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DOI 10.1186/s13023-023-02919-8

Медицинская библиотека · Orphanet journal of rare diseases · 2023

Safety outcomes and patients' preferences for home-based intravenous enzyme replacement therapy (ERT) in pompe disease and mucopolysaccharidosis type I (MPS I) disorder: COVID-19 and beyond.

Toscano Antonio, Musumeci Olimpia, Sacchini Michele, Ravaglia Sabrina, Siciliano Gabriele, Fiumara Agata, Verrecchia Elena, Maione Melania, Gentile Jennifer, Fischetto Rita, Crescimanno Grazia, Taurisano Roberta, Sechi Annalisa, Gasperini Serena, Cianci Vittoria, Maggi Lorenzo, Parini Rossella, Lupica Antonino, Scarpa Maurizio

Многоцентровое исследованиеСтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

BACKGROUND

The Italian Medicines Agency (AIFA) demands precise information on benefit/risk profile of home-based enzyme replacement therapy (ERT) for the treatment of patients with Pompe disease and Mucopolysaccharidosis type I (MPS I). This passage is necessary to obtain the authorization for ERT home therapy, even after the coronavirus disease-19 (COVID-19) pandemic period. This research intends to evaluate the safety, treatment satisfaction, and compliance of MPS I patients treated with laronidase (Aldurazyme®) and Pompe Disease patients treated with alglucosidase alfa (Myozyme®) in a homecare setting.

RESULTS

We report herein an early interim analysis of the HomERT (Home infusions of ERT) study, a multicenter, non-interventional, double-cohort study that retrospectively analyzed 38 patients from 14 sites in Italy: cohort A (Pompe disease - 32 patients) and cohort B (MPS I - 6 patients). Among the selected patients who started home therapy before enrollment, the average number of missed home-based infusions was 0.7 (1.3) in cohort A and 3.8 (6.4) in cohort B with no return to the hospital setting. Irrespective of the treatment location, 3 prior ADRs per cohort were reported. The majority of patients preferred home-based infusions (cohort A: 96.9%; cohort B: 100%): the main reason was attributed to treatment convenience (cohort A: 81.3%; cohort B: 83.3%). Despite the underlying conditions, most patients self-evaluated their health as "good" (cohort A: 50%; cohort B: 83.3%).

CONCLUSIONS

Evidence of favorable safety profile, improved treatment compliance and personal satisfaction validates the use of ERT with laronidase and alglucosidase alfa as a strong candidate for home therapy.

Для цитирования: Toscano Antonio, Musumeci Olimpia, Sacchini Michele, Ravaglia Sabrina, Siciliano Gabriele, Fiumara Agata, Verrecchia Elena, Maione Melania, Gentile Jennifer, Fischetto Rita, Crescimanno Grazia, Taurisano Roberta, Sechi Annalisa, Gasperini Serena, Cianci Vittoria, Maggi Lorenzo, Parini Rossella, Lupica Antonino, Scarpa Maurizio. Safety outcomes and patients' preferences for home-based intravenous enzyme replacement therapy (ERT) in pompe disease and mucopolysaccharidosis type I (MPS I) disorder: COVID-19 and beyond.. Orphanet journal of rare diseases. 2023. DOI: 10.1186/s13023-023-02919-8.

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