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Fibrous dysplasia (FD) of the bone is characterized by fibrotic, expansile bone lesions caused by activating mutations in GNAS. There are no effective therapies for FD. We previously showed that ColI(2.3)+\/Rs1+ mice, in which Gs-GPCR signaling was hyper-activated in osteoblastic cell lineages using an engineered receptor strategy, developed a fibrotic bone phenotype with trabecularization that could be reversed by normalizing Gs-GPCR signaling, suggesting that targeting the Gs-GPCR or components of the downstream signaling pathway could serve as a promising therapeutic strategy for FD. The Wnt signaling pathway has been implicated in the pathogenesis of FD-like bone, but the specific Wnts and which cells produce them remain largely unknown. Single-cell RNA sequencing on long-bone stromal cells of 9-wk-old male ColI(2.3)+\/Rs1+ mice and littermate controls showed that fibroblastic stromal cells in ColI(2.3)+\/Rs1+ mice were expanded. Multiple Wnt ligands were up- or downregulated in different cellular populations, including in non-osteoblastic cells. Treatment with the porcupine inhibitor LGK974, which blocks Wnt signaling broadly, induced partial resorption of the trabecular bone in the femurs of ColI(2.3)+\/Rs1+ mice, but no significant changes in the craniofacial skeleton. Bone fibrosis remained evident after treatment. Notably, LGK974 caused significant bone loss in control mice. These results provide new insights into the role of Wnt and Gs-signaling in fibrosis and bone formation in a mouse model of Gs-GPCR pathway overactivation.<\/jats:p>","DOI":"10.1093\/jbmrpl\/ziae011","type":"journal-article","created":{"date-parts":[[2024,1,21]],"date-time":"2024-01-21T22:19:50Z","timestamp":1705875590000},"source":"Crossref","is-referenced-by-count":8,"title":["Wnt pathway inhibition with the porcupine inhibitor LGK974 decreases trabecular bone but not fibrosis in a murine model with fibrotic bone"],"prefix":"10.1093","volume":"8","author":[{"given":"Hsuan","family":"Lung","sequence":"first","affiliation":[{"name":"Department of Medicine , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"},{"name":"University of California , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"},{"name":"Oral and Craniofacial Sciences Graduate Program , School of Dentistry, , San Francisco, CA 94143 , United States"},{"name":"University of California , School of Dentistry, , San Francisco, CA 94143 , United States"},{"name":"Department of Dentistry, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine , Kaohsiung 833 , Taiwan"},{"name":"School of Dentistry, Institute of Oral Medicine, College of Medicine, National Cheng Kung University , Tainan 701 , Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelly L","family":"Wentworth","sequence":"additional","affiliation":[{"name":"Department of Medicine , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"},{"name":"University of California , Division of Endocrinology 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Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"},{"name":"University of California , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gauri","family":"Ganesh","sequence":"additional","affiliation":[{"name":"Department of Medicine , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"},{"name":"University of California , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for Regeneration Medicine, , San Francisco, CA 94143 , United States"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Misun","family":"Kang","sequence":"additional","affiliation":[{"name":"Oral and Craniofacial Sciences Graduate Program , School of Dentistry, , San Francisco, CA 94143 , United States"},{"name":"University of California , School of Dentistry, , San Francisco, CA 94143 , United States"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sunita","family":"Ho","sequence":"additional","affiliation":[{"name":"Oral and Craniofacial Sciences Graduate Program , School of Dentistry, , San Francisco, CA 94143 , United States"},{"name":"University of California , School of Dentistry, , San Francisco, CA 94143 , United States"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward C","family":"Hsiao","sequence":"additional","affiliation":[{"name":"Department of Medicine , Division of Endocrinology and Metabolism, The Institute for Human Genetics, and the Eli and Edythe Broad Institute for 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