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AACE Clinical Case Reports 2024Although common in adults, primary hyperparathyroidism (PHPT) is a rare condition in children with the most common etiology being solitary parathyroid adenoma (PTA). The...
BACKGROUND/OBJECTIVE
Although common in adults, primary hyperparathyroidism (PHPT) is a rare condition in children with the most common etiology being solitary parathyroid adenoma (PTA). The typical presentation is symptomatic hypercalcemia. Management of PHTP secondary to PTA requires excision of the adenoma.
CASE REPORT
A 13-year-old adolescent boy presented because of orbital cellulitis and was noted to have hypercalcemia. Despite this, the patient was curiously asymptomatic. Further investigations yielded an elevated parathyroid hormone (PTH) level and a normal urine calcium-to-creatinine ratio making the most likely cause of hypercalcemia PHTP secondary to PTA. Imaging demonstrated PTA. The patient underwent parathyroidectomy with the pathology demonstrating PTA. Postoperatively, the PTH levels were undetectable; hence, the patient was treated with calcitriol and calcium supplementation for 1 month and 4 months, respectively. Genetic work-up for multiple endocrine neoplasia 1 and rearranged during transfection mutations was negative.
DISCUSSION
Solitary PTA is the most common cause of PHPT. Adenomas are mostly sporadic or may be a manifestation of an inheritable syndrome, such as multiple endocrine neoplasia. Although symptomatic disease is more common in children, our patient denied any hypercalcemia symptoms. The distinguishing biochemical feature of PHPT because of PTA is high or inappropriately normal PTH level in the context of high-normal or elevated serum calcium levels. Urinary calcium excretion is usually normal or high. PTAs are localized by ultrasound and Tc-99m-Sestamibi scintigraphy. Management includes parathyroidectomy and monitoring for postoperative hypocalcemia.
CONCLUSION
In a child or adolescent presenting with hypercalcemia and elevated PTH levels, it is important to consider PHPT secondary to PTA, because an early diagnosis will aid in preventing complications from hypercalcemia.
PubMed: 38799050
DOI: 10.1016/j.aace.2024.02.008 -
Medicina (Kaunas, Lithuania) May 2024Secondary hyperparathyroidism (SHPT) poses a common condition among patients with chronic kidney disease (CKD) due to the chronic stimulation of the parathyroid glands...
Secondary hyperparathyroidism (SHPT) poses a common condition among patients with chronic kidney disease (CKD) due to the chronic stimulation of the parathyroid glands as a result of persistently low calcium levels. As a first option for medical treatment, vitamin D receptor analogs (VDRAs) and calcimimetic agents are generally used. Apart from cinacalcet, which is orally taken, in recent years, another calcimimetic agent, etelcalcetide, is being administered intravenously during dialysis. In a 5-year retrospective study between 2018 and 2023, 52 patients undergoing dialysis were studied. The aim of this study is to highlight the possible effects and/or benefits that intravenously administered calcimimetic agents have on CKD patients. A total of 34 patients (65.4%) received cinacalcet and etelcalcetide while parathormone (PTH) and calcium serum levels were monitored on a monthly basis. A total of 29 out of 33 patients (87.9%) that received treatment with etelcalcetide showed a significant decrease in PTH levels, which rose up to 57% compared to the initial values. None of the included patients needed to undergo parathyroidectomy (PTx) due to either extremely high and persistent PTH levels or severe side effects of the medications. It is generally strongly advised that parathyroidectomies should be performed by an expert surgical team. In recent years, a significant decrease in parathyroidectomies has been recorded globally, a fact that is mainly linked to the constantly wider use of new calcimimetic agents. This decrease in parathyroidectomies has resulted in an important decrease in complications occurring in cervical surgeries (e.g., perioperative hemorrhage and nerve damage). Despite the fact that these surgical complications cannot be easily compared to the pharmaceutical side effects, the recorded decrease in parathyroidectomies is considered to be notable, especially in cases of relapse where a difficult reoperation would be considered based on previously published guidelines.
Topics: Humans; Hyperparathyroidism, Secondary; Retrospective Studies; Male; Female; Middle Aged; Cinacalcet; Aged; Calcimimetic Agents; Parathyroidectomy; Renal Dialysis; Peptides; Parathyroid Hormone; Renal Insufficiency, Chronic; Calcium; Adult
PubMed: 38792994
DOI: 10.3390/medicina60050812 -
Cancer Medicine May 2024This study aimed to establish the standardized procedure of trans-areola single site endoscopic parathyroidectomy (TASSEP), and to compare the performance of TASSEP with...
BACKGROUND
This study aimed to establish the standardized procedure of trans-areola single site endoscopic parathyroidectomy (TASSEP), and to compare the performance of TASSEP with that of conventional open parathyroidectomy (COP).
METHODS
This study enrolled 40 patients with primary hyperparathyroidism (PHPT) who underwent TASSEP, and included 40 of 176 PHPT patients who underwent COP based on propensity score matching. The retrospective analysis was conducted based on prospectively collected data. Perioperative outcomes, including surgical profile, surgical burden and cosmetic results and follow-up were reported. The learning curve was described using a cumulative sum (CUSUM) analysis.
RESULTS
40 TASSEPs were completed successfully without conversions or severe complications. There was no statistically significant difference in operation time between TASSEP and COP groups (80.83 ± 11.95 vs. 76.95 ± 7.30 min, p = 0.084). Experience of 17 cases was necessitated to reach the learning curve of TASSEP. Postoperative pain score and traumatic index (C-reactive protein and erythrocyte sedimentation rate) in TASSEP were apparently lower than those in COP group (p < 0.05). During the proliferation and stabilization phases, TASSEP was associated with significantly better incision recovery and cosmetic scores. Postoperative serum calcium and PTH levels throughout the follow-up period indicated satisfactory surgical qualities in both groups.
CONCLUSION
Based on precise preoperative localization and intraoperative planning facilitated by three-dimensional (3D) virtual modeling, TASSEP can be feasibly performed on selected patients with satisfactory success rates and low complication rates, providing preferable cosmetic results and alleviating the surgical burden to a certain extent.
Topics: Humans; Parathyroidectomy; Male; Female; Middle Aged; Parathyroid Neoplasms; Retrospective Studies; Adenoma; Endoscopy; Treatment Outcome; Adult; Hyperparathyroidism, Primary; Aged; Propensity Score; Operative Time
PubMed: 38770646
DOI: 10.1002/cam4.7290 -
Endocrinology and Metabolism (Seoul,... May 2024Parathyroid hormone (PTH) and fibroblast growth factor 23 (FGF23) each play a central role in the pathogenesis of chronic kidney disease (CKD)-mineral and bone disorder....
Parathyroid hormone (PTH) and fibroblast growth factor 23 (FGF23) each play a central role in the pathogenesis of chronic kidney disease (CKD)-mineral and bone disorder. Levels of both hormones increase progressively in advanced CKD and can lead to damage in multiple organs. Secondary hyperparathyroidism (SHPT), characterized by parathyroid hyperplasia with increased PTH secretion, is associated with fractures and mortality. Emerging evidence suggests that these associations may be partially explained by PTH-induced browning of adipose tissue and increased energy expenditure. Observational studies suggest a survival benefit of PTHlowering therapy, and a recent study comparing parathyroidectomy and calcimimetics further suggests the importance of intensive PTH control. The mechanisms underlying the regulation of FGF23 secretion by osteocytes in response to phosphate load have been unclear, but recent experimental studies have identified glycerol-3-phosphate, a byproduct of glycolysis released by the kidney, as a key regulator of FGF23 production. Elevated FGF23 levels have been shown to be associated with mortality, and experimental data suggest off-target adverse effects of FGF23. However, the causal role of FGF23 in adverse outcomes in CKD patients remains to be established. Further studies are needed to determine whether intensive SHPT control improves clinical outcomes and whether treatment targeting FGF23 can improve patient outcomes.
PubMed: 38752265
DOI: 10.3803/EnM.2024.1978 -
Transplant International : Official... 2024Tertiary hyperparathyroidism (THPT) is characterized by elevated parathyroid hormone and serum calcium levels after kidney transplantation (KTx). To ascertain whether...
Pre-Transplant Calcimimetic Use and Dose Information Improves the Accuracy of Prediction of Tertiary Hyperparathyroidism after Kidney Transplantation: A Retrospective Cohort Study.
Tertiary hyperparathyroidism (THPT) is characterized by elevated parathyroid hormone and serum calcium levels after kidney transplantation (KTx). To ascertain whether pre-transplant calcimimetic use and dose information would improve THPT prediction accuracy, this retrospective cohort study evaluated patients who underwent KTx between 2010 and 2022. The primary outcome was the development of clinically relevant THPT. Logistic regression analysis was used to evaluate pre-transplant calcimimetic use as a determinant of THPT development. Participants were categorized into four groups according to calcimimetic dose, developing two THPT prediction models (with or without calcimimetic information). Continuous net reclassification improvement (CNRI) and integrated discrimination improvement (IDI) were calculated to assess ability to reclassify the degree of THPT risk by adding pre-transplant calcimimetic information. Of the 554 patients, 87 (15.7%) developed THPT, whereas 139 (25.1%) received pre-transplant calcimimetic treatment. Multivariate logistic regression analysis revealed that pre-transplant calcimimetic use was significantly associated with THPT development. Pre-transplant calcimimetic information significantly improved the predicted probability accuracy of THPT (CNRI and IDI were 0.91 [ < 0.001], and 0.09 [ < 0.001], respectively). The THPT prediction model including pre-transplant calcimimetic information as a predictive factor can contribute to the prevention and early treatment of THPT in the era of calcimimetics.
Topics: Humans; Kidney Transplantation; Retrospective Studies; Male; Female; Middle Aged; Calcimimetic Agents; Adult; Calcium; Hyperparathyroidism; Parathyroid Hormone; Logistic Models
PubMed: 38751772
DOI: 10.3389/ti.2024.12704 -
EJNMMI Reports May 2024To determine the use of four-dimensional CT as first-line imaging compared to the traditional combination of ultrasound and [Tc]Tc-Sestamibi SPECT.
Four-dimensional computed tomography as first-line imaging in primary hyperparathyroidism, a retrospective comparison to conventional imaging in a predominantly single adenoma population.
BACKGROUND
To determine the use of four-dimensional CT as first-line imaging compared to the traditional combination of ultrasound and [Tc]Tc-Sestamibi SPECT.
MATERIALS AND METHODS
Retrospective review of preoperative imaging in patients with primary hyperparathyroidism, who underwent parathyroidectomy between 2012 and 2021. In one group, the combination ultrasound and [Tc]Tc-Sestamibi SPECT was used as first-line imaging (n = 54), in the other group four-dimensional CT was the first-line imaging modality (n = 51). Sensitivity and positive predictive value were calculated on patient, lateralisation and localisation level. The need for additional imaging was also assessed for both groups.
RESULTS
Four-dimensional CT had a significantly higher sensitivity compared to the combination of ultrasound/[Tc]Tc-Sestamibi SPECT on patient and localisation level (70.6% vs. 51.9%, p = 0.049 and 60.8% vs. 35.2%, p = 0.009 respectively). Sensitivity for lateralisation also appeared higher, but did not reach significance (62.7% vs. 44.4%, p = 0.060). Positive predictive value was not significantly higher for four-dimensional CT compared to ultrasound and [Tc]Tc-Sestamibi SPECT (88.9% vs. 85.7% for lateralisation and 86.1% vs. 67.9% for localisation respectively). Additional imaging was required in 14 patients with four-dimensional CT as first-line imaging (27.4%) consisting of 2 ultrasound/[Tc]Tc-Sestamibi SPECT and 13 [F]fluorocholine PET/CT, compared to 24 patients with ultrasound/[Tc]Tc-Sestamibi SPECT as first-line imaging (44.4%), requiring 22 four-dimensional CT and 9 [F]fluorocholine PET/CT.
CONCLUSIONS
Four-dimensional CT as the sole first-line parathyroid imaging modality had higher sensitivity than the combination of ultrasound and [Tc]Tc-Sestamibi SPECT, therefore requiring fewer additional procedures. Although the most costly, [F]fluorocholine PET/CT was the most effective technique to localise parathyroid adenoma in case all other imaging was negative.
PubMed: 38748330
DOI: 10.1186/s41824-024-00198-5 -
BJS Open May 2024Multicentre studies have previously reported on national outcomes of surgery for primary hyperparathyroidism, but not investigated whether management and outcome are... (Observational Study)
Observational Study
BACKGROUND
Multicentre studies have previously reported on national outcomes of surgery for primary hyperparathyroidism, but not investigated whether management and outcome are uniform among countries. This study investigated whether there are differences among European countries in operative management and outcome of surgery for primary hyperparathyroidism.
METHODS
Using data from Eurocrine®, a pan-European registry for endocrine surgeries, a retrospective observational cross-sectional multicentre study with 99 participating centres in 14 European countries was performed. Data on age, sex, calcium levels, operative strategy, conversion rate and rate of failed exploration were analysed for patients who underwent initial surgery for sporadic primary hyperparathyroidism. Primary outcome measures were intention to perform limited parathyroidectomy and the rate of hypercalcaemia at first follow-up.
RESULTS
A total of 9548 patients were registered between 2015 and 2020. There were 7642 (80%, range 74.5-93.2%) females. There was intention to perform limited parathyroidectomy in 7320 of 9548 (76.7%) operations, ranging from 498 of 1007 (49.5%) to 40 of 41 (97.6%) among countries. Hypercalcaemia at first follow-up (median time to follow-up 15 days) was found in 416 of 9548 (4.4%) operations, ranging from 0 of 119 (0%) to 3 of 38 (7.9%) among countries.
CONCLUSION
This study demonstrated large differences in the intention to perform limited parathyroidectomy for primary hyperparathyroidism among European countries, as well as differences in the rate of postoperative hypercalcaemia. Future studies are needed to evaluate the impact of these different healthcare practices on patient outcomes.
Topics: Humans; Hyperparathyroidism, Primary; Female; Male; Parathyroidectomy; Europe; Retrospective Studies; Middle Aged; Cross-Sectional Studies; Hypercalcemia; Aged; Adult; Treatment Outcome; Registries
PubMed: 38747104
DOI: 10.1093/bjsopen/zrae037 -
Cureus Apr 2024Secondary hyperparathyroidism is a prevalent complication of end-stage renal disease (ESRD), arising from chronic renal insufficiency leading to disturbed calcium...
Secondary hyperparathyroidism is a prevalent complication of end-stage renal disease (ESRD), arising from chronic renal insufficiency leading to disturbed calcium metabolism. This disruption triggers hypersecretion of the parathyroid gland, characterizing the condition. Osteitis fibrosa cystica (OFC), a rare complication of untreated secondary hyperparathyroidism, results in benign resorptive bone lesions and the formation of cystic cavities within bones. Our case report describes a 46-year-old incarcerated Hispanic male with a 17-year history of end-stage renal disease and secondary hyperparathyroidism. The patient initially presented with a traumatic right elbow injury. Further diagnostic evaluation revealed an 8 cm destructive process involving the distal humerus, initially suspected as malignancy but confirmed as OFC through bone biopsy. Management involved orthopedic surgery performing an open reduction and internal fixation (ORIF) of the affected limb, with subsequent consideration for inpatient parathyroidectomy. Imaging studies, including magnetic resonance imaging (MRI) and computed tomography (CT) scans, elucidated a 6 × 5.5 cm soft tissue mass, further confirmed as a brown tumor. The case underscores the complexities of diagnosing OFC, often misinterpreted in radiologic studies, and highlights the multidisciplinary approach involving orthopedics, otolaryngology, and nephrology in managing this intricate scenario. The objective is to explore clinical manifestations and treatment challenges of OFC and secondary hyperparathyroidism triggered by trauma in end-stage renal disease, emphasizing the need for continued awareness and precise diagnostic strategies in resource-rich areas.
PubMed: 38741796
DOI: 10.7759/cureus.58208 -
Radiology Case Reports Aug 2024An 18-year-old male with multiple endocrine neoplasm type 1 (MEN1) syndrome presented with hyperparathyroidism. Parathyroidectomy was performed. Patient complained of...
An 18-year-old male with multiple endocrine neoplasm type 1 (MEN1) syndrome presented with hyperparathyroidism. Parathyroidectomy was performed. Patient complained of bone pain afterwards, multiple imaging modalities revealed features of osteitis fibrosa cystica and biochemical profile showed features of hungry bone syndrome. Incidental suspicious pancreatic lesion was initially revealed by F-FDG PET/CT scan while MRI further characterized the possibility of insulinoma. Ultimately, the patient was diagnosed of MEN1 syndrome by genetic test. This case report demonstrates the utilization of various imaging modalities such as ultrasound, Tc-sestamibi parathyroid scintigraphy, bone scintigraphy, CT, PET/CT and MRI, which leads to ultimately the diagnosis of MEN1 syndrome.
PubMed: 38737174
DOI: 10.1016/j.radcr.2024.04.024 -
International Journal of Surgery Case... May 2024Maxillomandibular involvement with brown tumours is rare, especially in the paediatric population. We present a rare case of a young girl with brown tumour (BT)...
INTRODUCTION AND IMPORTANCE
Maxillomandibular involvement with brown tumours is rare, especially in the paediatric population. We present a rare case of a young girl with brown tumour (BT) occurring in the mandible as the first manifestation of hyperparathyroidism.
CASE PRESENTATION
A 7-year-old black female, presented with a 6-month history of a swelling on the left mandible. Patient had a history of intractable gastrointestinal symptoms such as nausea, vomiting and abdominal cramps. Biopsy confirmed the lesion as a giant cell lesion. Elevated parathyroid hormone (PTH) levels confirmed the diagnosis of a BT of hyperparathyroidism. The patient underwent surgical intervention involving parathyroidectomy. This was followed by segmental resection of the mandibular tumour two years later.
CLINICAL DISCUSSION
Brown Tumour is a rare, non-neoplastic lesion resulting from abnormal bone metabolism secondary to hyperparathyroidism. BT predominantly arise in long bones and the axial skeleton. Maxillomandibular involvement is very rare. In the present case, after parathyroidectomy, normal PTH and calcium levels were restored, and there was relief of gastrointestinal tract hypercalcaemic symptoms. However, there was no spontaneous regression of the mandibular tumour over a 2-year observation period. Hemimandibulectomy followed by reconstruction with a plate and costochondral graft was then performed.
CONCLUSION
It is difficult to differentiate BT from other giant cell lesions without blood chemistry revealing hyperparathyroidism. The distinction is imperative to avoid mutilating and aggressive treatment for BTs. Although BTs are amenable for conservative treatment, the present case illustrates that for bigger tumours, in the absence of spontaneous regression, aggressive surgical treatment may be required.
PubMed: 38735218
DOI: 10.1016/j.ijscr.2024.109735