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Genome vs. Exome Analysis for Rare Disease Diagnosis: What Every Provider Should Know

Written by Renée Pelletier, MS, CGC | Jan 29, 2026 2:55:59 AM


When a patient has a suspected rare or genetic condition, timing is everything. Moving from clinical uncertainty to a definitive answer can fundamentally shift care plans, prevent "diagnostic odysseys" and unnecessary testing, and provide families with much-needed clarity.

Today, leading medical organizations—including the American College of Medical Genetics and Genomics (ACMG)1, the American Academy of Pediatrics (AAP)2, and the National Society of Genetic Counselors (NSGC)3—recommend exome and genome sequencing early in the diagnostic journey for many clinical presentations. But for some providers, choosing the right path can feel complex.

 What Sets Exome/Genome Testing Apart from Other Genetic Tests

Traditionally,  providers relied on single- or multi-gene panels or chromosomal microarrays (CMA) as a first step. While useful, these strategies can often miss the underlying cause of complex conditions, as they are limited to analyzing a small portion of a person’s genetic material. Both genome and exome sequencing have demonstrated higher diagnostic yields for complex presentations like intellectual disability/developmental delay (ID/DD) and epilepsy1,4-6—making them more likely to lead to a clear diagnosis.

The key difference between the two is scope:

  • Exome sequencing analyzes the coding regions (exons) of genes (where many known disease-causing variants occur)
  • Genome sequencing analyzes the entire genome, including non-coding regions (introns), allowing for the ability to identify intronic changes and certain structural variations in the DNA. 

When to Consider Diagnostic Exome/Genome Testing

According to leading medical organizations1-3, if your patient presents with any of the following, exome or genome sequencing should be considered as a first-line test:

  • Developmental delay
  • Intellectual disability
  • Multiple congenital anomalies
  • Idiopathic epilepsy/seizures not explained by trauma, infection, or stroke

Sequencing can also help diagnose rare or unexplained conditions associated with:

  • Failure to thrive or growth concerns
  • Abnormal muscle tone (low or high tone)
  • Autism spectrum disorder
  • Cerebral palsy
  • Significant hearing or vision differences
  • Movement differences such as hypotonia, dystonia, or spasticity

Comprehensive exome or genome sequencing can be especially valuable when a patient has already been evaluated by multiple specialists, had a previous negative genetic test, or when their healthcare provider has concern about a possible genetic disorder, metabolic condition, or mitochondrial disease. In these cases, exome or genome sequencing can provide a broader view to help identify (or rule out) known genetic causes.

How to Choose Between Exome and Genome Sequencing

In general, genome sequencing is the most comprehensive option and typically offers the highest diagnostic yield. However, practical factors such as insurance coverage can often be the deciding factor—some state Medicaid programs and commercial plans are more likely to cover exome sequencing than genome sequencing.

If genome sequencing isn’t covered under your patient’s plan, exome sequencing is often the best place to start to move the diagnostic process forward quickly. And if exome results are negative or inconclusive, genome sequencing can be ordered next for a more complete analysis.

How MyOme Makes Exome and Genome Testing More Flexible

Most laboratories treat Exome and Genome as two entirely different workflows. MyOme makes it easier to choose between exome and genome testing by running both tests on a whole-genome backbone, even when an exome is ordered. This delivers three key benefits: (1) stronger exome performance and (2) built-in features to boost detection, and (3) simplified reflex workflows.

  1. Stronger exome performance: Traditional exomes use "capture" methods that can leave gaps. By using a genome backbone, we provide more consistent coverage across those tricky coding regions7. This allows MyOme to detect rare variants, Copy Number Variants (CNVs), and Tandem Repeat Expansions (TREs) that traditional exomes often miss, increasingthe likelihood of identifying a diagnostic finding.
  2. Built-in Advanced Detection: We automatically include analysis that usually requires separate orders:
    • CNVs (Copy Number Variants): Harder to see in traditional exomes; clear in genome-backed data.
    • TREs (Tandem Repeat Expansions): Critical for conditions like Fragile X or Ataxias.
    • Mitochondrial Analysis: Assessment for mitochondrial SNVs  is standard
  3. Simplified upgrade from exome to genome: If exome results are negative or inconclusive, you can “reflex” to a full genome analysis That means no restarting the process, no new sample, and no re-sequencing needed. We simply analyze the existing data further, often saving time and reducing cost.

The Bottom Line

If a patient has developmental delay, intellectual disability, congenital anomalies, epilepsy, or other unexplained neurologic or systemic findings, exome and genome testing should be considered early for an efficient path to answers.

  • Exome testing is often the first step due to coverage and accessibility
  • Genome testing offers the most comprehensive view and can capture more variant types
  • A whole-genome backbone approach can help you get the benefits of both—without forcing you to choose between flexibility and depth

References

  1. Manickam K, et al. Genet Med. 2021;23(11):2029-2037(ES). 
  2. Lance H. Rodan, Joan Stoler, Emily Chen, Timothy Geleske, Council on Genetics; Genetic Evaluation of the Child With Intellectual Disability or Global Developmental Delay: Clinical Report. Pediatrics July 2025; 156 (1): e2025072219. 10.1542/peds.2025-072219
  3. Smith L, et al. J Genet Couns. 2023 Apr;32(2):266-280.
  4. Mellone S, Puricelli C, Vurchio D, et al. Front Genet. 2022 Aug 11;13:875182. doi: 10.3389/fgene.2022.875182 
  5. Spataro N, Trujillo-Quintero JP, Manso C, et al. Genes (Basel). 2023 Mar 13;14(3):708. doi: 10.3390/genes14030708.
  6. Pekeles H, Accogli A, Boudrahem-Addour N, Russell L, Parente F, Srour M. Pediatr Neurol. 2019 Mar;92:32-3
  7. Meienberg, Janine, et al. "Clinical sequencing: is WGS the better WES?." Human genetics 135 (2016): 359-362.