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Case Reports in Nephrology 2024Urinary lithiasis constitutes a recurrent pathology affecting a relatively young population. The risk of progression to chronic renal failure and the cost of treatment...
BACKGROUND
Urinary lithiasis constitutes a recurrent pathology affecting a relatively young population. The risk of progression to chronic renal failure and the cost of treatment are the most important issues. Primary hyperparathyroidism (PHPT) is responsible for urolithiasis and nephrocalcinosis in 7% of patients, and it represents the 7th cause of urolithiasis in Tunisia. Unfortunately, it remains an underdiagnosed pathology although it is curable. We aim to determine the clinical, biological, therapeutic, and evolutionary particularities of urinary lithiasis associated with PHPT in a nephrology setting.
METHODS
This is a monocentric, retrospective, descriptive study which took place in our nephrology department during the period from January 2010 to January 2023. Ten patients were included. All of them underwent blood and urine tests and a morphoconstitutional study of the urinary stones if possible.
RESULTS
The median age at diagnosis of PHPT was 42 years (34-54). The median time from the onset of kidney stones to the diagnosis of PHPT was 6.2 years (1-17). The male/female gender ratio was 0.66. Five patients had hypertension, two patients had obesity, one patient had diabetes, and three patients had urinary tract infections. Kidney stones were bilateral in eight cases and unilateral in two cases. Nine patients underwent urological intervention: surgery in 5 cases associated with nephrectomy in one case, extracorporeal lithotripsy in 4 cases, and percutaneous nephrolithotomy in two cases. The diagnosis of PHPT was retained with high or uncontrolled PTH associated with hypercalcemia in 8 cases and normocalcemic PHPT was found in 2 patients. Two patients had parathyroid adenoma and one patient had mediastinal adenoma. Radiology exploration was normal for the others patients. Surgery was performed in 7 patients and histology revealed an adenoma in 5 cases and hyperplasia in one case. The predominant urinary risk factors in our study were hypercalciuria in 6 cases and insufficient diuresis in 4 cases.
CONCLUSION
This study underlines the role of the nephrologist in the exploration of urinary lithiasis and the prevention of recurrences, especially as PHPT is a curable aetiology of urolithiasis and affects a relatively young population. The determination of the epidemiological profile of patients with stones associated with primary PHPT and lithogenic risk factors allows the primary and secondary prevention of stone formation.
PubMed: 38419822
DOI: 10.1155/2024/1265364 -
Cureus Sep 2023Hyperparathyroidism usually presents asymptomatically with elevated levels of calcium and parathyroid hormone; this biochemical imbalance establishes the diagnosis. In...
Hyperparathyroidism usually presents asymptomatically with elevated levels of calcium and parathyroid hormone; this biochemical imbalance establishes the diagnosis. In 80-85% of cases of primary hyperparathyroidism, singular parathyroid adenomas occur. In rare cases, this problem occurs due to multiple adenomas, multiglandular hyperplasia, or parathyroid carcinoma. Recurrent primary hyperparathyroidism (R-PHPT), as demonstrated in this case, is defined as hypercalcemia that arises after six months of normocalcemia following initial surgery for PHPT. The aim of this report is to describe the diagnosis and management of three parathyroid adenomas in a patient, two of which occurred after an initial partial parathyroidectomy.
PubMed: 37809263
DOI: 10.7759/cureus.44849 -
Head & Neck Mar 2024Primary adenoma (PA) and multi-gland hyperplasia (MGH) account for 85% and 15% of primary hyperparathyroidism (PHPT) cases, respectively. Near-infrared autofluorescence...
BACKGROUND
Primary adenoma (PA) and multi-gland hyperplasia (MGH) account for 85% and 15% of primary hyperparathyroidism (PHPT) cases, respectively. Near-infrared autofluorescence (NIRAF) enhances intraoperative parathyroid identification. We hypothesized that PA would display a more heterogeneous NIRAF pattern compared to MGH.
METHODS
Patients undergoing surgery for sporadic PHPT were categorized based on the presence of PA or MGH. To quantify heterogeneity, we utilized ratios of (1) mean parathyroid gland (PG) NIRAF over background NIRAF (mean ratio), (2) minimum and (3) maximum PG NIRAF over mean PG NIRAF (minimum and maximum ratios). Additionally, a heterogeneity score was quantified using mean ratio (mean PG NIRAF over background NIRAF), and overall NIRAF (mean NIRAF of eight random 15 × 15 pixel areas). A point was assigned to ratios <0.8 or >1.2. Images were quantified by ImageJ software. Mann-Whitney test was performed for all comparisons.
RESULTS
Of 78 patients, 63 had a single PA and 15 had MGH, totaling 102 PGs. There was no difference between their mean ratios. PA had a lower minimum ratio compared to that of MGH (0.86 ± 0.01 vs. 0.93 ± 0.01, p = 0.001) and a brighter maximum ratio (1.21 ± 0.02 vs. 1.12 ± 0.01, p = 0.0008). PA also scored higher on their heterogeneity scores compared to MGH (1.27 ± 0.23 vs. 0.33 ± 0.15, p = 0.001).
CONCLUSION
Single parathyroid adenomas display a more heterogeneous autofluorescence pattern compared to that of multi-gland hyperplasia. Intraoperative characterization of PGs by real-time NIR imaging patterns may be a beneficial adjunct during parathyroid surgery.
Topics: Humans; Parathyroid Glands; Hyperparathyroidism, Primary; Hyperplasia; Optical Imaging; Parathyroidectomy; Adenoma
PubMed: 38133490
DOI: 10.1002/hed.27599 -
Cureus Feb 2024Cure rates following parathyroidectomy for primary hyperparathyroidism are excellent, with well-documented low short-term recurrence rates of hypercalcaemia. Rates of...
BACKGROUND
Cure rates following parathyroidectomy for primary hyperparathyroidism are excellent, with well-documented low short-term recurrence rates of hypercalcaemia. Rates of long-term recurrence have been investigated to a lesser extent, but recent studies have reported higher than anticipated rates. This study sought to evaluate recurrence rates at more than four years post seemingly corrective surgery and depending on the findings, propose whether recommendations of annual calcium monitoring post-parathyroidectomy are appropriate based on the limited data available at the time of formulating guidelines.
METHODS
Fifty-two sequential parathyroidectomies for primary hyperparathyroidism from 2014-2016 from a single unit were retrospectively followed up with serum calcium levels. A hospital computer system was used to collect data on pre-operative, immediate post-operative and most recent follow-up calcium levels. Patients were excluded if there was no minimum of 48 months between the operation date and most recent calcium. Recurrence was defined as hypercalcaemia more than six months after eucalcaemia post-parathyroidectomy.
RESULTS
Of the 52 cases analysed, two were lost to long-term follow-up, two patients died during the follow-up period while 10 did not meet the inclusion criteria of at least 48 months follow-up. This resulted in a cohort of 38 patients (mean age 66.4 years, 78.9% female). The median follow-up of 73.17 months (range 48.77-95.47 months) demonstrated a hypercalcaemia recurrence of 5.26% (2/38 patients). These cases were due to misdiagnosed parathyroid hyperplasia as opposed to suspected adenoma. Therefore, the long-term cure rate was 94.74% (36/38 patients).
CONCLUSION
These findings support the high cure rates and low recurrence rates of the numerous short-term studies already performed despite a longer follow-up period. This is in contrast to recent series which have documented a higher recurrence in the long-term. This study would, therefore, suggests recommendations of annual calcium monitoring are excessive and that less frequent calcium monitoring is necessary in the first few years post-operation. However, the 5.26% recurrence rate in this study is not insignificant and follow-up is still paramount. Therefore, following the initial post-operative assessment, the authors propose a follow-up at the five-year mark and an annual continuation from this point forward due to the evidenced delayed recurrence of hypercalcaemia.
PubMed: 38449925
DOI: 10.7759/cureus.53591 -
Technology and Health Care : Official... Jun 2024Secondary hyperparathyroidism (SHPT) is one of the common complications of end-stage renal disease-uremia, and is mainly manifested as parathyroid hyperplasia and...
BACKGROUND
Secondary hyperparathyroidism (SHPT) is one of the common complications of end-stage renal disease-uremia, and is mainly manifested as parathyroid hyperplasia and abnormal secretion of parathyroid hormone (PTH).
OBJECTIVE
To investigate the value and advantages of contrast-enhanced ultrasound (CEUS) in evaluating the survival of autografts after parathyroidectomy + parathyroid autotransplantation.
METHODS
In this study, 125 patients with renal failure due to polycystic kidney disease, chronic nephritis, diabetic nephropathy, lupus nephritis, and atherosclerotic nephropathy were enrolled as the participants and each of them had 4 secondary hyperactive parathyroid glands and underwent parathyroid autotransplantation. One parathyroid gland was taken from each patient and equally divided into 4 parts and placed in the subcutaneous fat of one forearm for transplantation. CEUS was performed 14 days after the transplantation to observe the micro blood supply of the graft and assess the survival and secretory function of the transplanted parathyroid. The grafts were divided into the partial survival group and the total survival group based on the enhancement characteristics. The survival of the grafts was determined by comparing the parathyroid hormone level in bilateral elbow cephalic veins 1 month after surgery.
RESULTS
Among the 125 patients, 112 had linear or punctate enhancement of 2-4 parathyroid glands 14 days after surgery, and 13 patients had linear or punctate enhancement of 0-1 parathyroid gland. There were statistically significant differences in the perfusion pattern, enhancement uniformity, and parathyroid hormone levels in the cephalic veins at the elbow on both the graft and non-graft sides among all groups (P< 0.05).
CONCLUSION
Compared to the detection of the difference in the parathyroid hormone level in the cephalic vein of bilateral elbows 1 month after surgery, CEUS can reflect the parathyroid survival after transplantation more quickly and accurately 2 weeks later, and provide a more rapid and agile non-invasive clinical diagnosis method.
PubMed: 38905065
DOI: 10.3233/THC-231191 -
Frontiers in Endocrinology 2023A germline mutation can be identified in up to 10% of patients with primary hyperparathyroidism (PHPT). In 2017, a high frequency of the [(NM_ 004752.4) c.1181A> C;...
CONTEXT
A germline mutation can be identified in up to 10% of patients with primary hyperparathyroidism (PHPT). In 2017, a high frequency of the [(NM_ 004752.4) c.1181A> C; p.Tyr394Ser; rs142287570] variant was reported in PHPT Ashkenazi Jews (AJ).
OBJECTIVE
To evaluate the presence of the p.Tyr394Ser variant in Israeli patients addressed for genetic evaluation to characterize their phenotype and clinical management.
METHOD
Patients with PHPT who underwent addressed for genetic screening for suspected familial hypocalciuric hypercalcemia (FHH), a family history of isolated hyperparathyroidism (FIHP), or failed parathyroidectomy with persistent PHPT were recruited. Those with normal initial selected gene sequencing or hyperparathyroid genetic panel completed the p.Tyr394Ser variant sequencing. The prevalence of this variant was evaluated using our local genomic database.
RESULTS
A total of 42 single individuals from unrelated kindreds were evaluated. A disease-causing mutation was found in 11 (26.1%) patients: 10 were diagnosed with FHH (eight and two mutations), and one patient had a CKN2B mutation. In 28 of the remaining patients, the p.Tyr394Ser variant was positive in three (10.7%), and all were AJ. Within AJ (15/28, 53.5%), the rate of the p.Tyr394Ser variant was 3/15 (20%), and of those, two had a history of familial isolated hyperparathyroidism. Multi-glandular parathyroid adenoma/hyperplasia was also observed in two of these patients. No clinical or laboratory findings could discriminate patients with the p.Tyr394Ser variant from those with FHH. Cinacalcet normalized the calcium levels in one patient. The prevalence of the p.Tyr394Ser variant in 15,407 tests in our local genomic database was 0.98%.
CONCLUSION
In contrast to previous observations, the p.Tyr394Ser variant-associated phenotype may be mild in AJ with FIHP, sometimes mimicking FHH. Because surgery may be curative, surgeons should be aware of the possibility of multiple gland diseases in these patients. The clinical spectrum and clinical utility of screening for this variant warrant further investigation.
Topics: Humans; Hyperparathyroidism, Primary; Israel; Parathyroid Hormone; Mutation; Nuclear Proteins; Transcription Factors
PubMed: 38130397
DOI: 10.3389/fendo.2023.1254156 -
Endocrine Connections Oct 2023Secondary hyperparathyroidism (SHPT) is a common complication of end-stage kidney disease (ESKD). Hungry bone syndrome (HBS) occurs frequently in patients on maintenance...
Secondary hyperparathyroidism (SHPT) is a common complication of end-stage kidney disease (ESKD). Hungry bone syndrome (HBS) occurs frequently in patients on maintenance dialysis receiving parathyroidectomy for refractory SHPT. However, there is scanty study investigating the clinical risk factors that predict postoperative HBS, and its outcome in peritoneal dialysis (PD) patients. We conducted a single-center retrospective study to analyze 66 PD patients who had undergone parathyroidectomy for secondary hyperparathyroidism at Chang Gung Memorial Hospital between 2009 and 2019. The patients were stratified into two groups based on the presence (n=47) or absence (n=19) of HBS after parathyroidectomy. Subtotal parathyroidectomy was the most common surgery performed (74.2%), followed by total parathyroidectomy with autoimplantation (25.8%). Pathological examination of all surgical specimens revealed parathyroid hyperplasia (100%). Patients with HBS had lower levels of postoperative nadir corrected calcium, higher alkaline phosphate (ALP), and higher potassium levels compared with patients without HBS (all P<0.05). A multivariate logistic regression model confirmed that lower preoperative serum calcium level (OR 0.354, 95% CI 0.133-0.940, P=0.037), higher ALP (OR 1.026, 95% CI 1.008-1.044, P=0.004), and higher potassium level (OR 6.894, 95% CI 1.806-26.317, P=0.005) were associated with HBS after parathyroidectomy. Patients were followed for 58.2±30.8 months after the surgery. There was no significant difference between HBS and non-HBS groups in persistence (P=0.496) or recurrence (P=1.000) of hyperparathyroidism. The overall mortality rate was 10.6% with no significant difference found between both groups (P=0.099). We concluded that HBS is a common complication (71.2%) of parathyroidectomy for SHPT and should be managed appropriately.
PubMed: 37606078
DOI: 10.1530/EC-23-0107 -
Cureus Oct 2023Multiple endocrine neoplasia (MEN) is an inherited, autosomal dominant condition characterized by primary parathyroid hyperplasia, medullary thyroid neoplasm, and...
Multiple endocrine neoplasia (MEN) is an inherited, autosomal dominant condition characterized by primary parathyroid hyperplasia, medullary thyroid neoplasm, and pheochromocytoma. It most commonly presents with medullary thyroid cancer and less frequently with other complaints. Pheochromocytoma can also manifest through gastrointestinal complaints such as abdominal pain, nausea, and constipation. We present a normotensive case of pheochromocytoma, initially featuring abdominal pain and vomiting, which was later found to be associated with neck swelling and medullary thyroid cancer. The patient underwent an adrenalectomy and has continued to visit our endocrinology clinic for ongoing monitoring and treatment of iatrogenic hypoparathyroidism and hypothyroidism. A brief review is also provided.
PubMed: 38022135
DOI: 10.7759/cureus.47063 -
The Journal of Clinical Endocrinology... Feb 2024
Response to Letter to the Editor From Jagannath and Mayilvaganan: "FCH-PET/CT in Primary Hyperparathyroidism With Discordant/Negative MIBI Scintigraphy and Ultrasonography".
Topics: Humans; Positron Emission Tomography Computed Tomography; Hyperparathyroidism, Primary; Radionuclide Imaging; Parathyroid Glands; Ultrasonography
PubMed: 37846771
DOI: 10.1210/clinem/dgad615 -
Scandinavian Journal of Surgery : SJS :... Mar 2024Studies on the outcomes of parathyroid surgery are scarce. The aim was to report the outcomes and to study the association between pre- and peri-operative information...
BACKGROUND AND OBJECTIVE
Studies on the outcomes of parathyroid surgery are scarce. The aim was to report the outcomes and to study the association between pre- and peri-operative information with the outcomes of patients operated for primary hyperparathyroidism.
METHODS
This was a retrospective, descriptive study with unselected patients treated surgically for primary hyperparathyroidism from a catchment population of 704,500 in Finland. Data were acquired from the electronic hospital registers based on parathyroid surgery procedure codes between 1 January 2017 and 31 December 2018. Preoperative data, surgical data, preoperative and postoperative laboratory values, histopathological findings, and postoperative clinical data were recorded.
RESULTS
During the 2-year study period, 149 patients with primary hyperparathyroidism were treated surgically with a 97% remission rate. Surgical complications included postoperative bleeding in two patients (1%) and vocal cord paralysis in one patient (0.6%). No postoperative infections were reported. Three patients (2%) developed postoperative hypoparathyroidism necessitating the use of alfacalcidol more than 1 month after surgery. Ionized calcium measured 0-1 days after surgery was not statistically significantly associated with remission or postoperative hypoparathyroidism. Serum parathyroid hormone (PTH) assessed 0-1 days postoperatively was associated with persistent disease, but not with postoperative hypoparathyroidism. The histopathological diagnosis was adenoma or hyperplasia in 112 patients (75%), atypical adenoma in 28 patients (19%), and carcinoma in five patients (3%). Patients with parathyroid carcinoma had higher preoperative ionized calcium and PTH values than those with adenoma or hyperplasia.
CONCLUSIONS
Most patients who were operated due to primary hyperparathyroidism achieved normocalcemia after surgery, and the frequency of complications was low. Ionized calcium taken 0-1 days after surgery was not associated with remission of hyperparathyroidism or postoperative hypoparathyroidism. High postoperative serum PTH predicted persistent disease.
PubMed: 38433618
DOI: 10.1177/14574969241228409