About the gene
No gene-level summary was recorded for this case.
No variant summary was recorded for this case.
Five-step scale from benign to pathogenic. This variant is uncertain. Select a step to read what it means.
Uncertain.The recorded classification is uncertain significance.
Gene-level background from the recorded interpretation.
Words with a dotted underline have a short definition. Hover or tap them.No gene-level summary was recorded for this case.
The recorded framework is Not recorded. The criteria and strengths shown below come from the saved assessment; this page does not recalculate the classification.
The applied criteria and their strengths are shown as assessed under the stated framework.
No pathogenic criterion marked as applied in the recorded assessment.
No benign criterion marked as applied in the recorded assessment.
VUS under ACMG/AMP 2015 combination rules: with no criterion met or applied, neither a pathogenic nor a benign classification threshold was reached.
Evidence summaries and captured source records, where available. Missing entries should not be interpreted as negative findings.
gnomAD v4.1 allele counts were not recorded for this variant.
No SpliceAI or REVEL score is recorded in the fields used for this tile.
A ClinVar laboratory submission summary was not recorded.
No standalone functional evidence summary is recorded in evidence_summary.functional.
No COSMIC evidence statement is recorded in this case.
ACMG/AMP criteria under Not recorded, with the data and sources behind each one.
28 criteria with recorded statuses: 0 met · 0 not met · 28 not assessed · 0 not applicable. Open any row for the full reasoning, the rule and the evidence behind it.
The governing framework for this case is the generic ACMG/AMP 2015 fallback (PMID 25741868): case_summary framework_mode 'generic_acmg', final_classification_framework found false with precedence_rank 3 and scoring_model 'ACMG/AMP 2015'; no ClinGen CSPEC or gene-specific VCEP materials exist (cspec skipped 'no gene'; vcep_materials domain_tables empty).
None of the five codon/residue-domain criteria could be assessed because the case lacks a usable variant: VariantValidator reported ReferenceMismatchError for NM_000264.5:c.3647A>G (reference base at c.3647 is G, not the submitted A; 1 LOVD-suggested correction), so the gene symbol, genomic coordinates, and predicted protein consequence were never established.
Every evidence source in the bundle is empty: ClinVar retrieval skipped and no variation IDs/submissions/PMIDs; gnomAD v2.1/v4.1/Canada evidence null; REVEL/BayesDel skipped; literature_pass total_pmids 0; paper_extracts.json and fulltext_map.json empty; pm5_candidates.json mode 'unknown' with 0 candidates.
Per the group instruction that insufficient evidence yields not_assessed with the gap identified, all five criteria (PS1, PM1, PM5, PP2, BP1) are returned not_assessed — not not_met — because the total absence of retrieved data stems from a failed normalization, and absence of data cannot be treated as evidence against any of these criteria.
Human review is required first to confirm or correct the variant notation (possible strand/reference misstatement such as c.3647G>A, or a transcript-version issue) and to re-run the pipeline before any codon/residue-domain criterion can be applied.
No criterion among PS2, PM6, PP1, or BS4 can be adjudicated from the supplied bundle because variant normalization is unresolved and no proband, parental, pedigree, meiosis, segregation, or non-segregation data are available.
The case uses the generic ACMG/AMP fallback because no applicable VCEP or gene-specific framework was retrieved.
No calibrated computational criterion was applied because the submitted HGVS has an unresolved reference mismatch and the case contains no normalized variant identity or relevant predictor output.
No criteria match.
PVS1 cannot be assessed because the submitted HGVS expression has a reference-base mismatch, leaving the transcript, gene, variant consequence, exon position, and NMD prediction unresolved. In addition, loss of function as a disease mechanism has not been established for the unidentified gene.
Generic ClinGen SVI PVS1 framework: do not assign PVS1 without a resolved predicted loss-of-function consequence and evidence that loss of function is a disease mechanism for the relevant gene.
PS1 requires the variant to produce the same amino-acid change as a previously established pathogenic variant (regardless of nucleotide change). This cannot be evaluated: normalization of NM_000264.5:c.3647A>G failed (VariantValidator ReferenceMismatchError: the reference base at c.3647 is G, not A as submitted), so no gene symbol, genomic coordinates, predicted protein consequence, or affected residue were established (protein_1l/protein_3l null; hgvs_predicted_protein_consequence empty). ClinVar retrieval was skipped ('no gene'), source_registry contains no ClinVar/PMID entries, literature_pass.json reports 0 PMIDs, and no established-pathogenic comparator of any kind is present in the case bundle. The governing framework is the generic ACMG/AMP 2015 fallback (no CSPEC/VCEP present), under which PS1 cannot be applied to an unconfirmed variant with an unknown residue. Returned not_assessed rather than not_met because the absence of data reflects a failed normalization, not evidence that the criterion's premise is false.
No usable proband-level de novo observation or documented parental testing is present. Variant normalization failed because the submitted reference allele did not match the available transcript sequence, leaving gene and genomic coordinates unresolved; the evidence bundle contains no case-level family data.
PS2 requires a confirmed de novo occurrence in a patient with the relevant phenotype and confirmed maternity and paternity; strength may be calibrated when an applicable specification provides sufficient confirmed de novo observations.
No validated functional assay evidence is available in the case bundle. Variant normalization failed because the submitted reference allele did not match the reference sequence, leaving the gene and consequence unresolved; no VCEP-specific framework, functional-study publications, or paper findings were retrieved. PS3 therefore cannot be assessed.
Generic ACMG/AMP PS3 requires well-established functional studies supportive of a damaging effect; no assay evidence is available here.
Case-control enrichment cannot be assessed because the submitted HGVS could not be normalized, leaving no resolved gene, genomic coordinate, or exact variant record; no case-control data were retrieved.
Generic ACMG/AMP PS4: increased prevalence in affected individuals compared with controls requires variant-specific case-control evidence.
PM1 requires the variant to lie in a mutational hot spot and/or a critical, well-established functional domain. Per the governing instructions, vcep_materials.json domain_tables is the authoritative domain list when an entry exists for the gene; here domain_tables is empty ({}) with no CSPEC URL available, and no gene or residue was identified (normalization failed: reference-allele mismatch at c.3647; protein consequence empty), so no residue-to-domain mapping is possible under either a VCEP table or the generic ACMG/AMP 2015 fallback. No ClinGen domain-table or hotspot evidence is present in any bundle file (hotspots.source_data empty, evidence.json results empty). Returned not_assessed: the variant's residue itself is unestablished, so absence of a domain entry cannot be equated with the variant being outside all critical domains.
PM2 was not assessed because normalization failed and no validated population database result established absence or rarity of the variant in an appropriate reference population.
not_assessed_pending_variant_normalization_and_validated_population_lookup; no applicable VCEP-specific rule was retrieved
PM3 could not be assessed because variant normalization failed: the supplied NM_000264.5:c.3647A>G has a reference-sequence mismatch, no gene or genomic coordinates were resolved, and no affected-proband, phase, inheritance, or trans/cis observations are available in the case evidence bundle.
Generic ACMG/AMP PM3 requires well-established observation of the variant in trans with a pathogenic variant (or appropriate recessive disease allelic evidence); no such evidence is available for this unresolved variant.
PM4 cannot be assessed because the variant did not normalize and there is no established protein consequence or protein length change. The submitted transcript-level substitution cannot be treated as an in-frame length-changing variant while the reference-base mismatch remains unresolved.
Generic ACMG/AMP PM4: evaluate only a confirmed protein length-changing variant, with repeat-region context considered to avoid overlap with BP3.
PM5 requires a novel missense change at a residue where a different missense change has already been established as pathogenic. The PM5 candidate artifact could not support this: pm5_candidates.json reports pm5_mode 'unknown', eligible_for_classic_pm5_search false ('Unable to confirm classic same-residue PM5 semantics safely'), residue context unparsed (reference_aa/position/observed_alt_aa all null), and 0 candidates of any kind. ClinVar retrieval was skipped ('no gene') and literature_pass.json contains 0 PMIDs, so no same-residue pathogenic comparator is available. Returned not_assessed pending confirmation of the variant's residue and a same-residue comparator search.
No case-level report of an apparently de novo variant without confirmed parental testing is present. The variant itself is not reliably normalized, and the bundle contains no proband, phenotype, or family report from which PM6 could be evaluated.
PM6 may be considered for an apparently de novo occurrence when maternity and paternity are not confirmed, but it requires credible case-level evidence and is weaker than confirmed de novo evidence.
No affected relatives, informative meioses, phase information, or family segregation observations are provided. Therefore cosegregation cannot be assessed.
PP1 supports pathogenicity when a variant cosegregates with disease in multiple affected family members; strength can be increased only when the number and informativeness of meioses meet an applicable calibrated rule.
PP2 requires gene-level evidence that the gene has a low rate of benign missense variation and that missense is a common mechanism of disease. Neither premise can be evaluated: the gene was never resolved (normalization failure at c.3647), gnomAD evidence sentences are null in case_summary for v2.1/v4.1/Canada and evidence.json gnomad results are empty, and no gene-specific framework exists. Generic ACMG/AMP 2015 (PMID 25741868) PP2 is therefore not assessable. Returned not_assessed rather than not_met because no gene-level data were retrieved at all.
PP3 cannot be assessed because the submitted transcript-level HGVS failed reference-sequence validation, leaving no normalized gene, genomic coordinates, molecular consequence, SpliceAI result, or calibrated missense predictor result.
Phenotype specificity cannot be assessed because neither a gene nor patient phenotype/disease context was resolved, and no phenotype literature was retrieved.
Generic ACMG/AMP PP4: the patient's phenotype or family history must be highly specific for a disease with a single genetic etiology.
PP5 cannot be applied because no exact-variant ClinVar record or ClinVar expert-panel Pathogenic/Likely pathogenic assertion was retrieved.
Apply PP5 at supporting strength only for an exact-variant ClinVar expert-panel Pathogenic or Likely pathogenic classification; do not use laboratory submissions, aggregate labels, or non-expert assertions.
BA1 was not assessed because the variant could not be normalized: VariantValidator reported a reference mismatch (submitted A, reference G), no gene or genomic coordinates were resolved, and no population-frequency dataset was retrieved.
not_assessed_pending_variant_normalization_and_population_frequency; no applicable VCEP-specific rule was retrieved
BS1 was not assessed because the variant identity was not resolved and no population frequency, ancestry-stratified frequency, disease prevalence, or penetrance data were retrieved.
not_assessed_pending_variant_normalization_and_frequency_threshold; no applicable VCEP-specific rule was retrieved
BS2 was not assessed because no reliable unaffected homozygote or hemizygote observations, phenotype information, inheritance context, disease prevalence, or penetrance assessment were available.
not_assessed_pending_variant_identity_and_unaffected_genotype_phenotype_data; no applicable VCEP-specific rule was retrieved
No validated functional assay evidence demonstrating a well-established benign effect is available in the case bundle. Variant normalization failed because the submitted reference allele did not match the reference sequence, leaving the gene and consequence unresolved; no VCEP-specific framework, functional-study publications, or paper findings were retrieved. BS3 therefore cannot be assessed.
Generic ACMG/AMP BS3 requires well-established functional studies showing no damaging effect on protein function or a biologically relevant assay; no assay evidence is available here.
No unaffected informative relatives with reliable genotype and phenotype data are provided. Absence of family data cannot establish non-segregation.
BS4 supports benignity when a variant does not segregate with disease in affected or expected-to-be-affected informative relatives; an absence of family information is not evidence of non-segregation.
BP1 requires a missense variant in a gene for which primarily truncating variants are known to cause disease (i.e., missense is not an established disease mechanism). The gene is unresolved (normalization failed: reference-allele mismatch at c.3647; protein consequence unknown, so even the missense class is unconfirmed), and the gene-mechanism premise is explicitly unresolved: pvs1_gene_context.json reports germline_disease_context_found false, lof_mechanism_supported false, status 'unresolved', with incomplete literature retrieval, and no gene-specific framework exists. Generic ACMG/AMP 2015 (PMID 25741868) BP1 therefore cannot be assessed. Returned not_assessed because neither the variant class nor the gene's truncating-only mechanism could be established.
BP2 could not be assessed because the variant identity and gene remain unresolved and the case evidence bundle contains no observations of the variant in cis or trans with another pathogenic variant, no family segregation data, and no inheritance information.
Generic ACMG/AMP BP2 requires observation of the variant in trans with a pathogenic variant for a dominant disorder or in cis with a pathogenic variant for a recessive disorder, as applicable; no qualifying observation is available.
BP3 cannot be assessed because the variant did not normalize and there is no evidence of an in-frame protein length change or of localization within a repetitive region without known function.
Generic ACMG/AMP BP3: evaluate only a confirmed in-frame deletion or insertion in a repetitive region without known function.
BP4 cannot be assessed because the submitted HGVS is not validated and no normalized consequence, SpliceAI result, or calibrated benign computational prediction is available.
BP5 cannot be assessed because no affected individual, phenotype, or independent molecular diagnosis is available for this unresolved variant.
Generic ACMG/AMP BP5: a variant may support a benign interpretation when it is found in a case with an alternate molecular basis for disease.
BP6 cannot be applied because no exact-variant ClinVar record or ClinVar expert-panel Benign/Likely benign assertion was retrieved.
Apply BP6 at supporting strength only for an exact-variant ClinVar expert-panel Benign or Likely benign classification; do not use laboratory submissions, aggregate labels, or non-expert assertions.
BP7 cannot be assessed because the submitted HGVS fails reference-sequence validation, so it is not possible to establish that the variant is synonymous or to evaluate predicted splice impact.
No population statements were recorded.
PP3 cannot be assessed because the submitted transcript-level HGVS failed reference-sequence validation, leaving no normalized gene, genomic coordinates, molecular consequence, SpliceAI result, or calibrated missense predictor result.
BP4 cannot be assessed because the submitted HGVS is not validated and no normalized consequence, SpliceAI result, or calibrated benign computational prediction is available.
BP7 cannot be assessed because the submitted HGVS fails reference-sequence validation, so it is not possible to establish that the variant is synonymous or to evaluate predicted splice impact.
0 paper records listed. No publication is explicitly linked to an applied criterion in the recorded metadata.