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Frontiers in Endocrinology 2022Primary hyperparathyroidism is a common disorder of the parathyroid glands. Parathyroid adenoma (PA) in pregnancy is a relatively rare disease, whose diagnosis and...
OBJECTIVE
Primary hyperparathyroidism is a common disorder of the parathyroid glands. Parathyroid adenoma (PA) in pregnancy is a relatively rare disease, whose diagnosis and treatment is a challenging task. The aim of the present study is to present a new case of parathyroid adenoma during pregnancy and to give a detailed account of all reported cases of parathyroid adenoma during pregnancy in the literature.
STUDY DESIGN
A bibliographic research was performed, and characteristics of parathyroid adenomas in pregnancy such as age, gestational week at diagnosis, ionized calcium levels, genetic testing result, symptomatology, radiological method of localization, treatment method, gestational week at operation, and maternal/fetal complications were recorded.
RESULTS
A 34-year-old woman at her 25 weeks' gestation was diagnosed with parathyroid adenoma and was referred to our Surgical Department due to contraindication for conservative treatment. A parathyroidectomy was performed, and the maternal and fetal postoperative period was uneventful. Two hundred eleven cases of parathyroid adenoma in pregnancy were recorded in the literature, and statistical analysis was performed. The median gestational week at diagnosis was 21 ± 9.61 weeks. The mean level of ionized calcium was 2.69 mmol/l [SD = 0.75 (2.55-2.84 95% CI)]. Most cases were familiar (72.4%), while surgery was the preferred treatment option (67.3%). The majority of cases were asymptomatic (21.7%), and the main radiological method applied for localization was ultrasound (63.4%).
CONCLUSION
Parathyroid adenoma in pregnancy is a rare condition. The early diagnosis is of great importance as surgical treatment at the second trimester of pregnancy outweighs the maternal and fetal risks.
Topics: Humans; Female; Pregnancy; Adult; Parathyroid Neoplasms; Calcium; Adenoma; Pregnancy Complications, Neoplastic; Parathyroidectomy
PubMed: 36325457
DOI: 10.3389/fendo.2022.975954 -
Nutrients Oct 2022Vitamin D deficiency and insufficiency as well as low serum calcium levels can trigger negative health outcomes in women of childbearing age. Therefore, we aimed to... (Meta-Analysis)
Meta-Analysis Review
Vitamin D deficiency and insufficiency as well as low serum calcium levels can trigger negative health outcomes in women of childbearing age. Therefore, we aimed to estimate the prevalence of serum vitamin D and calcium deficiencies and insufficiencies and associated risk factors in Brazilian women of childbearing age and to assess whether there are differences in prevalence according to regions of the country and the presence or absence of pregnancy. The systematic literature review was performed using the following databases: PubMed, LILACS, Embase, Scopus, and Web of Science. Cross-sectional, cohort, and intervention studies were included. Among pregnant women, the prevalence of vitamin D deficiency ranged from 0% to 27% and of vitamin D insufficiency from 33.9% to 70.4%. Among non-pregnant women, the prevalence of vitamin D deficiency ranged from 0% to 41.7% and of vitamin D insufficiency from 38.5% to 69.3%. We found a high prevalence of vitamin D deficiency and insufficiency in women of childbearing age, with insufficiency affecting more than half of these women. The highest prevalence of vitamin D deficiency and insufficiency was observed in the South region. It was not possible to assess the prevalence and factors associated with calcium deficiency.
Topics: Pregnancy; Female; Humans; Vitamin D; Calcium; Prevalence; Cross-Sectional Studies; Vitamin D Deficiency; Vitamins; Malnutrition; Calcium Metabolism Disorders; Parathyroid Diseases; Risk Factors
PubMed: 36297034
DOI: 10.3390/nu14204351 -
Journal of Personalized Medicine Sep 2022Neuroendocrine neoplasms (NEN) originate from neuroendocrine cells ubiquitously spread throughout the body. Hypercalcemia associated with cancer is the most common... (Review)
Review
BACKGROUND
Neuroendocrine neoplasms (NEN) originate from neuroendocrine cells ubiquitously spread throughout the body. Hypercalcemia associated with cancer is the most common life-threatening metabolic disorder in patients with advanced stage cancer. Paraneoplastic hypercalcemia is more commonly associated with hematological malignancies, renal and breast carcinomas, and squamous cell carcinomas, but it has also been described in patients with well-differentiated NEN, where it often remains undiagnosed. Among its causes, systemic secretion of parathyroid hormone-related protein (PTHrP) and ectopic production of 1,25-dihydroxyvitamin D and parathyroid hormone (PTH) may be considered paraneoplastic causes of hypercalcemia. In order to clarify the diagnostic work up of paraneoplastic hypercalcemia in patients with NEN, we perform a systematic review, which is lacking in the literature.
METHODS
We performed a data search using MEDLINE and SCOPUS including papers from 1961 to 2021. We selected articles on paraneoplastic hypercalcemia associated with well-differentiated NEN.
RESULTS
The search led to the selection of 78 publications for a total of 114 patients. Pooled data showed that the most frequent primary tumor site associated with paraneoplastic hypercalcemia was pancreatic NEN, followed by Pheochromocytoma. In most cases, paraneoplastic hypercalcemia was caused by PTHrP production and secretion. In more than two thirds of cases, paraneoplastic hypercalcemia was present at the time of NEN diagnosis and, in metachronous cases, was related to local recurrence, distant metastasis development, or tumor progression. In most patients, a combination of therapeutic approaches was employed, and reduction of the tumor burden was essential to control the paraneoplastic syndrome.
DISCUSSION
The onset of hypercalcemia associated with cancer in patients with well-differentiated NEN represents a major clinical challenge. The complex clinical and therapeutical management of paraneoplastic hypercalcemia implies the need for a multidisciplinary approach, aimed at controlling the clinical syndrome and tumor growth.
PubMed: 36294693
DOI: 10.3390/jpm12101553 -
Journal of Bone and Mineral Research :... Nov 2022The last international guidelines on the evaluation and management of primary hyperparathyroidism (PHPT) were published in 2014. Research since that time has led to new... (Review)
Review
The last international guidelines on the evaluation and management of primary hyperparathyroidism (PHPT) were published in 2014. Research since that time has led to new insights into epidemiology, pathophysiology, diagnosis, measurements, genetics, outcomes, presentations, new imaging modalities, target and other organ systems, pregnancy, evaluation, and management. Advances in all these areas are demonstrated by the reference list in which the majority of listings were published after the last set of guidelines. It was thus, timely to convene an international group of over 50 experts to review these advances in our knowledge. Four Task Forces considered: 1. Epidemiology, Pathophysiology, and Genetics; 2. Classical and Nonclassical Features; 3. Surgical Aspects; and 4. Management. For Task Force 4 on the Management of PHPT, Grading of Recommendations, Assessment, Development, and Evaluations (GRADE) methodology addressed surgical management of asymptomatic PHPT and non-surgical medical management of PHPT. The findings of this systematic review that applied GRADE methods to randomized trials are published as part of this series. Task Force 4 also reviewed a much larger body of new knowledge from observations studies that did not specifically fit the criteria of GRADE methodology. The full reports of these 4 Task Forces immediately follow this summary statement. Distilling the essence of all deliberations of all Task Force reports and Methodological reviews, we offer, in this summary statement, evidence-based recommendations and guidelines for the evaluation and management of PHPT. Different from the conclusions of the last workshop, these deliberations have led to revisions of renal guidelines and more evidence for the other recommendations. The accompanying papers present an in-depth discussion of topics summarized in this report. © 2022 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
Topics: Humans; Hyperparathyroidism, Primary
PubMed: 36245251
DOI: 10.1002/jbmr.4677 -
Journal of Bone and Mineral Research :... Dec 2022The approach utilized a systematic review of the medical literature executed with specifically designed criteria that focused on the etiologies and pathogenesis of...
The approach utilized a systematic review of the medical literature executed with specifically designed criteria that focused on the etiologies and pathogenesis of hypoparathyroidism. Enhanced attention by endocrine surgeons to new knowledge about parathyroid gland viability are reviewed along with the role of intraoperative parathyroid hormone (ioPTH) monitoring during and after neck surgery. Nonsurgical etiologies account for a significant proportion of cases of hypoparathyroidism (~25%), and among them, genetic etiologies are key. Given the pervasive nature of PTH deficiency across multiple organ systems, a detailed review of the skeletal, renal, neuromuscular, and ocular complications is provided. The burden of illness on affected patients and their caregivers contributes to reduced quality of life and social costs for this chronic endocrinopathy. © 2022 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
Topics: Humans; Hypoparathyroidism; Parathyroid Hormone; Quality of Life; Parathyroid Glands
PubMed: 36153665
DOI: 10.1002/jbmr.4714 -
Chinese Medical Journal Jul 2022The increasing burden of non-alcoholic fatty liver disease (NAFLD) worldwide imposes an emerging public health issue. We perform the current study to estimate the global... (Meta-Analysis)
Meta-Analysis
BACKGROUND
The increasing burden of non-alcoholic fatty liver disease (NAFLD) worldwide imposes an emerging public health issue. We perform the current study to estimate the global prevalence, incidence, disease progression, and clinical outcomes of NAFLD.
METHODS
A systematic search was conducted in Medline, Embase, Web of Science, Google Scholar, and Cochrane CENTRAL that screened articles in English language published from January 2000 to December 2021. NAFLD prevalence, incidence, rate of disease progression, and outcomes were calculated with the DerSimonian-Laird random effects model with arcsine transformation.
RESULTS
Our search identified 59,156 records, of which 578 studies fulfilled our inclusion criteria. The overall prevalence of NAFLD was 29.38% (95% confidence interval [CI] 28.09-30.69) regardless of the diagnostic techniques. Looking at the group in which the diagnosis was made by ultrasound exclusively, the pooled prevalence was 30.49% (95% CI 29.55-31.43). NAFLD has become more prevalent during the year 2011-2021 (31.63%, 95% CI 30.23-33.04) compared with year 2000-2010 (27.94%, 95% CI 26.23-29.69). The pooled estimation of non-alcoholic steatohepatitis prevalence was 8.26% (95% CI 1.13-21.01), 46.49% (95% CI 35.93-57.20), and 46.72% (95% CI 37.57-55.98) in general population, NAFLD patients, and severe/morbidly obese patients, respectively. Based on a total of 110,142 newly developed NAFLD patients, the pooled incident rate was estimated as 46.24 cases per 1000 person-years (95% CI 43.21-49.30). In patients with NAFLD, the incident rate of hepatocellular carcinoma was 1.46 (95% CI 0.90-2.03) cases per 1000 person-years. The overall pooled estimate of NAFLD related mortality was 23.91 (95% CI 13.55-37.18) death per 1000 person-years.
CONCLUSIONS
The prevalence of NAFLD is increasing globally. It is contributing to poor clinical outcomes including hepatocellular carcinoma and death. Rising awareness and urgent actions are warranted to control the NAFLD pandemic across the globe.
REGISTRATION
PROSPERO, No. CRD42020171104.
Topics: Carcinoma, Hepatocellular; Disease Progression; Humans; Incidence; Liver Neoplasms; Non-alcoholic Fatty Liver Disease; Obesity, Morbid; Prevalence
PubMed: 36070463
DOI: 10.1097/CM9.0000000000002277 -
Journal of Bone and Mineral Research :... Nov 2022Both medical and surgical therapy represent potential management options for patients with asymptomatic primary hyperparathyroidism (PHPT). Because uncertainty remains... (Meta-Analysis)
Meta-Analysis
The Efficacy and Safety of Medical and Surgical Therapy in Patients With Primary Hyperparathyroidism: A Systematic Review and Meta-Analysis of Randomized Controlled Trials.
Both medical and surgical therapy represent potential management options for patients with asymptomatic primary hyperparathyroidism (PHPT). Because uncertainty remains regarding both medical and surgical therapy, this systematic review addresses the efficacy and safety of medical therapy in asymptomatic patients or symptomatic patients who decline surgery and surgery in asymptomatic patients. We searched Medline, Embase, Cochrane Central Register of Controlled Trials, and PubMed from inception to December 2020, and included randomized controlled trials in patients with PHPT that compared nonsurgical management with medical therapy versus without medical therapy and surgery versus no surgery in patients with asymptomatic PHPT. For surgical complications we included observational studies. Paired reviewers addressed eligibility, assessed risk of bias, and abstracted data for patient-important outcomes. We conducted random-effects meta-analyses to pool relative risks and mean differences with 95% confidence intervals and used Grading of Recommendations, Assessment, Development, and Evaluation (GRADE) to assess quality of evidence for each outcome. For medical therapy, 11 trials reported in 12 publications including 438 patients proved eligible: three addressed alendronate, one denosumab, three cinacalcet, two vitamin D, and two estrogen therapy. Alendronate, denosumab, vitamin D, and estrogen therapy all increased bone density. Cinacalcet probably reduced serum calcium and parathyroid hormone (PTH) levels. Cinacalcet and vitamin D may have a small or no increase in overall adverse events. Very-low-quality evidence raised the possibility of an increase in serious adverse events with alendronate and denosumab. The trials also provided low-quality evidence for increased bleeding and mastalgia with estrogen therapy. For surgery, six trials presented in 12 reports including 441 patients proved eligible. Surgery achieved biochemical cure in 96.1% (high quality). We found no convincing evidence supporting an impact of surgery on fracture, quality of life, occurrence of kidney stones, and renal function, but the evidence proved low or very low quality. Surgery was associated with an increase in bone mineral density. For patients with symptomatic and asymptomatic PHPT, who are not candidates for parathyroid surgery, cinacalcet probably reduced serum calcium and PTH levels; anti-resorptives increased bone density. For patients with asymptomatic PHPT, surgery usually achieves biochemical cure. These results can help to inform patients and clinicians regarding use of medical therapy and surgery in PHPT. © 2022 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
Topics: Humans; Cinacalcet; Hyperparathyroidism, Primary; Alendronate; Calcium; Quality of Life; Denosumab; Randomized Controlled Trials as Topic; Parathyroid Hormone; Vitamin D; Estrogens
PubMed: 36053960
DOI: 10.1002/jbmr.4685 -
BJS Open Sep 2022Postsurgical hypoparathyroidism (PH) is the most frequent complication after thyroid surgery. The aim of this systematic review and meta-analysis is to summarize a... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Postsurgical hypoparathyroidism (PH) is the most frequent complication after thyroid surgery. The aim of this systematic review and meta-analysis is to summarize a unifying definition of PH and to elucidate the best possible approach for early detection of PH.
METHODS
A systematic review of the literature according to the PICO framework using Embase, PUBMED and the Cochrane library was carried out on 1 December 2021 followed by analysis for risk of bias, data extraction and meta-analysis. All studies addressing the definition of postoperative hypoparathyroidism and/or diagnostic approaches for early detection and diagnosis were included. Case reports, commentaries, non-English articles, book chapters and pilot studies and reviews were excluded.
RESULTS
From 13 704 articles, 188 articles were eligible for inclusion and further analysis. These articles provided heterogeneous definitions of PH. Meta-analysis revealed that postoperative measurements of parathormone (PTH) levels have a higher sensitivity and specificity than intraoperative PTH measurements to predict PH after thyroid surgery. None of the timeframes analysed after surgery within the first postoperative day (POD1) was superior to predict the onset of PH. PTH levels of less than 15 pg/ml and less than 10 pg/ml are both reliable threshold levels to predict the postoperative onset of PH. A relative reduction of mean(s.d.) PTH levels from pre- to postoperative values of 73 (standard deviation 11) per cent may also be predictive for the development of PH. The estimation of calcium levels on POD1 are recommended.
CONCLUSION
PH is best defined as an undetectable or inappropriately low postoperative PTH level in the context of hypocalcaemia with or without hypocalcaemic symptoms. PTH levels should be measured after surgery within 24 h. Both threshold levels below 10 and 15 pg/ml or relative loss of PTH before/after thyroid surgery are reliable to predict the onset of PH.
Topics: Humans; Hypocalcemia; Hypoparathyroidism; Parathyroid Glands; Parathyroid Hormone; Postoperative Complications; Thyroid Gland; Thyroidectomy
PubMed: 36050906
DOI: 10.1093/bjsopen/zrac102 -
World Journal of Surgery Nov 2022
Meta-Analysis
Topics: Humans; Hyperparathyroidism, Primary; Multiple Endocrine Neoplasia Type 1; Multiple Endocrine Neoplasia Type 2a; Neoplasm Recurrence, Local; Parathyroid Glands; Parathyroidectomy
PubMed: 36042033
DOI: 10.1007/s00268-022-06707-6 -
Journal of Bone and Mineral Research :... Oct 2022Autosomal dominant hypocalcemia type 1 (ADH1) is a rare form of hypoparathyroidism due to activating variants of the calcium-sensing receptor gene (CASR). Inherited or...
Autosomal dominant hypocalcemia type 1 (ADH1) is a rare form of hypoparathyroidism due to activating variants of the calcium-sensing receptor gene (CASR). Inherited or de novo activating variants of the CASR alter the set point for extracellular calcium, resulting in inadequate parathyroid hormone (PTH) secretion and inappropriate renal calcium excretion leading to hypocalcemia and hypercalciuria. Conventional therapy includes calcium and activated vitamin D, which can worsen hypercalciuria, resulting in renal complications. A systematic literature review, using published reports from 1994 to 2021, was conducted to catalog CASR variants, to define the ADH1 clinical spectrum, and to determine the effect of treatment on patients with ADH1. There were 113 unique CASR variants reported, with a general lack of genotype/phenotype correlation. Clinical data were available in 191 patients; 27% lacked symptoms, 32% had mild/moderate symptoms, and 41% had severe symptoms. Seizures, the most frequent clinical presentation, occurred in 39% of patients. In patients with blood and urine chemistries available at the time of diagnosis (n = 91), hypocalcemia (99%), hyperphosphatemia (59%), low PTH levels (57%), and hypercalciuria (34%) were observed. Blood calcium levels were significantly lower in patients with severe symptoms compared with asymptomatic patients (6.8 ± 0.7 versus 7.6 ± 0.7 mg/dL [mean ± SD]; p < 0.0001), and the age of presentation was significantly lower in severely symptomatic patients (9.1 ± 15.0 versus 19.3 ± 19.4 years; p < 0.01). Assessments for complications including nephrocalcinosis, nephrolithiasis, renal impairment, and brain calcifications in 57 patients on conventional therapy showed that 75% had at least one complication. Hypercalciuria was associated with nephrocalcinosis, nephrolithiasis, renal impairment, or brain calcifications (odds ratio [OR] = 9.3; 95% confidence interval [CI] 2.4-37.2; p < 0.01). In 27 patients with urine calcium measures before and after starting conventional therapy, the incidence of hypercalciuria increased by 91% (p < 0.05) after therapy initiation. ADH1 is a condition often associated with severe symptomatology at presentation with an increase in the risk of renal complications after initiation of conventional therapy. © 2022 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).
Topics: Humans; Hypercalciuria; Hypocalcemia; Receptors, Calcium-Sensing; Calcium; Nephrocalcinosis; Hypoparathyroidism; Parathyroid Hormone; Nephrolithiasis; Vitamin D
PubMed: 35879818
DOI: 10.1002/jbmr.4659