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Nusinersen in type 0 spinal muscular atrophy: should we treat?

Authors :
Tiberi, Eloisa
Costa, Simonetta
Pane, Marika
Priolo, Francesca
De Sanctis, Roberto
Romeo, Domenico Marco
Tiziano, Francesco Danilo
Conti, Giorgio
Vento, Giovanni
Mercuri, Eugenio Maria
Tiberi E.
Costa S.
Pane M. (ORCID:0000-0002-4851-6124)
Priolo F.
de Sanctis R.
Romeo D. (ORCID:0000-0002-6229-1208)
Tiziano F. D. (ORCID:0000-0002-5545-6158)
Conti G. (ORCID:0000-0002-8566-9365)
Vento G. (ORCID:0000-0002-8132-5127)
Mercuri E. (ORCID:0000-0002-9851-5365)
Tiberi, Eloisa
Costa, Simonetta
Pane, Marika
Priolo, Francesca
De Sanctis, Roberto
Romeo, Domenico Marco
Tiziano, Francesco Danilo
Conti, Giorgio
Vento, Giovanni
Mercuri, Eugenio Maria
Tiberi E.
Costa S.
Pane M. (ORCID:0000-0002-4851-6124)
Priolo F.
de Sanctis R.
Romeo D. (ORCID:0000-0002-6229-1208)
Tiziano F. D. (ORCID:0000-0002-5545-6158)
Conti G. (ORCID:0000-0002-8566-9365)
Vento G. (ORCID:0000-0002-8132-5127)
Mercuri E. (ORCID:0000-0002-9851-5365)
Publication Year :
2020

Abstract

A male infant affected by type 0 SMA with one copy of SMN2 received early treatment with Nusinersen at the age of 13 days. He showed mild motor improvement 2 months after treatment started but despite also showing some minimal respiratory improvement, required tracheostomy at the age of 4 months and had increasing cardiac and autonomic dysfunction leading to exitus at 5 months. Our findings, expanding the results available on Nusinersen, confirm its relative efficacy in the most severely affected infants and provide clinical evidence to be used at the time requests for treating severe infants are discussed.

Details

Database :
OAIster
Notes :
English
Publication Type :
Electronic Resource
Accession number :
edsoai.on1242038017
Document Type :
Electronic Resource