SPECT/CT was shown to be helpful in parathyroid adenoma (PA) localization, it may not have optimal resolution as pinhole. This study directly compared diagnostic accuracies and confidences of various imaging protocols on same patients.One hundred fifty-five patients with pathologically confirmed diagnosis were included. Pinhole DP, pinhole DT, pinhole DP SPECT/CT, pinhole DT SPECT/CT, and SPECT/CT with only pinhole-delayed MIBI (D) were reviewed for accuracies and certainties of PA diagnosis/localization. Parathyroid adenomas were classified as clearly or unclearly distinguishable from thyroid. Furthermore, the contribution of pinhole DP to pinhole DT SPECT/CT was assessed.Of 153 PAs, the correct diagnosis/localization was significantly higher by pinhole DT SPECT/CT than pinhole DP SPECT/CT, SPECT/CT D, pinhole DT alone, and DP alone. Parathyroid adenomas were clearly more distinguished from thyroid in pinhole DT than DP with/without SPECT/CT. Consequently, PA diagnosis certainty was higher in pinhole DT than DP, whereas PA localization certainty was higher in both with SPECT/CT. In pinhole DT SPECT/CT, the pinhole DP addition confirmed diagnosis/localization of only 24 uncertain PAs.In this large patient group, the accuracy and certainty of PA diagnosis/localization were higher in pinhole DT SPECT/CT than all other parathyroid scintigraphy protocols. Pinhole DT better identified PA than pinhole DP, whereas SPECT/CT improved PA localization in both protocols. Pinhole DP showed limited contribution and thus should be only considered when PA diagnosis/localization is uncertain by pinhole DT SPECT/CT.

Purpose Patients with primary hyperparathyroidism routinely undergo preoperative imaging to localize the abnormal gland to facilitate a guided parathyroidectomy. These techniques include neck ultrasound (US), dual phase planar technetium-99 m (99mTC) sestamibi (MIBI) scans, single photon emission computed tomography (SPECT), combined SPECT/CT, and four dimensional CT scans (4D CT). Despite appropriate preoperative imaging, non-localization of abnormal glands does occur. This study aims to determine whether non-localization is the result of radiologic interpretive error or a representation of a subset of truly non-localizing parathyroid adenomas. Materials and methods A retrospective study was performed; two senior radiologists reinterpreted the preoperative imaging (US and MIBI scans) of 30 patients with initially non-localizing studies. All patients underwent parathyroidectomy for primary hyperparathyroidism at a tertiary referral center. Both radiologists were blinded to the scores of his colleague. The results were compared for inter-reader reliability using Cohen’s kappa test. Results Twenty-nine of thirty nuclear studies were found to be negative on reinterpretation. The readers agreed in 86.67% of their observations, with a kappa (κ) value of 0.706 (SE = ± 0.131, 95% confidence interval for κ = 0.449–0.962). One of eighteen ultrasounds had positive localizations on reexamination, however, the inter-observer agreement was only 55.6%, with a kappa value of 0.351 (SE = ± 0.139, and 95% confidence interval for κ = 0.080–0.623). Overall, no statistically significant difference in preoperative and retrospective interpretation was found. Conclusion This study identifies a subset of parathyroid adenomas that do not localize on preoperative imaging despite sound radiographic evaluation.

Objective: Parathyroid gland malignancies are considered rare. The most common of these tumor types is primary parathyroid carcinoma. Metastatic spread from other cancers may also occur with up to 10% of cancers from other sites showing parathyroid involvement at autopsy. Tumor-to-tumor metastases (metastatic spread to parathyroid neoplasm) from remote cancers to the parathyroid gland have been described. Methods: We did a PubMed literature review and analysis of our own experience of 392 consecutive parathyroidectomies. Results: Primary and secondary parathyroid malignancies can be grouped into three categories: primary parathyroid carcinoma (PPCa), spread of carcinoma into parathyroid glands by contiguous extension from the thyroid gland or other head and neck cancer, and metastatic disease to the parathyroid gland from distant cancers. Studies of tumor-to-tumor spread indicate a predilection of spread to endocrine tumors possibly because of the rich blood supply that is present in endocrine tumors. Two of our 392 parathyroidectomies (0.5%) had cancer: one metastatic (thymic neuroendocrine tumor) and another PPCa. Conclusion: Metastatic disease to the parathyroid gland is poorly documented. When performing surgery for primary thyroid cancer, the search for parathyroid gland metastases is often overlooked because of the desire to preserve parathyroid function. Metastatic disease from other cancers to a benign parathyroid gland or to a parathyroid adenoma probably suggests a grave prognosis because it likely indicates widespread metastatic disease; however, isolated metastases to the parathyroid may occur. Although these lesions may be uncommon they may not be as rare as once thought.

Precise preoperative localization is essential for focussed parathyroidectomy. The imaging standard consists of cervical ultrasonography (cUS) and 99mTc-MIBI-SPECT (MIBI-SPECT). 11C-methionine positron emission tomography/computed tomography (Met-PET/CT) is a promising method for localizing parathyroid adenomas. The objective of our study was to elucidate whether additional Met-PET/CT increases the rate of focussed parathyroidectomy. Fourteen patients with primary hyperparathyroidism (HPT) and three patients with tertiary HPT underwent cUS and MIBI-SPECT. Met-PET/CT was carried out in patients with negative MIBI results. Subsequent surgical strategy was adapted according to imaging results. cUS localized a single parathyroid adenoma in 10/17 patients (59 %), while MIBI-SPECT/CT identified 11/17 single adenomas (65 %). In the remaining six patients, Met-PET/CT identified five single adenomas. This step-up approach correctly identified single adenomas in 16/17 patients (94 %). Met-PET/CT raises the rate of correctly localized single parathyroid adenomas in patients with negative cUS and MIBI-SPECT/CT and increases the number of focussed surgical approaches.

Components of the tumour microenvironment initiate and promote cancer development. In this study, we investigated the stromal component of parathyroid neoplasia. Immunohistochemistry for alpha-smooth muscle actin (α-SMA) showed an abundant periacinar distribution of α-SMA+ cells in normal parathyroid glands (n=3). This pattern was progressively lost in parathyroid adenomas (PAds; n=6) where α-SMA+cells were found to surround new microvessels, as observed in foetal parathyroid glands (n=2). Moreover, in atypical adenomas (n=5) and carcinomas (n=4), α-SMA+ cells disappeared from the parenchyma and accumulated in the capsula and fibrous bands. At variance with normal glands, parathyroid tumours (n=37) expressed high levels of fibroblast-activation protein (FAP) transcripts, a marker of tumour-associated fibroblasts. We analysed the ability of PAd-derived cells to activate fibroblasts using human bone-marrow mesenchymal stem cells (hBM-MSCs). PAd-derived cells induced a significant increase in FAP and vascular endothelial growth factor A (VEGFA) mRNA levels in co-cultured hBM-MSCs. Furthermore, the role of the calcium-sensing receptor (CASR) and of the CXCL12/CXCR4 pathway in the PAd-induced activation of hBM-MSCs was investigated. Treatment of co-cultures of hBM-MSCs and PAd-derived cells with the CXCR4 inhibitor AMD3100 reduced the stimulated VEGFA levels, while CASR activation by the R568 agonist was ineffective. PAd-derived cells co-expressing parathyroid hormone (PTH)/CXCR4 and PTH/CXCL12 were identified by FACS, suggesting a paracrine/autocrine signalling. Finally, CXCR4 blockade by AMD3100 reduced PTH gene expression levels in PAd-derived cells. In conclusion, i) PAd-derived cells activated cells of mesenchymal origin; ii) PAd-associated fibroblasts were involved in tumuor neoangiogenesis and iii) CXCL12/CXCR4 pathway was expressed and active in PAd cells, likely contributing to parathyroid tumour neoangiogenesis and PTH synthesis modulation.

Background Hyperparathyroidism-jaw tumor syndrome (HPT-JT) is a rare autosomal dominant disease secondary to germline inactivating mutations of the tumor suppressor gene HRPT2/CDC73. The aim of the present study is to determine the optimal surgical approach to parathyroid disease in patients with HPT-JT.

A 7½-yr-old male cougar (Puma concolor) was presented with a 2-wk history of progressive hindlimb abnormalities. An abdominal mass was palpated on physical examination. Computed tomography of the abdomen showed a mass surrounding the left ureter. A postmortem diagnosis of paraganglioma was established.

Background: The intraoperative parathyroid hormone (IOPTH) assay is widely used in patients with primary hyperparathyroidism (PHPT). We investigated the usefulness of the IOPTH assay in Korean patients with PHPT. Methods: We retrospectively reviewed the data of 33 patients with PHPT who underwent parathyroidectomy. Neck ultrasonography (US) and 99m Tc-sestamibi scintigraphy (MIBI scan) were performed preoperatively and IOPTH assays were conducted. Results: The sensitivity of neck US and MIBI scans were 91% and 94%, respectively. A 50% decrease in parathyroid hormone (PTH) levels 10 minutes after excision of the parathyroid gland was obtained in 91% (30/33) of patients and operative success was achieved in 97% (32/33) of patients. The IOPTH assay was 91% true-positive, 3% true-negative, 0% false-positive, and 6% falsenegative. The overall accuracy of the IOPTH assay was 94%. In five cases with discordant neck US and MIBI scan results, a sufficient decrease in IOPTH levels helped the surgeon confirm the complete excision of the parathyroid gland with no additional neck exploration. Conclusion: The IOPTH assay is an accurate tool for localizing hyperfunctioning parathyroid glands and is helpful for evaluating cases with discordant neck US and MIBI scan results.

The conventional approach for preoperative parathyroid localization with the dual 99mTc-sestamibi (99mTc-MIBI) and 99mTc-pertechnetate (99mTcO 4 − ) scintigraphy technique obtains the differential image by subtracting images from the two scans; the former depicts both the thyroid and parathyroid glands while the latter depicts the thyroid only. In this study, we developed a novel method based on Poisson noise modeling and maximum-likelihood estimation to generate the differential image in an iterative fashion using both planar images jointly. We demonstrated improved performance of our joint method as compared with the subtraction method in both phantom and patient studies. In the phantom study, we acquired two noise-free planar datasets using 99mTc on an in-house thyroid phantom and a “lesion” bead (representing a parathyroid gland) with the same attenuation background as the thyroid phantom. These two planar datasets were combined and scaled to simulate noise-free clinical MIBI (four lesion-to-background contrast (LBC) values: 1.2, 1.3, 1.4 and 1.5), and 99mTcO 4 − datasets. One-hundred Poisson noise realizations were generated for each datasets. The mean and standard deviation (SD) of the lesion contrast in the differential images were computed for both the subtraction and the joint methods. We also applied both the subtraction and the joint methods to one parathyroid patient dataset. The voxel-wise mean-to-SD ratios in four hyperfunctioning parathyroid lesions were calculated. The phantom results showed that the joint method at the 50th iteration yielded a significant SD reduction compared with the subtraction method ranging from 20% to 45% (p < 0.05). Similarly, the voxel-wise mean-to-SD ratios were substantially improved in the patient study from 0.40–1.60 (subtraction) to 2.68–3.16 (joint).

International audience; PURPOSE:To evaluate the sensitivity of F18-choline (FCH) PET/CT for parathyroid adenoma detection prior to surgery in patients with primary hyperparathyroidism and negative or inconclusive cervical ultrasound and Tc99m-sestaMIBI SPECT/CT.METHODS:We conducted a prospective bicentric study (NCT02432599). All patients underwent FCH PET/CT. The result was scored positive, inconclusive or negative. The number of uptakes and their sites were recorded. The FCH PET/CT result guided the surgical procedure (minimally invasive parathyroidectomy, bilateral cervical exploration, or other in case of multiple or ectopic foci). FCH PET/CT results were compared to the surgical and pathological findings and the follow-up.RESULTS:Twenty-five patients were included. Mean calcium and PTH levels prior to surgery were 2.76 ± 0.17 mmol/l and 94.8 ± 37.4 ng/l. Nineteen (76%) FCH PET/CTs were scored positive, 3 (12%) inconclusive and 3 (12%) negative, showing 21 cases of uniglandular disease, including 1 ectopic localization and 1 case of multiglandular (3 foci) disease. Mean lesion size was 13.1 ± 8.6 mm. Twenty-four patients underwent surgery. FCH PET/CT guided surgery in 22 (88%) patients, allowing for 17 minimally invasive parathyroidectomies, 1 bilateral cervical exploration for multifocality and 4 other surgical procedures. Two patients with negative FCH-PET/CT underwent bilateral cervical exploration. When dichotomizing the FCH PET/CT results, thereby classifying the inconclusive FCH PET/CT results as positive, the per lesion and per patient sensitivities were 91.3% (95%CI: 72.0-98.9) and 90.5% (95%CI: 69.6-98.8) and the corresponding positive predictive values were 87.5% (95%CI: 67.6-97.3) and 86.4% (95%CI: 65.1-97.1), respectively. Twenty-one (88%) patients were considered cured after surgery. Their mean calcium level after surgery was 2.36 ± 0.17 mmol/l.CONCLUSIONS:Preoperative FCH PET/CT has a high sensitivity and positive predictive value for parathyroid adenoma detection in patients with primary hyperparathyroidism and negative or inconclusive conventional imaging results. Bilateral cervical exploration could be avoided in the majority (75%) of patients.

ContextA subset of PHPT patients exhibit a more severe disease phenotype characterized by bone loss, fractures, recurrent nephrolithiasis, and other dysfunctions, but the underlying reasons for this disparity in clinical presentation remain unknown.ObjectiveWe sought to identify new mechanistic indices that could inform more personalized management of PHPT.DesignPre-, peri-, and postoperative data and demographic, clinical, and pathological information from patients undergoing parathyroidectomy for PHPT were collected. Univariate and partial Spearman correlation was used to estimate the association of parathyroid tumor calcium sensing capacity with select variables.Patients or other participantsAn unselected series of 237 patients aged >18years and undergoing parathyroidectomy for PHPT were enrolled.Main outcome measuresCalcium sensing capacity, expressed as the concentration required for half-maximal biochemical response (EC50), was evaluated in parathyroid tumors from an unselected series of 74 patients and assessed for association with clinical parameters. The hypothesis was that greater disease severity would be associated with attenuated calcium sensitivity and biochemically autonomous parathyroid tumor behavior.ResultsParathyroid tumors segregated into two distinct groups of calcium responsiveness (EC50

INTRODUCTION Patients with primary hyperparathyroidism generally have a single parathyroid adenoma that causes excessive excretion of parathyroid hormone. For about 2% to 15% of these patients, a double adenoma is present that involves one lesion on each side of the neck. CASE PRESENTATION We describe a case of double parathyroid adenoma causing asymptomatic hypercalcemia. A presurgical technetium99m (Tc99m) sestamibi scan suggested an ipsilateral double adenoma in the left thyroid lobe. An intraoperative parathyroid hormone assay confirmed its successful removal. DISCUSSION Although double adenomas are not yet widely acknowledged, presurgical imaging and nuclear scans can help to localize multiple lesions, and intraoperative parathyroid hormone assays can confirm the diagnosis and cure.

Wstęp: Rak przytarczycy jest jednym z rzadziej występujących nowotworów układu endokrynnego i odpowiada za mniej niż 1% wszystkich przypadków pierwotnej nadczynności przytarczyc. W porównaniu do zmian łagodnych, objawy kliniczne (m.in. obecność guza zzajęciem okolicznych węzłów chłonnych lub odległych przerzutów), parametry biochemiczne i aktywność metaboliczna raka przytarczycy są z reguły bardziej widoczne. W przedstawionym przypadku nie było objawów, które uzasadniałyby przedoperacyjne rozpoznanie raka przytarczycy.Opis przypadku: Przedstawiono przypadek 52-letniej pacjentki z nawrotowa kamicą nerkową, umiarkowanym nadciśnieniem tętniczym, podwyższonym stężeniem wapnia całkowitego i zjonizowanego, niskim stężeniem potasu i podwyższonym poziomym kompletnego parathormonu. Obraz kliniczny i badania obrazowe (badanie ultrasonograficzne, scyntygrafia SPECT/CT) wskazywały na gruczolaka przytarczycy, jednakże za pomocą badania histopatologicznego usuniętej zmiany wykazano raka przytarczycy. Podczas reoperacji nie stwierdzono zajęcia okolicznych węzłów chłonnych. Uzyskano normalizację parametrów biochemicznych i redukcję nadciśnienia tętniczego z umiarkowanego do łagodnego stopnia podczas rocznej obserwacji chorej.Wnioski: 1. U pacjentów z pierwotną nadczynnością przytarczyc spowodowaną rakiem przytarczycy skuteczne leczenie operacyjne nie tylko doprowadza do normalizacji stężenia wapnia, fosforanów i parathormonu, ale również może poprawić skuteczność leczenia nadciśnienia tętniczego. 2. Rak przytarczycy może uwidocznić się jako ognisko o śladowej aktywności metabolicznej w badaniu SPECT/CT i równocześnie wykazywać sekrecję parathormonu w zakresie wartości bardziej charakterystycznych dla łagodnej patologii przytarczyc.

Cushing's syndrome is a rare disease. Cushing's syndrome presenting as acute psychosis is an exceptional occurrence. We present the case of a 37-year-old woman who was admitted with acute confusion associated with mild hypercalcaemia and was subsequently diagnosed with parathyroid and adrenal adenomas. Our hospital sees approximately 6000 endocrine patients per year, with an incidence of around four Cushing's cases annually. This is the first such case to occur in our hospital and one of few described in the literature.

Parathyroid glands of young adults consist primarily of chief cells. However, with age or after excessive functional stress, another cell type increases progressively—the oxyphil cell. There is evidence for a chief-to-oxyphil cell transdifferentiation in chronic kidney disease. The latter may represent a defense mechanism, transforming the actively secreting chief cells to a less actively secreting cell type. However, even if this strategy is able to delay the development of secondary hyperparathyroidism, it cannot prevent it.