After receiving a genetic diagnosis, many families want to understand more about what the test found. This page explains the key terms on a genetic test report and introduces the databases where researchers track information about SETD5 variants. You do not need to understand all of this. But if you are someone who feels better with more information, this page is for you.

Walk Through a Sample Report

Click any row to see what it means in plain language. Fields with multiple possible values show all the options your report might contain.

Sample Genetic Test Report Clinical Whole Exome Sequencing
Report Date: XX/XX/XXXX
Accession: SAMPLE-00000
Patient: [Sample] DOB: XX/XX/XXXX Ordering Physician: [Sample], MD
Gene
SETD5
+

This is the gene where the lab found a change. SETD5 provides instructions for making a protein involved in brain development and gene regulation. It sits on chromosome 3 at position 3p25.3. Changes in this gene are associated with intellectual disability and developmental delays - a condition sometimes called SETD5 Syndrome or MRD23.

Variant
c.2025_2026delAG (p.Gly676Valfs*2)
+

This is the exact "address" of the change in the DNA. The c. notation tells the lab where in the gene the change occurred. The p. notation describes how the protein is affected. You do not need to memorize this - but it's the key identifier when talking to a genetic counselor or searching databases like ClinVar.

Variant Type
Frameshift (deletion)
+

This describes what kind of change happened to the DNA. Different types of changes affect the gene in different ways. Your report will list one of the following:

Types of genetic changes seen in SETD5

Frameshift

A letter was added or deleted, scrambling all the instructions that follow. Like removing one letter from a sentence so every word after it becomes garbled.

Nonsense

One letter changed in a way that creates a premature stop signal. The protein gets cut short and usually cannot function.

Missense

One letter changed, swapping one protein building block for another. The protein is still made but may not work correctly.

Splice-site

A change at the boundary where the gene's instructions are edited together. Can disrupt how the protein is assembled.

Deletion

A piece of the genetic code is missing. Can be small (a few letters) or large (a section of chromosome). Larger deletions may include neighboring genes.

Duplication

A section of genetic code is copied, appearing twice instead of once.

Classification
Likely Pathogenic
+

This is the lab's assessment of whether this change is responsible for your child's condition. Labs use a five-tier system defined by the ACMG (American College of Medical Genetics):

The five classification levels

Pathogenic

This change is known to cause disease. Strong evidence from multiple sources. The clearest result.

Likely Pathogenic

Strong evidence this change causes disease, but seen in fewer people. Most clinicians treat this similarly to Pathogenic.

Variant of Uncertain Significance (VUS)

Not enough evidence yet. Doesn't confirm or rule out a diagnosis. Can be reclassified as more data becomes available.

Likely Benign

Probably harmless. Evidence suggests this change does not cause disease.

Benign

Definitely harmless. Common in the general population and not associated with disease.

Inheritance
De novo (autosomal dominant)
+

This tells you where the change came from and how it's passed through families. Most SETD5 cases are de novo, but not all reports will say this - it depends on whether parental testing was done.

Inheritance patterns you may see

De novo

The change occurred spontaneously in your child - it was not inherited from either parent. This is how most SETD5 cases happen.

Inherited (autosomal dominant)

The change was passed from a parent who also carries it. The parent may have milder or no symptoms - this is rare but documented in SETD5.

Unknown / Not tested

Parental testing hasn't been done yet, so the lab can't determine if it's de novo or inherited. Parental (trio) testing can clarify this.

Zygosity
Heterozygous
+

This describes how many copies of the gene are affected. You have two copies of most genes - one from each parent.

Zygosity options

Heterozygous

One copy is changed, one is working. This is expected for SETD5 Syndrome - the condition is caused by haploinsufficiency, meaning one working copy isn't enough.

Homozygous

Both copies carry the same change. This would be extremely rare for SETD5 and is not typical of this condition.

Hemizygous

Only one copy of the gene exists (relevant for genes on the X chromosome). SETD5 is on chromosome 3, so this term would not typically apply.

ACMG Criteria
PVS1, PM2, PP3
+

These are the specific evidence codes the lab used to arrive at the classification above. Each code represents one piece of evidence. You don't need to understand these - they're primarily for clinicians and genetic counselors. But if you're curious:

PVS1

The variant causes loss of gene function - a strong indicator of pathogenicity for genes where loss-of-function is a known disease mechanism.

PM2

The variant is absent or extremely rare in population databases like gnomAD, meaning it's not a common harmless variation.

PP3

Computer algorithms predict the variant is damaging to the protein. This is supporting evidence, not a standalone determination.

Click any row to expand - fields with multiple options show all possibilities

This is a sample report for educational purposes only. It does not represent any individual's actual genetic test results.


Interpreting your results

What the Classification Means

Pathogenic

This genetic change is known to cause SETD5 Syndrome. There is strong evidence from multiple sources. This is the clearest result.

Likely Pathogenic

Strong evidence that this change causes SETD5 Syndrome, but hasn't been seen in as many people yet. Many clinicians treat a likely pathogenic result similarly to a pathogenic one, but your genetics team will guide how this applies to your child's specific situation.

Variant of Uncertain Significance (VUS)

The lab found a change in SETD5, but there isn't enough evidence yet to say whether it causes the condition or is harmless. A VUS doesn't confirm a diagnosis, but it doesn't rule one out either. It means more information is needed. Classifications can and do change over time as research grows.

Likely Benign / Benign

This change is probably or definitely harmless. Common in the general population and not thought to cause disease.

If you received a VUS result

Some families find the following approaches helpful in working with their genetics team over time: parental testing (to see if either parent carries the same change); enrolling in research registries, which helps researchers gather data — see the Research Registries guide for options open to SETD5-related disorder families; and asking the lab whether reclassification may be possible as new evidence becomes available. Your genetic counselor can advise on what makes sense for your situation.


What happened in the DNA

Types of Genetic Changes

Missense mutation

One letter in the genetic code changed, which changes one building block of the protein. The protein is still made but may not work correctly. Like a typo that changes the meaning of a word.

Nonsense mutation

One letter changed in a way that creates a premature stop signal. The protein gets cut short and usually cannot do its job. Like a sentence that stops in the middle.

Frameshift mutation

A letter was added or deleted, throwing off the reading of everything that follows. The genetic code is read in groups of three, so shifting by one scrambles all the instructions that come after it.

Deletion

A piece of the genetic code is missing. Can be small (a few letters) or large (a section of chromosome). Larger deletions involving neighboring genes alongside SETD5 may cause additional features.

Duplication

A section of genetic code is copied, appearing twice instead of once.

Important note

The type of variant your child has can sometimes give clues about severity, but it is not a reliable predictor. Two people with the exact same variant can look very different. Researchers are still working to understand why.


Variable expression

Why Every Person with SETD5 Syndrome Looks Different

Variable expressivity describes the fact that the same genetic change can cause a wide range of outcomes. Two people in the same family with the identical SETD5 variant can have very different experiences — one might have relatively mild learning differences, while another has more significant support needs across multiple areas.

Genetic background plays a major role. Each person carries roughly 20,000 genes that all interact with one another. These other genes (sometimes called "modifier genes") can influence how SETD5 Syndrome shows up — which is part of why no two children look exactly alike. Your child's unique combination of genes from both parents shapes their outcome in ways researchers are still working to understand.

Variant type and location matter, but not in a simple way. Where in the SETD5 gene the change occurs can affect how much protein is made and how it functions. But the relationship between variant location and severity is not yet well-understood. This is an active area of research.


Where the research lives

A Guide to Genetics Databases

These are the databases researchers, genetic counselors, and clinicians use. You do not need to use them. But if you want to look up your child's specific variant, here is what each one does.

Databases You May Already Know

Additional Databases Worth Knowing


Support available

Getting Help Understanding Your Report

Free genetic counseling available

At the time this page was last updated, families enrolled in Simons Searchlight could access free genetic counseling. Many families find this to be a helpful resource. A genetic counselor can walk you through your child's specific report and answer questions that no website can. Check the Simons Searchlight website for current availability.

Want a broader overview? For a comprehensive look at what SETD5 Syndrome is and how it affects development, see our Understanding SETD5 Syndrome page.


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