Recurrent Hyperparathyroidism Research Articles

Histologic hypercellularity in a biopsied normal parathyroid gland does not correlate with hyperfunction in primary hyperparathyroidism

BackgroundAbout 15% of patients with primary hyperparathyroidism have multiglandular disease, thus during resection of an apparent single adenoma, a visibly normal parathyroid may be identified and biopsied. Using long-term biochemical follow-up, we examined whether normal parathyroid hypercellularity correlates with multiglandular disease or primary hyperparathyroidism recurrence. MethodsWe reviewed all patients who from 2001 to 2015 had an initial operation for sporadic primary hyperparathyroidism with removal of 1 gland, routine normal parathyroid biopsy, intraoperative parathyroid hormone monitoring, and follow-up of ≥3 years. Recurrence was defined by hypercalcemia after documented cure at 6 months, and hypercellularity by standard histologic criteria. ResultsOf 134 patients with mean follow-up of 9.4 years (range, 3.1−15.9), 132 (98.5%) exhibited cure at 6 months. Two had initial failure, and 8 of 132 (6.1%) developed recurrent hyperparathyroidism (mean 5.8 y, range 4−10.6). The normal parathyroid was hypercellular in 14 of 132 (10.6%) of the cured patients, and this rate did not differ for those with long-term cure (12/124, 9.7%) versus recurrence (2/8, 25%, P = .2). The positive predictive value of normal parathyroid hypercellularity for recurrence was low (14.3%), and the negative predictive value of normal parathyroid normocellularity was high (94.9%). ConclusionDuring the initial operation for primary hyperparathyroidism, 10% of normal parathyroids are hypercellular, but this does not signify missed multiglandular disease. In contrast, normal parathyroid normocellularity has high predictive value for durable cure (95%), slightly better than visual identification of a second normal parathyroid (94%).

Early biochemical response to parathyroidectomy for primary hyperparathyroidism and its predictive value for recurrent hypercalcemia and recurrent primary hyperparathyroidism

BackgroundThe traditional definition of cure after parathyroidectomy (PTX) for primary hyperparathyroidism is normocalcemia. Our hypothesis was that early postoperative levels of serum calcium and parathyroid hormone after PTX would have predictive value for later recurrence. MethodsWe performed a retrospective study of 1,146 patients with primary hyperparathyroidism who underwent PTX and had long-term biochemical follow-up. The first postoperative serum level of calcium and parathyroid hormone values were used to categorize patients into the following four early biochemical response groups: (1) complete response (normal calcium and normal parathyroid hormone), (2) partial response with hyperparathormonemia (normal calcium and increased parathyroid hormone), (3) partial response with hypercalcemia (increased calcium and normal parathyroid hormone), and (4) non-response (increases in both calcium and parathyroid hormone). Incidences of recurrent hypercalcemia and recurrent primary hyperparathyroidism >6 months after operation were then analyzed. ResultsThe overall rate of any elevated serum levels of calcium and any increase in serum levels of parathyroid hormone during >6-month follow-up was 9.8% (112 of 1146), with 6.6% (57 of 861) for group 1, 27% (35 of 129) for group 2, and 16% (20 of 127) for group 3 (P < .02). Partial biochemical responses with either increased serum calcium or increased parathyroid hormone levels were the strongest predictors of any episode of increased serum levels of calcium after 6 months and was associated with 2.7× to 4.3× the risk of recurrent primary hyperparathyroidism, respectively. ConclusionThis study demonstrates the importance of measuring parathyroid hormone in the early postoperative period to better predict later recurrent primary hyperparathyroidism.

Prevalence of canine primary hyperparathyroidism recurrence in Keeshond and non-Keeshond dogs after curative parathyroidectomy

BackgroundPrimary hyperparathyroidism (PHPT) is an uncommon condition in dogs, for which there is a documented genetic predisposition in Keeshonden and sporadic cases in other breeds. Secondary literature reports a 10...

Prevalence, Diagnostic Imaging and Treatment Strategies of Intrathyroid Parathyroid Adenomas in Patients Surgically Treated for Hyperparathyroidism: A Review of the Literature

Single parathyroid adenoma is the most common cause of primary hyperparathyroidism. Surgery is the definitive treatment. Intrathyroid parathyroid adenomas (ITPAs) can result in failure of the surgical procedure and persistence or recurrence of hyperparathyroidism. The aim of this review is to assess the prevalence of ITPAs, the efficacy of imaging studies in ITPA localisation as well as the efficacy of various surgical strategies. We conducted our search using the Medline database and searched for the keywords “intrathyroid parathyroid adenoma”. Eligibility criteria specified the availability of abstracts or full-text articles in English. Twenty five articles were considered relevant and were included in the synthesis of this article. The prevalence of ITPA in patients with primary hyperparathyroidism varies between 0.5% – 22.68%, with most studies reporting a frequency of 0.5% – 4.3%. The prevalence of ITPA in patients re-operated for persistent or recurrent hyperparathyroidism was 8.6% – 11%. The imaging modalities that are most commonly used are ultrasonography (US) and Technetium-99m sestamibi scintigraphy. A combination of both methods is associated with high diagnostic accuracy (81% – 92.3%). Thorough bilateral cervical exploration followed by thyroidotomy or thyroid lobectomy is the most common surgical strategy, although there are data that support low success rates. ITPAs are a rare cause of hyperparathyroidism that should not be neglected. Preoperative imaging is useful in the localisation of ITPAs. Bilateral cervical exploration followed by tumour enucleation or thyroid lobectomy is considered the best surgical strategy.

Re-operative Parathyroidectomy in Patients With Mild Primary Hyperparathyroidism

BackgroundSurgery is the definitive management of primary hyperparathyroidism and the only curative therapy. However, many surgeons are hesitant to operate on individuals with mild primary hyperparathyroidism, with an even greater reluctance to operate on those who underwent a previous parathyroidectomy. We hypothesize that patients with mild primary hyperparathyroidism who undergo a re-operation have equivalent outcomes compared with those who undergo a first-time (FT) operation. MethodsWe reviewed a prospective database of 459 patients with mild primary hyperparathyroidism who underwent surgery by one endocrine surgeon. Of these patients, 59 had a re-operative (RE-OP) parathyroid surgery. We compared these patients to those with mild primary hyperparathyroidism who had FT surgery (n = 400) using either the Pearson chi-square, Fisher's exact test, or Student's t-test where appropriate. ResultsThe mean age of our cohort was 60 ± 14 y, with 86% females. Patients in the RE-OP group had similar preoperative calcium and parathyroid hormone levels compared with those in the FT group. Most patients who underwent a RE-OP surgery had four gland hyperplasia on pathology (49.2%). Patients in the RE-OP and FT groups both had high and similar cure rates (100% versus 99.8%, P = 0.70). RE-OP patients had a higher rate of recurrent hyperparathyroidism (10.3% versus 3.3%, P = 0.025). ConclusionsIn patients with mild primary hyperparathyroidism, those who undergo RE-OP parathyroidectomy have a high cure rate that is similar to FT surgery. Therefore, we recommend that these patients with recurrence of mild hyperparathyroid disease be considered for parathyroidectomy.

Long-term results of a randomized study comparing parathyroidectomy with cinacalcet for treating tertiary hyperparathyroidism.

Tertiary hyperparathyroidism is a common cause of hypercalcemia after kidney transplantation (KT) and has been associated with renal dysfunction, bone mineral density loss, and increased risk of fracture and cardiovascular events. In a previous 12-month clinical trial, we demonstrated that subtotal parathyroidectomy was more effective than cinacalcet for controlling hypercalcemia. In the current study, we retrospectively evaluate whether this effect is maintained after 5years of follow-up. In total, 24 patients had data available at 5years, 13 in the cinacalcet group and 11 in the parathyroidectomy group. At 5years, 7 of 11 patients (64%) in the parathyroidectomy group and 6 of 13 patients (46%) in the cinacalcet group (P=.44) showed normocalcemia. However, recurrence of hypercalcemia was only observed in the cinacalcet group (P=.016). Subtotal parathyroidectomy retained a greater reduction in intact parathyroid hormone (iPTH) compared with cinacalcet group. No differences were observed in kidney function and incidence of fragility fractures between both groups. Cinacalcet was discontinued in 5 out of 13 patients. In conclusion, in kidney transplant patients with tertiary hyperparathyroidism recurrence of hypercalcemia after 5-year follow-up is more frequent in cinacalcet than after subtotal parathyroidectomy.

MON-509 RET Mutations in the MEN1 Syndrome: Is It an Innocent Bystander?

IntroductionMENIN and RET mutations in MEN1 families, are rare, and, when they co-exist either mutation may predominate the clinical picture.We report a family, with both mutations, and, suspect that the RET mutation may not be an innocent bystander.Clinical CasesGM (36M): 2009: Presented with skin lesions and a lactotroph adenoma causing chiasmal compression. Treatment: Hypophysectomy and Cabergoline. This resulted in restoration of sexual function and fertility.2013: Developed hyperparathyroidism [Calcium 10.6mg% (8.5-10.5); PTH 610pg/ml (10-65); MIBG Scan: parathyroid adenomas]. Treatment: Subtotal parathyroidectomy with allotransplant.2015: Developed Zollinger-Ellison Syndrome [multiple gastric ulcers; S Gastrin: 516pg/ml; (0-180)]. Treatment: Pantoprazole.MRI Abdomen: Calcific atrophic pancreas and bilateral non-functioning adrenal adenomas.HM (32M): 2008: Skin lesions and Lactotroph adenoma. Treatment with Cabergoline resulted in restoration of sexual function and a reduction in breast and tumour size.2013: Hyperparathyroidism [Calcium 10.9mg%; PTH: 166pg/ml; Calcium excretion: 1160mg/24hrs (100-250); BMD: Osteopenia. MIBG 123 Scan: Avid uptake in Right Inferior parathyroid gland]. He underwent a subtotal parathyroidectomy with allotransplant.2015: Recurrent Hyperparathyroidism (Calcium 10.7mg%; PTH: 116pg/ml; MIBG 123 scan: Hyperfunction of the transplanted gland). Stable on treatment with Cinacalcet.Their mother, AM (42F): 1980: Detected to have a lactotroph adenoma when investigated for primary infertility and galactorrhoea. She was treated with Bromocriptine and delivered the boys in 1980 and 1984 respectively. She had recurrent renal calculi and hydronephrosis (? hyperparathyroidism). She succumbed to renal failure following surgery for a benign pancreatic cystadenoma in 1990.DiscussionWhole exome sequencing of GM and HM showed pathogenic mutations of both, MENIN and RET gene. The precise mutation was a stop gain mutation at exon 3 MENIN.C511T:p.Q171X . They (GM/HM) also harboured a mutation in the RET gene at exon 14 c. G2492T: p.G831V; g. chr10. This may be the first family in which the rare combination of these two mutations has been reported.The genomics and the clinical presentation suggest that the MENIN mutation predominates in the family, but the presence of bilateral adrenal adenomas in GM are significant on account of RET mutation. The latter may be a harbinger of serious disease in the future. The situation is further compounded by the recurrence of hyperparathyroidism in the allograft of HM. This may be caused due to chance or an unknown genetic/ epigenetic phenomenon in the two MEN mutated genes and RNA sequencing of the tumour tissue may explain the genetic phenomenon. The latter two events suggest that the RET mutations in MEN1 may not be an innocent bystander.

MON-923 MENIN Gene Mutation: Unity Amidst Diversity

IntroductionThe MEN 1 syndrome is an enigmatic disorder, manifesting a wide spectrum of disorders, in members of a family, harbouring the same gene mutation. We present one such family, with a MENIN gene mutation with marked diversity in the clinical presentationClinical cases1992: RB (Age: 13) presented with accelerated puberty, galactorrhoea and a lactotroph adenoma. Treated with Bromocriptine, followed by hypophysectomy and radiotherapy. He was on hormone replacement for hypopituitarism.1996: He gained 21 kg of weight and had recurrent episodes of convulsions with unconsciousness.He had hypoglycaemia (13mg/dl; N >70), with hyperinsulinemia (58uIU/ml; N<25). MRI abdomen showed a mass (3.4 x 3.0 cm) over the tail of the pancreas for which a distal pancreatectomy was done (HPE: Neuroendocrine tumour).2013: He had pain in the lower limbs with hypercalcemia (11.4mg%; N: 8.5-10.5) and hyperparathyroidism (329 pg/ml; N: 10-61) (MIBG Scan: parathyroid adenomas treated by bilateral inferior parathyroidectomy).2016: He had hypoglycaemia with hyperinsulinemia with multifocal pancreatic NETs. These were enucleated at surgery.2017: He developed Zollinger Ellison syndrome with raised basal gastrin levels (Gastrin: >200ng/ml; N<180) and multiple duodenal ulcers (Treatment: Pantoprazole). He simultaneously had recurrent hyperparathyroidism and underwent a total parathyroidectomy with allograft. The allograft initially showed evidence of hyperparathyroidism followed by hypoparathyroidism.His father VB (Age: 56) was seen by us in 1996 for skin lesions and a malignant thymic carcinoid, to which he succumbed to, shortly after the surgery. His aunt, SB (Age: 18), has a lactotroph adenoma with severe insulin resistance characterised by obesity, acanthosis nigricans and hyperandrogenism.2009: His paternal uncle PB (Age: 54) had a pituitary macroadenoma. He underwent a hypophysectomy and was lost to follow up.2018: PB had massive haemoptysis. A bronchoscopy showed nodules in the right lung which on biopsy revealed a NET. The whole body scan showed a hilar and mediastinal mass along with metastatic disease to the adrenals, liver, spine, skull and rectum. The histopathology examination revealed a malignant carcinoidClinical lessonWhole exome sequencing of the two of the affected members showed mutations of the MENIN gene at exon 2 c.G2492T:p.G831V; g. chr10. This common mutation in the family was associated with a wide spectrum of diverse clinical manifestations which include the classic disease, malignant carcinoids of the lung and acromegaloid variant of Type A insulin resistance with hyperandrogenism. These observations suggest the unity amidst diversity in the enigmatic syndrome that encompasses MEN1.

SAT-386 Inconsistent Bilateral Arm Serum PTH Testing in Recurrent Hyperparathyroidism in a Patient Status-Post Parathyroid Auto-Transplantation

Background: More than 95% of patients with primary hyperparathyroidism will be cured with the initial operation by an experienced surgeon. However, localization of hyperparathyroid recurrences, especially after extensive surgery becomes challenging. For patients with transplanted parathyroid glands into the forearm, there may be utility in bilateral arm serum PTH testing to help with localization.Clinical Case:A 65-year-old woman presented to the clinic with primary hyperparathyroidism in 2008. After a localization study, she had a partial parathyroidectomy but continued to have persistent biochemical hyperparathyroidism despite negative localization studies. She was then referred to another institution for further studies. Follow up Sestamibi scans were negative but 4D-CT scans assisted in localizing the presence of a superior parathyroid gland adenoma which was later removed in 2011. During this time, the left inferior parathyroid gland was auto-transplanted into the left forearm. Again, her calcium and PTH levels rose despite negative Sestamibi scans showing no abnormalities in post-operative beds or in the forearm. Review of previous labs revealed elevated PTH levels in the ranges of 80-110 pg/mL since 2012-2019. The patient’s most recent PTH was 2408 pg/mL. At that point, the decision was made to repeat the labs on the left and right forearms simultaneously and labs showed PTH levels of 1283 pg/mL and 118 pg/mL, respectively. Repeat Sestamibi scan following these labs demonstrated evidence of increased radiotracer uptake in the region of the prior transplanted parathyroid tissue with no neck uptake concerning for hyperparathyroidism due to auto-transplanted hyperplastic tissue.Conclusion:This case demonstrates the utility of bilateral arm serum PTH testing in the evaluation of recurrent hyperparathyroidism in patient’s status-post parathyroid auto-transplantation.

Abstract #806991: Case Report: Papillary Thyroid Cancer with Recurrent Primary Hyperparathyroidism Due to a Rare Ectopic Parathyroid Adenoma in the Carotid Sheath

Abstract #806991: Case Report: Papillary Thyroid Cancer with Recurrent Primary Hyperparathyroidism Due to a Rare Ectopic Parathyroid Adenoma in the Carotid Sheath

Safety and effectiveness of reoperation for persistent or recurrent drug refractory secondary hyperparathyroidism.

Drug-refractory secondary hyperparathyroidism (SHPT) is the most common complication in patients with chronic renal failure (CRF). Although surgery is the most effective and safe method for drug-refractory SHPT, the condition may persist or recur after the primary surgery, and reoperation is often required in these patients. The purpose of our current study was to evaluate the safety and effectiveness of reoperation for drug-refractory SHPT. The clinical data of 15 patients requiring reoperation after a surgery for drug-refractory SHPT in our hospital from 2010 to 2019 were retrospectively analyzed. Changes in biochemical markers including intact parathyroid hormone (iPTH), blood calcium (Ca), blood phosphorus (P), alkaline phosphatase (ALP), and blood calcium phosphorus product (Ca*P) were compared before and after the surgery, and the effectiveness and complications of the reoperation were summarized. The reoperation was successful in all the 15 patients after a single attempt. Routine pathological examinations identified a total of 25 parathyroid glands, of which 10 were in the neck in situ, 5 were ectopic in the neck, and 10 were in the forearm. The ectopic parathyroid glands were located inside the thyroid gland (n=1), anterior superior mediastinum (n=1), or thymus (n=3). Surgical treatment significantly improved clinical symptoms such as skin pruritus and bone pain. Blood iPTH, Ca, P, ALP, and Ca*P were significantly reduced (P<0.05 or P<0.01) after surgery. Hypothyroidism occurred in 1 patient; 4 patients undergoing orthotopic neck surgery developed transient hoarseness, which were alleviated within 6 months; no severe complications such as bleeding or death were noted. No recurrence occurred during the 6-month follow-up. Reoperation is safe and effective for drug-refractory SHPT. Preoperative imaging should be performed to achieve accurate positioning, and the recurrent laryngeal nerve should be closely monitored during surgery. The purpose of the reoperation is to remove all possible parathyroid tissues to avoid recurrence.

Results of 150 Subtotal Parathyroidectomies for Secondary Hyperparathyroidism

We evaluated our experience of subtotal parathyroidectomy (SP) for secondary hyperparathyroidism (SHP) cases. We evaluated retrospectively our 150 SP operations due to SHP. Serious hypocalcemia (< 6.5 mg/dL) developed in 23 (15.3%) patients, and we performed cryopreserved parathyroid cell autotransplantation in four cases with successful results. Recurrent hyperparathyroidism developed in 3 (2%) cases, and persistent hyperparathyroidism was observed in 22 (14.6%) cases. Upon reoperation in nine persistent hyperparathyroidism cases, we found supernumerary parathyroid glands in three cases, remnant tissue hyperplasia in four cases, and ectopic parathyroid glands in two cases. Among cases in which four glands cannot be found during surgery, the permanent HP rate is high. Serious hypocalcemia may develop postoperatively, and cryopreserved parathyroid autotransplantation may be inevitable in some of these cases.

Surgical Outcomes of Subtotal Parathyroidectomy for Renal Hyperparathyroidism.

ObjectivesThe aim of this study was to evaluate the effectiveness of subtotal parathyroidectomy for patients with renal hyperparathyroidism.MethodsWe studied 25 patients with renal hyperparathyroidism who underwent subtotal parathyroidectomy from October 2002 to October 2017. We analyzed serum intact parathyroid hormone (iPTH), calcium, and inorganic phosphorus levels before and at multiple time points following surgery, and evaluated the surgical outcomes and complications.ResultsOf the 25 patients, 13 (52%) were male and 12 (48%) were female, and the mean age was 53.4±9.3 years. The mean duration of dialysis before parathyroidectomy was 156.8±79.5 months. Mean preoperative serum iPTH and calcium levels were 1,199.0±571.3 pg/mL and 10.5±1.0 mg/dL, respectively. At 6 months postoperatively, the mean iPTH and calcium levels decreased to 49.2±47.6 pg/mL (P<0.01) and 8.0±1.0 mg/dL (P<0.01), respectively. Recurrent hyperparathyroidism occurred in two patients: one subsequently underwent kidney transplantation and the other continued hemodialysis and maintained normal calcium levels. One patient developed postoperative permanent hypoparathyroidism.ConclusionSubtotal parathyroidectomy is a safe and effective surgical treatment for renal hyperparathyroidism.

Parathyromatosis in a patient with parathyroid carcinoma and hypercalcemic crisis: Findings on MIBI scan and 4D CT

Parathyroid carcinoma is a rare tumor associated with significantly high levels of parathyroid hormone (PTH) and primary hyperparathyroidism (HPT) and is difficult to distinguish from parathyroid adenoma, which is a much more common cause of HPT. Parathyromatosis is a rare condition in which several nodules of hyperfunctioning parathyroid tissue form in the neck and mediastinum, and it presents as recurrent hyperparathyroidism. We describe the case of a 44-year-old woman with recurrent hyperparathyroidism who presented with hypercalcemic crisis and biochemical investigations showed a severely raised serum intact parathyroid hormone (iPTH) level of 2513 pg/ml and serum calcium level of 17 mg/dl. A four-dimensional computed tomography (4D CT) of the neck was done, which showed few arterially enhancing nodular lesions in the right paratracheal and retrotracheal location at the level of thoracic inlet showing washout in delayed images suggestive of recurrent/residual parathyroid lesions. A technetium-99 m methoxyisobutylisonitrile (Tc-99 m MIBI) scan was done, and the planar neck anterior early images showed an area of increased radiotracer uptake in the right lower neck region with persistent uptake in the delayed images. Single photon emission computed tomography/ computed tomography (SPECT/CT) images showed a soft-tissue lesion in the right paratracheal region at the level of C7 vertebra showing increased radiotracer uptake suggestive of a recurrent/residual parathyroid lesion. Exploration and excision of the recurrent parathyroid mass was planned, and the right paratracheal mass with nodes above the aortic arch along with the subcutaneous and intramuscular lesions were excised. Postoperative serum calcium was 7.8 mg/dl, and serum iPTH was 135 pg/ml. Histopathological examination of the excised tissue showed the features of parathyroid carcinoma. Thus, Tc-99 m MIBI scan and 4D CT were critical in evaluating this case of parathyroid carcinoma with recurrent hyperparathyroidism and planning its management.

Intraoperative Monitoring of Parathormone in the Surgical Treatment of Renal Hyperparathyroidism.

Intraoperative monitoring of parathyroid hormone can confirm the complete excision of hyperfunctional parathyroid tissue, as the plasma half-life of PTH is approximately 5 minutes. The purpose of this study was to analyse the values of parathormon (PTH) and the intraoperative impact in patients with secondary hyperparathyroidism of renal cause (sHPT). A series of 86 patients who were hospitalised in our clinic between February 2015 to December 2018, were included in the study rom. All patients underwent surgery with PTH monitoring. PTH was determined preoperatively, intraoperatively 15 minutes after parathyroidectomy and postoperatively. Out of a total of 86 patients, 6 patients had non-functional renal transplant. 81 patients were operated on per primam and 5 patients were operated for disease recurrence. There were 77 total parathyroidectomies and 4 subtotal parathyroidectomies. One patient had 5 parathyroid glands. There were 4 patients with recurrent hyper-plastic tissue excision. Blood samples were collected intraoperatively through the puncture of the jugular vein. The PTH value was determined by the Elecsys PTH STATÃÂî test. The mean value of preoperative PTH was 1658 pg / mL and decreased to 46.5 pg / mL at the end of the operation. Subsequently, the level of PTH harvested at 3-6 months increased slightly to 59.8 pg / mL. 80 (93%) of patients had elevated preoperative calcium values. Recurrent hyperparathyroidism was found in 1 of the 4 patients who underwent subtotal parathyroidectomy. IPTH value is influenced by the intraoperative manipulation of the parathyroid glands, the individual variability of PTH half-life and the physiological state of the patient. The decrease of PTH measured intraoperatively at 15 minutes after harvest with at least 90% of the preoperative value indicates the success of a total parathyroidectomy, with normalisation of calcium and PTH.

Challenges in recurrent hyperparathyroidism

Challenges in recurrent hyperparathyroidism

Recurrent hyperparathyroidism in a patient with multiple endocrine neoplasia type 1 (MEN1) after total parathyroidectomy with autotransplantation unmasked by depression and vague abdominal pain

Recurrent hyperparathyroidism in a patient with multiple endocrine neoplasia type 1 (MEN1) after total parathyroidectomy with autotransplantation unmasked by depression and vague abdominal pain

Parathyromatosis: A Rare Case of Recurrent Hyperparathyroidism Localized by Four-Dimensional Computed Tomography

To present a rare case of parathyromatosis. We present the clinical, laboratory, and imaging findings, along with a review of the literature. A 33-year-old man with a history of right upper parathyroid adenoma removal 5 years prior due to hyperparathyroidism was admitted for severe hypercalcemia (15.6 mg/dL; normal, 8.5 to 10.5 mg/dL) with elevated plasma parathyroid hormone (PTH) (882 pg/mL; normal, 15 to 65 pg/mL). Ultrasound, computed tomography (CT), sestamibi, and positron emission tomography scans were unremarkable; however, a four-dimensional CT (4DCT) of the neck showed an area of increased signal enhancement and hypervascularity without discrete nodule in the posterior right thyroid region. The patient underwent parathyroid surgical exploration with right hemithyroidectomy and compartment neck dissection to remove the affected tissue. PTH levels dropped to 208 pg/mL postoperatively; calcium decreased but remained elevated at 12.7 mg/dL. Pathology revealed the presence of several small nodular foci of atypical hyperplastic parathyroid tissue in the right thyroid and soft tissue in the left central neck compartment consistent with parathyromatosis. This case report represents the first-time use of 4DCT to localize parathyromatosis. Parathyromatosis is a rare but problematic cause of recurrent hyperparathyroidism. Ultrasound and 4DCT may represent the best imaging modalities for identification and perioperative management to remove all affected tissue without reseeding.

Pre-operative Localisation of the Parathyroid Glands in Secondary Hyperparathyroidism: A Retrospective Cohort Study

Complete parathyroidectomy (PTx) is essential during total PTx for secondary hyperparathyroidism (SHPT) to prevent recurrent and persistent hyperparathyroidism. Pre-operative imaging evaluations, including computed tomography (CT), ultrasonography (US), and Tc-99m sestamibi (MIBI) scans, are commonly performed. Between June 2009 and January 2016, 291 patients underwent PTx for SHPT after pre-operative evaluations involving CT, US, and MIBI scans, and the diagnostic accuracies of these imaging modalities for identifying the parathyroid glands were evaluated in 177 patients whose intact parathyroid hormone (PTH) levels were <9 pg/mL after the initial PTx. Additional PTx procedures were performed on 7 of 114 patients whose intact PTH levels were >9 ng/mL after PTx, and the diagnostic validities of the imaging modalities for the remnant parathyroid glands were evaluated. A combination of CT, US, and MIBI scans achieved the highest diagnostic accuracy (75%) for locating bilateral upper and lower parathyroid glands before initial PTx. The accuracies of CT, US, and MIBI scans with respect to locating remnant parathyroid glands before additional PTx were 100%, 28.6%, and 100%, respectively. A combination of CT, US, and MIBI scans is useful for initial PTx for SHPT, and CT and MIBI scans are useful imaging modalities for additional PTx procedures.

Factors associated with late recurrence after parathyroidectomy for primary hyperparathyroidism

BackgroundA recent study with unusually lengthy follow-up after surgery for primary hyperparathyroidism reported higher recurrence rates than previously appreciated. We sought to identify specific factors associated with late recurrence after seemingly curative parathyroidectomy. MethodsProspectively collected data were retrieved for patients who had surgical treatment of sporadic primary hyperparathyroidism with ≥ 3-year follow-up (3–17.6 years). Recurrence was defined by 6 months of eucalcemia with subsequent hypercalcemia and a high or unsuppressed parathyroid hormone. Recurrent patients were compared with cured patients (defined by consistent eucalcemia). ResultsAmong 261 patients, 28 (10.7%) had recurrence and 233 (89.3%) were cured. The mean time to recurrence was 77 months (range 13–170). The mean final intraoperative parathyroid hormone (49.0 pg/mL vs 37.5 pg/mL, P < .01), 6-month calcium levels (9.6 mg/dL vs 9.2 mg/dL, P = .02) and mean 6-month parathyroid hormone levels (86.5 pg/mL vs 59.6 pg/mL, P = .04) were higher for recurrence. By multivariable analysis, 6-month calcium ≥ 9.7 and eucalcemic elevation of the parathyroid hormone at 6 months were independently associated with recurrent primary hyperparathyroidism. ConclusionLong-term follow-up after apparent curative surgery for primary hyperparathyroidism identified a high late recurrence rate (10.7%), up to 17 years later. A 6-month calcium >9.7 mg/dL and eucalcemic parathyroid hormone elevation at 6 months may be associated with recurrence, and such findings may help guide management.