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Variant interpretation report

SRSF2 c.284C>A, p.Pro95His

NM_003016.4:c.284C>A NP_003007.2:p.(Pro95His) Missense · exon 1 View identifiers
Classification
Uncertain significance

Variant description:

The SRSF2 NM_003016.4:c.284C>A (NP_003007.2:p.(Pro95His)) missense variant has previously been reported in somatic cancers (COSMIC; COSV57969816, n = 500 times), and lies in a statistically significant hotspot (cancerhotspots.org). Experimental evidence: P95H caused 548 misregulated splicing events in CRISPR knock-in cells and concordant RNA-binding, cellular, and in vivo hematopoietic abnormalities across four studies. Based on available guidelines, this variant is considered to be a Variant of Uncertain Significance (VUS).

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.

In plain language

Gene-level background from the recorded interpretation.

About SRSF2

SRSF2 encodes a spliceosome protein that helps process pre-mRNA into mature messenger RNA and also supports mRNA transport and use by cells. Changes affecting SRSF2 are associated with myelodysplastic syndromes, acute myeloid leukemia, and other myeloid disorders; in these cancers, abnormal RNA splicing can disrupt blood-cell development, cell growth, and survival.

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: 1
Pathogenic criteria applied · 2
PS3Strong
PS3 · strong

Met, strong: P95H caused 548 misregulated splicing events in CRISPR knock-in cells and concordant RNA-binding, cellular, and in vivo hematopoietic abnormalities across four studies.

PM2Supporting
PM2 · supporting

Met at supporting strength: gnomAD v4.1 grpmax FAF is 0.00009783, just below the 0.0001 PM2 threshold, with zero homozygotes.

Benign criteria applied · 1
BP5Supporting
BP5 · supporting

Met, supporting: the exact variant occurred with homozygous JAK2 V617F and ASXL1 c.1720-2A>G, providing an alternate molecular basis for the reported disease progression.

Recorded classification: Uncertain significance

VUS: PS3 (strong), PM2 (supporting), and BP5 (supporting) do not satisfy a generic ACMG/AMP pathogenic or benign combination rule.

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
157 / 1,610,816 alleles

gnomAD v4.1: 157 / 1,610,816 alleles. Recorded AF 9.74661e-05. Recorded homozygote count 0.

gnomAD v2.1

This variant is present in gnomAD v2.1 (AF= 0.000103396; MAF= 0.01034%, 28/270804 alleles, homozygotes = 0) and has highest observed frequency in the European (non-Finnish) population (AF= 0.000172332; MAF= 0.01723%, 21/121858 alleles, homozygotes = 0); grpmax FAF= 0.00013143.

gnomAD-Canada

Not available in gnomAD-Canada v1.0.

No screenshot for this evidence
REVEL score
0.419 recorded score

Recorded REVEL score: 0.419. Refer to the recorded PP3/BP4 rationale for interpretation.

No listed criterion applied
ClinVar submissions
1 reported laboratory submissions

1 Pathogenic. ClinVar API review status: criteria provided, single submitter.

Context only ClinVar ↗
Functional evidence
Statement recorded

OncoKB identified variant-specific curated literature and context relevant to functional review; biological-effect context: Loss-of-function; curated oncogenicity label: Oncogenic.

COSMIC
Cancer Hotspots
Somatic evidence
500 recorded samples

This variant has previously been reported in somatic cancers (COSMIC; COSV57969816, n = 500 times).

Cancer Hotspots

This variant lies 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: 3 met · 7 not met · 14 not assessed · 4 not applicable. Open any row for the full reasoning, the rule and the evidence behind it.

Met Not met Not assessed Not applicable

No criterion in this group is assigned because the available evidence does not fully satisfy the governing requirements for an independent same-amino-acid pathogenic comparator, a benign-variation-free PM1 hotspot/domain, a pathogenic alternate same-residue comparator, or the gene-level PP2/BP1 mechanisms.

Variant-specific, biologically relevant cellular, biochemical, and in vivo studies support PS3 and provide no support for BS3.

The exact variant is observed in myeloid-neoplasm cases, including 31 cases in PMID:21909114, but the available literature does not establish the PS4 case-control OR/RR and confidence interval requirement.

PP4 cannot be assessed because a sufficiently specific patient phenotype or family history is not available.

PP5 and BP6 are not met because ClinVar provides only a single-submitter laboratory assertion and no expert-panel classification.

BP5 is supported by a case report describing the exact variant alongside homozygous JAK2 V617F and ASXL1 c.1720-2A>G, although its source-registry registration requires human review.

Both criteria are applicable to this missense variant through the REVEL path only; the REVEL score is below PP3 supporting and above BP4 supporting thresholds.

SpliceAI is not used for a missense variant, and BayesDel is not available for generic ACMG-strength assignment.

Met · 3

PS3 StrongACMG_2015Met, strong: P95H caused 548 misregulated splicing events in CRISPR knock-in cells and concordant RNA-binding, cellular, and in vivo hematopoietic abnormalities across four studies. Details ▾Hide ▴
Reasoning

PS3 is met at strong strength. Multiple independent, variant-specific functional studies evaluated SRSF2 p.Pro95His (P95H), corresponding to NM_003016.4:c.284C>A, using orthogonal systems and wild-type comparators. CRISPR knock-in heterozygous K562 cells showed 548 misregulated splicing events and sequence-dependent changes in RNA-binding affinity (PMID:26261309). Conditional endogenous P95H mouse models demonstrated hematopoietic dysplasia, impaired differentiation, altered transcriptome/splicing, and changed RNA-binding specificity (PMID:25965569); additional mouse and human-cell studies independently showed altered splicing and hematopoietic effects (PMID:26124281, PMID:27135740, PMID:31040863). The evidence is biologically relevant, concordant, and reproducible across cellular, biochemical, and in vivo models, although it is not described as a validated clinical diagnostic-laboratory assay.

Rule
Well-established in vitro or in vivo functional studies supportive of a damaging effect on the gene or gene product.
Evidence considered
  • Kim et al. 2015 (PMID:25965569): endogenous conditional Srsf2P95H knock-in mice showed cytopenias, dysplasia, impaired hematopoietic differentiation, altered transcriptome/splicing, and changed CCNG/GGNG RNA-binding specificity.
  • Lee et al. 2015 (PMID:26124281): in an MDS-L cell model with endogenous SRSF2 depletion and shRNA-resistant rescue, P95H caused growth arrest, apoptosis, and 210 additional alternative-splicing events beyond those shared with the deletion control.
  • Zhang et al. 2015 (PMID:26261309): CRISPR/Cas9 knock-in heterozygous P95H K562 clones showed 548 altered splicing events and sequence-dependent changes in SRSF2 RNA-binding affinity, with wild-type comparator experiments.
  • Shirai et al. 2016 (PMID:27135740): inducible heterozygous Srsf2P95H mouse hematopoietic models showed abnormal splicing and transcriptome effects, with severe hematopoietic failure after loss of the remaining wild-type allele.
  • Masaki et al. 2019 (PMID:31040863): P95H was evaluated in HeLa localization, in-vitro IgM pre-mRNA splicing, RNA immunoprecipitation, and stable K562 RNA-sequencing assays; mutant and wild-type proteins differed in exon-selection patterns and EZH2 exon inclusion.
Sources
PM2 SupportingACMG_2015Met at supporting strength: gnomAD v4.1 grpmax FAF is 0.00009783, just below the 0.0001 PM2 threshold, with zero homozygotes. Details ▾Hide ▴
Reasoning

PM2 is met at supporting strength using the current all-comers gnomAD v4.1 dataset. The filtering grpmax FAF is 0.00009783, which is below the supplied generic PM2 threshold of 0.0001, with 157/1,610,816 alleles and no homozygotes. gnomAD v2.1 also shows an extremely low frequency but its grpmax FAF is 0.00013143, slightly above the threshold; therefore the conclusion depends on the larger, more current v4.1 dataset rather than an assertion that the variant is absent. The available non-cancer v2.1 exome subset has grpmax FAF 0.00012656 and does not meet the supplied cutoff, but no governing VCEP rule requires that subset here.

Rule
Absent from controls (or at extremely low frequency if recessive) in Exome Sequencing Project, 1000 Genomes or Exome Aggregation Consortium.
Evidence considered
  • gnomAD v4.1 all-comers: 157/1,610,816 alleles, total AF 0.0000974661, homozygotes 0, grpmax FAF 0.00009783, meeting the supplied PM2 cutoff <=0.0001.
  • gnomAD v2.1 all-comers: 28/270,804 alleles, homozygotes 0, grpmax FAF 0.00013143, slightly above the supplied cutoff.
  • gnomAD v2.1 non-cancer exomes: 26/228,334 alleles, homozygotes 0, grpmax FAF 0.00012656; this subset is not the governing source under generic rules in this case.
  • The generic PM2 calibration threshold supplied for this case is <=0.0001 for supporting PM2 (ClinGen SVI recommendation, PMID:25741868).
Still missing
  • The gnomAD v2.1 and non-cancer v2.1 FAF values are slightly above the generic PM2 cutoff, so cross-version robustness is limited.
Sources
BP5 SupportingACMG_2015Met, supporting: the exact variant occurred with homozygous JAK2 V617F and ASXL1 c.1720-2A>G, providing an alternate molecular basis for the reported disease progression. Details ▾Hide ▴
Reasoning

A case report explicitly identifies the exact SRSF2 c.284C>A (p.Pro95His) variant in a patient with polycythemia vera progressing to secondary myelofibrosis who also carried homozygous JAK2 V617F and an ASXL1 splice-site variant. The abstract states that the rapid progression was probably due to coexistence of these alterations and that ASXL1 cooperated with JAK2 V617F, providing an alternate molecular basis for the disease course. This supports BP5 at supporting strength. The source was retrieved in the case's PVS1 article material but is not present as a key in source_registry.json, so a human should verify and register the citation before relying on this assertion operationally.

Rule
Variant found in a case with an alternate molecular basis for disease.
Evidence considered
  • Kanduła et al., PMID:35435261, abstract: a PV patient with rapid progression to secondary myelofibrosis carried homozygous JAK2 V617F, SRSF2 c.284C>A p.(Pro95His), and ASXL1 c.1720-2A>G.
  • The abstract states that rapid progression was probably due to coexistence of the JAK2, SRSF2, and ASXL1 alterations and concludes that ASXL1 alteration cooperates with JAK2 V617F, supporting an alternate molecular basis for the reported disease course.
  • The article was directly reviewed from output/pvs1_articles/PMID_35435261.md; no source_registry key exists for this PMID, so evidence_refs is intentionally empty.
Still missing
  • PMID:35435261 is not present in source_registry.json; citation registration and independent review are required before final operational use.

Not met · 7 · evaluated, threshold not reached

PP3 ACMG_2015Not met: REVEL 0.419 is below the generic PP3 supporting threshold of >= 0.644 for this missense variant. Details ▾Hide ▴
Reasoning

The variant is a missense substitution, so the generic rule selects the REVEL path only; splice prediction is not used for this situation. REVEL is 0.419, below the generic PP3 supporting threshold of >= 0.644 calibrated by ClinGen SVI (Pejaver et al. 2022, PMID:36413997). BayesDel is not used because no generic ACMG-strength calibration is established, and no other applicable calibrated computational evidence meets PP3.

Rule
Multiple lines of computational evidence support a deleterious effect on the gene or gene product (conservation, evolutionary, splicing impact, etc.).
Evidence considered
  • NM_003016.4:c.284C>A is classified as missense and produces NP_003007.2:p.(Pro95His), so REVEL is the sole applicable PP3/BP4 predictor path.
  • Local REVEL lookup reports a score of 0.419 for NM_003016.4:c.284C>A.
  • The supplied ClinGen SVI REVEL calibration (Pejaver et al. 2022, PMID:36413997) sets generic PP3 supporting at >= 0.644; 0.419 does not meet it.
  • BayesDel score -0.151583 is not available for generic PP3/BP4 strength assignment because no generic ACMG-strength calibration is established.
Sources
revel
PP5 ACMG_2015Not met: ClinVar lists one exact-variant Pathogenic laboratory submission, but no expert-panel classification supports PP5. Details ▾Hide ▴
Reasoning

The exact variant has a ClinVar Pathogenic submission from one clinical laboratory with a single-submitter review status, not an expert-panel classification. Under the required generic PP5 rule, ordinary laboratory submissions and non-expert assertions do not trigger PP5, so the ClinVar Pathogenic label cannot establish this criterion.

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 3765039 exactly matches NM_001195427.2(SRSF2):c.284C>A (p.Pro95His), but the record is criteria provided, single submitter; the audit reports zero expert-panel submissions.
  • The ClinVar classification is Pathogenic from one clinical laboratory and is therefore excluded from PP5 under the instructed expert-panel-only rule.
Sources
BA1 ACMG_2015Not met: gnomAD v4.1 grpmax FAF is 0.00009783, far below the 0.05 stand-alone BA1 threshold. Details ▾Hide ▴
Reasoning

BA1 is not met. The applicable filtering frequency is the gnomAD v4.1 grpmax FAF of 0.00009783, based on 157/1,610,816 total alleles; this is far below the generic BA1 threshold of 0.05 (5%). The gnomAD v2.1 grpmax FAF is also only 0.00013143. The higher Ashkenazi Jewish gnomAD v4.1 point estimate (0.000169308 from 5/29,532 alleles) is not used for BA1 because the required filtering FAF, not the popmax point estimate, is the relevant metric.

Rule
Allele frequency is above 5% in Exome Sequencing Project, 1000 Genomes or Exome Aggregation Consortium.
Evidence considered
  • gnomAD v4.1 all-comers: 157/1,610,816 alleles, total AF 0.0000974661, homozygotes 0, grpmax FAF 0.00009783.
  • gnomAD v2.1 all-comers: 28/270,804 alleles, total AF 0.000103396, homozygotes 0, grpmax FAF 0.00013143.
  • The generic BA1 calibration threshold supplied for this case is >=0.05 (ACMG/AMP 2015 stand-alone benign allele frequency, PMID:25741868); the filtering FAF is used rather than the population point estimate.
Sources
BS1 ACMG_2015Not met: the highest applicable filtering FAF is 0.00013143, well below the generic 0.01 BS1 default. Details ▾Hide ▴
Reasoning

BS1 is not met on the available population data. The gnomAD v4.1 filtering grpmax FAF is 0.00009783 and the gnomAD v2.1 filtering grpmax FAF is 0.00013143, both well below the supplied generic default threshold of 0.01. The frequency is not elevated in a qualifying population; the highest gnomAD v4.1 population point estimate is only 0.000169308 in Ashkenazi Jewish individuals and is not substituted for FAF. No disease prevalence, penetrance, or allelic-heterogeneity calculation indicates that a lower frequency should be interpreted as greater than expected for the disorder.

Rule
Allele frequency is greater than expected for the disorder.
Evidence considered
  • gnomAD v4.1 all-comers: grpmax FAF 0.00009783; highest population point estimate 0.000169308 in Ashkenazi Jewish individuals from 5/29,532 alleles.
  • gnomAD v2.1 all-comers: grpmax FAF 0.00013143; highest population point estimate 0.000172332 in European non-Finnish individuals from 21/121,858 alleles.
  • The generic BS1 calibration threshold supplied for this case is >=0.01; the filtering FAF is used rather than the popmax point estimate, consistent with ClinGen SVI/ Ghosh et al. 2018 (PMID:30311383).
Sources
BS3 ACMG_2015Not met: no benign functional result was reported, while P95H produced 548 altered splicing events and concordant RNA-binding and hematopoietic abnormalities. Details ▾Hide ▴
Reasoning

BS3 is not met. The reviewed variant-specific studies do not show absence of damaging effect on SRSF2 function or splicing; instead, they report altered RNA-binding specificity, hundreds of abnormal splicing events, altered exon selection, and hematopoietic abnormalities in cellular and in vivo models. No well-established benign functional or splicing result for P95H was identified.

Rule
Well-established in vitro or in vivo functional studies show no damaging effect on protein function or splicing.
Evidence considered
  • The reviewed P95H studies consistently report altered splicing, RNA-binding, exon selection, or hematopoietic phenotypes rather than preserved protein function or normal splicing; therefore they do not support BS3.
Sources
BP4 ACMG_2015Not met: REVEL 0.419 exceeds the generic BP4 supporting threshold of <= 0.29 for this missense variant. Details ▾Hide ▴
Reasoning

The variant is a missense substitution, so the generic rule selects the REVEL path only; SpliceAI cannot substitute for REVEL and is not used for this situation. REVEL is 0.419, above the generic BP4 supporting threshold of <= 0.29 calibrated by ClinGen SVI (Pejaver et al. 2022, PMID:36413997), so it does not support no impact. BayesDel is not used because no generic ACMG-strength calibration is established.

Rule
Multiple lines of computational evidence suggest no impact on gene or gene product (conservation, evolutionary, splicing impact, etc)
Evidence considered
  • NM_003016.4:c.284C>A is classified as missense and produces NP_003007.2:p.(Pro95His), so REVEL is the sole applicable PP3/BP4 predictor path.
  • Local REVEL lookup reports a score of 0.419 for NM_003016.4:c.284C>A.
  • The supplied ClinGen SVI REVEL calibration (Pejaver et al. 2022, PMID:36413997) sets generic BP4 supporting at <= 0.29; 0.419 does not meet it.
  • The absence of a returned SpliceAI score is not used to assess BP4 because SpliceAI is not the applicable path for a missense variant.
  • BayesDel score -0.151583 is not available for generic PP3/BP4 strength assignment because no generic ACMG-strength calibration is established.
Sources
revel
BP6 ACMG_2015Not met: no exact-variant ClinVar expert-panel Benign or Likely benign classification was identified for BP6. Details ▾Hide ▴
Reasoning

No exact-variant ClinVar expert-panel classification of Benign or Likely benign is present. ClinVar contains a Pathogenic single-submitter laboratory assertion and no expert-panel submission, which cannot trigger BP6 and does not provide the required benign reputable-source assertion under the instructed 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 3765039 exactly matches the variant but has only a single-submitter Pathogenic laboratory assertion and zero expert-panel submissions.
  • Because ordinary laboratory submissions, aggregate labels, and non-expert assertions cannot trigger BP6, the available ClinVar record does not support BP6.
Sources

Not assessed · 14 · no assessment recorded

PS1 ACMG_2015Not assessed: no independent previously established pathogenic variant with the same SRSF2 Pro95His amino-acid change but a different nucleotide change was identified. Details ▾Hide ▴
Reasoning

The variant is SRSF2 p.Pro95His, but the available evidence does not identify a previously established pathogenic variant producing the same amino-acid change through a different nucleotide change. The ClinVar record is for this same nucleotide change and has single-submitter Pathogenic status, so it does not establish the required independent PS1 comparator.

Rule
Same amino acid change as a previously established pathogenic variant regardless of nucleotide change. ... Caveat: Beware of changes that impact splicing rather than at the amino acid/protein level.
Evidence considered
  • ClinVar variation 3765039 is the same NM_003016.4:c.284C>A (p.Pro95His) nucleotide change and is classified Pathogenic with criteria provided by a single submitter; it is not an independent same-amino-acid comparator.
Still missing
  • An independently established pathogenic SRSF2 Pro95His variant caused by a different nucleotide change is not documented.
Sources
PS2 ACMG_2015Not assessed: no documented germline proband with confirmed maternity and paternity, disease phenotype, and absence of family history is available. Details ▾Hide ▴
Reasoning

The available evidence identifies SRSF2 c.284C>A (p.Pro95His) in functional and somatic myeloid-neoplasm studies but provides no proband-level evidence establishing a germline de novo occurrence, no confirmed maternity and paternity, and no documented absence of family history. Therefore the ACMG/AMP PS2 definition cannot be assessed.

Rule
De novo (both maternity and paternity confirmed) in a patient with the disease and no family history.
Evidence considered
  • PMID:26261309 experimentally studies heterozygous SRSF2 P95H in K562 cells and explicitly reports no patient-level case, segregation, or population-frequency data for the exact variant.
  • The consolidated case evidence contains no parental testing, maternity confirmation, paternity confirmation, or family-history documentation for this variant.
Still missing
  • A germline proband report documenting the disease phenotype, no family history, and molecular confirmation of both maternity and paternity.
Sources
PS4 ACMG_2015Not assessed: affected cases were reported, but no valid exact-variant case-control odds ratio and confidence interval were available for comparison with the >5.0 PS4 threshold. Details ▾Hide ▴
Reasoning

The literature contains affected-case observations but does not provide the required case-control comparison for the exact variant. PMID:21909114 reports SRSF2 P95H in 31 cases within a 582-specimen myeloid-neoplasm cohort, but it does not report unaffected or population controls for P95H, an odds ratio, or a confidence interval. The governing ACMG/AMP note requires an OR or RR >5.0 with a confidence interval excluding 1.0; because no valid matched-ancestry case-control estimate and CI are available, the threshold cannot be evaluated. The gnomAD v4.1 frequency is 157/1,610,816 alleles (AF 0.0000974661), but the affected cohort's case frequency cannot be paired with a matched-ancestry control frequency to produce a reliable estimate here. The literature_pass PS4 leads PMID:20963938 and PMID:23970018 were checked as leads; the case packet supplies neither an abstract nor full text establishing exact-variant case-control counts for either source.

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
  • Yoshida et al., PMID:21909114, Figure 2: SRSF2 P95H was reported in 31 cases; the study screened 582 myeloid-neoplasm specimens and did not provide unaffected/population control counts for this exact variant.
  • The same paper reports that SRSF2 mutations were highly specific to myelodysplastic-feature diseases, but this is disease-category enrichment rather than an exact-variant case-control comparison with an OR and 95% CI.
  • gnomAD v4.1 reports 157/1,610,816 alleles, AF 0.0000974661; this population frequency is not sufficient here for a matched-ancestry exact-variant OR/RR estimate from the available affected-case data.
  • The governing PS4 threshold is OR/RR >5.0 with a confidence interval excluding 1.0; no valid OR/RR or CI is available, so the threshold is not evaluable.
Still missing
  • An exact-variant affected-case and unaffected/matched-ancestry control table or frequencies are needed to calculate OR/RR and its 95% CI.
  • PMID:20963938 and PMID:23970018 require accessible abstract or full text verification of any exact-variant case-control data.
Sources
PM1 ACMG_2015Not assessed: P95 is a statistically significant recurrent hotspot, but evidence that the hotspot lacks benign variation is not available. Details ▾Hide ▴
Reasoning

P95 is reported as a recurrent SRSF2 hotspot: PMID:21909114 reports 31 P95H cases among 56 SRSF2 mutations and places P95 between the RNA recognition motif and arginine/serine-rich domains; the Cancer Hotspots result also states that the variant lies in a statistically significant hotspot. However, the available evidence does not establish that the hotspot or surrounding region is free of benign variation, which is required by the generic PM1 definition.

Rule
Located in a mutational hot spot and/or critical and well-established functional domain (e.g. active site of an enzyme) without benign variation.
Evidence considered
  • Frequent pathway mutations of splicing machinery in myelodysplasia, Results/Figure 2: SRSF2 mutations exclusively occurred at P95, with P95H reported in 31 cases among 56 SRSF2 mutations; P95 lies between the RNA recognition motif and arginine/serine-rich domains.
  • Cancer Hotspots result for SRSF2 P95: This variant lies in a statistically significant hotspot.
Still missing
  • A documented assessment showing absence of benign variation in the relevant hotspot or functional region is required.
Sources
PM3 ACMG_2015Not assessed: No parent or offspring phase data document NM_003016.4:c.284C>A in trans with a pathogenic variant in an affected proband. Details ▾Hide ▴
Reasoning

PM3 cannot be assessed because the available case evidence does not establish a recessive SRSF2 disorder, a pathogenic allele in the same affected proband, or phase showing NM_003016.4:c.284C>A in trans. No parent or offspring testing, compound-heterozygous observation, or affected-proband trans observation is documented. The available SRSF2 literature describes heterozygous somatic cancer models and does not provide qualifying germline PM3 evidence for this variant.

Rule
For recessive disorders, detected in trans with a pathogenic variant...Note: This requires testing of parents (or offspring) to determine phase.
Evidence considered
  • Case evidence identifies NM_003016.4:c.284C>A as SRSF2 p.(Pro95His), but provides no parent/offspring genotypes, phase result, compound-heterozygous observation, or affected-proband trans observation.
  • The case summary reports no specified inheritance mode for SRSF2 and the available variant-specific literature concerns heterozygous somatic cancer models rather than qualifying recessive germline phase evidence.
Still missing
  • Documented recessive SRSF2 disease context and inheritance mode
  • An affected proband carrying a second pathogenic variant
  • Parent or offspring testing establishing the variants are in trans
PM5 ACMG_2015Not assessed: P95L and P95R are reported at residue 95, but neither is established as pathogenic by the available evidence. Details ▾Hide ▴
Reasoning

The evidence documents other SRSF2 substitutions at residue 95, including P95L and P95R, but does not establish either alternate missense change as pathogenic under the governing generic rule. Therefore, the required pathogenic same-residue comparator for PM5 is not confirmed.

Rule
Novel missense change at an amino acid residue where a different missense change determined to be pathogenic has been seen before. ... Caveat: Beware of changes that impact splicing rather than at the amino acid/protein level.
Evidence considered
  • Myelodysplastic Syndrome-Associated SRSF2 Mutations Cause Splicing Changes by Altering Binding Motif Sequences, Abstract and study description: the study characterized SRSF2 P95H, P95L, and P95R, but the extracted evidence does not provide a pathogenic classification for P95L or P95R.
Still missing
  • An alternate missense variant at Pro95 with an independently established pathogenic determination is required.
Sources
PM6 ACMG_2015Not assessed: no proband-level assumed de novo report with the relevant parental-testing and family-history context is available. Details ▾Hide ▴
Reasoning

The available evidence identifies SRSF2 c.284C>A (p.Pro95His) in functional and somatic myeloid-neoplasm studies but provides no proband-level evidence establishing an assumed de novo occurrence, and no parental testing or family-history information is reported. Therefore the ACMG/AMP PM6 definition cannot be assessed.

Rule
Assumed de novo, but without confirmation of paternity and maternity.
Evidence considered
  • PMID:26261309 experimentally studies SRSF2 P95H in K562 cells and does not report a patient-level de novo observation or parental testing.
  • The consolidated case evidence contains no parental testing, maternity confirmation, paternity confirmation, or family-history documentation for this variant.
Still missing
  • A proband report supporting de novo status while documenting whether maternity and paternity were or were not confirmed.
Sources
PP1 ACMG_2015Not assessed: zero informative affected relatives or documented co-segregating meioses are available for this variant. Details ▾Hide ▴
Reasoning

No affected relatives, pedigree, genotype results in family members, phenotype concordance, or meioses are documented for SRSF2 c.284C>A (p.Pro95His). The available variant-specific literature is functional or somatic and does not provide co-segregation evidence, so PP1 cannot be assessed.

Rule
Co-segregation with disease in multiple affected family members in a gene definitively known to cause the disease.
Evidence considered
  • PMID:26261309 reports a heterozygous cell-line knock-in study and explicitly provides no patient-level segregation data.
  • The consolidated case evidence contains no affected-family-member testing, pedigree, phenotype concordance assessment, or meiosis count.
Still missing
  • A pedigree with clinically affected relatives, their phenotype assessments, variant genotypes, and the number of informative co-segregating meioses.
Sources
PP2 ACMG_2015Not assessed: recurrent somatic SRSF2 missense mutations are shown, but low benign missense variation and a common disease missense mechanism are not both established. Details ▾Hide ▴
Reasoning

The available evidence shows recurrent somatic SRSF2 missense mutations and functional effects at P95, but it does not establish both required gene-level conditions for PP2: a low rate of benign missense variation and missense variants as a common disease mechanism in the applicable disease context.

Rule
Missense variant in a gene that has a low rate of benign missense variation and where missense variants are a common mechanism of disease.
Evidence considered
  • PMID:21909114 reports recurrent somatic SRSF2 mutations concentrated at P95, but this does not by itself establish the generic PP2 gene-level requirements.
  • gnomAD v4.1 reports the queried p.Pro95His allele at AF 9.74661e-05; this single-variant frequency does not quantify the gene-wide benign missense rate or establish the disease mechanism.
Still missing
  • Gene-level evidence quantifying a low benign missense rate is missing.
  • Disease-context evidence that missense variation is a common mechanism of disease is missing.
Sources
PP4 ACMG_2015Not assessed: no sufficiently specific patient phenotype or family history was available to establish a single-genetic-etiology disease under PP4. Details ▾Hide ▴
Reasoning

The available evidence does not provide a patient phenotype or family history sufficiently specific for a disease with a single genetic etiology. The case materials describe a somatic myeloid-neoplasm context and recurrent SRSF2 P95H biology, but no exact patient's diagnostic phenotype or phenotype-specific differential is supplied for applying PP4.

Rule
Patient’s phenotype or family history is highly specific for a disease with a single genetic etiology.
Evidence considered
  • The available case evidence concerns SRSF2 P95H in myeloid neoplasms and does not provide a specific patient phenotype or family history for PP4 assessment.
Still missing
  • A documented patient phenotype and family history, including the specific disease diagnosis and differential etiologies, are needed.
BS2 ACMG_2015Not assessed: no established germline inheritance, penetrance, or early-onset full-penetrance model supports interpreting database controls under BS2. Details ▾Hide ▴
Reasoning

BS2 cannot be assessed from the available evidence. gnomAD reports no homozygotes, but the BS2 rule requires an observed healthy adult individual in the relevant inheritance model and assumes full penetrance with early-age disease; the case does not establish a governing germline disease, inheritance pattern, expected age of onset, or penetrance for SRSF2 p.Pro95His. Heterozygous presence in a population database alone is therefore insufficient to apply BS2.

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 v4.1 reports 157/1,610,816 alleles and 0 homozygotes; gnomAD v2.1 reports 28/270,804 alleles and 0 homozygotes.
  • The available case context describes recurrent somatic SRSF2 P95H findings and does not establish the inheritance, penetrance, or age-of-onset assumptions required for a healthy-adult BS2 interpretation.
Still missing
  • A defined germline disorder and inheritance model for SRSF2 p.Pro95His.
  • Disease age of onset and penetrance sufficient to determine whether gnomAD participants qualify as healthy adults for BS2.
Sources
BS4 ACMG_2015Not assessed: no affected relatives with documented negative variant testing are available to demonstrate lack of segregation. Details ▾Hide ▴
Reasoning

No affected family members with documented absence of the variant are reported for SRSF2 c.284C>A (p.Pro95His). The available variant-specific literature does not provide a family pedigree or non-segregation results, so lack of segregation cannot be evaluated and BS4 cannot be assessed.

Rule
Lack of segregation in affected members of a family.
Evidence considered
  • PMID:26261309 explicitly reports no patient-level segregation data for the exact variant.
  • The consolidated case evidence contains no affected-relative genotypes, pedigree, phenotype assessment, or documentation of a phenocopy or alternative pathogenic variant.
Still missing
  • Affected relatives with reliable clinical phenotyping and molecular testing showing absence of the variant, with pedigree context sufficient to evaluate phenocopies and alternative pathogenic variants.
Sources
BP1 ACMG_2015Not assessed: no evidence establishes that SRSF2 disease is caused primarily by truncating variants rather than missense variants. Details ▾Hide ▴
Reasoning

The available evidence does not show that SRSF2-associated disease is caused primarily by truncating variants. The literature instead documents recurrent somatic missense mutations at Pro95, but that observation does not establish the converse BP1 gene-level pattern.

Rule
Missense variant in a gene for which primarily truncating variants are known to cause disease.
Evidence considered
  • PMID:21909114 reports 56 SRSF2 mutations with concentration at codon P95 in myeloid neoplasms, but it does not establish that primarily truncating variants cause disease.
Still missing
  • A gene-level disease-variant spectrum demonstrating a primarily truncating disease mechanism is missing.
Sources
BP2 ACMG_2015Not assessed: No phase or affected-proband allele-pair data show the variant in trans or cis with a pathogenic variant. Details ▾Hide ▴
Reasoning

BP2 cannot be assessed because the available case evidence does not document NM_003016.4:c.284C>A in trans with a pathogenic variant in a fully penetrant dominant disorder or in cis with a pathogenic variant in any inheritance pattern. No affected-proband allele pair, phase result, or parent/offspring testing establishes either configuration.

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
  • Case evidence provides no documented cis/trans relationship between NM_003016.4:c.284C>A and another pathogenic variant, and no parent or offspring testing establishes phase.
  • No affected-proband allele-pair observation meeting the BP2 definition is reported in the available case evidence.
Still missing
  • A documented pathogenic allele paired with this variant in the same individual
  • Phase testing establishing cis or trans configuration
  • For a trans observation, evidence that the disorder is fully penetrant and dominant
Not applicable · 4· recorded as not applicable Show reasons ▾Hide reasons ▴
PVS1 N/ANot applicable: PVS1 requires a null variant (nonsense, frameshift, canonical splice, start-loss or stop-loss), and this is a missense variant. Details ▾Hide ▴
Reasoning

PVS1 (ClinGen SVI PVS1 recommendations, PMC6185798, pvs1_generic_framework) applies only to null variant classes — nonsense, frameshift, canonical ±1/2 splice-consensus, translation-initiation-codon loss, and stop-loss variants — each expected to trigger nonsense-mediated decay or otherwise abolish normal protein function. NM_003016.4:c.284C>A in SRSF2 is a missense substitution predicted to produce NP_003007.2:p.(Pro95His). As a result, no null-variant mechanism (nonsense-mediated decay, truncation, or canonical splice disruption) is triggered by this variant class, so PVS1's structural prerequisite is not met by this variant class. No VCEP criteria specification covers PVS1 for SRSF2, 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_null_variant_class
Evidence considered
  • Deterministic consequence classification: missense (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
pvs1_generic_framework ↗generic_acmg_combination_rules
PM4 N/ANot applicable: PM4 requires a protein-length change, and this is a missense 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_003016.4:c.284C>A in SRSF2 is a missense substitution predicted to produce NP_003007.2:p.(Pro95His). 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 SRSF2, 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: missense (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
pvs1_generic_framework ↗generic_acmg_combination_rules
BP3 N/ANot applicable: BP3 requires an in-frame indel in a repeat region, and this is a missense 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_003016.4:c.284C>A in SRSF2 is a missense substitution predicted to produce NP_003007.2:p.(Pro95His). 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 SRSF2, 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: missense (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
pvs1_generic_framework ↗generic_acmg_combination_rules
BP7 N/ANot applicable: BP7 requires a synonymous variant, and this is a missense 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_003016.4:c.284C>A in SRSF2 is a missense substitution predicted to produce NP_003007.2:p.(Pro95His). 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 SRSF2, 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: missense (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules

Identifiers

Gene
SRSF2
Transcript
NM_003016.4
Coding (HGVS c.)
NM_003016.4:c.284C>A
Protein (HGVS p.)
NP_003007.2:p.(Pro95His)
GRCh38
chr17:76736877 G>T
GRCh37
chr17:74732959 G>T
Location and type
Exon 1 · missense
ClinVar
3765039
COSMIC
COSV57969816

Gene diagram

Every interpreted variant in SRSF2, mapped onto the exon structure of NM_003016.4. Select a row to find it on the plot.

SRSF2NM_003016.4
Fetching transcript structure from UCSC…
gnomAD v4.1 157 / 1,610,816 alleles AF 9.75e-05 · 0 homozygotes · FAF 9.78e-05
gnomAD v2.1 28 / 270,804 alleles AF 0.000103 · 0 homozygotes · FAF 0.000131
gnomAD-Canada v1.0 Allele count data not recorded

By genetic ancestry

gnomAD v4.1

AF 9.7e-05 · 157 / 1,610,816 alleles · 0 hom · FAF 9.8e-05
Ashkenazi Jewish5 / 29,532 0.00017
European (non-Finnish)134 / 1,178,968 0.00011
Remaining individuals5 / 62,344 8e-05
European (Finnish)4 / 62,376 6.4e-05
Admixed American3 / 59,920 5e-05
African/African American3 / 74,864 4e-05
South Asian3 / 90,988 3.3e-05
0 alternate alleles recorded in 3 other groups: Amish, East Asian, Middle Eastern.

gnomAD v2.1

AF 0.0001 · 28 / 270,804 alleles · 0 hom · FAF 0.00013
European (non-Finnish)21 / 121,858 0.00017
Remaining individuals1 / 6,960 0.00014
Ashkenazi Jewish1 / 10,070 9.9e-05
South Asian2 / 30,446 6.6e-05
East Asian1 / 19,166 5.2e-05
African/African American1 / 22,836 4.4e-05
Admixed American1 / 35,080 2.9e-05
0 alternate alleles recorded in 1 other group: European (Finnish).

Statements

gnomAD v4.1

This variant is present in gnomAD v4.1 (AF= 9.74661e-05; MAF= 0.00975%, 157/1610816 alleles, homozygotes = 0) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.000169308; MAF= 0.01693%, 5/29532 alleles, homozygotes = 0); grpmax FAF= 9.783e-05.

gnomAD v2.1

This variant is present in gnomAD v2.1 (AF= 0.000103396; MAF= 0.01034%, 28/270804 alleles, homozygotes = 0) and has highest observed frequency in the European (non-Finnish) population (AF= 0.000172332; MAF= 0.01723%, 21/121858 alleles, homozygotes = 0); grpmax FAF= 0.00013143.

gnomAD-Canada

Not available in gnomAD-Canada v1.0.

Other predictors

REVEL
0.419
BayesDel
-0.152

How the predictions were used

PP3 ACMG_2015Not met: REVEL 0.419 is below the generic PP3 supporting threshold of >= 0.644 for this missense variant. Details ▾Hide ▴
Reasoning

The variant is a missense substitution, so the generic rule selects the REVEL path only; splice prediction is not used for this situation. REVEL is 0.419, below the generic PP3 supporting threshold of >= 0.644 calibrated by ClinGen SVI (Pejaver et al. 2022, PMID:36413997). BayesDel is not used because no generic ACMG-strength calibration is established, and no other applicable calibrated computational evidence meets PP3.

Rule
Multiple lines of computational evidence support a deleterious effect on the gene or gene product (conservation, evolutionary, splicing impact, etc.).
Evidence considered
  • NM_003016.4:c.284C>A is classified as missense and produces NP_003007.2:p.(Pro95His), so REVEL is the sole applicable PP3/BP4 predictor path.
  • Local REVEL lookup reports a score of 0.419 for NM_003016.4:c.284C>A.
  • The supplied ClinGen SVI REVEL calibration (Pejaver et al. 2022, PMID:36413997) sets generic PP3 supporting at >= 0.644; 0.419 does not meet it.
  • BayesDel score -0.151583 is not available for generic PP3/BP4 strength assignment because no generic ACMG-strength calibration is established.
Sources
revel
BP4 ACMG_2015Not met: REVEL 0.419 exceeds the generic BP4 supporting threshold of <= 0.29 for this missense variant. Details ▾Hide ▴
Reasoning

The variant is a missense substitution, so the generic rule selects the REVEL path only; SpliceAI cannot substitute for REVEL and is not used for this situation. REVEL is 0.419, above the generic BP4 supporting threshold of <= 0.29 calibrated by ClinGen SVI (Pejaver et al. 2022, PMID:36413997), so it does not support no impact. BayesDel is not used because no generic ACMG-strength calibration is established.

Rule
Multiple lines of computational evidence suggest no impact on gene or gene product (conservation, evolutionary, splicing impact, etc)
Evidence considered
  • NM_003016.4:c.284C>A is classified as missense and produces NP_003007.2:p.(Pro95His), so REVEL is the sole applicable PP3/BP4 predictor path.
  • Local REVEL lookup reports a score of 0.419 for NM_003016.4:c.284C>A.
  • The supplied ClinGen SVI REVEL calibration (Pejaver et al. 2022, PMID:36413997) sets generic BP4 supporting at <= 0.29; 0.419 does not meet it.
  • The absence of a returned SpliceAI score is not used to assess BP4 because SpliceAI is not the applicable path for a missense variant.
  • BayesDel score -0.151583 is not available for generic PP3/BP4 strength assignment because no generic ACMG-strength calibration is established.
Sources
revel
BP7 N/ANot applicable: BP7 requires a synonymous variant, and this is a missense 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_003016.4:c.284C>A in SRSF2 is a missense substitution predicted to produce NP_003007.2:p.(Pro95His). 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 SRSF2, 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: missense (prefetch.json normalized protein consequence)
  • VCEP criterion policy: absent
Sources
generic_acmg_combination_rules

Statement

In silico

REVEL score = 0.419. BayesDel score = -0.151583.

9 paper records listed. 5 cited in the classification.

Cited in the classification

  • SRSF2 Mutations Contribute to Myelodysplasia by Mutant-Specific Effects on Exon Recognition.
    Kim E et al. · 2015 May 11
    The paper explicitly studies the SRSF2 P95H substitution, corresponding to the provided NP_003007.2:p.(P95H) variant (c.284C>A). Using an endogenous conditional Srsf2P95H knock-in mouse model, the authors found that heterozygous P95H expression caused cytopenias, macrocytic anemia, myeloid and erythroid dysplasia, expansion of hematopoietic stem/progenitor cells, impaired hematopoietic differentiation and stem-cell reconstitution, and increased apoptosis. RNA-seq, RNA-binding, ITC, and NMR experiments showed that P95H alters SRSF2 sequence-specific RNA-binding preference and splicing, including EZH2 poison-exon inclusion.
    → PS3 · strong Names this variant
    PMID 25965569 ↗
    Source detail
    Here we studied the biological and transcriptional effects of somatic expression of the common SRSF2P95H mutation in the hematopoietic compartment.

    Location Introduction, final paragraph; Results sections titled ‘Srsf2P95H Mutant Mice Develop MDS...’ and ‘SRSF2 Proline 95 Mutations Alter RNA Binding Specificity...’ (pp. 618–625)

    Context Conditional endogenous Srsf2P95H knock-in mice with hematopoietic transplantation and phenotyping; RNA-seq; K562 expression; ITC and NMR binding assays; EZH2 isoform-specific RT-PCR and nonsense-mediated-decay assays.

  • SRSF2 Is Essential for Hematopoiesis, and Its Myelodysplastic Syndrome-Related Mutations Dysregulate Alternative Pre-mRNA Splicing.
    The paper explicitly describes the SRSF2 P95H missense mutation, corresponding to NP_003007.2:p.(P95H). In a human MDS-L cell-line model, endogenous SRSF2 was reduced with an shRNA while an shRNA-resistant P95H construct was expressed. Relative to wild-type SRSF2, P95H caused growth arrest, apoptosis, and significant alternative-splicing changes, with combined loss- and gain-of-function effects.
    → PS3 · strong Names this variant
    PMID 26124281 ↗
    Source detail
    the P95H missense mutation and P95 to R102 in-frame 8-amino-acid deletion caused significant changes in alternative splicing.

    Location Abstract; Results, ‘SRSF2 Mutants altered a large RNA splicing program,’ paragraphs beginning at lines 323 and 344; Discussion, paragraphs beginning at lines 451 and 455

    Context Human MDS-L cells with doxycycline-inducible endogenous SRSF2 shRNA and shRNA-resistant wild-type or P95H rescue; RASL-seq profiling of 5,530 alternatively spliced mRNA isoforms; murine bone-marrow overexpression experiments.

  • Disease-associated mutation in SRSF2 misregulates splicing by altering RNA-binding affinities.
    Zhang J et al. · 2015 Aug 25
    This paper experimentally studies heterozygous SRSF2 P95H in CRISPR knock-in K562 leukemia-cell clones and reports altered RNA binding and 548 misregulated splicing events; it does not provide a patient pedigree, parental testing, segregation, or non-segregation data.
    → PS3 · strong Names this variant
    PMID 26261309 ↗
    Source detail
    We found that SRSF2 (P95H) misregulates 548 splicing events (<1% of total).

    Location Abstract; Results, "Mutant SRSF2 Misregulates Hundreds of Splicing Events"; Fig. 3 and Fig. 6

    Context CRISPR/Cas9 knock-in of heterozygous SRSF2 P95H into K562 leukemia cells; RNA-seq and RT-PCR splicing analysis; transient expression in 293T cells; RNA gel-shift and coimmunoprecipitation assays.

  • Modulation of splicing catalysis for therapeutic targeting of leukemia with mutations in genes encoding spliceosomal proteins.
    The paper explicitly studies SRSF2 P95H in inducible mouse hematopoietic models. Heterozygous Srsf2P95H cells showed transcriptome and splicing abnormalities, while loss of the remaining wild-type allele caused severe hematopoietic stem/progenitor-cell loss and bone-marrow failure.
    → PS3 · strong Names this variant
    PMID 27135740 ↗
    Source detail
    We engineered mice that express hemizygous Srsf2P95H mutant alleles in an inducible manner.

    Location Results, first experimental paragraph; Fig. 1 and Supplementary Fig. 1

    Context Inducible conditional Srsf2P95H mouse hematopoietic models, bone-marrow transplantation, RNA-seq and differential-splicing analysis, leukemia models, and human hematopoietic cell experiments.

  • Myelodysplastic Syndrome-Associated SRSF2 Mutations Cause Splicing Changes by Altering Binding Motif Sequences.
    Masaki S et al. · 2019
    The paper explicitly studies SRSF2 P95H and characterizes the additional P95L and P95R substitutions in functional assays, demonstrating that alternate missense changes at residue 95 were investigated; the extracted evidence does not establish those alternates as pathogenic.
    → PS3 · strong Names this variant
    PMID 31040863 ↗
    Source detail
    In this study, we characterized MDS-associated SRSF2 mutants (P95H, P95L, and P95R).

    Location Abstract

    Context HeLa-cell localization; in-vitro IgM pre-mRNA splicing and RNA co-immunoprecipitation; stable K562-cell expression followed by RNA sequencing, motif analysis, and EZH2 RT-PCR validation.

Other recorded publications

  • Spliceosome Mutations Induce R Loop-Associated Sensitivity to ATR Inhibition in Myelodysplastic Syndromes.
    No paper-specific finding is recorded.
    PMID 30054334 ↗
  • Frequent pathway mutations of splicing machinery in myelodysplasia.
    Yoshida K et al. · 2011
    The paper explicitly reports the SRSF2 p.P95H mutation, shown as P95H (31) in Figure 2, indicating that it was observed in 31 cases in the studied myeloid-neoplasm cohort. Across the cohort, SRSF2 mutations were recurrently concentrated at codon P95, located in the intervening region between the RNA recognition motif (RRM) and arginine/serine-rich (RS) domains.
    Names this variant
    PMID 21909114 ↗
    Quoted passage
    SRSF2 mutations exclusively occurred at P95 within an intervening sequence between the RNA recognition motif (RRM) and arginine/serine-rich (RS) domains (Fig. 2 and Supplementary Figs 8 and 9).

    Results, "Frequent mutations in splicing machinery"; Figure 2

  • CEBPA-Associated Familial Acute Myeloid Leukemia.
    Adam MP et al. · 1993
    This literature_pass PS4/PP5 lead could not be independently verified from the available case materials because its abstract is empty and no full text is present in source_registry.json.
    Does not name this variant
    PMID 20963938 ↗
  • American College of Medical Genetics and Genomics technical standards and guidelines: microarray analysis for chromosome abnormalities in neoplastic disorders.
    No paper-specific finding is recorded.
    PMID 23619274 ↗

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= 9.74661e-05; MAF= 0.00975%, 157/1610816 alleles, homozygotes = 0) and has highest observed frequency in the Ashkenazi Jewish population (AF= 0.000169308; MAF= 0.01693%, 5/29532 alleles, homozygotes = 0); grpmax FAF= 9.783e-05.

gnomAD v2.1

This variant is present in gnomAD v2.1 (AF= 0.000103396; MAF= 0.01034%, 28/270804 alleles, homozygotes = 0) and has highest observed frequency in the European (non-Finnish) population (AF= 0.000172332; MAF= 0.01723%, 21/121858 alleles, homozygotes = 0); grpmax FAF= 0.00013143.

gnomAD-Canada

Not available in gnomAD-Canada v1.0.

ClinVar

This variant has been reported in ClinVar as Pathogenic (1 clinical laboratory). (ClinVarID = 3765039)

In silico

REVEL score = 0.419. BayesDel score = -0.151583.

Functional

OncoKB identified variant-specific curated literature and context relevant to functional review; biological-effect context: Loss-of-function; curated oncogenicity label: Oncogenic.

Somatic

This variant lies in a statistically significant hotspot. This variant has previously been reported in somatic cancers (COSMIC; COSV57969816, n = 500 times).

Cancer Hotspots

This variant lies in a statistically significant hotspot.