Mereo, Ultragenyx’s setrusumab falls short in Phase 3 against osteogenesis imperfecta

Ultragenyx Pharmaceutical Inc. and Mereo BioPharma Group plc have reported topline results from the pivotal Phase 3 ORBIT and COSMIC trials evaluating setrusumab (UX143) in patients with osteogenesis imperfecta (OI), a rare genetic disorder characterised by brittle bones and a high risk of fracture.

The Phase 2/3 ORBIT study enrolled 159 pediatric and young adult participants aged 5 to 25 in its Phase 3 portion, while the Phase 3 COSMIC study enrolled 69 children aged 2 to under 7.

In both trials, the primary endpoint — reduction in annualized clinical fracture rate — did not reach statistical significance versus comparator arms (placebo in ORBIT and intravenous bisphosphonates in COSMIC). However, secondary endpoints demonstrated statistically significant improvements in bone mineral density (BMD) across both studies, consistent with treatment effects observed in earlier Phase 2 data. Safety findings were in line with prior experience, with no new safety signals reported.

In ORBIT, BMD improvements versus placebo were clear, but a lower-than-expected fracture rate in the placebo arm limited the ability to demonstrate a statistically significant reduction in fractures. In the younger COSMIC population, higher baseline fracture rates and numerically meaningful reductions in fracture incidence were observed with setrusumab compared with active control, although these did not meet prespecified statistical thresholds.

Outlook and implications

Commenting on the readout, Mereo CEO Denise Scots-Knight said that while the results were disappointing with respect to the primary endpoint, the companies plan to conduct further analyses to better understand treatment effects across patient subgroups and clinical measures beyond fractures — particularly in pediatric populations where unmet need remains high.

To preserve cash, Mereo is scaling back pre-commercial and manufacturing investments and shifting focus toward maximizing value across its broader rare-disease portfolio, including partnered assets such as alvelestat in alpha-1 antitrypsin deficiency–associated lung disease. As of Sept. 30, 2025, the company reported approximately $48.7 million in cash reserves, which it said should support continued operations as strategic priorities are refined.

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The Phase 3 programme had been closely watched following previously reported smooth trial conduct and acceptable safety at interim analyses. Setrusumab holds orphan designation in both the US and EU, Breakthrough Therapy designation from the US FDA, and PRIME designation in Europe.

Research context

Setrusumab is a fully human monoclonal antibody designed to inhibit sclerostin, a negative regulator of bone formation, with the aim of increasing bone mass and strength in patients with OI — a condition for which no globally approved therapies currently exist.

The molecule, originally known as BPS-804, was discovered by Novartis and out-licensed to UK-based Mereo BioPharma in a 2015 deal. Mereo subsequently licensed the asset to Ultragenyx, which is now the primary sponsor of ongoing clinical development.

Sclerostin is a glycoprotein predominantly produced by osteocytes in bone. By binding to LRP5/6 co-receptors, it inhibits the Wnt/β-catenin signalling pathway, suppressing osteoblast activity and acting as a key negative regulator of bone mass.

Beyond osteoporosis, sclerostin inhibition is being explored in indications such as bone healing, periodontitis, and metabolic disorders, reflecting its broader role in energy metabolism and adipose tissue function.

As of late 2025, the sclerostin-targeted therapy landscape includes one globally approved product and several clinical-stage candidates, although no other programmes are currently targeting OI:

  • Amgen’s romosozumab (Evenity), approved in the US and EU in 2019 for postmenopausal osteoporosis at high fracture risk
  • Eli Lilly’s blosozumab, which completed Phase 2 in postmenopausal osteoporosis
  • Jiangsu Hengrui’s SHR-1222, which completed a Phase 1 study
  • Angitia Bio’s AGA2118, a bispecific antibody targeting both sclerostin and DKK1, at the Phase 1 stage and again under development for postmenopausal osteoporosis