One-Year Italian Experience with Off-Label rhGH Treatment in SHOX-Deficient Children to Overcome the Shortage of Authorized rhGH Somatropin
Background/Objectives: The global landscape of growth hormone (GH) therapy is increasingly affected by supply shortages, posing risks to treatment continuity, particularly in rare diseases with a single authorized GH brand. In Italy, a prolonged shortage of the only approved recombinant human GH (rhGH) for SHOX-deficient (SHOXD) patients (Somatropin, Humatrope®) raised concerns about treatment interruption. As growth impairment is a key clinical feature of SHOXD, uninterrupted rhGH therapy is essential. To address this issue, the Italian Medicines Agency (AIFA) temporarily authorized the off-label use of alternative rhGH formulations. This study aimed to evaluate growth outcomes and safety over one year of off-label rhGH treatment in SHOXD patients and to compare these data with prior Humatrope® treatment. Methods: Fifty SHOXD patients (25 females), aged 2–17 and still in the growth phase, previously treated with Humatrope®, were switched to an alternative rhGH therapy. Height standard deviation scores (HSDS) and height velocity standard deviation scores (HVSDS) were recorded 12 and 6 months before and after the switch. Growth trends were analyzed using repeated-measures ANOVA. Results: No statistically significant differences in HSDS or HVSDS were observed after switching to alternative rhGH compared with prior treatment (p > 0.05). IGF-1 SDS values remained stable within age- and puberty-appropriate reference ranges. No adverse events were reported. Conclusions: In this real-world pediatric cohort, one year of off-label rhGH treatment during a drug shortage was not associated with relevant changes in growth parameters or safety concerns. These findings may support treatment continuity during temporary disruptions in rhGH availability.
Authors
- Małgorzata Waśniewska (ORCID: https://orcid.org/0000-0002-8299-2677)
- Chiara Mozzato
- Maria Felicia Faienza (ORCID: https://orcid.org/0000-0002-1899-8337)
- Rossella Gaudino (ORCID: https://orcid.org/0000-0002-6292-0246)
- Marco Cappa (ORCID: https://orcid.org/0000-0001-5647-7519)
- Laura Guazzarotti (ORCID: https://orcid.org/0000-0002-5648-0109)
- Mariacarolina Salerno (ORCID: https://orcid.org/0000-0003-1310-3300)
- Maria Cristina Maggio (ORCID: https://orcid.org/0000-0002-7777-1239)
- Gabriella Pozzobon
- Francesco Gallo
Institutions
- University of Messina (IT)
- University of Verona (IT)
- University of Padua (IT)
- Vita-Salute San Raffaele University (IT)
- Ospedale A. Perrino (IT)
- Federico II University Hospital (IT)
- Bambino Gesù Children's Hospital (IT)
- Azienda Ospedaliera Universitaria Policlinico "G. Martino" (IT)
- University of Bari Aldo Moro (IT)
- University of Naples Federico II (IT)
- University of Palermo (IT)
Publication Details
- Journal
- Endocrines
- Published
- 2026-07-02
- DOI
- https://doi.org/10.3390/endocrines7030033
- Primary Topic
- Growth Hormone and Insulin-like Growth Factors
- Type
- article
- Field-Weighted Citation Impact
- 0.00