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Frontiers in Endocrinology 2023Autosomal dominant hypocalcemia (ADH1) is a genetic disorder characterized by low serum calcium and low or inappropriately normal levels of parathyroid hormone. The...
Autosomal dominant hypocalcemia (ADH1) is a genetic disorder characterized by low serum calcium and low or inappropriately normal levels of parathyroid hormone. The disease is caused by a heterozygous activating mutation of the calcium-sensing receptor () gene, encoding a G-Protein-coupled cell membrane sensor of extracellular calcium concentration mainly expressed by parathyroid glands, renal tubules, and the brain. ADH1 has been linked to 113 unique germline mutations, of which nearly 96% are missense mutations. There is often a lack of a clear genotype/phenotype correlation in the reported literature. Here, we described a case series of 6 unrelated ADH1 probands, each one bearing a gain-of-function mutation, and two children of one of these cases, matching our identified mutations to the same ones previously reported in the literature, and comparing the clinical and biochemical characteristics, as well as the complication profile. As a result of these genetic and clinical comparisons, we propose that a genotype/phenotype correlation may exist because our cases showed similar presentation, characteristics, and severity, with respect to published cases with the same or similar mutations. We also contend that the severity of the presentation is highly influenced by the specific variant. These findings, however, require further evaluation and assessment with a systematic review.
Topics: Gain of Function Mutation; Receptors, Calcium-Sensing; Calcium; Research; Mutation
PubMed: 37654565
DOI: 10.3389/fendo.2023.1215036 -
Frontiers in Endocrinology 2023This study aimed to systematically review research on cinacalcet and secondary hyperparathyroidism (SHPT) using machine learning-based statistical analyses. (Meta-Analysis)
Meta-Analysis
INTRODUCTION
This study aimed to systematically review research on cinacalcet and secondary hyperparathyroidism (SHPT) using machine learning-based statistical analyses.
METHODS
Publications indexed in the Web of Science Core Collection database on Cinacalcet and SHPT published between 2000 and 2022 were retrieved. The R package "Bibliometrix," VOSviewer, CiteSpace, meta, and latent Dirichlet allocation (LDA) in Python were used to generate bibliometric and meta-analytical results.
RESULTS
A total of 959 articles were included in our bibliometric analysis. In total, 3753 scholars from 54 countries contributed to this field of research. The United States, Japan, and China were found to be among the three most productive countries worldwide. Three Japanese institutions (Showa University, Tokai University, and Kobe University) published the most articles on Cinacalcet and SHPT. Fukagawa, M.; Chertow, G.M.; Goodman W.G. were the three authors who published the most articles in this field. Most articles were published in , , and . Research on Cinacalcet and SHPT has mainly included three topics: 1) comparative effects of various treatments, 2) the safety and efficacy of cinacalcet, and 3) fibroblast growth factor-23 (FGF-23). Integrated treatments, cinacalcet use in pediatric chronic kidney disease, and new therapeutic targets are emerging research hotspots. Through a meta-analysis, we confirmed the effects of Cinacalcet on reducing serum PTH ( = -0.56, 95% = -0.76 to -0.37, = 0.001) and calcium ( = -0.93, 95% = -1.21to -0.64, = 0.001) and improving phosphate ( = 0.17, 95% = -0.33 to -0.01, = 0.033) and calcium-phosphate product levels ( = -0.49, 95% = -0.71 to -0.28, = 0.001); we found no difference in all-cause mortality ( = 0.97, 95% = 0.90 to 1.05, = 0.47), cardiovascular mortality ( = 0.69, 95% = 0.36 to 1.31, = 0.25), and parathyroidectomy ( = 0.36, 95% = 0.09 to 1.35, = 0.13) between the Cinacalcet and non-Cinacalcet users. Moreover, Cinacalcet was associated with an increased risk of nausea ( = 2.29, 95% = 1.73 to 3.05, = 0.001), hypocalcemia ( = 4.05, 95% = 2.33 to 7.04, = 0.001), and vomiting ( = 1.90, 95% = 1.70 to 2.11, = 0.001).
DISCUSSION
The number of publications indexed to Cinacalcet and SHPT has increased rapidly over the past 22 years. Literature distribution, research topics, and emerging trends in publications on Cinacalcet and SHPT were analyzed using a machine learning-based bibliometric review. The findings of this meta-analysis provide valuable insights into the efficacy and safety of cinacalcet for the treatment of SHPT, which will be of interest to both clinical and researchers.
Topics: Child; Humans; Calcimimetic Agents; Calcium; Cinacalcet; Hyperparathyroidism, Secondary; Phosphates; United States; Machine Learning
PubMed: 37538795
DOI: 10.3389/fendo.2023.1146955 -
Nutrients Jul 2023Vitamin D has been shown to have multiple pleiotropic effects beyond bone and mineral metabolism, with purported roles in cardiovascular disease, cancer, and host... (Review)
Review
Vitamin D has been shown to have multiple pleiotropic effects beyond bone and mineral metabolism, with purported roles in cardiovascular disease, cancer, and host immunity. Vitamin D deficiency is common in patients with end-stage kidney disease (ESKD); however, current clinical practice has favored the use of the active hormone. Whether vitamin D deficiency should be corrected in patients with ESKD remains unclear, as few randomized trials have been conducted. In this systematic review, we summarize the current evidence examining whether vitamin D supplementation improves outcomes, beyond mineral metabolism, in patients with ESKD. Data from randomized controlled trials of adults with ESKD were obtained by searching Ovid MEDLINE, Embase, the Cochrane Central Register of Controlled Trials, and the Web of Science Core Collection from inception to February 2023. Twenty-three trials composed of 2489 participants were identified for inclusion. Data were synthesized by two independent reviewers and summarized in tables organized by outcome. Outcomes included measures of mortality, cardiovascular disease, inflammation, muscle strength/function, nutrition, patient well-being, and outcomes specific to ESKD including erythropoietin usage, pruritus, and dialysis access maturation. The Cochrane risk of Bias Tool (RoB 2, 2019) was used to assess study quality. Overall, our findings indicate a minimal and varied benefit of native vitamin D supplementation. From the largest studies included, we determine that vitamin D has no demonstrated effect on patient-reported measures of well-being or utilization of erythropoietin, nor does it change levels of the inflammation biomarker -reactive protein. Included trials were heterogeneous with regards to outcomes, and the majority studied small participant populations with a relatively short follow-up. We conclude that vitamin D supplementation corrects vitamin D deficiency and is safe and well-tolerated in humans with ESKD. However, it is not clear from clinical trials conducted to date that a causal pathway exists between 25(OH)D and pleiotropic effects that is responsive to vitamin D treatment.
Topics: Adult; Humans; Vitamin D; Cardiovascular Diseases; Renal Dialysis; Randomized Controlled Trials as Topic; Vitamins; Kidney Failure, Chronic; Vitamin D Deficiency; Dietary Supplements; Erythropoietin; Minerals
PubMed: 37447398
DOI: 10.3390/nu15133072 -
Cureus Jun 2023Major depressive disorder (MDD) is a common neuropsychiatry manifestation that is more prevalent lately. Many contributing factors are present (for example,... (Review)
Review
Major depressive disorder (MDD) is a common neuropsychiatry manifestation that is more prevalent lately. Many contributing factors are present (for example, neurochemical, physiological, pathophysiological, and endocrinological factors). Patients with increased serum parathyroid levels are usually linked to psychosis symptoms but not to depressive symptoms. We conducted this systematic review to explore a correlation between depressive disorder and increased serum parathyroid levels, a major endocrinological pathology, and help establish mental wellness in patients suffering from hyperparathyroidism. We conducted a thorough literature search using five major databases, MEDLINE, PubMed, PubMed Central (PMC), ScienceDirect, and Google Scholar, using three keywords-MDD, depression, and hyperparathyroidism. We included mixed method studies, including observational studies, non-randomized controlled trials, case reports, and review articles published in the last ten years, focusing on the adult and geriatric population (>18 years) and on depressive and anxiety symptoms associated with patients with hyperparathyroidism. We included 11 articles (seven observational studies + four case reports) for qualitative synthesis after screening the literature. The reviewed studies showed an association between high serum parathyroid level, high serum calcium level, high serum alkaline phosphatase level, low serum phosphorous level, and increased depressive neurocognitive symptoms. After a patient with hyperparathyroidism is treated for hypercalcemia or undergoes parathyroidectomy and the serum parathyroid levels are lowered, a decrease in severe depressive symptoms is noted. The qualitative analysis of the reviewed literature showed an association between major depressive disorder and hyperparathyroidism. This paper can guide clinicians to assess patients with increased serum parathyroid levels for depressive neuropsychiatric symptoms and plan treatment, as treatment of their hyperparathyroidism can significantly lower their depressive symptoms. More randomized controlled trials should be conducted to find the treatment effectiveness of depression in patients with hyperparathyroidism.
PubMed: 37425517
DOI: 10.7759/cureus.40150 -
Medicine Jun 2023Correction of calcium, phosphorus, and parathyroid hormone disorders is the standard of treatment in nondialysis patients with chronic kidney disease-mineral and bone... (Meta-Analysis)
Meta-Analysis
A meta-analysis of randomized controlled trials of tonifying kidney and strengthen bone therapy on nondialysis patients with chronic kidney disease-mineral and bone disorder.
BACKGROUND
Correction of calcium, phosphorus, and parathyroid hormone disorders is the standard of treatment in nondialysis patients with chronic kidney disease-mineral and bone disorder (CKD-MBD). However, the side effects and adverse reactions are still the main problems. Moreover, the lack of protection of kidney function in the treatment dramatically affects patients' health. Although Traditional Chinese Medicine, specifically tonifying kidney and strengthen bone (TKSB) therapy, is wildly applied to patients with CKD-MBD in China, the evidence of TKSB therapy in the treatment of CKD-MBD is limited. Thus, we conducted this meta-analysis to evaluate the efficacy and safety of TKSB therapy combined with Western medicine (WM) for nondialysis patients with CKD-MBD.
METHODS
Two investigators conducted systematic research of randomized controlled trials of TKSB therapy for CKD-MBD from 7 electronic databases. Methodological quality evaluations were performed using the Cochrane collaboration tool, and data analysis was conducted by RevMan v5.3 software and STATA v15.0.
RESULTS
In total, 8 randomized controlled trials involving 310 patients met the criteria of meta-analysis. The complete results showed that compared with WM alone, TKSB treatment could improve the clinical efficacy rate (risk ratio = 4.49, 95% confidence interval [CI]: [2.64, 7.61], P .00001), calcium (weighted mean difference [WMD] = 0.11, 95% CI: [0.08, 0.14], P < .00001), serum creatinine (WMD = 45.58, 95% CI: [32.35, 58.8], P < .00001) phosphorus (WMD = 0.11, 95% CI: [0.08, 0.13], P < .00001), parathyroid hormone (WMD = 16.72, 95% CI: [12.89, 20.55], P < .00001), blood urea nitrogen levels (WMD = 0.95, 95% CI: [0.26, 1.64], P = .007) on nondialysis patients with CKD-MBD, which was beneficial to improve the patients' bone metabolic state and renal function. In addition, evidence shows that, compared with WM alone, TKSB treatment is safe and does not increase side effects.
CONCLUSION
The systematic review found that TKSB therapy combined with WM has a positive effect on improving renal function and correcting bone metabolism disorder in nondialysis patients with CKD-MBD, which shows that Traditional Chinese Medicine is effective and safe in treating CKD-MBD. However, more high-quality, large-sample, multicenter clinical trials should be conducted to assess the safety and efficacy of TKSB therapy in treating nondialysis patients with CKD-MBD.Systematic review registration: INPLASY2020120086.
Topics: Humans; Calcium; Calcium, Dietary; Chronic Kidney Disease-Mineral and Bone Disorder; Drug-Related Side Effects and Adverse Reactions; Kidney; Multicenter Studies as Topic; Parathyroid Hormone; Randomized Controlled Trials as Topic
PubMed: 37352066
DOI: 10.1097/MD.0000000000034044 -
The Journal of Clinical Endocrinology... Oct 2023Secondary hyperparathyroidism (SHPT) is a complication of chronic kidney disease (CKD) affecting mineral and bone metabolism and characterized by excessive parathyroid... (Meta-Analysis)
Meta-Analysis
CONTEXT
Secondary hyperparathyroidism (SHPT) is a complication of chronic kidney disease (CKD) affecting mineral and bone metabolism and characterized by excessive parathyroid hormone (PTH) production and parathyroid hyperplasia.
OBJECTIVE
The objective of this analysis was to compare the efficacy and adverse effects of extended-release calcifediol (ERC) and paricalcitol (PCT) by assessing their effect on the biomarkers PTH, calcium, and phosphate in patients with non-dialysis CKD (ND-CKD).
METHODS
A systematic literature research was performed in PubMed to identify randomized control trials (RCTs). Quality assessment was done with the GRADE method. The effects of ERC vs PCT were compared using random effects in a frequentist setting.
RESULTS
Nine RCTs comprising 1426 patients were included in the analyses. The analyses were performed on 2 overlapping networks, due to nonreporting of outcomes in some of the included studies. No head-to-head trials were identified. No statistically significant differences in PTH reduction were found between PCT and ERC. Treatment with PCT showed statistically significant increases in calcium compared with ERC (0.2 mg/dL increase; 95% CI, -0.37 to -0.05 mg/dL). No differences in effects on phosphate were observed.
CONCLUSION
This network meta-analysis showed that ERC is comparable in lowering PTH levels vs PCT. ERC displayed avoidance of potentially clinically relevant increases in serum calcium, offering an effective and well-tolerated treatment option for the management of SHPT in patients with ND-CKD.
Topics: Humans; Calcifediol; Calcium; Ergocalciferols; Hyperparathyroidism, Secondary; Network Meta-Analysis; Parathyroid Hormone; Phosphates; Renal Insufficiency, Chronic; Randomized Controlled Trials as Topic
PubMed: 37235771
DOI: 10.1210/clinem/dgad289 -
BMJ (Clinical Research Ed.) May 2023To review the comparative effectiveness of osteoporosis treatments, including the bone anabolic agents, abaloparatide and romosozumab, on reducing the risk of fractures... (Meta-Analysis)
Meta-Analysis
Fracture risk reduction and safety by osteoporosis treatment compared with placebo or active comparator in postmenopausal women: systematic review, network meta-analysis, and meta-regression analysis of randomised clinical trials.
OBJECTIVE
To review the comparative effectiveness of osteoporosis treatments, including the bone anabolic agents, abaloparatide and romosozumab, on reducing the risk of fractures in postmenopausal women, and to characterise the effect of antiosteoporosis drug treatments on the risk of fractures according to baseline risk factors.
DESIGN
Systematic review, network meta-analysis, and meta-regression analysis of randomised clinical trials.
DATA SOURCES
Medline, Embase, and Cochrane Library to identify randomised controlled trials published between 1 January 1996 and 24 November 2021 that examined the effect of bisphosphonates, denosumab, selective oestrogen receptor modulators, parathyroid hormone receptor agonists, and romosozumab compared with placebo or active comparator.
ELIGIBILITY CRITERIA FOR SELECTING STUDIES
Randomised controlled trials that included non-Asian postmenopausal women with no restriction on age, when interventions looked at bone quality in a broad perspective. The primary outcome was clinical fractures. Secondary outcomes were vertebral, non-vertebral, hip, and major osteoporotic fractures, all cause mortality, adverse events, and serious cardiovascular adverse events.
RESULTS
The results were based on 69 trials (>80 000 patients). For clinical fractures, synthesis of the results showed a protective effect of bisphosphonates, parathyroid hormone receptor agonists, and romosozumab compared with placebo. Compared with parathyroid hormone receptor agonists, bisphosphonates were less effective in reducing clinical fractures (odds ratio 1.49, 95% confidence interval 1.12 to 2.00). Compared with parathyroid hormone receptor agonists and romosozumab, denosumab was less effective in reducing clinical fractures (odds ratio 1.85, 1.18 to 2.92 for denosumab parathyroid hormone receptor agonists and 1.56, 1.02 to 2.39 for denosumab romosozumab). An effect of all treatments on vertebral fractures compared with placebo was found. In the active treatment comparisons, denosumab, parathyroid hormone receptor agonists, and romosozumab were more effective than oral bisphosphonates in preventing vertebral fractures. The effect of all treatments was unaffected by baseline risk indicators, except for antiresorptive treatments that showed a greater reduction of clinical fractures compared with placebo with increasing mean age (number of studies=17; β=0.98, 95% confidence interval 0.96 to 0.99). No harm outcomes were seen. The certainty in the effect estimates was moderate to low for all individual outcomes, mainly because of limitations in reporting, nominally indicating a serious risk of bias and imprecision.
CONCLUSIONS
The evidence indicated a benefit of a range of treatments for osteoporosis in postmenopausal women for clinical and vertebral fractures. Bone anabolic treatments were more effective than bisphosphonates in the prevention of clinical and vertebral fractures, irrespective of baseline risk indicators. Hence this analysis provided no clinical evidence for restricting the use of anabolic treatment to patients with a very high risk of fractures.
SYSTEMATIC REVIEW REGISTRATION
PROSPERO CRD42019128391.
Topics: Humans; Female; Bone Density Conservation Agents; Network Meta-Analysis; Postmenopause; Denosumab; Receptor, Parathyroid Hormone, Type 1; Osteoporosis; Osteoporotic Fractures; Diphosphonates; Spinal Fractures; Risk Reduction Behavior; Osteoporosis, Postmenopausal; Randomized Controlled Trials as Topic
PubMed: 37130601
DOI: 10.1136/bmj-2021-068033 -
Asian Journal of Surgery Sep 2023Permanent hypoparathyroidism is a postoperative complication of thyroid and parathyroid surgery and can be cured by cryopreserved parathyroid autotransplantation (CPAT).... (Review)
Review
Permanent hypoparathyroidism is a postoperative complication of thyroid and parathyroid surgery and can be cured by cryopreserved parathyroid autotransplantation (CPAT). However, due to the lack of unified and standardized guidelines, the limited ability of the parathyroid tissue itself to withstand cryopreservation, and some yet-to-be-defined processes or technologies, the success rate of cryopreserved parathyroid autotransplantation varies between institutions; it is low for some institutions and high for others. Due to the sparsity of data, views vary on which factors most influence the success rate of cryopreserved parathyroid autotransplantation. In this review, we analyzed the following probable influencing factors: ischemic period before cryopreservation; processes of cryopreservation and thawing, including freezing medium; freezing and thawing methods; duration of cryopreservation; examination of the graft before transplantation; graft site; mass of transplanted tissue fragments; blood calcium level; and the evaluation criteria for cryopreserved parathyroid autotransplantation success. Although the effects of these factors are debatable, we hypothesized that examining them in the above-given order to determine whether they affect the success rate of cryopreserved parathyroid autotransplantation could be beneficial to maximizing the success rate. Our findings led us to conclude that cryopreserved parathyroid autotransplantation operations should be standardized. Standardized guidelines for cryopreserved parathyroid autotransplantation that include such factors as ischemic period time, freezing and thawing methods, and recipient status should be established based on a comprehensive analysis of these factors.
Topics: Humans; Transplantation, Autologous; Parathyroid Glands; Hypoparathyroidism; Cryopreservation; Postoperative Complications
PubMed: 37105818
DOI: 10.1016/j.asjsur.2023.04.032 -
American Journal of Otolaryngology 2023Parathyroid carcinoma is rarely encountered in clinical practice. When faced with this clinical challenge, there is currently a paucity of evidence available for the... (Review)
Review
OBJECTIVE
Parathyroid carcinoma is rarely encountered in clinical practice. When faced with this clinical challenge, there is currently a paucity of evidence available for the optimal management of patients with parathyroid carcinoma. This systematic review synthesizes the available literature to evaluate the optimal management approach, thus providing guidance for future management.
METHODS
A systematic review was conducted according to PRISMA guidelines using Ovid MEDLINE, EMBASE, and PubMed databases for studies, published in English, reporting on parathyroid carcinoma. Full text of potentially eligible articles were reviewed by two authors independently and eligible studies selected. Treatment options and associated outcomes were evaluated. Descriptive statistics were used to describe pooled patient cohorts.
RESULTS
3203 articles were initially identified using the search criteria with 59 full-text articles then screened for eligibility. Seven articles, all retrospective studies, concerning 2307 patients (median 224/study). Parathyroidectomy alone was the most frequently utilised surgical approach across all studies, followed by en-bloc resection (with adjacent thyroid and/or nodal tissue). There was no difference in post-operative morbidity, mortality or survival between surgical approaches (p < 0.005). Patients who underwent either form of surgery had longer overall survival than those managed non-operatively (p < 0.005).
CONCLUSION
Surgical resection is the optimal treatment of parathyroid carcinoma. However there remains no consensus on the optimal extent of surgery, and as such future randomised prospective studies are necessary to evaluate the effects of different surgical approaches on morbidity, mortality and oncologic outcomes. Following resection, long-term surveillance with PTH is advised.
Topics: Humans; Parathyroid Neoplasms; Prospective Studies; Retrospective Studies
PubMed: 36989753
DOI: 10.1016/j.amjoto.2023.103843 -
Medicine Mar 2023Glucocorticoid-induced osteoporosis (GIOP) is the most common secondary osteoporosis. Bushen Jiangu (BSJG), a classic traditional Chinese medicine (TCM) therapy, is... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Glucocorticoid-induced osteoporosis (GIOP) is the most common secondary osteoporosis. Bushen Jiangu (BSJG), a classic traditional Chinese medicine (TCM) therapy, is widely used for treatment of GIOP. We conducted a meta-analysis to evaluate the effectiveness and safety of BSJG therapy on the treatment of GIOP.
METHODS
We searched randomized controlled trials (RCTs) of BSJG therapy for GIOP in 10 databases. Methodological quality assessment was performed by using the Cochrane collaboration tool. RevMan v5.3 and Stata v14.0 software were used for performing data analysis. This study was conducted and reported following the PRISMA checklist.
RESULTS
Overall, 14 RCTs with 988 participants met the inclusion criteria. Pooled results indicated that BSJG therapy contributed to a betterment in improving the clinical efficacy rate of GIOP (risk ratio [RR] = 1.22, 95% confidence interval [CI]: 1.14, 1.30, P < .00001). The pooled results also indicated that BSJG therapy increased lumbar spine bone mineral density (LS-BMD) (weighted mean difference [WMD] = 0.21, 95% CI: 0.08, 0.33, P = .001), total hip bone mineral density (TH-BMD) (WMD = 0.16, 95% CI: 0.09, 0.24, P < .0001), and femoral neck bone mineral density (FN-BMD) (WMD = 0.07, 95% CI: 0.03, 0.10, P = .0001). Furthermore, our results indicated that BSJG therapy improved visual analogue scale (VAS) score (WMD = -0.60, 95% CI: -0.97, -0.23, P = .002), parathyroid hormone (PTH) (standardized mean difference [SMD] = -0.93, 95% CI: -1.58, -0.27, P = .006), and N-terminal propeptide of type I precollagen (PINP) (SMD = 0.69, 95% CI: 0.44, 0.95, P < .00001). In terms of safety, there was no significant difference in the adverse events (AEs) between the 2 groups (RR = 1.45, 95% CI: 0.63, 3.31, P = .38).
CONCLUSION
Our analysis indicates that BSJG therapy has a valid and safe effect on the treatment of GIOP in the clinic. However, the results need to be confirmed in more well-designed and large-scale RCTs.
Topics: Humans; Glucocorticoids; Randomized Controlled Trials as Topic; Osteoporosis; Bone Density; Bone Density Conservation Agents
PubMed: 36930100
DOI: 10.1097/MD.0000000000033278