SMA Research Platform

Evidence graph for Spinal Muscular Atrophy

Biology-first target discovery
Christian Fischer / Bryzant Labs
14,766Targets
453Trials
84Drugs
7Datasets
6,987Sources
64,683Claims
72,052Evidence
29,649Hypotheses
DRUGapprovedaso

nusinersen

Brand names: Spinraza

Mechanism

Antisense oligonucleotide targeting SMN2 pre-mRNA ISS-N1 to promote exon 7 inclusion.

Approved indications

type1type2type3presymptomatic

Related claims (50)

TypePredicateConfSource
drug efficacyNusinersen is approved for the treatment of spinal muscular atrophy (clinical trial result)100%34621235
drug efficacyNusinersen is a medication that treats Spinal Muscular Atrophy.100%39690144
safetyCardiac arrest, autism spectrum disorder, and epilepsy emerged as potential new side effects of Nusinersen not previously listed.90%39690144
drug efficacyDelay in nusinersen infusions may have a relevant impact on the course of spinal muscular atrophy pathology (observed in patient cohort)80%34621235
drug efficacyNusinersen treatment was associated with a significant improvement in EK2 (OR = 0.81, p = 0.001) (observed in patient cohort).69%35872571
drug efficacyNusinersen treatment is associated with improvement in patient condition after one year in 84% of patients (observed in patient cohort).65%40451126
motor functionHigh baseline motor function is associated with the probability of acquiring a sitting position in patients with SMA1 treated with nusinersen (clinical trial result).65%31799720
splicing eventNusinersen is an antisense oligonucleotide (ASO) targeting the splicing inhibitory sequence in the intron downstream of exon 7 from SMN2, thus increasing exon 7 inclusion.65%36408582
biomarkerNPTX2 emerges as a potential biomarker of treatment response to nusinersen in pwSMA suggesting its significant pathophysiological role in late-onset SMA (observed in patient cohort).65%W4411855648
drug efficacyApproximately 70% of infants appear to have a clinically significant response to nusinersen with improved motor function (clinical trial result).65%30414815
drug efficacyNusinersen treatment is associated with a statistically significant (p = 0.042) improvement of HFMSE (clinical trial result).65%35832502
drug efficacyA total of 70% of patients in the pediatric cohort and 72% of patients in the adult cohort achieved a clinically meaningful improvement in motor function following nusinersen treatment (clinical trial result).65%38921951
splicing eventAnalysis of autopsy tissue from patients exposed to nusinersen showed increased SMN2 mRNA exon 7 inclusion in the spinal cord.65%27939059
biomarkerNPTX2 emerges as a potential biomarker of treatment response to nusinersen in pwSMA suggesting its significant pathophysiological role in late-onset SMA.65%40868076
splicing eventNusinersen acts through prevention of skipping of exon 7 of *Survival Motor Neuron 2* (*SMN2*) by sequestering Intronic Splicing Silencer N1 (ISS-N1), located within *SMN2* intron 7.65%41551726
splicing eventNusinersen alters SMN2 pre-RNA splicing so exon 7 is included (clinical trial result).65%31093018
drug efficacyNusinersen treatment was associated with a significant improvement in HFMSE (odds ratio [OR] 1.15, p = 0.006) (observed in patient cohort).65%35872571
biomarkerNeurofilament heavy chain, tau protein, S100B protein, and neuron-specific enolase do not serve as potential biomarkers during the loading phase of nusinersen (observed in patient cohort).65%31671515
splicing eventNusinersen promotes the inclusion of exon 7 in SMN2 transcripts (observed in mouse model).65%32612161
drug efficacyNusinersen treatment improved motor scores as measured by the Hammersmith Infant Neurological Examination-2 by 2.6% (p = .008) over the 22-month study period (clinical trial result).65%33608138
biomarkerMuscle quantitative MRI (qMRI) may be a biomarker of disease progression in adult SMA3 patients during nusinersen treatment.65%35832502
motor functionIntrathecal nusinersen treatment improves or stabilizes motor function in 90% of young children with spinal muscular atrophy types 1c-3a (clinical trial result).65%36382221
neuroprotectionIncreased MCP1/CCL2 levels indicate a possible neuroprotective mechanism associated with nusinersen therapy (observed in patient cohort).65%36525882
gene expressionAn increase in SMN expression by either SMN gene therapy replacement or the antisense oligonucleotide (ASO), Nusinersen, significantly upregulated the endogenous levels of GEMIN5 in mammalian cells and mutant GEMIN5-derived iPSC neurons.65%37369805
drug efficacyNusinersen treatment was associated with a significant improvement in the 6-min walk test (OR = 1.07, p < 0.001) (observed in patient cohort).65%35872571
gene expressionIn tissues isolated from nusinersen-treated SMA patients, antisense oligonucleotide (ASO) concentration and full-length (exon 7 including) SMN2 (SMN2-FL) mRNA level increases were highest in lumbar and thoracic spinal cord.65%31589162
motor functionTreatment with Nusinersen in SMA3 improves motor function in longstanding disease even in clinically advanced stages.65%33682724
gene expressionHA2-ApoE(131-150), when conjugated to Nusinersen, significantly increased the level of full-length functional mRNA of the survival motor neuron 2 (SMN2) gene in SMA patient-derived fibroblasts (demonstrated in vitro)65%33640427
gene expressionNusinersen augmented the expression of full-length SMN transcripts and proteins in SMA mice (observed in mouse model).65%40180233
drug efficacyNusinersen treatment has led to dramatic improvements in respiratory failure and survival in SMA1 patients (observed in patient cohort)65%34921596
drug efficacyNusinersen treatment resulted in an improvement, with increased mean scores between baseline (T0) and 12 months (T12) on both the Hammersmith Functional Motor Scale Expanded and the Revised Upper Limb Module (clinical trial result).65%34099377
splicing eventNusinersen binds to the pre-mRNA of SMN2 to prevent exon skipping and produce functional SMN protein.65%34515035
splicing eventThe combined treatment of SMA cells with sub-optimal doses of LBH589 and of an antisense oligonucleotide that mimic Nusinersen (ASO_ISSN1) elicits additive effects on SMN2 splicing (demonstrated in vitro).65%31811660
drug efficacyNusinersen treatment improved motor scores as measured by the Hammersmith Functional Motor Scale-Expanded by 3.3% (p = .00005) over the 22-month study period (clinical trial result).65%33608138
drug efficacyNusinersen treatment is associated with improvement in patient condition after four years in 54% of patients (observed in patient cohort).65%40451126
splicing eventNusinersen modifies premessenger RNA splicing of exon 7, leading to stable SMN protein expression.65%30617569
biomarkerSMN circ4-2b-3 is a potential biomarker to predict the therapeutic response of type I SMA patients to Nusinersen (observed in patient cohort).65%39592557
drug efficacyNusinersen treatment supports efficacy and safety in adult patients with SMA2 and SMA3 (observed in patient cohort).65%35527201
drug efficacyDuring the 18 months of nusinersen treatment, CK decreased (∆CK = -17.56%, p < 0.0001) (observed in patient cohort).64%33792208
motor functionGrip strength increased by 44.9% (P = 0.031) in treatment-naive SMA patients treated with nusinersen via SIC (clinical trial result).64%34606118
drug efficacyMedian symptom severity decreased during nusinersen treatment (median observational period 6.1 months, 0.5-16 months) from 3.7 to 3.3 MYMOP2 score points (p < 0.001) (clinical trial result).64%33960080
drug efficacyDuring the 18 months of nusinersen treatment, Crn slightly increased (∆Crn = +4.75%, p < 0.05) (observed in patient cohort).64%33792208
splicing eventNusinersen binds to intron splicing silencer-N1 (ISS-N1; a site present ten nucleotides down to the junction of exon 7 and intron 7), modulating the splicing of SMN2 pre-mRNA transcript to increase the inclusion of exon 7, thereby increasin...60%28755059
drug efficacyA significant clinical improvement was seen on all domains between T0 (prior to administration of nusinersen) and T1 (immediately before the first administration of maintenance doses, 6 months after the first administration of nusinersen) (...60%31939268
motor functionSeventy-one per cent of nusinersen-treated infants versus 3% of infants in the control group were responders on the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND) measure of motor disability (clinic...60%31825542
drug efficacyExpedited therapy initiation, including reversible treatments like nusinersen or risdiplam, is recommended without waiting for the completion of the molecular testing, thus minimizing delays in crucial therapeutic interventions.59%40900970
gene expressionNusinersen induced modulation of apolipoprotein A1, apolipoprotein E, and transthyretin may support SMN improved-expression effects (observed in patient cohort).59%33919289
drug efficacyNusinersen treatment was associated with stability in bulbar function in 40% of SMA type 1 patients with symptom onset >2 weeks and <3 months (observed in patient cohort).59%40504745
safetyNusinersen has the same safety and tolerability profile as in the clinical trials (clinical trial result).59%32456992
biomarkerHigh-throughput CSF proteomics data prior and after nusinersen therapy provide possible biomarkers that may help in identification of more accurate prognosis59%32128827

Off-Target Findings (9)

T4: 4untiered: 5
TargetRoleTierBoltz-2 iPTMChai-1 iPTMVerdict
HSP70_NBDOTHERT40.774Low relevance
LRP4_propellerOTHERT40.767Low relevance
LIMK2_aCOTHERT40.616Low relevance
CHRNG_fetalOTHERT40.541Low relevance
CHRNA1_ECDOTHER0.859
PFN2SMA0.615
STMN1SMA0.594
KCNA2_poreOTHER0.473
PERP_ECL1OTHER0.452
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