Therapy with PTH 1–34 or calcitriol and calcium in diverse etiologies of hypoparathyroidism over 27 years at a single tertiary care center

Winer, Karen K.; Ye, Shangyuan; Ferré, Elise M. N.; Schmitt, Monica M.; Zhang, Bo; Cutler, Gordon B.; Lionakis, Michail S

doi:10.1016/j.bone.2021.115977

Cited by 12 publications

(11 citation statements)

References 45 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The goals of replacement treatment include the prevention of hypocalcemic crises but also avoidance of the long-term development of nephrocalcinosis, nephrolithiasis, and renal failure resulting from excessive replacement. To that end, maintaining serum calcium levels within the lower range of normal or just below the normal range is favored ( 161 ). The recent introduction of recombinant PTH in clinical practice for the treatment of adult patients with hypoparathyroidism ( 162 ) may help a subset of APECED patients with hypoparathyroidism to achieve optimal calcium homeostasis, particularly in the setting of pre-existing renal impairment and/or intestinal malabsorption.…”

Section: Clinical Management Of Apeced Patientsmentioning

confidence: 99%

Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy

2021

Self Cite

View full text Add to dashboard Cite

Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED), also known as autoimmune polyglandular syndrome type-1 (APS-1), is a rare monogenic autoimmune disease caused by loss-of-function mutations in the autoimmune regulator (AIRE) gene. AIRE deficiency impairs immune tolerance in the thymus and results in the peripheral escape of self-reactive T lymphocytes and the generation of several cytokine- and tissue antigen-targeted autoantibodies. APECED features a classic triad of characteristic clinical manifestations consisting of chronic mucocutaneous candidiasis (CMC), hypoparathyroidism, and primary adrenal insufficiency (Addison's disease). In addition, APECED patients develop several non-endocrine autoimmune manifestations with variable frequencies, whose recognition by pediatricians should facilitate an earlier diagnosis and allow for the prompt implementation of targeted screening, preventive, and therapeutic strategies. This review summarizes our current understanding of the genetic, immunological, clinical, diagnostic, and treatment features of APECED.

show abstract

Section: Clinical Management Of Apeced Patientsmentioning

confidence: 99%

Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy

2021

Self Cite

View full text Add to dashboard Cite

show abstract

“…Furthermore, the data could not be analyzed in relation to different etiologies of hypoparathyroidism, which could be a confounding factor. A recent study by Winer and colleagues (63) found that patients with genetic mutations required higher doses of PTH 1À34 than did patients with postsurgical or idiopathic hypoparathyroidism. Finally, most of the studies were sponsored, with only 10 nonprofit studies included.…”

Section: Discussionmentioning

confidence: 97%

Safety and efficacy of PTH 1-34 and 1-84 therapy in chronic hypoparathyroidism: a meta-analysis of prospective trials

Hasenmajer¹,

Puliani²,

Sada³

et al. 2021

EJEA

View full text Add to dashboard Cite

Hypoparathyroidism is the only endocrine deficiency for which hormone replacement therapy is not the standard of care. Although conventional treatments may control hypocalcaemia, other complications such as hyperphosphatemia, kidney stones, peripheral calcifications, and bone disease remain unmet needs. This meta-analysis (PROSPERO registration number CRD42019126881) aims to evaluate and compare the efficacy and safety of PTH 1À34 and PTH 1À84 in restoring calcium metabolism in chronic hypoparathyroidism. EMBASE, PubMed, and CENTRAL databases were searched for randomized clinical trials or prospective studies published between January 1996 and March 2021. English-language trials reporting data on replacement with PTH 1À34 or PTH 1À84 in chronic hypoparathyroidism were selected. Three authors extracted outcomes, one author performed quality control, all assessed the risk of biases. Overall, data from 25 studies on 588 patients were analyzed. PTH therapy had a neutral effect on calcium levels, while lowering serum phosphate (À0.21 mmol/L; 95% confidence interval [CI], À0.31 to À0.11 mmol/L; p < 0.001) and urinary calcium excretion (À1.21 mmol/24 h; 95% CI, À2.03 to À0.41 mmol/24 h; p = 0.003). Calcium phosphate product decreased under PTH 1À84 therapy only. Both treatments enabled a significant reduction in calcium and calcitriol supplementation. PTH therapy increased bone turnover markers and lumbar spine mineral density. Quality of life improved and there was no difference in the safety profile between PTH and conventionally treated patients. Results for most outcomes were similar for the two treatments. Limitations of the study included considerable population overlap between the reports, incomplete data, and heterogeneity in the protocol design. In conclusion, the meta-analysis of data from the largest collection to date of hypoparathyroid patients shows that PTH therapy is safe, welltolerated, and effective in normalizing serum phosphate and urinary calcium excretion, as well as enabling a reduction in calcium and vitamin D use and improving quality of life.

show abstract

“…A recent study demonstrated an increased prevalence of renal complications in nonsurgical versus surgical HypoPT, suggesting that more vigilant monitoring of nonsurgical patients for complications is necessary. ( 21 ) In addition, the SCPS identified considerable variation in practice patterns, highlighting areas in need of future research. Our survey recognizes the lack of agreement with respect to the need for initial assessment and follow‐up monitoring of complications associated with chronic HypoPT.…”

Section: Discussionmentioning

confidence: 99%

Initial Assessment and Monitoring of Patients with Chronic Hypoparathyroidism: A Systematic Current Practice Survey

Uum

Shrayyef

M'Hiri³

et al. 2020

Journal of Bone and Mineral Research

Self Cite

View full text Add to dashboard Cite

Chronic hypoparathyroidism (HypoPT) is associated with significant morbidity and impaired quality of life (QoL). The goals of management for chronic HypoPT include improvement in QoL and the prevention of both hypo‐ and hypercalcemia symptoms and long‐term complications. Several groups have provided consensus statements and guidelines on the management of HypoPT; however, due to limited evidence, these recommendations have largely been based on literature reviews, expert opinion, and consensus statements. The objective of this study was to use a systematic approach to describe current practice for the initial assessment and follow‐up of patients with chronic HypoPT. We developed a survey asking experts in the field to select the responses that best reflect their current practice. The survey found no differences in responses between nonsurgical and postsurgical patient assessment. For new patients, respondents usually performed an assessment of serum lab profile (calcium [either albumin‐adjusted or ionized], magnesium, creatinine, phosphate, 25‐hydroxyvitamin D), 24‐hour urine (creatinine, calcium), and a renal ultrasound to evaluate for the presence of nephrocalcinosis or nephrolithiasis. For follow‐up patients, most respondents perform blood tests and urine tests every 6 months or less frequently. The reported clinical practice patterns for monitoring for complications of chronic HypoPT vary considerably among respondents. Based on the responses in this systematic expert practice survey, we provide practice suggestions for initial assessment and follow‐up of patients with chronic HypoPT. In addition, we highlight areas with significant variation in practice and identify important areas for future research. © 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).

show abstract

Therapy with PTH 1–34 or calcitriol and calcium in diverse etiologies of hypoparathyroidism over 27 years at a single tertiary care center

Cited by 12 publications

References 45 publications

Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy

Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy

Safety and efficacy of PTH 1-34 and 1-84 therapy in chronic hypoparathyroidism: a meta-analysis of prospective trials

Initial Assessment and Monitoring of Patients with Chronic Hypoparathyroidism: A Systematic Current Practice Survey

Contact Info

Product

Resources

About