Real-World Growth Outcomes of Vosoritide in Children with Achondroplasia: A Single-Center Cohort Study

Abstract Introduction Vosoritide, a C-type natriuretic peptide analogue, is the first approved therapy targeting the pathophysiology of achondroplasia. While clinical trials demonstrate efficacy, individuals with prior orthopedic surgeries are typically excluded, and real-world data remain limited. Materials and Methods This retrospective analysis included 25 children with achondroplasia (17 boys, age range 2.9-14.3 years) treated with vosoritide for a mean duration of 12.7 months at a multidisciplinary center. Z-scores were calculated using an AI-assisted growth assessment tool previously validated for children with achondroplasia. Δ z-scores for height, sitting height, arm span, and body mass index (BMI) were calculated from baseline to follow-up. Multiple linear regression analyses assessed predictors of response. Results Mean height z-scores improved from -0.62 ± 1.09 to -0.24 ± 1.20 (p < 0.001). Mean arm span z-score increased from -1.28 ± 0.93 to -0.96 ± 0.91 (p=0.007). Sitting height z-scores showed a non-significant trend toward improvement; BMI z-scores remained stable. Mean Δ height and Δ arm span z-score gains were +0.38 ± 0.45 and +0.32 ± 0.48, respectively. A subgroup analysis of 8 patients with versus 17 without prior limb-lengthening surgery showed no significant differences. Regression analyses adjusted for sex, age, BMI z-scores, and surgical history were likely underpowered to detect meaningful predictors of response. There were no serious treatment-related adverse events. Conclusions Vosoritide therapy enhanced linear and appendicular growth in a real-world cohort of children with achondroplasia, including post-surgical patients. These findings align with clinical trial data and support the therapy’s applicability across diverse clinical profiles.

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Journal
Journal of the Endocrine Society
Published
2026-03-26
DOI
https://doi.org/10.1210/jendso/bvag072
Primary Topic
Connective tissue disorders research
Type
article
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article

Real-World Growth Outcomes of Vosoritide in Children with Achondroplasia: A Single-Center Cohort Study

Ravit Regev, Avivit Brener, Maya Gal, Leonid Zeitlin et al.
Journal of the Endocrine Society
Connective tissue disorders research
article

Real-World Growth Outcomes of Vosoritide in Children with Achondroplasia: A Single-Center Cohort Study

Ravit Regev, Avivit Brener, Maya Gal, Leonid Zeitlin, Yael Lebenthal, Nechama Dechovich, Eyal Cohen-Sela, Ophir Borger, Yarden Waksman
article en

Abstract

Abstract Introduction Vosoritide, a C-type natriuretic peptide analogue, is the first approved therapy targeting the pathophysiology of achondroplasia. While clinical trials demonstrate efficacy, individuals with prior orthopedic surgeries are typically excluded, and real-world data remain limited. Materials and Methods This retrospective analysis included 25 children with achondroplasia (17 boys, age range 2.9-14.3 years) treated with vosoritide for a mean duration of 12.7 months at a multidisciplinary center. Z-scores were calculated using an AI-assisted growth assessment tool previously validated for children with achondroplasia. Δ z-scores for height, sitting height, arm span, and body mass index (BMI) were calculated from baseline to follow-up. Multiple linear regression analyses assessed predictors of response. Results Mean height z-scores improved from -0.62 ± 1.09 to -0.24 ± 1.20 (p < 0.001). Mean arm span z-score increased from -1.28 ± 0.93 to -0.96 ± 0.91 (p=0.007). Sitting height z-scores showed a non-significant trend toward improvement; BMI z-scores remained stable. Mean Δ height and Δ arm span z-score gains were +0.38 ± 0.45 and +0.32 ± 0.48, respectively. A subgroup analysis of 8 patients with versus 17 without prior limb-lengthening surgery showed no significant differences. Regression analyses adjusted for sex, age, BMI z-scores, and surgical history were likely underpowered to detect meaningful predictors of response. There were no serious treatment-related adverse events. Conclusions Vosoritide therapy enhanced linear and appendicular growth in a real-world cohort of children with achondroplasia, including post-surgical patients. These findings align with clinical trial data and support the therapy’s applicability across diverse clinical profiles.

Journal of the Endocrine Society
Tel Aviv University (IL), Tel Aviv Sourasky Medical Center (IL), Finisar (Israel) (IL), Children's Hospital Association (US), Institute of Nutrition, Metabolism and Diabetes (CA)
Openalex Percentile: Top 4%
Connective tissue disorders research
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