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Variant interpretation report

NOD2 c.2938dup, p.Leu980ProfsTer2

NM_001370466.1:c.2938dup NP_001357395.1:p.(Leu980ProfsTer2) Frameshift · exon 11 View identifiers
Classification
Pathogenic

Variant description:

The NOD2 NM_001370466.1:c.2938dup (NP_001357395.1:p.(Leu980ProfsTer2)) frameshift variant has not previously been reported in somatic cancers (COSMIC). This variant is predicted to result in an absent or disrupted protein product. Based on available guidelines, this variant is considered to be Pathogenic.

Five-step scale from benign to pathogenic. This variant is pathogenic. Select a step to read what it means.

Pathogenic.A pathogenic classification under the recorded framework.

In plain language

Gene-level background from the recorded interpretation.

About NOD2

NOD2 encodes an immune-system protein that recognizes components of bacterial cell walls and helps activate inflammatory signaling. Changes in NOD2 are associated with Crohn disease and Blau syndrome. NOD2 is not known to have a specific established role in cancer.

LYFE Sciences is an AI system, and it can make mistakes. Criteria may be applied incorrectly, sources may be misread, and a confident-looking classification can still be wrong. Double-check every criterion and its underlying evidence before relying on any call.
01The reasoning

How it was classified

The recorded framework is Generic ACMG/AMP 2015 with ClinGen SVI recommendations. The criteria and strengths shown below come from the saved assessment; this page does not recalculate the classification.

Recorded applied criteria

The applied criteria and their strengths are shown as assessed under the stated framework.

Pathogenic evidence criteria: 2 Benign evidence criteria: 0
Pathogenic criteria applied · 2
PVS1Strong
PVS1 · strong

Met at strong: the p.(L980Pfs*2) frameshift has a stop 32 nucleotides before the exon-11/exon-12 junction, triggering the NMD-escape downgrade.

PS4Strong
PS4 · strong

Met: homozygous genotype relative risk 42.1 with 95% CI 4.3–infinity exceeds the PS4 >5 threshold and excludes 1 in Crohn disease cases versus controls.

Benign criteria applied · 0

No benign criterion marked as applied in the recorded assessment.

Recorded classification: Pathogenic

Pathogenic: PVS1 strong plus PS4 strong satisfy the generic ACMG/AMP rule requiring two strong pathogenic criteria.

02The sources

Evidence at a glance

Evidence summaries and captured source records, where available. Missing entries should not be interpreted as negative findings.

gnomAD v4.1
gnomAD v2.1
Population frequency
29,257 / 1,613,186 alleles

gnomAD v4.1: 29,257 / 1,613,186 alleles. Recorded AF 0.0181362. Recorded homozygote count 607.

gnomAD v2.1

This variant is present in gnomAD v2.1 (AF= 0.0152001; MAF= 1.52001%, 4298/282762 alleles, homozygotes = 115) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.0342532; MAF= 3.42532%, 355/10364 alleles, homozygotes = 16); grpmax FAF= 0.0239652.

gnomAD-Canada

This variant is present in gnomAD-Canada v1.0 (AF= 0.01857282502443793, 342/18414 alleles, homozygotes = 3).

SpliceAI score
0.055 maximum delta

Recorded SpliceAI maximum delta score: 0.055. Refer to the recorded PP3/BP4 rationale for interpretation.

No listed criterion applied SpliceAI ↗
ClinVar submissions
16 reported laboratory submissions

6 Uncertain significance · 3 Likely benign · 2 Likely Pathogenic · 1 Benign · 1 association · 1 Pathogenic · 1 Likely risk allele · 1 Established risk allele. ClinVar API review status: criteria provided, conflicting classifications.

Context only ClinVar ↗
No screenshot for this evidence
Functional evidence
Not recorded

No standalone functional evidence summary is recorded in evidence_summary.functional.

No listed criterion applied OncoKB ↗
COSMIC
Cancer Hotspots
Somatic evidence
No matching record reported

This variant has not previously been reported in somatic cancers (COSMIC).

Cancer Hotspots

This variant does not lie in a statistically significant hotspot.

For specialists

Technical detail

ACMG/AMP criteria under Generic ACMG/AMP 2015 with ClinGen SVI recommendations, with the data and sources behind each one.

28 criteria with recorded statuses: 2 met · 4 not met · 12 not assessed · 10 not applicable. Open any row for the full reasoning, the rule and the evidence behind it.

Met Not met Not assessed Not applicable

PS2, PM6, PP1, and BS4 are not assessed because the case lacks a documented proband-parent trio, affected-relative genotypes and phenotypes, informative meioses, or exact-variant family segregation results.

Reviewed NOD2 family studies concern different variants and cannot be transferred to NM_001370466.1:c.2938dup.

Neither PS3 nor BS3 is assessed because the available functional literature concerns different NOD2 variants and does not provide direct validated functional evidence for c.2938dup/p.(Leu980ProfsTer2).

PS4 is met from a verified case-control study of the same genomic NOD2 insertion allele: homozygous Crohn disease cases had reported genotype relative risk 42.1 (95% CI 4.3–infinity), satisfying RR >5 with a confidence interval excluding 1.

PP4 and BP5 were not assessed because the available record lacks the patient-level phenotype/family-history specificity and alternate-molecular-basis documentation required by their definitions.

PP5 and BP6 were not met because ClinVar variation 4691 has no expert-panel submission; ordinary laboratory assertions and aggregate classifications are excluded.

Neither PM3 nor BP2 can be assessed because the supplied evidence lacks a pathogenic partner allele and family-level phase information.

Population evidence does not support BA1 or PM2: the variant is common in the specified gnomAD control datasets, but its required grpmax FAF remains below 0.05.

BS1 requires disorder-specific maximum-credible-frequency assessment because NOD2/Crohn disease is a susceptibility phenotype with ancestry-related frequency enrichment and incomplete penetrance; BS2 cannot be established from gnomAD homozygote counts without healthy-adult and penetrance data.

Met · 2

PVS1 StrongACMG_2015Met at strong: the p.(L980Pfs*2) frameshift has a stop 32 nucleotides before the exon-11/exon-12 junction, triggering the NMD-escape downgrade. Details ▾Hide ▴
Reasoning

NM_001370466.1:c.2938dup is a frameshift in the MANE Select NOD2 transcript, producing NP_001357395.1:p.(L980Pfs*2). NOD2 loss of function is supported as a germline disease mechanism in the case evidence. The variant is in exon 11, and the predicted premature termination occurs 32 nucleotides before the exon-11/exon-12 junction, placing it in the generic ClinGen SVI NMD-escape region and triggering a one-tier downgrade from very strong to strong. The truncation affects the C-terminal leucine-rich-repeat region rather than an established non-critical terminal segment, so no additional non-critical-exon or distal-region downgrade is applied. Direct RNA evidence of transcript decay or escape was not available, so the inferred NMD behavior warrants human review.

Rule
PVS1: Null variant (nonsense, frameshift, canonical +/−1 or 2 splice sites, initiation codon, single or multi-exon deletion) in a gene where loss of function (LOF) is a known mechanism of disease.
Evidence considered
  • Variant normalization: NM_001370466.1:c.2938dup produces NP_001357395.1:p.(L980Pfs*2) / p.(Leu980ProfsTer2).
  • Variant annotation identifies a frameshift_variant in NOD2 transcript variant 3, which is marked MANE Select and RefSeq Select.
  • Transcript exon coordinates place c.2938dup in exon 11 (c.2886-c.2969), with exon 12 beginning at c.2970; the predicted termination is therefore 32 nucleotides before the downstream exon junction.
  • The case-level NOD2 gene-context assessment records loss of function as an eligible germline disease mechanism under the generic PVS1 framework.
  • The ClinGen SVI PVS1 framework is the governing generic framework for evaluating frameshift variants and includes downgrade consideration for predicted NMD escape or a non-critical distal region.
Still missing
  • No direct RNA or protein assay confirms whether the transcript escapes nonsense-mediated decay.
  • The functional criticality of the affected C-terminal leucine-rich-repeat segment is inferred from transcript/domain context rather than a variant-specific assay.
Sources
PS4 StrongACMG_2015Met: homozygous genotype relative risk 42.1 with 95% CI 4.3–infinity exceeds the PS4 >5 threshold and excludes 1 in Crohn disease cases versus controls. Details ▾Hide ▴
Reasoning

The Hampe et al. case-control study evaluated the same recurrent NOD2 exon-11 C-insertion allele represented for this case as NC_000016.9:g.50763781dup / chr16:50729867 G>GC (rs2066847; alternate transcript nomenclature NM_022162.3:c.3019dup, p.Leu1007fs). Among sporadic Crohn disease cases, 20/304 were homozygous compared with 0/272 controls; the study reports genotype relative risk 42.1 with 95% CI 4.3–infinity, which excludes 1. For all carriers, the extracted counts were 77/304 cases versus 24/272 controls; I computed the carrier OR as 3.51 (95% CI 2.14–5.73), below the >5 threshold, but the prespecified PS4 rule permits RR or OR and the homozygous genotype relative risk meets the required >5 and CI-not-including-1 condition. The literature_pass packet had no ai_classified_papers, so there were no classifier PS4 leads requiring separate verification; the full text of PMID:11425413 was directly verified.

Rule
The prevalence of the variant in affected individuals is significantly increased compared to the prevalence in controls. ... Relative risk (RR) or odds ratio (OR), as obtained from case-control studies, is >5.0 and the confidence interval around the estimate of RR or OR does not include 1.0.
Evidence considered
  • Hampe et al. 2001, Table 2: 20/304 Crohn disease cases were homozygous for the exon-11 C-insertion versus 0/272 controls; reported genotype relative risk was 42.1 (95% CI 4.3–infinity).
  • Hampe et al. 2001, Table 2: carriers were 77/304 cases (57 heterozygotes plus 20 homozygotes) versus 24/272 controls; computed carrier OR = 3.51, 95% CI 2.14–5.73.
  • The paper's exon-11 NOD2 C-insertion is the recurrent allele represented in this case by NC_000016.9:g.50763781dup / chr16:50729867 G>GC and ClinVar variation 4691 (rs2066847; alternate transcript NM_022162.3:c.3019dup).
  • The literature_pass packet contains ai_classified_papers: []; no classifier-derived PS4 assertion was used.
Sources

Not met · 4 · evaluated, threshold not reached

PM2 ACMG_2015Not met: observed control frequencies are 0.0146597–0.0181362, far above the <=0.0001 PM2 threshold. Details ▾Hide ▴
Reasoning

PM2 is not met. The generic supporting threshold supplied for this case is <=0.0001, whereas the specified population sources show the variant at AF 0.0146597 in gnomAD v2.1 non-cancer exomes, AF 0.014827 in gnomAD v3.1 non-cancer genomes, and AF 0.0181362 in gnomAD v4.1 all-comers. These frequencies are far above the PM2 threshold and the variant is not absent from controls.

Rule
Absent from controls (or at extremely low frequency if recessive) in Exome Sequencing Project, 1000 Genomes or Exome Aggregation Consortium.
Evidence considered
  • gnomAD v2.1 non-cancer exomes: AF 0.0146597, 3472/236840 alleles.
  • gnomAD v3.1 non-cancer genomes: AF 0.014827, 2193/147906 alleles.
  • gnomAD v4.1 all-comers: AF 0.0181362, 29257/1613186 alleles.
  • The generic PM2 supporting threshold supplied for this case is <=0.0001.
Sources
PP5 ACMG_2015Not met: ClinVar reports zero expert-panel submissions, so conflicting laboratory and aggregate classifications cannot trigger PP5. Details ▾Hide ▴
Reasoning

PP5 is not met because ClinVar variation 4691 has no expert-panel submissions; the record contains conflicting ordinary laboratory submissions and aggregate classifications, which are explicitly excluded from triggering PP5.

Rule
Reputable source recently reports variant as pathogenic, but the evidence is not available to the laboratory to perform an independent evaluation.
Evidence considered
  • ClinVar variation 4691 submission audit: expert_panel_submissions = 0; review status is criteria provided, conflicting classifications, with ordinary clinical-laboratory submissions only.
Sources
BA1 ACMG_2015Not met: required grpmax FAF values are 0.02141027–0.0239652, all below the 0.05 BA1 threshold. Details ▾Hide ▴
Reasoning

BA1 is not met. The governing default threshold is >=0.05, and the required filtering frequency is the grpmax FAF rather than a popmax point estimate. In the specified non-cancer exome-only gnomAD v2.1 data, grpmax FAF is 0.0239652; in gnomAD v4.1 all-comers, grpmax FAF is 0.02141027; and in gnomAD v3.1 non-cancer genomes, grpmax FAF is 0.02212918. All are below 0.05. The high Ashkenazi Jewish popmax point estimates do not replace the required grpmax FAF metric.

Rule
Allele frequency is above 5% in Exome Sequencing Project, 1000 Genomes or Exome Aggregation Consortium.
Evidence considered
  • gnomAD v2.1 non-cancer exomes: 3472/236840 alleles, AF 0.0146597, homozygotes 103, grpmax FAF 0.0239652.
  • gnomAD v4.1 all-comers: 29257/1613186 alleles, AF 0.0181362, homozygotes 607, grpmax FAF 0.02141027.
  • gnomAD v3.1 non-cancer genomes: 2193/147906 alleles, AF 0.014827, homozygotes 28, grpmax FAF 0.02212918.
  • The generic BA1 threshold supplied for this case is >=0.05; the required metric is filtering grpmax FAF, not a population point estimate.
Sources
BP6 ACMG_2015Not met: ClinVar reports zero expert-panel submissions, so ordinary laboratory benign assertions cannot trigger BP6. Details ▾Hide ▴
Reasoning

BP6 is not met because ClinVar variation 4691 has no expert-panel submissions; ordinary laboratory benign submissions and aggregate ClinVar labels are not sufficient under the stated BP6 rule.

Rule
Reputable source recently reports variant as benign, but the evidence is not available to the laboratory to perform an independent evaluation.
Evidence considered
  • ClinVar variation 4691 submission audit: expert_panel_submissions = 0; benign and likely-benign entries are ordinary clinical-laboratory submissions, not expert-panel classifications.
Sources

Not assessed · 12 · no assessment recorded

PS2 ACMG_2015Not assessed: no documented de novo occurrence with confirmed maternity and paternity is available for NM_001370466.1:c.2938dup. Details ▾Hide ▴
Reasoning

No proband-level de novo observation is documented, and there is no evidence that both maternity and paternity were confirmed. The available literature concerns other NOD2 variants and does not establish de novo occurrence of NM_001370466.1:c.2938dup.

Rule
De novo (both maternity and paternity confirmed) in a patient with the disease and no family history.
Evidence considered
  • The case evidence contains no proband pedigree, parental testing results, or documented confirmed maternity and paternity for NM_001370466.1:c.2938dup.
  • The reviewed papers describe other NOD2 variants and do not report de novo occurrence of NM_001370466.1:c.2938dup.
Still missing
  • A documented affected proband carrying NM_001370466.1:c.2938dup is needed.
  • Both maternity and paternity must be confirmed for the proband.
  • Family history and parental variant results are not documented.
PS3 ACMG_2015Not assessed: available functional assays studied different NOD2 variants, and no direct validated assay result for c.2938dup (p.(Leu980ProfsTer2)) was identified. Details ▾Hide ▴
Reasoning

No well-established functional assay of NOD2 c.2938dup (p.(Leu980ProfsTer2)) was identified. The available functional studies evaluated other NOD2 variants, especially the distinct L1007fs/3020insC frameshift, and their results cannot be transferred to this variant without direct assay evidence; therefore PS3 is not assessed.

Rule
Well-established in vitro or in vivo functional studies supportive of a damaging effect on the gene or gene product.
Evidence considered
  • PMID:11425413, Methods and Table 3: the reported exon 11 C-insertion produces a premature stop at amino acid 1007 and is not c.2938dup/p.(Leu980ProfsTer2); the study reports association data rather than a direct functional assay of c.2938dup.
  • PMID:12673278, Introduction and Results: functional context concerns the distinct 3020insC/SNP13 frameshift, not c.2938dup.
  • PMID:15024686, Introduction: the functional statement concerns the common 1007fs mutation and other NOD2 variants, not c.2938dup.
  • PMID:22684479, Results: assays of NOD2-L1007fs showed altered vimentin interaction, localization, autophagy, and bacterial survival, but the tested variant was not c.2938dup/p.(Leu980ProfsTer2).
Still missing
  • A validated in vitro or in vivo assay directly testing NOD2 c.2938dup (p.(Leu980ProfsTer2)) is needed.
  • Assay controls, reproducibility, and biological relevance for the exact variant are not available.
Sources
PM3 ACMG_2015Not assessed: no parent or offspring testing establishes c.2938dup in trans with a pathogenic allele in a recessive disorder. Details ▾Hide ▴
Reasoning

The available case evidence does not provide parent or offspring testing, affected-proband observations, or any other phasing result showing NM_001370466.1:c.2938dup in trans with a pathogenic variant. The inheritance context for applying a recessive PM3 assessment is also not established in the supplied evidence.

Rule
For recessive disorders, detected in trans with a pathogenic variant ... This requires testing of parents (or offspring) to determine phase.
Evidence considered
  • The case evidence identifies NOD2 c.2938dup as a frameshift variant but contains no documented parent/offspring genotypes, phase result, affected-proband compound-heterozygous observation, or pathogenic allele in trans.
  • Population homozygote counts do not establish phase with a pathogenic variant in an affected proband and therefore do not satisfy PM3.
Still missing
  • Documented recessive disease context for NOD2 c.2938dup
  • Parent or offspring testing establishing phase
  • Affected-proband observation with a second pathogenic allele in trans
PM6 ACMG_2015Not assessed: no apparently de novo occurrence without confirmed maternity and paternity is documented for this variant. Details ▾Hide ▴
Reasoning

No assumed de novo occurrence is documented for NM_001370466.1:c.2938dup. The available evidence does not identify an affected proband with an apparently absent parental variant or provide parental testing information.

Rule
Assumed de novo, but without confirmation of paternity and maternity.
Evidence considered
  • The case evidence contains no proband-level parental testing results or documented assumed de novo finding for NM_001370466.1:c.2938dup.
  • The reviewed literature does not report an assumed de novo occurrence of this exact variant.
Still missing
  • A documented affected proband carrying NM_001370466.1:c.2938dup is needed.
  • Parental variant results or a documented assumption of de novo status are needed.
  • Family history is not documented.
PP1 ACMG_2015Not assessed: zero informative affected-relative meioses or exact-variant segregation results are documented for NM_001370466.1:c.2938dup. Details ▾Hide ▴
Reasoning

No affected family members carrying NM_001370466.1:c.2938dup and no meioses or segregation analysis for this exact variant are documented. The reviewed NOD2 literature concerns other variants, including 3020insC/1007fs, and therefore cannot establish co-segregation for c.2938dup; one reviewed study describes Crohn disease as a multifactorial trait.

Rule
Co-segregation with disease in multiple affected family members in a gene definitively known to cause the disease. ... May be used as stronger evidence with increasing segregation data.
Evidence considered
  • Bairead et al. (PMID:12673278) studied NOD2 SNP13/3020insC in Irish Crohn disease families; the paper explicitly concerns that different variant, not NM_001370466.1:c.2938dup.
  • Rosenstiel et al. (PMID:15024686) studied CARD15/NOD2 variants including 1007fs in Jewish Crohn disease families; the paper states that Crohn disease is a multifactorial trait and does not report segregation of NM_001370466.1:c.2938dup.
  • Neither reviewed paper provides informative meioses or affected-relative co-segregation data for NM_001370466.1:c.2938dup.
Still missing
  • The disease phenotype and pedigree structure for the case are not documented.
  • Genotypes and phenotypes of affected relatives are not documented.
  • Informative meioses and exact-variant segregation results are not available.
Sources
PP4 ACMG_2015Not assessed: the patient's phenotype and family history are insufficient to establish a highly specific phenotype for a single genetic etiology. Details ▾Hide ▴
Reasoning

The available case evidence does not provide the patient's phenotype or family history in sufficient detail to determine whether it is highly specific for a single-gene disease. NOD2-associated Crohn disease susceptibility is not, by itself, a highly specific phenotype with a single genetic etiology.

Rule
Patient’s phenotype or family history is highly specific for a disease with a single genetic etiology.
Still missing
  • Patient-level phenotype and family-history details are needed to assess specificity for a single genetic etiology.
BS1 ACMG_2015Not assessed: grpmax FAF is 0.02141027, but reduced/incomplete penetrance and missing maximum-credible-frequency inputs prevent using the 1% default alone. Details ▾Hide ▴
Reasoning

BS1 is not assessed from the generic 1% default alone. The required filtering frequencies exceed 1% (grpmax FAF 0.0239652 in non-cancer gnomAD v2.1 exomes, 0.02212918 in non-cancer gnomAD v3.1 genomes, and 0.02141027 in gnomAD v4.1 all-comers), but NOD2-associated Crohn disease is a susceptibility phenotype with ancestry-related enrichment and incomplete penetrance rather than an established fully penetrant early-onset Mendelian disorder. No case-specific maximum-credible-frequency calculation with quantified prevalence, penetrance, allelic heterogeneity, and inheritance inputs is available, so BS1 cannot be applied solely because the frequency exceeds 1%.

Rule
Allele frequency is greater than expected for the disorder.
Evidence considered
  • Required filtering frequencies are above the generic 0.01 default: gnomAD v2.1 non-cancer exome grpmax FAF 0.0239652, gnomAD v3.1 non-cancer genome grpmax FAF 0.02212918, and gnomAD v4.1 all-comers grpmax FAF 0.02141027.
  • NOD2 variation is associated with Crohn disease susceptibility, and the frequency pattern includes Ashkenazi Jewish enrichment; this does not establish full penetrance or provide a disorder-specific maximum credible allele frequency.
  • The generic BS1 default supplied for this case is >=0.01; a disorder-specific maximum-credible-frequency calculation requires quantified prevalence, penetrance, allelic heterogeneity, and inheritance inputs that are not available.
Still missing
  • A quantified maximum-credible-frequency calculation using disorder prevalence, penetrance, allelic heterogeneity, and inheritance assumptions is needed before applying BS1.
Sources
BS2 ACMG_2015Not assessed: gnomAD reports 28–607 homozygotes, but healthy-adult status and full early-age penetrance are not established. Details ▾Hide ▴
Reasoning

BS2 is not assessed. The population datasets report homozygotes (103 in gnomAD v2.1 non-cancer exomes, 28 in gnomAD v3.1 non-cancer genomes, and 607 in gnomAD v4.1 all-comers), but those records do not establish that the individuals were healthy adults, clinically unaffected for Crohn disease, or observed in a disorder expected to be fully penetrant at an early age. Population homozygote counts alone therefore do not satisfy the BS2 definition.

Rule
Observed in a healthy adult individual for a recessive (homozygous), dominant (heterozygous), or X-linked (hemizygous) disorder, with full penetrance expected at an early age.
Evidence considered
  • gnomAD v2.1 non-cancer exomes reports 103 homozygotes; gnomAD v3.1 non-cancer genomes reports 28 homozygotes; gnomAD v4.1 all-comers reports 607 homozygotes.
  • gnomAD allele-count records do not provide the clinical phenotype, age, or penetrance status needed to classify each homozygote as a healthy adult for BS2.
  • The available NOD2/Crohn disease context is susceptibility-associated rather than evidence that this specific variant causes a fully penetrant early-onset disorder.
Still missing
  • Healthy-adult clinical status, age, and penetrance information for the reported homozygotes is unavailable.
Sources
BS3 ACMG_2015Not assessed: no direct validated assay showed normal protein function or splicing for NOD2 c.2938dup (p.(Leu980ProfsTer2)). Details ▾Hide ▴
Reasoning

No well-established functional assay of NOD2 c.2938dup (p.(Leu980ProfsTer2)) demonstrating no damaging effect on protein function or splicing was identified. The available studies evaluated other NOD2 variants, especially the distinct L1007fs/3020insC frameshift, so they cannot establish BS3 for this variant; therefore BS3 is not assessed.

Rule
Well-established in vitro or in vivo functional studies show no damaging effect on protein function or splicing.
Evidence considered
  • PMID:11425413, Methods and Table 3: the reported exon 11 C-insertion produces a premature stop at amino acid 1007 and is not c.2938dup/p.(Leu980ProfsTer2); no benign functional result for c.2938dup is reported.
  • PMID:12673278, Introduction and Results: the functional context concerns the distinct 3020insC/SNP13 frameshift, not c.2938dup.
  • PMID:15024686, Introduction: the functional statement concerns the common 1007fs mutation and other NOD2 variants, not c.2938dup.
  • PMID:22684479, Results: assays of NOD2-L1007fs reported abnormal functional findings and did not test c.2938dup/p.(Leu980ProfsTer2); they provide no evidence for BS3.
Still missing
  • A validated in vitro or in vivo assay directly testing normal protein function or splicing for NOD2 c.2938dup (p.(Leu980ProfsTer2)) is needed.
  • Assay controls, reproducibility, and biological relevance for the exact variant are not available.
Sources
BS4 ACMG_2015Not assessed: no affected relatives with documented genotypes are available to demonstrate lack of segregation for this variant. Details ▾Hide ▴
Reasoning

No affected family members have been documented for this case, and there are no genotype results showing that NM_001370466.1:c.2938dup fails to segregate with disease. Therefore lack of segregation cannot be evaluated, including the possibility of phenocopies or more than one pathogenic variant in a family.

Rule
Lack of segregation in affected members of a family.
Evidence considered
  • The case evidence contains no affected-relative genotype/phenotype dataset or family segregation analysis for NM_001370466.1:c.2938dup.
  • The available literature does not provide a non-segregation observation for this exact variant in an affected family.
Still missing
  • Affected relatives and their clinical phenotypes are not documented.
  • Genotypes of affected relatives are not documented.
  • Potential phenocopies and additional familial pathogenic variants cannot be evaluated.
BP2 ACMG_2015Not assessed: no family-level phase data show c.2938dup in trans with a dominant pathogenic allele or in cis with any pathogenic allele. Details ▾Hide ▴
Reasoning

The available case evidence does not document NM_001370466.1:c.2938dup in trans with a pathogenic variant in a fully penetrant dominant disorder, or in cis with a pathogenic variant in any inheritance pattern. No family-level phase data are supplied.

Rule
Observed in trans with a pathogenic variant for a fully penetrant dominant gene/disorder or observed in cis with a pathogenic variant in any inheritance pattern.
Evidence considered
  • The case evidence contains no family-level phasing, cis/trans observation, or affected-proband report pairing NOD2 c.2938dup with another pathogenic variant.
  • Population database observations, including homozygotes, do not establish cis or trans phase with a separate pathogenic variant and do not satisfy BP2.
Still missing
  • Parent, offspring, or other family testing establishing phase
  • A documented pathogenic variant paired with c.2938dup
  • Disease inheritance and penetrance context for any trans observation
BP5 ACMG_2015Not assessed: no patient-level evidence identifies an alternate molecular basis for the disease in a case carrying this variant. Details ▾Hide ▴
Reasoning

No case-level evidence in the available materials establishes that this variant was found in a patient with an alternate molecular basis for disease. Population frequency and conflicting ClinVar submissions do not satisfy BP5.

Rule
Variant found in a case with an alternate molecular basis for disease.
Still missing
  • A documented case with this variant and an independently established alternate molecular diagnosis is required.
Not applicable · 10· recorded as not applicable Show reasons ▾Hide reasons ▴
PS1 N/ANot applicable: PS1 requires a missense variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

PS1 (Richards et al. 2015, PMID:25741868) requires the same amino acid change as a previously established pathogenic variant, evaluated regardless of the underlying nucleotide change. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, no altered amino acid exists at this position to compare against any previously established pathogenic missense variant, so PS1's structural prerequisite is not met by this variant class. No VCEP criteria specification covers PS1 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_missense
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules
PM1 N/ANot applicable: PM1 requires a missense variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

PM1 (Richards et al. 2015, PMID:25741868) applies to a missense variant located in a mutational hot spot and/or a critical, well-established functional domain without benign variation. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, no altered residue exists to evaluate for mutational hot-spot or critical-domain membership, so PM1's structural prerequisite is not met by this variant class. No VCEP criteria specification covers PM1 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_missense
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules
PM4 N/ANot applicable: PM4 requires a protein-length change, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

PM4 (Richards et al. 2015, PMID:25741868) applies to protein length changes: in-frame insertions/deletions in a non-repetitive region, or stop-loss variants that extend the protein past the reference stop codon. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, no change in protein length occurs, so there is nothing for this criterion to evaluate, so PM4's structural prerequisite is not met by this variant class. No VCEP criteria specification covers PM4 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_stop_loss_or_nonrepeat_indel
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
pvs1_generic_framework ↗generic_acmg_combination_rules
PM5 N/ANot applicable: PM5 requires a missense variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

PM5 (Richards et al. 2015, PMID:25741868) requires a novel missense change at an amino acid residue where a different missense change has previously been determined to be pathogenic. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, no missense change exists at this residue to compare against a different pathogenic missense change at the same position, so PM5's structural prerequisite is not met by this variant class. No VCEP criteria specification covers PM5 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_missense
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules
PP2 N/ANot applicable: PP2 requires a missense variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

PP2 (Richards et al. 2015, PMID:25741868) applies to a missense variant in a gene with a low rate of benign missense variation, where missense variants are a common mechanism of disease. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, the gene's missense-constraint properties are irrelevant because no missense change is present to evaluate, so PP2's structural prerequisite is not met by this variant class. No VCEP criteria specification covers PP2 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_missense
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules
PP3 N/ANot applicable: c.2938dup is a frameshift (p.L980Pfs*2), outside PP3's missense or intronic/splice-region scope. Details ▾Hide ▴
Reasoning

PP3 is not applicable because NM_001370466.1:c.2938dup is a frameshift variant, predicted as NP_001357395.1:p.(L980Pfs*2). Under the governing scope, PP3 applies only to missense or intronic/splice-region variants, not frameshift or other protein-length-changing variants. The available SpliceAI max delta of 0.055 is therefore not used to assign PP3, and no REVEL score is applicable.

Rule
Multiple lines of computational evidence support a deleterious effect on the gene or gene product (conservation, evolutionary, splicing impact, etc.). ... PP3/BP4 apply only to a missense variant, or to a splice-region/intronic variant via the SpliceAI path - never to a frameshift, nonsense, or other truncating/protein-length-changing variant.
Evidence considered
  • Variant consequence: frameshift_variant, NM_001370466.1:c.2938dup, producing NP_001357395.1:p.(L980Pfs*2).
  • SpliceAI reports max delta 0.055 (displayed as 0.06), but a splice prediction is not used for PP3 when the variant is a frameshift/protein-length-changing variant.
  • REVEL is unavailable and is not an applicable path for this frameshift variant.
Sources
BP1 N/ANot applicable: BP1 requires a missense variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

BP1 (Richards et al. 2015, PMID:25741868) applies to a missense variant in a gene for which primarily truncating variants are known to cause disease. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, the gene's truncating-variant mechanism is irrelevant because no missense change is present to evaluate, so BP1's structural prerequisite is not met by this variant class. No VCEP criteria specification covers BP1 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_missense
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules
BP3 N/ANot applicable: BP3 requires an in-frame indel in a repeat region, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

BP3 (Richards et al. 2015, PMID:25741868) applies to in-frame insertions/deletions located in a repetitive region without a known function. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, the variant does not alter protein length within a repeat region, so there is nothing for this criterion to evaluate, so BP3's structural prerequisite is not met by this variant class. No VCEP criteria specification covers BP3 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_repeat_region_indel
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
pvs1_generic_framework ↗generic_acmg_combination_rules
BP4 N/ANot applicable: c.2938dup is a frameshift (p.L980Pfs*2), so SpliceAI max delta 0.055 cannot support BP4. Details ▾Hide ▴
Reasoning

BP4 is not applicable because NM_001370466.1:c.2938dup is a frameshift variant, predicted as NP_001357395.1:p.(L980Pfs*2). Under the governing scope, BP4 applies only to missense or intronic/splice-region variants, not frameshift or other protein-length-changing variants; a no-impact splice prediction cannot negate the truncating protein effect. The available SpliceAI max delta of 0.055 is therefore not used to assign BP4.

Rule
Multiple lines of computational evidence suggest no impact on gene or gene product (conservation, evolutionary, splicing impact, etc) ... PP3/BP4 apply only to a missense variant, or to a splice-region/intronic variant via the SpliceAI path - never to a frameshift, nonsense, or other truncating/protein-length-changing variant.
Evidence considered
  • Variant consequence: frameshift_variant, NM_001370466.1:c.2938dup, producing NP_001357395.1:p.(L980Pfs*2).
  • SpliceAI reports max delta 0.055 (displayed as 0.06), but BP4 is not applicable to a frameshift variant because no predicted splice impact does not remove the truncating protein effect.
Sources
BP7 N/ANot applicable: BP7 requires a synonymous variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

BP7 (Richards et al. 2015, PMID:25741868) is defined for a synonymous (silent) variant for which splicing prediction algorithms predict no impact to the splice consensus sequence and the nucleotide is not highly conserved. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, the encoded protein sequence is altered, so the criterion's silent-variant premise does not hold regardless of any splicing prediction, so BP7's structural prerequisite is not met by this variant class. No VCEP criteria specification covers BP7 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_synonymous
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules

Identifiers

Gene
NOD2
Transcript
NM_001370466.1
Coding (HGVS c.)
NM_001370466.1:c.2938dup
Protein (HGVS p.)
NP_001357395.1:p.(Leu980ProfsTer2)
GRCh38
chr16:50729867 G>GC
GRCh37
chr16:50763778 G>GC
Location and type
Exon 11 · frameshift
ClinVar
4691

Gene diagram

Every interpreted variant in NOD2, mapped onto the exon structure of NM_001370466.1. Select a row to find it on the plot.

NOD2NM_001370466.1
Fetching transcript structure from UCSC…
gnomAD v4.1 29,257 / 1,613,186 alleles AF 0.0181 · 607 homozygotes · FAF 0.0214
gnomAD v2.1 4,298 / 282,762 alleles AF 0.0152 · 115 homozygotes · FAF 0.024
gnomAD-Canada v1.0 Allele count data not recorded

By genetic ancestry

gnomAD v4.1

AF 0.018 · 29257 / 1,613,186 alleles · 607 hom · FAF 0.021
Ashkenazi Jewish940 / 29,570 0.03234 hom
Amish28 / 912 0.031
European (non-Finnish)25512 / 1,179,300 0.022527 hom
European (Finnish)977 / 63,982 0.01512 hom
Remaining individuals861 / 62,438 0.01425 hom
Admixed American560 / 60,002 0.00936 hom
African/African American222 / 75,020 0.0032 hom
Middle Eastern16 / 6,052 0.0026
South Asian138 / 91,054 0.00151 hom
East Asian3 / 44,856 6.7e-05

gnomAD v2.1

AF 0.015 · 4298 / 282,762 alleles · 115 hom · FAF 0.024
Ashkenazi Jewish355 / 10,364 0.03416 hom
European (non-Finnish)3012 / 129,102 0.02392 hom
European (Finnish)398 / 25,124 0.0165 hom
Remaining individuals114 / 7,224 0.016
Admixed American301 / 35,420 0.00852 hom
African/African American71 / 24,966 0.0028
South Asian47 / 30,614 0.0015
0 alternate alleles recorded in 1 other group: East Asian.

gnomAD-Canada

AF not recorded · ? / ? alleles

Statements

gnomAD v4.1

This variant is present in gnomAD v4.1 (AF= 0.0181362; MAF= 1.81362%, 29257/1613186 alleles, homozygotes = 607) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.031789; MAF= 3.17890%, 940/29570 alleles, homozygotes = 34); grpmax FAF= 0.0214103.

gnomAD v2.1

This variant is present in gnomAD v2.1 (AF= 0.0152001; MAF= 1.52001%, 4298/282762 alleles, homozygotes = 115) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.0342532; MAF= 3.42532%, 355/10364 alleles, homozygotes = 16); grpmax FAF= 0.0239652.

gnomAD-Canada

This variant is present in gnomAD-Canada v1.0 (AF= 0.01857282502443793, 342/18414 alleles, homozygotes = 3).

Open the gnomAD-Canada detail page →

SpliceAI and Pangolin

SpliceAI acceptor lossat +369 bp
0.025
SpliceAI acceptor gainat -20 bp
0.000
SpliceAI donor lossat +34 bp
0.055
SpliceAI donor gainat +220 bp
0.000
Pangolin splice lossmagnitude shown
-0.040
Pangolin splice gain
0.012

Other predictors

REVEL
Not available
BayesDel
Not available

How the predictions were used

PP3 N/ANot applicable: c.2938dup is a frameshift (p.L980Pfs*2), outside PP3's missense or intronic/splice-region scope. Details ▾Hide ▴
Reasoning

PP3 is not applicable because NM_001370466.1:c.2938dup is a frameshift variant, predicted as NP_001357395.1:p.(L980Pfs*2). Under the governing scope, PP3 applies only to missense or intronic/splice-region variants, not frameshift or other protein-length-changing variants. The available SpliceAI max delta of 0.055 is therefore not used to assign PP3, and no REVEL score is applicable.

Rule
Multiple lines of computational evidence support a deleterious effect on the gene or gene product (conservation, evolutionary, splicing impact, etc.). ... PP3/BP4 apply only to a missense variant, or to a splice-region/intronic variant via the SpliceAI path - never to a frameshift, nonsense, or other truncating/protein-length-changing variant.
Evidence considered
  • Variant consequence: frameshift_variant, NM_001370466.1:c.2938dup, producing NP_001357395.1:p.(L980Pfs*2).
  • SpliceAI reports max delta 0.055 (displayed as 0.06), but a splice prediction is not used for PP3 when the variant is a frameshift/protein-length-changing variant.
  • REVEL is unavailable and is not an applicable path for this frameshift variant.
Sources
BP4 N/ANot applicable: c.2938dup is a frameshift (p.L980Pfs*2), so SpliceAI max delta 0.055 cannot support BP4. Details ▾Hide ▴
Reasoning

BP4 is not applicable because NM_001370466.1:c.2938dup is a frameshift variant, predicted as NP_001357395.1:p.(L980Pfs*2). Under the governing scope, BP4 applies only to missense or intronic/splice-region variants, not frameshift or other protein-length-changing variants; a no-impact splice prediction cannot negate the truncating protein effect. The available SpliceAI max delta of 0.055 is therefore not used to assign BP4.

Rule
Multiple lines of computational evidence suggest no impact on gene or gene product (conservation, evolutionary, splicing impact, etc) ... PP3/BP4 apply only to a missense variant, or to a splice-region/intronic variant via the SpliceAI path - never to a frameshift, nonsense, or other truncating/protein-length-changing variant.
Evidence considered
  • Variant consequence: frameshift_variant, NM_001370466.1:c.2938dup, producing NP_001357395.1:p.(L980Pfs*2).
  • SpliceAI reports max delta 0.055 (displayed as 0.06), but BP4 is not applicable to a frameshift variant because no predicted splice impact does not remove the truncating protein effect.
Sources
BP7 N/ANot applicable: BP7 requires a synonymous variant, and this is a frameshift variant. Details ▾Hide ▴
Reasoning

BP7 (Richards et al. 2015, PMID:25741868) is defined for a synonymous (silent) variant for which splicing prediction algorithms predict no impact to the splice consensus sequence and the nucleotide is not highly conserved. NM_001370466.1:c.2938dup in NOD2 is a frameshift variant predicted to produce NP_001357395.1:p.(Leu980ProfsTer2). As a result, the encoded protein sequence is altered, so the criterion's silent-variant premise does not hold regardless of any splicing prediction, so BP7's structural prerequisite is not met by this variant class. No VCEP criteria specification covers BP7 for NOD2, so the generic ACMG/AMP 2015 definition applies. This determination was made deterministically from the variant's consequence class, prior to LLM adjudication.

Rule
deterministic_prefilter: requires_synonymous
Evidence considered
  • Deterministic consequence classification: frameshift (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules

Statement

In silico

SpliceAI predicts no significant splice impact for this variant (max delta score = 0.06).

8 paper records listed. 2 cited in the classification. None of the listed paper records explicitly indicates that the variant is named.

Cited in the classification

  • Association between insertion mutation in NOD2 gene and Crohn's disease in German and British populations.
    Hampe J et al. · 2001 Jun 16
    This study describes a different NOD2 exon 11 C-insertion frameshift producing a premature stop at amino acid 1007 and reports case-control association data; it does not directly assay c.2938dup/p.(Leu980ProfsTer2).
    → PS4 · strong Does not name this variant
    PMID 11425413 ↗
    Source detail
    A C-insertion mutation identified in exon 11 in the region coding for the leucine-rich region leads to a premature stop codon at aminoacid 1007 of the encoded protein.

    Location Methods, paragraph describing mutation selection; Table 3; Discussion

    Context NOD2 coding-region mutation screening and genotyping in affected individuals, families, trios, and normal controls; association analyses for Crohn disease and ulcerative colitis.

  • Standards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology.
    The ACMG/AMP paper provides the PVS1 null-variant framework and notes that frameshift variants can disrupt gene function through absence of gene product or nonsense-mediated decay; it does not mention NOD2 c.2938dup or p.(L980Pfs*2).
    → PVS1 · strong Does not name this variant
    PMID 25741868 ↗
    Source detail
    Certain types of variants (e.g. nonsense, frameshift, canonical +/−1 or 2 splice sites, initiation codon, single exon or multi-exon deletion) can often be assumed to disrupt gene function by leading to complete absence of the gene product by lack of transcription or nonsense-mediated decay of an altered transcript.

    Location Proposed Criteria for Interpretation of Sequence Variants, PVS1 Null variants

    Context ACMG/AMP sequence-variant interpretation guideline; general framework rather than a variant-specific experiment.

Other recorded publications

  • Crohn's disease-associated NOD2 variants share a signaling defect in response to lipopolysaccharide and peptidoglycan.
    Bonen DK et al. · 2003
    No paper-specific finding is recorded.
    PMID 12512038 ↗
  • Association of NOD2 with Crohn's disease in a homogenous Irish population.
    Bairead E et al. · 2003
    The paper does not explicitly mention NOD2 c.2938dup or NP_001357395.1:p.(L980Pfs*2). It reports findings for a different NOD2 frameshift, SNP13/3020insC, which truncates the leucine-rich repeat region and was associated with Crohn’s disease in an Irish cohort; these findings do not establish segregation of the specified c.2938dup variant.
    Does not name this variant
    PMID 12673278 ↗
    Quoted passage
    Following a frameshift mutation (caused by a cytosine insertion and known as 3020insC or single-nucleotide polymorphism (SNP)13) leading to the truncation of the leucine-rich repeat region of NOD2, responsiveness to bacterial lipopolysaccharide (LPS) was observed to be greatly diminished.

    Introduction; Results, ‘NOD2 SNP analysis’ and ‘NOD2 SNPs and predominant disease distribution’

  • Crohn disease: frequency and nature of CARD15 mutations in Ashkenazi and Sephardi/Oriental Jewish families.
    The paper does not explicitly mention NOD2/CARD15 c.2938dup or NP_001357395.1:p.(L980Pfs*2). It studied CARD15/NOD2 variation in Crohn disease families, focusing on other variants including 1007fs, and describes Crohn disease as a multifactorial trait; it does not report segregation of the specified variant.
    Does not name this variant
    PMID 15024686 ↗
    Quoted passage
    The LRR domain of the gene has a binding activity for bacterial peptidoglycans, and mutations in this domain abrogate nuclear factor-kB–mediated signaling (Inohara et al. 1999, 2000; Chamaillard et al. 2002).

    Introduction, page 624

  • Genotyping for NOD2 genetic variants and crohn disease: a metaanalysis.
    No paper-specific finding is recorded.
    PMID 19713276 ↗
  • The intermediate filament protein, vimentin, is a regulator of NOD2 activity.
    Functional assays of the distinct NOD2-L1007fs variant found impaired vimentin interaction, altered localization, defective autophagy response, and impaired bacterial survival control; c.2938dup/p.(Leu980ProfsTer2) was not tested.
    Does not name this variant
    PMID 22684479 ↗
    Quoted passage
    However the NOD2-R702W and NOD2-L1007fs variant proteins seemingly lose their ability to interact with vimentin (Figure 2C).

    Results, ‘The LRR domain of NOD2 is the major determinant for binding with vimentin and common CD-associated polymorphisms disrupt this interaction’

  • Dysfunctional Crohn's Disease-Associated NOD2 Polymorphisms Cannot be Reliably Predicted on the Basis of RIPK2 Binding or Membrane Association.
    No paper-specific finding is recorded.
    PMID 26500656 ↗

Screenshots represent source records captured during evidence collection. Select one to view the recorded source content.

All source links

Statements for reports

The evidence statements recorded by the interpretation pipeline. They have not been reinterpreted by the page.

gnomAD v4.1

This variant is present in gnomAD v4.1 (AF= 0.0181362; MAF= 1.81362%, 29257/1613186 alleles, homozygotes = 607) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.031789; MAF= 3.17890%, 940/29570 alleles, homozygotes = 34); grpmax FAF= 0.0214103.

gnomAD v2.1

This variant is present in gnomAD v2.1 (AF= 0.0152001; MAF= 1.52001%, 4298/282762 alleles, homozygotes = 115) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.0342532; MAF= 3.42532%, 355/10364 alleles, homozygotes = 16); grpmax FAF= 0.0239652.

gnomAD-Canada

This variant is present in gnomAD-Canada v1.0 (AF= 0.01857282502443793, 342/18414 alleles, homozygotes = 3).

ClinVar

This variant has been reported in ClinVar as Uncertain significance (6 clinical laboratories) and as Likely benign (3 clinical laboratories) and as Likely Pathogenic (2 clinical laboratories) and as Benign (1 clinical laboratory) and as association (1 clinical laboratory) and as Pathogenic (1 clinical laboratory) and as Likely risk allele (1 clinical laboratory) and as Established risk allele (1 clinical laboratory). (ClinVarID = 4691)

In silico

SpliceAI predicts no significant splice impact for this variant (max delta score = 0.06).

Somatic

This variant does not lie in a statistically significant hotspot. This variant has not previously been reported in somatic cancers (COSMIC).

Cancer Hotspots

This variant does not lie in a statistically significant hotspot.