Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma

Background: Osteosarcoma (OS) is a primary malignant bone tumor predominantly affecting adolescents. Chemotherapeutic agents, such as cisplatin, are commonly used in OS treatment; however, drug resistance markedly undermines treatment efficacy and contributes to reduced patient survival. The mechani...

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Main Authors: Tianlong Huang, Yifan Chen, Qiangqiang Zhao, Xin Wu, Hongxing Li, Xin Luo, Yang Su, Shengqun Zhang, Pan Liu, Ning Tang
Format: Article
Language:English
Published: American Association for the Advancement of Science (AAAS) 2025-01-01
Series:Research
Online Access:https://spj.science.org/doi/10.34133/research.0708
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author Tianlong Huang
Yifan Chen
Qiangqiang Zhao
Xin Wu
Hongxing Li
Xin Luo
Yang Su
Shengqun Zhang
Pan Liu
Ning Tang
author_facet Tianlong Huang
Yifan Chen
Qiangqiang Zhao
Xin Wu
Hongxing Li
Xin Luo
Yang Su
Shengqun Zhang
Pan Liu
Ning Tang
author_sort Tianlong Huang
collection DOAJ
description Background: Osteosarcoma (OS) is a primary malignant bone tumor predominantly affecting adolescents. Chemotherapeutic agents, such as cisplatin, are commonly used in OS treatment; however, drug resistance markedly undermines treatment efficacy and contributes to reduced patient survival. The mechanisms underlying cisplatin resistance remain poorly understood. Recently, palmitoyl-protein thioesterase 1 (PPT1), a depalmitoylation enzyme, has attracted attention for its role in tumorigenesis and drug resistance. Investigating the mechanisms of PPT1 may offer new strategies to overcome resistance. Methods: This study analyzed multiple Gene Expression Omnibus datasets and utilized the OncoPredict tool to demonstrate the elevated expression of PPT1 in OS and its critical role in cisplatin resistance. By combining single-cell analysis with in vitro and in vivo experiments, we explored how PPT1 influences OS development through depalmitoylation and assessed the antitumor effects of the PPT1 inhibitor Ezurpimtrostat (GNS561), as well as its synergistic effects when combined with cisplatin. Results: We demonstrated that Sprouty 4 (SPRY4) undergoes a dynamic palmitoylation cycle regulated by zinc finger DHHC-type palmitoyl transferase 7 (ZDHHC7) and PPT1, which modulates mitogen-activated protein kinase (MAPK) signaling and subsequently affects tumor cell proliferation, migration, apoptosis, and drug resistance. Further validation confirmed the effectiveness of the PPT1 inhibitor GNS561 in overcoming cisplatin resistance. Notably, GNS561 exhibited a significant synergistic effect when used in combination with cisplatin, greatly enhancing the sensitivity of cisplatin-resistant cells. Conclusion: This study highlights the pivotal role of PPT1 in OS resistance mechanisms. PPT1 and ZDHHC7 regulate SPRY4 through a dynamic palmitoylation–depalmitoylation cycle that modulates MAPK signaling activation and contributes to OS cell proliferation, migration, and drug resistance. As a PPT1 inhibitor, GNS561 not only inhibits OS cell proliferation but also demonstrates synergistic effects with cisplatin, significantly enhancing cisplatin sensitivity in resistant cells and promoting apoptosis. Our findings offer a novel approach for targeting PPT1 in therapeutic strategies. GNS561 holds promise as an adjunctive therapy when combined with cisplatin, potentially overcoming resistance and improving efficacy, thereby enhancing the prognosis for OS patients. Future studies should further investigate the clinical potential of GNS561 and optimize OS treatment strategies.
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spelling doaj-art-69f24d920e6442c8ada8a4c14a4715202025-08-20T02:26:00ZengAmerican Association for the Advancement of Science (AAAS)Research2639-52742025-01-01810.34133/research.0708Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant OsteosarcomaTianlong Huang0Yifan Chen1Qiangqiang Zhao2Xin Wu3Hongxing Li4Xin Luo5Yang Su6Shengqun Zhang7Pan Liu8Ning Tang9Orthopaedic Department, The Second Xiangya Hospital of Central South University, Changsha, Hunan, China.Orthopaedic Department, The Second Xiangya Hospital of Central South University, Changsha, Hunan, China.Department of Hematology, Liuzhou People’s Hospital affiliated to Guangxi Medical University, Liuzhou, Guangxi, China.Department of Spine Surgery, Third Xiangya Hospital, Central South University, Changsha, Hunan, China.Department of Orthopaedic, The Central Hospital of Shaoyang, Shaoyang, China.Orthopaedic Department, The Third Xiangya Hospital of Central South University, Changsha, Hunan, China.Orthopaedic Department, The Second Xiangya Hospital of Central South University, Changsha, Hunan, China.Orthopaedic Department, The Second Xiangya Hospital of Central South University, Changsha, Hunan, China.Orthopaedic Department, The Second Xiangya Hospital of Central South University, Changsha, Hunan, China.Orthopaedic Department, The Second Xiangya Hospital of Central South University, Changsha, Hunan, China.Background: Osteosarcoma (OS) is a primary malignant bone tumor predominantly affecting adolescents. Chemotherapeutic agents, such as cisplatin, are commonly used in OS treatment; however, drug resistance markedly undermines treatment efficacy and contributes to reduced patient survival. The mechanisms underlying cisplatin resistance remain poorly understood. Recently, palmitoyl-protein thioesterase 1 (PPT1), a depalmitoylation enzyme, has attracted attention for its role in tumorigenesis and drug resistance. Investigating the mechanisms of PPT1 may offer new strategies to overcome resistance. Methods: This study analyzed multiple Gene Expression Omnibus datasets and utilized the OncoPredict tool to demonstrate the elevated expression of PPT1 in OS and its critical role in cisplatin resistance. By combining single-cell analysis with in vitro and in vivo experiments, we explored how PPT1 influences OS development through depalmitoylation and assessed the antitumor effects of the PPT1 inhibitor Ezurpimtrostat (GNS561), as well as its synergistic effects when combined with cisplatin. Results: We demonstrated that Sprouty 4 (SPRY4) undergoes a dynamic palmitoylation cycle regulated by zinc finger DHHC-type palmitoyl transferase 7 (ZDHHC7) and PPT1, which modulates mitogen-activated protein kinase (MAPK) signaling and subsequently affects tumor cell proliferation, migration, apoptosis, and drug resistance. Further validation confirmed the effectiveness of the PPT1 inhibitor GNS561 in overcoming cisplatin resistance. Notably, GNS561 exhibited a significant synergistic effect when used in combination with cisplatin, greatly enhancing the sensitivity of cisplatin-resistant cells. Conclusion: This study highlights the pivotal role of PPT1 in OS resistance mechanisms. PPT1 and ZDHHC7 regulate SPRY4 through a dynamic palmitoylation–depalmitoylation cycle that modulates MAPK signaling activation and contributes to OS cell proliferation, migration, and drug resistance. As a PPT1 inhibitor, GNS561 not only inhibits OS cell proliferation but also demonstrates synergistic effects with cisplatin, significantly enhancing cisplatin sensitivity in resistant cells and promoting apoptosis. Our findings offer a novel approach for targeting PPT1 in therapeutic strategies. GNS561 holds promise as an adjunctive therapy when combined with cisplatin, potentially overcoming resistance and improving efficacy, thereby enhancing the prognosis for OS patients. Future studies should further investigate the clinical potential of GNS561 and optimize OS treatment strategies.https://spj.science.org/doi/10.34133/research.0708
spellingShingle Tianlong Huang
Yifan Chen
Qiangqiang Zhao
Xin Wu
Hongxing Li
Xin Luo
Yang Su
Shengqun Zhang
Pan Liu
Ning Tang
Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma
Research
title Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma
title_full Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma
title_fullStr Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma
title_full_unstemmed Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma
title_short Dual Regulation of Sprouty 4 Palmitoylation by ZDHHC7 and Palmitoyl-Protein Thioesterase 1: A Potential Therapeutic Strategy for Cisplatin-Resistant Osteosarcoma
title_sort dual regulation of sprouty 4 palmitoylation by zdhhc7 and palmitoyl protein thioesterase 1 a potential therapeutic strategy for cisplatin resistant osteosarcoma
url https://spj.science.org/doi/10.34133/research.0708
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