Multiple pathological fractures and muscle atrophy caused by a parathyroid carcinoma with postoperative hungry bone syndrome: A case report.

BACKGROUND: Parathyroid carcinoma (PC) is a rare endocrine malignancy causing pathological changes such as abnormal bone metabolism, elevated serum calcium, and impaired renal function, and uncontrollable hypercalcemia is the main cause of death in PC patients. The diagnosis of PC is challenging and relying on postoperative histopathology. Radical surgery at the first time is the only effective therapy to cure PC. Hungry bone syndrome (HBS) is a relatively uncommon complication of parathyroidectomy characterized by profound and prolonged hypocalcemia, timely electrolyte monitoring and alternative interventional protocols can prevent symptomatic hypocalcemia. CASE: A 57-year-old man presented with multiple pathological fractures and muscle atrophy as the main symptoms accompanied by bone pain, hypercalcemia, elevated parathyroid hormone (PTH), and an enlarged left-sided neck mass. After consultation of multidisciplinary team, he was treated conservatively with plaster bandage fixation and infusion of intravenous zoledronic acid; and then complete resection of parathyroid mass + removal of involved tissue structures + left thyroid and isthmus lobectomy + lymph node dissection in the VI region in left neck were performed. The postoperative histopathology suggested a diagnosis of parathyroid carcinoma. Calcium and fluid supplementation and oral levothyroxine tablets were given postoperatively. Unexpectedly, the patient's PTH level decreased rapidly at 24 h postoperative, and serum calcium and phosphorus decreased continuously, and he felt numb around perioral sites and fingertips, which considered to be postoperative HBS complicated by parathyroidectomy. Then, a large amount of calcium supplementation and vitamin D were given timely and the patient got better at 1 month postoperatively. At 9-month postoperative, his bone pain and fatigue were significantly relieved compared with before with calcium, phosphorus, and PTH levels at normal range. CONCLUSION: The possibility of parathyroid disease, particularly PC, should be considered in the presence of multiple pathological fractures, muscle atrophy, generalized bone pain, hypercalcemia, and clear neck mass. Radical resection of the tumor lesions at the first surgery is a key element affecting the prognosis of PC, and the effective management of preoperative hypercalcemia and postoperative HBS is also of great significance for improving prognosis.

Persistent hypercalcaemia associated with two pathogenic variants in the CYP24A1 gene and a parathyroid adenoma-a case report and review.

Introduction: 24-Hydroxylase, encoded by the CYP24A1 gene, is a crucial enzyme involved in the catabolism of vitamin D. Loss-of-function mutations in CYP24A1 result in PTH-independent hypercalcaemia with high levels of 1,25(OH)2D3. The variety of clinical manifestations depends on age, and underlying genetic predisposition mutations can lead to fatal infantile hypercalcaemia among neonates, whereas adult symptoms are usually mild. Aim of the study: We report a rare case of an adult with primary hyperparathyroidism and loss-of-function mutations in the CYP24A1 gene and a review of similar cases. Case presentation: We report the case of a 58-year-old woman diagnosed initially with primary hyperparathyroidism. Preoperatively, the suspected mass adjoining the upper pole of the left lobe of the thyroid gland was found via ultrasonography and confirmed by 99mTc scintigraphy and biopsy as the parathyroid gland. The patient underwent parathyroidectomy (a histopathology report revealed parathyroid adenoma), which led to normocalcaemia. After 10 months, vitamin D supplementation was introduced due to deficiency, and the calcium level remained within the reference range. Two years later, biochemical tests showed recurrence of hypercalcaemia with suppressed parathyroid hormone levels and elevated 1,25(OH)2D3 concentrations. Further investigation excluded the most common causes of PTH-independent hypercalcaemia, such as granulomatous disease, malignancy, and vitamin D intoxication. Subsequently, vitamin D metabolites were measured using LC-MS/MS, which revealed high levels of 25(OH)D3, low levels of 24,25(OH)2D3 and elevated 25(OH)2D3/24,25(OH)2D3 ratios, suggesting a defect in vitamin D catabolism. Molecular analysis of the CYP24A1 gene using the NGS technique revealed two pathogenic variants: p.(Arg396Trp) and p.(Glu143del) (rs114368325 and rs777676129, respectively). Conclusions: The diagnostic process for hypercalcaemia becomes complicated when multiple causes of hypercalcaemia coexist. The measurement of vitamin D metabolites using LC-MS/MS may help to identify carriers of CYP24A1 mutations. Subsequent molecular testing may contribute to establishing the exact frequency of pathogenic variants of the CYP24A1 gene and introducing personalized treatment.

Parathyroid carcinoma: impact of preoperative diagnosis on the choice of surgical procedure.

The operative procedure in the surgical treatment of parathyroid carcinoma differs from that of benign hyperparathyroidism. However, preoperative differentiation is often difficult. This study elucidated how clinicians diagnose parathyroid carcinoma and the relationship between preoperative diagnosis and the operative course. Using a retrospective chart review, twenty cases of parathyroid carcinoma from nine participating centers were examined. In 11 cases with preoperative suspicion of malignancy, at least one of these three features was found: elevated serum calcium level (>14 mg/dL), palpable mass, and irregular margin on ultrasonography. Although an intact parathyroid hormone (iPTH) threshold to suspect malignancy has not been established, six cases showed marked iPTH elevation exceeding 8.0 times the upper limit of normal. One case was excluded from analysis due to hemodialysis. Compared with the four cases that showed calcium elevation, the iPTH threshold might represent better sensitivity. Among 9 cases of benign preoperative diagnosis, six cases were performed with pericapsular resection. In three cases where malignancy was suspected in the middle of the operation, the recommended en bloc resection with ipsilateral thyroid lobectomy was not performed but a parathyroidectomy with surrounding soft tissue. In contrast, 10 preoperatively suspected cases underwent en bloc resection, and one case underwent pericapsular resection followed by supplementary ipsilateral hemithyroidectomy due to the uncertain pre- and intraoperative findings to determine the diagnosis. In conclusion, the surgical procedure for parathyroid carcinoma strongly depends on the preoperative diagnosis. The presence of excessive iPTH levels might contribute to improved preoperative diagnostic sensitivity for parathyroid carcinoma.

The Clinical Features and Treatment Strategy of Parathyroid Cancer: A Retrospective Analysis.

Objective: To review the features and treatment of parathyroid cancer in our series. Explore the suitable extent of initial surgery and the effect of adjuvant radiotherapy in local recurrence. Methods: Seven cases of parathyroid cancer presented from 2014 to 2021. The presenting features, diagnosis, and treatment are presented. Results: Only two patients had multiple manifestations of hypercalcemia. Marked hypercalcemia, which was revealed to be an average of 13.9 mg/dl (range from 11.8 mg/dl to 15.8 mg/dl), was observed in four patients (57%). The others' serum calcium levels were in the normal range with an average of 9.9 mg/dl (range from 8.6 mg/dl to 10.8 mg/dl). All seven patients had hyperparathyroidism with an average of 733 pg/ml (range from 113 pg/ml to 3193 pg/ml). En bloc resection was performed in two patients with neighboring structure invasion, and four patients with complete tumor capsules underwent tumor resection with limited resection of the thyroid gland. Postoperative adjuvant radiotherapy appeared unsuccessful for local recurrence. Conclusion: High calcium, high PTH, parathyroid occupation by ultrasound, and intraoperative invasion should be considered to have the possibility of parathyroid cancer. Open surgery is recommended and protecting tumor integration is the elementary surgery principle. The initial surgical extent should be decided by the invasion of the tumor. When PC has a local recurrence, the debulking surgery and adjuvant radiotherapy are always fake.

Successful Control of Hypercalcemia with Sorafenib, Evocalcet, and Denosumab Combination Therapy for Recurrent Parathyroid Carcinoma.

Parathyroid carcinoma (PC) is a rare type of endocrine cancer. Recurrence and metastasis are common after surgery, and refractory hypercalcemia often leads to a poor prognosis. However, there are currently no specific strategies for PC recurrence. We herein report a 61-year-old Japanese man with metastatic PC who was treated with sorafenib, a multikinase inhibitor. In this case, the serum calcium level was under control for 10 months after the initiation of sorafenib. This case suggests that combination therapy with sorafenib, evocalcet, and denosumab may be an alternative, stronger management option for refractory hypercalcemia in recurrent PC.

Parathyroid carcinoma: Single centre experience.

OBJECTIVE: Parathyroid Carcinoma is a rare malignant neoplasm, accounting for less than 1% of primary hyperparathyroidism cases. Parathyroid carcinomas are characterized by markedly elevated levels of PTH, severe hypercalcemia and established target organ damage. The authors report the experience of a single centre regarding the management and outcome of patients with parathyroid carcinomas and revise relevant literature. DESIGN: Retrospective review of all patients with parathyroid carcinoma evaluated at a tertiary oncologic centre from 1991 until 2021. RESULTS: Seventeen patients were identified (10 males), with a mean age at diagnosis of 53 ± 16 years and a median follow-up of 16.5 years. Most patients presented with hypercalcemia (n = 15), with a mean serum calcium concentration of 13.5 mg/dl (9.6-16.5) and mean PTH of 1173 pg/ml (276-2500). Hyperparathyroidism-mediated organ damage was observed in most patients (n = 16), with predominant renal (n = 12) and skeletal (n = 9) complications. En bloc surgical resection was performed in nine patients. Three patients underwent adjuvant radiotherapy. Recurrence was observed in 8 cases (47.1%) after a median of 24 months following surgery and no independent predictors of recurrence were identified. The overall survival and disease specific survival at 5-year was 88% and 94%, respectively. CDC73 mutations were present in 38.5% of analysed patients and one patient was diagnosed with MEN1. CONCLUSION: Parathyroid carcinoma is associated with a significant rate of recurrence and limited effective treatment beyond initial complete surgical resection. Therefore, preoperatively high index of suspicion is paramount to optimize patient care. This is, to our knowledge, the largest Portuguese cohort published so far.

Incidence, survival time, and surgical treatment of parathyroid carcinomas in dogs: 100 cases (2010-2019).

OBJECTIVE: To evaluate outcomes of dogs with parathyroid carcinoma (PTC) treated by surgical excision and to describe the incidence of postoperative hypocalcemia, degree of hypocalcemia, duration of hospitalization, duration of calcium supplementation, and survival time. ANIMALS: 100 client-owned dogs with PTC admitted to academic, referral veterinary institutions. PROCEDURES: In a retrospective multi-institutional study, medical records of dogs undergoing surgical excision of PTC between 2010 to 2019 were reviewed. Signalment, relevant medical history, clinical signs, clinicopathologic testing, imaging, surgical findings, intraoperative complications, histologic examination, and survival time were recorded. RESULTS: 100 dogs with PTC were included, and 96 dogs had clinical or incidental hypercalcemia. Common clinical signs included polyuria (44%), polydipsia (43%), hind limb paresis (22%), lethargy (21%), and hyporexia (20%). Cervical ultrasonography detected a parathyroid nodule in 91 of 91 dogs, with a single nodule in 70.3% (64/91), 2 nodules in 25.3% (23/91), and ≥ 3 nodules in 4 (4/91)% of dogs. Hypercalcemia resolved in 89 of 96 dogs within 7 days after surgery. Thirty-four percent of dogs developed hypocalcemia, on the basis of individual analyzer ranges, within 1 week after surgery. One dog had metastatic PTC to the prescapular lymph node, and 3 dogs were euthanized for refractory postoperative hypocalcemia. Estimated 1-, 2-, and 3-year survival rates were 84%, 65%, and 51% respectively, with a median survival time of 2 years. CONCLUSIONS AND CLINICAL RELEVANCE: Excision of PTC results in resolution of hypercalcemia and excellent long-term tumor control. Surgical excision of PTC is recommended because of resolution of hypercalcemia and a good long-term prognosis. Future prospective studies and long-term follow-up are needed to further assess primary tumor recurrence, metastasis, and incidence of postoperative hypocalcemia.

A germline c.1546dupC MEN1 mutation in an MEN1 family: A case report.

RATIONALE: Multiple endocrine neoplasia type 1 (MEN1) is a rare tumor syndrome with an autosomal dominant inheritance, and genetic testing for MEN1 gene is important for both affected individuals and their relatives. We present a 2-person family affected by a germline c.1546dupC MEN1 mutation, and one of them had a full-spectrum of MEN-related endocrine tumors. PATIENT CONCERNS: A female patient aged 32 years presented with jejunal ulcer perforation due to gastrinoma. DIAGNOSES: We conducted genetic analysis and extensive biochemical/radiological evaluation for detecting other endocrine tumors. Multiple pancreatic neuroendocrine tumors (NETs), prolactinoma and primary hyperparathyroidism were diagnosed, and a frame-shift mutation, NM_130799.1:c.1546dupC (p.Arg516Profs∗15), was detected. One daughter of the proband, aged 12 years, had the same mutation for MEN1. INTERVENTION: She underwent pancreatic surgery for pancreatic NETs and total parathyroidectomy for primary hyperparathyroidism. OUTCOMES: After pancreatic surgery, long-term symptoms of epigastric soreness, acid belching, sweating, and palpitation in fasting were improved. Hypercalcemia was improved after parathyroidectomy and she was supplemented with oral calcium and vitamin D. Her daughter showed normal biochemical surveillance until 15 years of age. LESSONS: We report 2 people in a family affected by MEN1 with the heterozygous germline c.1546dupC mutation, a variant that should be surveilled for early development of full-blown MEN1-associated endocrine tumors.

Nonfunctional parathyroid cancer a case report.

INTRODUCTION: Parathyroid cancer is a rare endocrine malignancy. These tumors are typically functional, causing severe hypercalcemia due to primary hyperparathyroidism. Nonfunctional parathyroid cancer with normal serum calcium and parathyroid hormone levels is extremely rare. The disease is usually indolent but progressive with a tendency to metastasize. It is very difficult to diagnose this malignancy. The definitive diagnosis is made by histopathological examination. Radical surgery with ipsilateral lobectomy and en bloc neck dissection is considered to be the most appropriate therapeutic approach. There is no evidence of efficiency of adjuvant cancer therapy and its indication has not been defined. Disease recurrence is common. CASE REPORT: We report the case of a 26-year-old female patient who underwent left hemithyroidectomy for growth progression of a hypoechoic lesion behind the left thyroid lobe detected by ultrasonography. Preoperative cytology and imaging assessments were not suspicious for malignancy. Serum parathyroid hormone and calcium levels were normal. The diagnosis of nonfunctional parathyroid carcinoma was determined based on histopathological examination. No further surgery or adjuvant therapy was indicated. No signs of recurrence or generalization have been observed at 36 months after the surgery. CONCLUSION: Nonfunctional parathyroid cancer is extremely rare. In many cases, the diagnosis is made in advanced stages of the disease. No formal classification or treatment protocol has been established so far. A new staging system has been proposed in the 8th edition of AJCC/UICC. Early detection, radical surgery and close follow-up are crucial aspects to affect the mortality and morbidity of patients with this type of malignancy.

[Parathyroid carcinoma: a rare and aggressive tumor]. / Nebenschilddrüsenkarzinom: ein seltener und aggressiver Tumor.

Parathyroid carcinoma is a very rare disease that is accompanied by a poor prognosis. Diagnosis is based on laboratory tests (calcium, parathyroid hormone), sonography, and scintigraphy. Complete surgical resection is the treatment of choice. Adjuvant therapy is controversial. The lack of treatment guidelines and experience with this disease often delay timely therapy. In the following article, we report on two female patients with parathyroid carcinoma who underwent surgery in our clinic.

Sestamibi Single-Positron Emission Computed Tomography/Diagnostic-quality Computed Tomography for the localization of abnormal parathyroid glands in patients with primary hyperparathyroidism: What clinicopathologic factors affect its accuracy?

PURPOSE: Sestamibi Single-Positron Emission Computed Tomography/Diagnostic-quality Computed Tomography (MIBI-SPECT/CT) is a common technology used for primary hyperparathyroidism (PHPT) localization in clinical practice. However, the clinicopathologic factors affecting the accuracy of MIBI-SPECT/CT and the potential limitations remain unclear. METHODS: Retrospectively enrolled PHPT patients (n = 280) were analyzed from August 2017 to December 2019. RESULTS: Of 96 patients with PHPT (mean age, 54 years; 63 females), 17 had discordance between MIBI-SPECT/CT and intraoperative findings. Among the 17 patients with discordance, 58.8% had major discordance, which occurred in most patients with multigland disease (MGD). Compared with concordant patients, discordant patients exhibited increased frequencies of autoimmune thyroid disease (29.4% vs 10.1%, p = 0.035), MDG (41.2% vs 3.8%, p = 0.035), higher PTH (296 pg/mL vs 146 pg/mL; p = 0.012),and lower phosphorus levels (0.77 mmol/L vs 0.90 mmol/L; p = 0.024). MDG (odds ratio [OR], 16.95; 95% CI 2.10-142.86), parathyroid lesion size of 12 mm or less (OR, 6.93; 95% CI 1.41-34.10), and a PTH level higher than 192.5 pg/mL (OR, 12.66; 95% CI 2.17-71.43) were independently associated with discordant MIBI-SPECT/CT results. CONCLUSION: MGD was most strongly associated with discordance between MIBI-SPECT/CT and intraoperative findings followed by a PTH level higher than 192.5 pg/mL and parathyroid lesion size of 12 mm or less. Surgeons should recognize these potential limitations, which may improve the preoperative procedure by encouraging further localization imaging and promptly facilitate intraoperative troubleshooting.

Contemporary Evaluation and Management of Parathyroid Carcinoma.

Parathyroid carcinoma is a rare malignancy, representing 0.005% of all cancers and 0.5%-1% of all parathyroid disorders. Parathyroid carcinoma occurs equally in males and females, as opposed to primary hyperparathyroidism, which has a female predominance. Patients with parathyroid carcinoma present with symptoms of hypercalcemia, similar to those with benign primary hyperparathyroidism. Parathyroid carcinoma should be suspected when calcium or parathyroid hormone levels are high. Because of the difficulty of discerning parathyroid carcinoma from adenoma preoperatively, the diagnosis of carcinoma is often made only after parathyroidectomy. The goals of surgery are resection with negative margins because surgery represents the only opportunity for cure. Adjuvant therapy with chemotherapy or external beam radiation has not been proven to affect disease-free or overall survival for these patients. Recurrence is common, with reoperation recommended for resectable recurrent disease. Palliation with calcimimetic pharmacotherapy can aid with management of symptomatic hypercalcemia in recurrent or persistent disease after parathyroidectomy. Ultimately, patients succumb to sequelae of hypercalcemia rather than tumor burden.

Correlation of perioperative biochemical variables with single adenoma weight in patients with primary hyperparathyroidism.

BACKGROUND: Single parathyroid adenoma is the main cause of primary hyperparathyroidism (PHPT), with surgery remaining the gold standard for its treatment. The ability to preoperatively predict the parathyroid adenoma size and could facilitate the decision about the extent of surgical exploration. It is reasonable to hypothesize that the perioperative levels of PHPT-related variables (i.e. calcium, parathormone, phosphate) may predict the adenoma weight or/and demonstrate whether the adenoma is successfully removed or not. Aim of this study is to explore the relationship between perioperative biochemical values and adenoma weight. Secondarily, we investigated the relationship between adenoma weight and uni-/bilateral neck exploration. METHODS: Retrospective study of all patients undergone surgery for primary hyperparathyroidism due to single adenoma in a tertiary university hospital in Germany during a 6-year period. Following variables were analyzed: preoperative serum calcium, phosphorus and parathormone, intraoperative parathormone before and after adenoma excision, intraoperative PTH decrease, postoperative serum calcium and parathormone (PTHpostop-pg/ml), calcium and PTH decrease. Bivariate correlations were calculated by the Spearman's correlation test at the 95% significance level. RESULTS: A total of 339 patients were included in the study. The median age of the patients was 60 years (range 21-90) and 77% were females. The median adenoma weight was 1 g (range 0.1-11). Adenoma weight correlated strong with maximum adenoma diameter (r = 0.72, p < 0.05), moderate with preoperative parathormone (r = 0.44) and parathormone decrease (r = 0.27), whereas there was no correlation with the intraoperative PTH decrease (r = 0.02). There was also a borderline (moderate to weak) correlation with pre- and postoperative calcium levels (r = 0.21 and r = 0.23 respectively) and a negative borderline correlation with phosphorus (r = - 0.21). Patients who required bilateral neck exploration, had significantly lighter adenomas (median weight 0.8 g vs 1.1 g, p = 0.005). CONCLUSIONS: We conclude that preoperative PTH levels may only serve as an approximate guide to adenoma weight, as direct preoperative prediction is not possible. Serum calcium levels, PTH and calcium decrease correlate only weak with adenoma weight. Patients who require bilateral neck exploration, have significantly (20-25%) lighter adenomas.

Pitfalls of using denosumab preoperatively to treat refractory severe hypercalcaemia.

A 40-year-old man, with a history of metastatic parathyroid carcinoma, status post primary tumour resection and lung metastasectomy, was hospitalised for persistent severe hypercalcaemia and elevated parathyroid hormone levels despite conventional management and escalating doses of cinacalcet. A single dose (120 mg) of denosumab was given and his calcium level plummeted from 14.8 mg/dL to 5.5 mg/dL. After second lung metastasectomy, he developed prolonged hypocalcaemia that required calcium and vitamin D supplements for more than 3 years. In patients with severe hypercalcaemia refractory to conventional therapies, denosumab has been used off-label with some success. A known side effect of denosumab is hypocalcaemia, which is often short-lived. The risk of prolonged hypocalcaemia should be fully evaluated before using denosumab preoperatively, especially in patients with renal insufficiency, prolonged hyperparathyroidism or anticipated tumour debulking surgery.

Can postoperative calcemia kinetics predict the risk of hypocalcemia for the management of parathyroid adenoma in ambulatory surgery?

INTRODUCTION: The goal of this study was to analyse the kinetics of corrected calcemia levels (Cac) after parathyroid excision and to determine the percentage of variation (ΔCa) in the initial hours after surgery, in order to entertain an early discharge. POPULATION AND METHODS: Were included in this study, patients undergoing operation for parathyroid adenoma responsible for primary hyperparathyroidism (PHP). The Cac was measure preoperatively and four hours after surgery, and then every day until patient discharge. Group A included patients for whom the Cac was inferior to 2.2mmol/L at least once postoperatively while group B included patients for whom the Cac was always equal or superior to 2.2mmol/L. The ΔCa represented the percentage of the fall in postoperative Cac with respect to preoperative Cac. RESULTS: Between 2010 and 2017, 156 patients fulfilled the inclusion criteria (women 80.8%, [sex ratio 1:4], median age 64 years old). Preoperative Cac was statistically significantly lower in group A compared to group B (2.67 vs. 2.82mmol/L; P<0.0001). In total, 9.6% of patients had calcium supplementation for hypocalcemia, symptomatic or not. Postoperative Cac reached its nadir value on postoperative day 2. At four hours postoperative, the risk of postoperative calcelmia falling below 2.2mmol/L appeared when the ΔCa was superior to 6% with a sensitivity of 92.9% and a negative predictive value of 97.4%. CONCLUSION: After excision of a parathyroid adenoma for PHP, the Cac falls rapidly and reaches its nadir value on day 2. If the fall in calcemia is less than 6% four hours after surgery (vs. preoperative level), early discharge within the framework of ambulatory surgery is possible.

Unusual recurrent renal secondary hyperparathyroidism caused by hyperplastic autograft with supernumerary parathyroid adenoma: A case report.

RATIONALE: Secondary hyperparathyroidism (SHPT) is often complicated with chronic renal failure. Though the total parathyroidectomy (TPTX) with forearm autotransplantation (FAT) has been commonly used to treatment refractory renal SHPT, the recurrence of SHPT is not infrequent, resulting from hyperplastic autograft, remnant parathyroid tissues, and supernumerary parathyroid gland (SPG). PATIENT CONCERNS: A 67-year-old man undergoing TPTX+FAT 4 years previously for renal SHPT, who received regular hemodialysis with active vitamin D supplements of Rocaltrol treatment postoperatively, was admitted to our hospital with progressively elevated serum intact parathyroid hormone (iPTH) from 176 to 1266 pg/mL for 8 months and bilateral ankle joints pain for 1 month. Tc-sestamibi dual-phase imaging with single positron emission tomography (SPECT)/computed tomography (CT) revealed a nodule in suprasternal fossa, besides a nodule in autografted site, accompanied with intense radioactivity. DIAGNOSIS: Recurrent SHPT was easily diagnosed based on previous medical history, painful joints, increased serum iPTH level and positive findings of Tc-sestamibi imaging. Routine postoperative pathology showed that the nodules were consistent with an adenomatoid hyperplasic autograft and a supernumerary parathyroid adenoma in suprasternal fossa, respectively. INTERVENTIONS: Reoperation for removing nodules in suprasternal fossa and autografted site was performed 1 month later. Then regular hemodialysis 3 times a week with Rocaltrol was continued. OUTCOMES: During 12 months of follow-up, the joints pain improved obviously and the serum iPTH level ranged from 30.1 to 442 pg/mL. LESSONS: Although rare, recurrent renal SHPT may be caused by a coexistence of both hyperfunctional autograft and SPG after TPTX+FAT. The Tc-sestamibi parathyroid imaging with SPECT/CT is helpful to locate the culprits of recurrent renal SHPT before reoperation. To prevent recurrence of renal SHPT, the present initial surgical procedures should be further optimized in patient on permanent hemodialysis.

A Case of Pregnancy Complicated by Primary Hyperparathyroidism Due to a Parathyroid Adenoma.

BACKGROUND Primary hyperparathyroidism is most common in women during the menopause and its occurrence in pregnant women is rare. However, because neonatal mortality is associated with maternal hyperparathyroidism, early diagnosis is essential. This report describes the case of a late diagnosis of primary hyperparathyroidism in a 28-year-old pregnant woman and describes the effects on the mother and neonate. CASE REPORT During her second pregnancy, a 28-year-old woman presented with symptoms of general weakness, bone and joint pain, multiple fractures with bone deformity, muscle weakness, and gait disturbance. Due to the high risk of perinatal pathology, a cesarean section was performed. Several weeks later, she underwent thoracoscopic removal of an ectopic parathyroid gland located at the aortic arch. Hypocalcemia in the newborn infant required treatment with calcium and magnesium supplements. CONCLUSIONS This case demonstrates that primary hyperparathyroidism during pregnancy requires timely diagnosis and treatment to reduce potential maternal and fetal complications. Screening for primary hyperparathyroidism should be undertaken in pregnant women with any symptoms associated with hypercalcemia. Treatment should be individualized and includes conservative management, parathyroidectomy in the second trimester, or parathyroidectomy performed in the early postpartum period.

Parkinsonism as presenting symptom of primary hyperparathyroidism: Improvement after surgery. / Parkinsonismo como síntoma principal de hiperparatiroidismo: recuperación tras cirugía

Introduction: Hypercalcemia can cause different neurological disorders, depending on the calcium level. We report an exceptional case of primary hyperparathyroidism presenting as neurological alteration and it has favourable outcome after parathyroidectomy. Case report: A 74-year-old woman presented with progressive cognitive deterioration and impaired motor function. The complementary tests showed hypercalcemia due to a parathyroid adenoma. Parathyroidectomy was performed with symptomatic improvement. Conclusion: Cognitive impairment of the elderly due to a parathyroid adenoma is underdiagnosed, behavioral changes and alterations of motor functions are attributed to age, dementia and frailty, representing a diagnostic challenge.

Introducción: La hipercalcemia puede causar diferentes trastornos neurológicos, dependiendo de las concentraciones de calcio. Aportamos un caso excepcional de hiperparatiroidismo primario que se manifestó con deterioro neurológico rápidamente evolutivo y se resolvió mediante paratiroidectomía. Caso clínico: Mujer de 74 años que consultó por deterioro cognitivo progresivo y alteración de las funciones motoras. Las pruebas complementarias evidenciaron hipercalcemia debida a un adenoma paratiroideo. Se realizó paratiroidectomía, con mejoría sintomática. Conclusión: El deterioro cognitivo del anciano por un adenoma paratiroideo está infradiagnosticado, pues los cambios de conducta y las alteraciones de las funciones motoras se atribuyen a la edad, la demencia y la fragilidad, suponiendo un reto diagnóstico.

Ambulatory surgery under local anesthesia for parathyroid adenoma: Feasibility and outcome.

The aim of this study was to evaluate the results of ambulatory parathyroid resection performed under local anesthesia (LA). MATERIAL AND METHODS: Outpatients undergoing parathyroid adenoma resection by a focused approach under LA were included. Results were evaluated by intraoperative serum parathormone levels (ioPTH) and the balance of phosphate and calcium postoperatively, at 3 months, 1 year and at the point date. The quality of ambulatory care was evaluated by the number of cancelled interventions, the number of patients hospitalized after surgery or during the first postoperative month. The patient data manager of the institution carried out a medico-economic analysis. RESULTS: From 2005 to 2014, 129 patients met the inclusion criteria [women: 82% (sex ratio 1:5), median age: 72 years]. There was no morbidity for 98% of patients. Twelve patients had no statistically significant drop in ioPTH: two had persistent primary hyperparathyroidism (PHP). LA failed in four patients and PTH was late to normalize in six patients. Six patients had recurrent PHP (4.6%), of which two occurred four years after excision. Outpatient treatment was successful in 95%, without deprogramming or rehospitalization. The cost of the treatment under LA and on an outpatient basis was € 2014.90 (vs. € 2581.47 under general anesthesia and traditional hospitalization) CONCLUSION: Excision of single parathyroid adenomas can be performed under LA in an ambulatory setting without any major risk for the patient. The risk of recurrence after the focused approach requires regular laboratory monitoring for at least five years.