Interactive Genomic Exploration · Lower motor neuron

The Genetics of Spinal Muscular Atrophy (SMA)

An interactive journey through the human genome to understand why loss of the SMN1 gene weakens the muscles — and how the backup gene SMN2 and three new therapies have transformed the disease.

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Main causal gene (SMN1)
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Backup modifier gene (SMN2)
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Approximate incidence at birth
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Approved disease-modifying therapies
The story at a glance

From the missing gene to the muscle that weakens

The whole journey of this page, summarized in steps.

Starting point
SMN1 gene missing or mutated
The deficit
SMN protein is lacking
The backup
SMN2 makes little (exon 7 skipped)
The target cell
Lower motor neurons die
The muscle
Muscle with no signal
Outcome
Weakness and atrophy
The disease

What is SMA?

Before diving into the genetics, it helps to know the disease that these genes explain.

Spinal muscular atrophy (SMA) is an inherited neuromuscular disease caused by the degeneration of the lower motor neurons in the anterior horn of the spinal cord. The muscle stops receiving the command to move, and proximal, symmetric muscle weakness and atrophy appear (the muscles closer to the trunk are affected first). One important feature: cognition is preserved — intelligence and the senses are not affected.

~1/10,000
Affected births (approximate incidence)
~1/50
Healthy carriers in the population
Recessive
Autosomal recessive inheritance (loss of SMN1)
SMN2
The backup gene's copy number modulates severity
Origin

From Werdnig and Hoffmann to genetics

The Austrian physician Guido Werdnig (1891) and the German Johann Hoffmann (1893) described the most severe infantile form, today known as Werdnig-Hoffmann disease. For a century it was a clinical entity with no known cause; the genetic era began in 1995 with the identification of the SMN1 gene. Before today's therapies, SMA was the leading genetic cause of death in infancy.

Symptoms

When the muscle is left without its command

Symptoms depend on the type and age of onset, but share a common pattern:

Core sign

Weakness and hypotonia

Muscle weakness and hypotonia (low tone, "floppy baby"), predominantly proximal and symmetric. It is the hallmark of lower motor neuron damage.

Severe forms

Breathing and swallowing

In the most severe types, weakness affects the respiratory muscles and swallowing, which historically determined the prognosis.

What is spared

Mind and senses intact

SMA preserves cognition, sensation, and vision and hearing. Children with SMA usually have normal or above-average intellectual development.

Other signs

Areflexia and tremor

Reduced or absent reflexes, tongue fasciculations and, in forms that reach sitting or walking, scoliosis and contractures.

Profiles · Types

Not all SMA is the same: Types 0–4

SMA is classified into types according to the age of onset and the maximum motor milestone reached. The SMN2 copy number is the main modulator.

Type 1 · Werdnig-Hoffmann

Onset before 6 months, very severe. The baby never sits without support. Usually associated with 2 copies of SMN2.

Type 2 · Intermediate

Onset between 6 and 18 months. The child sits but never walks independently. Usually associated with 3 copies.

Type 3 · Kugelberg-Welander

Onset after the first year or in childhood. The person walks, though may lose that ability over time. 3–4 copies.

Type 4 · Adult

Onset in adulthood, the mildest form. Slowly progressive proximal weakness. Usually associated with 4 or more copies.

There is also a prenatal Type 0, the most severe form, with very little movement already before birth and usually 1 copy of SMN2.

Treatment

From fatal disease to treatable disease

SMA is today the great success story of gene therapy: there are three approved therapies that modify its course, especially when started early.

Antisense oligonucleotide

Nusinersen (Spinraza)

Nusinersen (2016) is an intrathecal ASO that makes SMN2 include exon 7 and produce more functional SMN protein.

Gene therapy

Onasemnogene abeparvovec (Zolgensma)

A gene therapy (2019) that delivers a functional copy of SMN1 via an AAV9 vector in a single dose.

Oral drug

Risdiplam (Evrysdi)

Risdiplam (2020) is an oral drug that modifies SMN2 splicing so it produces more full-length protein.

Educational content with a scientific basis (Werdnig 1891; Hoffmann 1893; SMN1 gene, Lefebvre et al. 1995; nusinersen, Finkel et al. 2017; Zolgensma, Mendell et al. 2017; risdiplam, 2020). It does not replace assessment by a healthcare professional.

Foundation

What is DNA?

DNA (deoxyribonucleic acid) is the molecule that stores the genetic instructions of every living thing, spread across about 3 billion base pairs.

Four bases — A, T, C and G — form the double helix. In SMA, the problem lies in a region of chromosome 5 that contains two nearly identical genes, SMN1 and SMN2: when SMN1 is lost, the motor neuron is left without enough SMN protein.

A — Adenine
T — Thymine
C — Cytosine
G — Guanine
Interactive

Explore the genome

The genetic heart of SMA is on chromosome 5, but there are genes that accompany or modulate it on other chromosomes. Click a chromosome to see its regions, the evidence, and the genes involved.

Gene atlas

Gene catalog

The causal gene, the backup modifier gene, and the SMA complex and modifier genes. Search and filter by cellular mechanism; click a card to see its function and the studies.

Functional convergence

Cellular mechanisms

The SMA genes revolve around the SMN protein and the lower motor neuron. Hover over a node to identify the gene; click to see the detail.

Interactive

From exon 7 to the motor neuron

Two key pieces of SMA: how many copies of SMN2 there are (and how much protein they make) and the lower motor neuron → muscle circuit. Explore them.

The severity modulator

The SMN2 copy number (1–4)

When SMN1 is missing, the only source of SMN protein is SMN2. But a single base change (c.840C>T) makes most of its RNA skip exon 7 and produce an unstable protein; only a small fraction is full-length. That is why, the more copies of SMN2, the more functional protein and the milder the disease. Slide to see the effect.

2SMN2 copies
Type 1–2

Indicative. The SMN2 copy number explains much of the severity, but it is not an exact rule: there are genetic modifiers (such as PLS3) and other factors. The definitive type is established by the clinical course.

The motor circuit

From the lower motor neuron to the muscle

In SMA the lower motor neuron (the one in the anterior horn of the cord) degenerates; it connects directly to the muscle. Click each step of the diagram to compare the healthy and degenerated states.

More than a century of science

Timeline of discoveries

From Werdnig and Hoffmann's description to the SMN1 gene and the three therapies that have changed the disease.

Biology

Biological processes involved

How the lack of SMN protein damages the lower motor neurons that control movement.

What the data say

Is SMA inherited?

Yes. SMA is autosomal recessive: it requires inheriting a missing or mutated copy of SMN1 from each parent. When both are healthy carriers, each pregnancy has a 25% chance of an affected child, 50% carrier, and 25% unaffected.

Father Aa Mother Aa AA Unaffected 25% Aa Carrier 25% Aa Carrier 25% aa Affected 25%
Unaffected (AA)
Carrier (Aa)
Affected (aa)
0%
Risk of an affected child (two carriers)
0%
Healthy carrier children (like their parents)

Around 1 in 50 people is a healthy carrier of an SMN1 loss, without knowing it and without symptoms. That is why many countries offer carrier screening to couples and newborn screening of newborns, which makes it possible to detect and treat SMA before symptoms appear.

Carrier frequency in the general population~1/50

About 2% of the population are carriers. Because inheritance is recessive, both parents must be carriers for a child to be affected.

Takeaways

What do we know for certain?

The essentials about the genetics of spinal muscular atrophy:

The most important point: SMA is the great success story of gene therapy. In just a few years it went from being the leading genetic cause of infant death to having three approved therapies (nusinersen, Zolgensma, and risdiplam). Treating early — ideally before symptoms, thanks to newborn screening — dramatically changes the prognosis.

Therapeutic pathways: where does each one stand?
Already in clinical use

Three disease-modifying therapies

  • Nusinersen (intrathecal ASO): includes exon 7 of SMN2
  • Zolgensma (AAV9 gene therapy): single-dose copy of SMN1
  • Risdiplam (oral): corrects SMN2 splicing
  • Respiratory and nutritional support and rehabilitation
In clinical trials

Improving even further

  • Combination therapies (SMN + muscle) to add benefits
  • Apitegromab and other agents aiming to improve muscle strength
  • Biomarkers to measure the response to treatment
Preclinical research / future

Optimize and arrive earlier

  • Improve the delivery and durability of gene therapy
  • Take advantage of the presymptomatic window (newborn screening)
  • Targeting modifiers such as PLS3 (protective)
Myths

What SMA is NOT

SMA is a genetic motor neuron disease. It is worth dispelling a few misconceptions:

It is not contagious It does not affect intelligence It does not affect vision or hearing It is not caused by anything the parents did It is not the same as muscular dystrophy

Being an SMA carrier causes no symptoms: it is completely normal and compatible with a healthy life. It only matters for family planning, if the partner is also a carrier.

The frontier

The latest and what's coming

With the disease now treatable, the frontier shifts to treating earlier and better.

Recent advances

What is changing the field

Early diagnosis

Newborn screening

Detecting the loss of SMN1 with a drop of blood from the newborn makes it possible to start treatment before symptoms, when there are more motor neurons to protect.

Presymptomatic treatment

The golden window

Studies in babies treated before symptoms show near-normal motor development: SMA teaches that in these diseases timing is almost everything.

Adding mechanisms

Combination therapies

Combining those that restore SMN with drugs that improve the muscle (such as apitegromab) aims to squeeze even more strength recovery.

Future directions

Where the research is heading

Better delivery

Optimize gene therapy

Better vectors and routes of administration so that the SMN1 copy reaches more motor neurons and lasts longer over time.

Muscle

Improve strength

Beyond protecting the motor neuron, regaining muscle mass and strength with drugs targeting the muscle itself.

Modifiers

Harness protectors

Explore natural modifiers such as PLS3, which protects some carriers within a family, as a possible therapeutic target.

Research is advancing quickly and some of these results are preliminary: the dates and specific data may change as the trials mature.

Frequently asked questions

Common questions

The questions that come up most when learning about SMA.

Is SMA inherited?
Yes. SMA is autosomal recessive: the child inherits a missing or mutated copy of SMN1 from each parent. If both are healthy carriers, each pregnancy has a 25% chance of an affected child, 50% carrier, and 25% unaffected.
What does it mean to be a carrier?
A carrier has one normal copy of SMN1 and one missing or mutated copy. They have no symptoms and their life is completely normal. About 1 in 50 people is one. It only matters if their partner is also a carrier, when it comes to having children.
What is SMN2 and why does it matter?
SMN2 is a gene almost identical to SMN1 that acts as a "backup." Because of a base change (c.840C>T), most of its RNA skips exon 7 and produces unstable protein; only a fraction is functional. That is why the more copies of SMN2 a person has, the more SMN protein they produce and the milder the disease tends to be.
Is there a treatment for SMA?
Yes, and it is good news: there are three approved therapies that modify the course of the disease — nusinersen (Spinraza), onasemnogene abeparvovec (Zolgensma, gene therapy), and risdiplam (Evrysdi, oral). They work best the earlier they are started, ideally before symptoms.
Is newborn screening worthwhile?
Very much. Newborn screening detects the loss of SMN1 in a drop of blood from the newborn and makes it possible to treat before symptoms appear. Babies treated presymptomatically reach far better motor development; that is why more and more countries are adopting it.
Does SMA affect intelligence?
No. SMA affects the lower motor neurons and the muscle, but preserves cognition, sensation, and the senses. Children with SMA have normal or above-average intellectual development.
Sources and glossary

Where this comes from

Milestones and scientific sources on which this page is based.

Founding milestones
1891Werdnig G. Description of the severe infantile form of spinal muscular atrophy (Werdnig-Hoffmann disease).
1893Hoffmann J. Clinical characterization of infantile spinal muscular atrophy, completing Werdnig's description.
1995Lefebvre S et al. Identification and characterization of a spinal muscular atrophy-determining gene. Cell. The SMN1 gene and its role.
The therapies that changed the disease
2017Finkel RS et al. Nusinersen versus sham control in infantile-onset spinal muscular atrophy. N Engl J Med. (ENDEAR trial.)
2017Mendell JR et al. Single-dose gene-replacement therapy for spinal muscular atrophy. N Engl J Med. The basis of onasemnogene abeparvovec (Zolgensma).
2020Approval of risdiplam (Evrysdi), an oral SMN2 splicing modifier (FIREFISH and SUNFISH trials).
Databases
DatabasesOMIM #253300 and resources from Cure SMA and GeneReviews.

A synthesizing educational page; not a primary clinical source. For medical decisions, consult professionals and the official resources of SMA associations.

Comparator

SMA among other motor neuron diseases

How SMA sits relative to other genetic atlases in this collection.

DiseaseNeuron affectedInheritanceKey gene
SMA (this page)Lower motor neuronAutosomal recessiveSMN1 / SMN2
ALSUpper and lower motor neuronMostly sporadicC9orf72, SOD1, TARDBP
Duchenne dystrophyMuscle (not the neuron)X-linked recessiveDMD (dystrophin)

Links to other atlases are indicative; they may not be available in this collection.

Glossary

Key terms

SMNThe "Survival Motor Neuron" protein.
A protein essential for the survival of the motor neuron. It takes part in assembling the machinery that processes RNA. When it is lacking, the lower motor neuron degenerates and SMA appears.
SMN1The causal gene of SMA.
Located at 5q13.2, it is the main source of full-length SMN protein. Its absence or mutation in both copies causes SMA. It is the gene targeted by gene therapy.
SMN2The backup gene that modulates severity.
Almost identical to SMN1. A base change (c.840C>T) makes most of its RNA skip exon 7, producing unstable protein. Its copy number is the main modulator of SMA severity.
Exon / splicingThe RNA fragments that are assembled.
Genes are transcribed into RNA, from which parts are cut out (splicing) to join the exons that encode the protein. In SMN2, exon 7 is usually left out; several therapies aim to have it included.
Lower motor neuronThe neuron that connects to the muscle.
It lives in the anterior horn of the spinal cord. Its axon reaches the muscle directly. It is the cell that degenerates in SMA; its loss leaves the muscle without a signal and causes weakness and atrophy.
RecessiveBoth altered copies are required.
In autosomal recessive inheritance, the disease appears only if both copies of the gene are inherited altered (one from each parent). With a single altered copy, a person is a healthy carrier.
CarrierHas one altered copy, without symptoms.
A person with one normal copy of SMN1 and one missing or mutated. Does not develop the disease. If their partner is also a carrier, their children have a 25% risk of being affected.
Gene therapyDelivering a functional copy of the gene.
A strategy that introduces a healthy copy of a gene into cells. In SMA, onasemnogene abeparvovec uses an AAV9 vector to deliver SMN1 in a single dose: the proof of concept that gene therapy works.
Test what you've learned

Interactive quiz

Six questions to check what you take away. It grades itself: tap an answer and you'll instantly see whether you're right, with the explanation.

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