Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice

Osteoporosis, characterized by reduced bone density and strength, increases fracture risk, pain, and limits mobility. Established therapies of parathyroid hormone (PTH) analogs effectively promote bone formation and reduce fractures in severe osteoporosis, but their use is limited by potential adver...

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Main Authors: Minsoo Noh, Xiangguo Che, Xian Jin, Dong-Kyo Lee, Hyun-Ju Kim, Doo Ri Park, Soo Young Lee, Hunsang Lee, Thomas J Gardella, Je-Yong Choi, Sihoon Lee
Format: Article
Language:English
Published: eLife Sciences Publications Ltd 2025-03-01
Series:eLife
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Online Access:https://elifesciences.org/articles/97579
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author Minsoo Noh
Xiangguo Che
Xian Jin
Dong-Kyo Lee
Hyun-Ju Kim
Doo Ri Park
Soo Young Lee
Hunsang Lee
Thomas J Gardella
Je-Yong Choi
Sihoon Lee
author_facet Minsoo Noh
Xiangguo Che
Xian Jin
Dong-Kyo Lee
Hyun-Ju Kim
Doo Ri Park
Soo Young Lee
Hunsang Lee
Thomas J Gardella
Je-Yong Choi
Sihoon Lee
author_sort Minsoo Noh
collection DOAJ
description Osteoporosis, characterized by reduced bone density and strength, increases fracture risk, pain, and limits mobility. Established therapies of parathyroid hormone (PTH) analogs effectively promote bone formation and reduce fractures in severe osteoporosis, but their use is limited by potential adverse effects. In the pursuit of safer osteoporosis treatments, we investigated R25CPTH, a PTH variant wherein the native arginine at position 25 is substituted by cysteine. These studies were prompted by our finding of high bone mineral density in a hypoparathyroidism patient with the R25C homozygous mutation, and we explored its effects on PTH type-1 receptor (PTH1R) signaling in cells and bone metabolism in mice. Our findings indicate that R25CPTH(1–84) forms dimers both intracellularly and extracellularly, and the synthetic dimeric peptide, R25CPTH(1–34), exhibits altered activity in PTH1R-mediated cyclic AMP (cAMP) response. Upon a single injection in mice, dimeric R25CPTH(1–34) induced acute calcemic and phosphaturic responses comparable to PTH(1–34). Furthermore, repeated daily injections increased calvarial bone thickness in intact mice and improved trabecular and cortical bone parameters in ovariectomized (OVX) mice, akin to PTH(1–34). The overall results reveal a capacity of a dimeric PTH peptide ligand to activate the PTH1R in vitro and in vivo as PTH, suggesting a potential path of therapeutic PTH analog development.
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spelling doaj-art-0ae91dba372449488adaf05aa06be3392025-08-20T03:42:10ZengeLife Sciences Publications LtdeLife2050-084X2025-03-011310.7554/eLife.97579Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female miceMinsoo Noh0https://orcid.org/0000-0002-1239-3783Xiangguo Che1Xian Jin2Dong-Kyo Lee3Hyun-Ju Kim4Doo Ri Park5Soo Young Lee6Hunsang Lee7Thomas J Gardella8Je-Yong Choi9Sihoon Lee10https://orcid.org/0000-0002-9444-5849Department of Internal Medicine and Laboratory of Genomics and Translational Medicine, Gachon University College of Medicine, Incheon, Republic of Korea; Department of Life Sciences, Korea University, Seoul, Republic of KoreaDepartment of Biochemistry and Cell Biology, Cell and Matrix Research Institute, School of Medicine, Kyungpook National University, Daegu, Republic of KoreaDepartment of Biochemistry and Cell Biology, Cell and Matrix Research Institute, School of Medicine, Kyungpook National University, Daegu, Republic of KoreaDepartment of Biochemistry and Cell Biology, Cell and Matrix Research Institute, School of Medicine, Kyungpook National University, Daegu, Republic of KoreaDepartment of Biochemistry and Cell Biology, Cell and Matrix Research Institute, School of Medicine, Kyungpook National University, Daegu, Republic of KoreaDepartment of Life Sciences, Multitasking Macrophage Research Center, Ewha Womans University, Seoul, Republic of KoreaDepartment of Life Sciences, Multitasking Macrophage Research Center, Ewha Womans University, Seoul, Republic of KoreaDepartment of Life Sciences, Korea University, Seoul, Republic of KoreaEndocrine Unit, Massachusetts General Hospital and Harvard Medical School, Boston, United StatesDepartment of Biochemistry and Cell Biology, Cell and Matrix Research Institute, School of Medicine, Kyungpook National University, Daegu, Republic of KoreaDepartment of Internal Medicine and Laboratory of Genomics and Translational Medicine, Gachon University College of Medicine, Incheon, Republic of KoreaOsteoporosis, characterized by reduced bone density and strength, increases fracture risk, pain, and limits mobility. Established therapies of parathyroid hormone (PTH) analogs effectively promote bone formation and reduce fractures in severe osteoporosis, but their use is limited by potential adverse effects. In the pursuit of safer osteoporosis treatments, we investigated R25CPTH, a PTH variant wherein the native arginine at position 25 is substituted by cysteine. These studies were prompted by our finding of high bone mineral density in a hypoparathyroidism patient with the R25C homozygous mutation, and we explored its effects on PTH type-1 receptor (PTH1R) signaling in cells and bone metabolism in mice. Our findings indicate that R25CPTH(1–84) forms dimers both intracellularly and extracellularly, and the synthetic dimeric peptide, R25CPTH(1–34), exhibits altered activity in PTH1R-mediated cyclic AMP (cAMP) response. Upon a single injection in mice, dimeric R25CPTH(1–34) induced acute calcemic and phosphaturic responses comparable to PTH(1–34). Furthermore, repeated daily injections increased calvarial bone thickness in intact mice and improved trabecular and cortical bone parameters in ovariectomized (OVX) mice, akin to PTH(1–34). The overall results reveal a capacity of a dimeric PTH peptide ligand to activate the PTH1R in vitro and in vivo as PTH, suggesting a potential path of therapeutic PTH analog development.https://elifesciences.org/articles/97579parathyroid hormonePTH type-1 receptorbone formationosteoporosis
spellingShingle Minsoo Noh
Xiangguo Che
Xian Jin
Dong-Kyo Lee
Hyun-Ju Kim
Doo Ri Park
Soo Young Lee
Hunsang Lee
Thomas J Gardella
Je-Yong Choi
Sihoon Lee
Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice
eLife
parathyroid hormone
PTH type-1 receptor
bone formation
osteoporosis
title Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice
title_full Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice
title_fullStr Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice
title_full_unstemmed Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice
title_short Dimeric R25CPTH(1–34) activates the parathyroid hormone-1 receptor in vitro and stimulates bone formation in osteoporotic female mice
title_sort dimeric r25cpth 1 34 activates the parathyroid hormone 1 receptor in vitro and stimulates bone formation in osteoporotic female mice
topic parathyroid hormone
PTH type-1 receptor
bone formation
osteoporosis
url https://elifesciences.org/articles/97579
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