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EPM1 details, HTT min path, and gnomAD links (#227)
Added EPM1 details field, updating Huntington's so motif ranges match, added new GnomAD links (#222). sort age plot ## Description Fixes: #222 ## Minor Changes - Added a details field for EPM1 - Made HTT motif ranges match (using most recent study, [](pmid:39572770)[pmid:39572770](https://www.ncbi.nlm.nih.gov/pubmed/39572770)) - Added missing/new GnomAd links ## Checklist - [x] All changes are well summarized - [x] Check all tests pass - [x] Check that the website preview looks good - [x] Update the STRchive version in `CITATION.cff`, format X.Y.Z. If any major changes, increment Y. If only minor changes, increment Z. If the breaking change (rare), increment X. - [x] Ask someone to review this PR --------- Co-authored-by: Macayla-weiner <205837004+Macayla-weiner@users.noreply.github.com> Co-authored-by: hdashnow <3794821+hdashnow@users.noreply.github.com> Co-authored-by: Harriet Dashnow <h.dashnow@gmail.com>
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CITATION.cff

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title: STRchive
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version: 2.5.1
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date-released: "2025-07-08"
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version: 2.6.0
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date-released: "2025-07-10"
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url: https://github.com/dashnowlab/STRchive
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authors:
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- family-names: Dashnow

data/STRchive-disease-loci.T2T-chm13.bed

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@@ -13,7 +13,7 @@ chr3 63956302 63956333 SCA7_ATXN7 ATXN7 CAG CAG 37 AD Spinocerebellar ataxia typ
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chr3 131917482 131917557 DM2_CNBP CNBP CAGG CAGG 75 AD Myotonic dystrophy type 2
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chr3 141687011 141687054 BPES_FOXL2 FOXL2 NGC NGC 15 AD,AR Blepharophimosis, epicanthus inversus, and ptosis
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chr3 186521667 186521706 FAME4_YEATS2 YEATS2 TTTTA TTTCA 1000 AD Familial adult myoclonic epilepsy 4
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chr4 3073603 3073687 HD_HTT HTT CAG CAG 40 AD Huntington disease
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chr4 3073603 3073687 HD_HTT HTT CAG CAG 36 AD Huntington disease
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chr4 39318077 39318136 CANVAS_RFC1 RFC1 AAAAG AAGGG,ACAGG,AGGGC,AAGGC,AGAGG 400 AR Cerebellar ataxia, neuropathy, and vestibular areflexia syndrome
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chr4 41719745 41719805 CCHS_PHOX2B PHOX2B GCN GCN 26 AD Congenital central hypoventilation syndrome
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chr4 162693303 162693405 FAME7_RAPGEF2 RAPGEF2 TTTTA TTTCA 60 AD Familial adult myoclonic epilepsy type 7

data/STRchive-disease-loci.hg19.bed

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@@ -13,7 +13,7 @@ chr3 63898360 63898391 SCA7_ATXN7 ATXN7 CAG CAG 37 AD Spinocerebellar ataxia typ
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chr3 128891419 128891499 DM2_CNBP CNBP CAGG CAGG 75 AD Myotonic dystrophy type 2
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chr3 138664861 138664904 BPES_FOXL2 FOXL2 NGC NGC 15 AD,AR Blepharophimosis, epicanthus inversus, and ptosis
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chr3 183429975 183430014 FAME4_YEATS2 YEATS2 TTTTA TTTCA 1000 AD Familial adult myoclonic epilepsy 4
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chr4 3076603 3076660 HD_HTT HTT CAG CAG 40 AD Huntington disease
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chr4 3076603 3076660 HD_HTT HTT CAG CAG 36 AD Huntington disease
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chr4 39350044 39350103 CANVAS_RFC1 RFC1 AAAAG AAGGG,ACAGG,AGGGC,AAGGC,AGAGG 400 AR Cerebellar ataxia, neuropathy, and vestibular areflexia syndrome
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chr4 41747989 41748049 CCHS_PHOX2B PHOX2B GCN GCN 26 AD Congenital central hypoventilation syndrome
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chr4 160263678 160263770 FAME7_RAPGEF2 RAPGEF2 TTTTA TTTCA 60 AD Familial adult myoclonic epilepsy type 7

data/STRchive-disease-loci.hg38.bed

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@@ -13,7 +13,7 @@ chr3 63912684 63912715 SCA7_ATXN7 ATXN7 CAG CAG 37 AD Spinocerebellar ataxia typ
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chr3 129172576 129172656 DM2_CNBP CNBP CAGG CAGG 75 AD Myotonic dystrophy type 2
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chr3 138946019 138946062 BPES_FOXL2 FOXL2 NGC NGC 15 AD,AR Blepharophimosis, epicanthus inversus, and ptosis
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chr3 183712187 183712226 FAME4_YEATS2 YEATS2 TTTTA TTTCA 1000 AD Familial adult myoclonic epilepsy 4
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chr4 3074876 3074933 HD_HTT HTT CAG CAG 40 AD Huntington disease
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chr4 3074876 3074933 HD_HTT HTT CAG CAG 36 AD Huntington disease
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chr4 39348424 39348483 CANVAS_RFC1 RFC1 AAAAG AAGGG,ACAGG,AGGGC,AAGGC,AGAGG 400 AR Cerebellar ataxia, neuropathy, and vestibular areflexia syndrome
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chr4 41745972 41746032 CCHS_PHOX2B PHOX2B GCN GCN 26 AD Congenital central hypoventilation syndrome
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chr4 159342526 159342618 FAME7_RAPGEF2 RAPGEF2 TTTTA TTTCA 60 AD Familial adult myoclonic epilepsy type 7

data/STRchive-loci.json

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"prevalence": null,
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"prevalence_details": "Found in individuals of European, Japanese, and Chinese ancestry [@pmid:38876750; @pmid:34047774].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["ABCD3"],
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"genereviews": [],
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"mondo": ["0025193"],
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"year": "2023 [@pmid:39068203]",
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"prevalence": null,
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"prevalence_details": "1/862 (1/654-1266) population prevalence of methylated AFF3 expansions (mild cognitive disability) [@pmid:39313615]. Disease cases observed in South Australia [@pmid:24763282].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["AFF3"],
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"genereviews": [],
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"mondo": [],
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"year": "2014 [@pmid:24763282]",
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"prevalence": null,
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"prevalence_details": "1/100,000 births; female/male ratio 2:1 [@pmid:19267933]. Found across ancestries/ethnicities [@omim:147791].",
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"stripy": ["CBL"],
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"gnomad": [],
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"gnomad": ["CBL"],
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"genereviews": [],
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"mondo": ["0007838"],
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"year": "1995 [@pmid:7603564]",
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"location_in_gene": null,
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"benign_min": 2.0,
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"benign_max": 3.0,
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"intermediate_min": 12.0,
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"intermediate_max": 17.0,
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"intermediate_min": null,
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"intermediate_max": null,
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"pathogenic_min": 30.0,
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"pathogenic_max": 81.0,
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"pathogenic_max": 125.0,
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"ref_copies": null,
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"motif_len": 12,
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"age_onset": "Typical: 6-15 [@genereviews:NBK1142]; Range: 6-18 [@pmid:18325013].",
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"typ_age_onset_max": 15.0,
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"novel": null,
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"mechanism": "LoF",
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"mechanism_detail": "LoF [@pmid:38467784].",
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"mechanism_detail": "The repeat expanison causes significantly reduced expression of cystatin-B protein [@genereviews:NBK1142].",
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"source": [],
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"details": null,
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"details": "Affected individuals have an unstable 12-nucleotide (dodecomer) repeat expansion. Alleles containing 2-3 motifs are considered benign, while alleles with 30-125 repeats are fully penetrant [@pmid:18325013]. Alleles in the range 12-17 repeats have been observed, however the individuals carrying them have not undergone clinical evaluation. Alleles in the range 4-11 and 18-29 repeats have not been reported to date.",
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"omim": ["254800"],
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"prevalence": null,
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"prevalence_details": "Worldwide prevalence unknown; Finland prevalence 2-4/100,000. Found across ethnicities/ancestries, with population-dependent prevalence; highest in Tunisia, Algeria, Morocco, and Finland [@genereviews:NBK1142].",
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"prevalence": null,
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"prevalence_details": "Intermediate expansions 1-2% of population, but non-GAA-pure without relation to ataxia [@genereviews:NBK599589]. Found in multiple ethnicities [@pmid:38876750]; diagnosed patients in America, Brazil, Japan, Germany, Spain, Canada, France, Austria, Australia, and Italy [@genereviews:NBK599589; @pmid:38886208; @pmid:37267898].",
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"stripy": ["FGF14"],
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"gnomad": [],
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"gnomad": ["FGF14"],
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"genereviews": ["NBK599589"],
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"mondo": ["0859340"],
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"year": "2023 [@pmid:36493768]",
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"benign_min": 6.0,
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"benign_max": 26.0,
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"intermediate_min": 27.0,
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"intermediate_max": 39.0,
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"pathogenic_min": 40.0,
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"intermediate_max": 35.0,
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"pathogenic_min": 36.0,
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"pathogenic_max": 250.0,
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"ref_copies": 21.3,
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"motif_len": 3,
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"mechanism": "GoF/LoF",
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"mechanism_detail": "While the primary pathogenic mechanism is gain of function of the protein product, pathogenesis is complex and multifactorial [@pmid:27940602].",
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"source": [],
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"details": "27-35 motifs are unstable/premutations, while 36-39 motifs are associated with reduced penetrance and mild phenotypes [@pmid:39572770]. >60 motifs associated with onset age <20 years [@genereviews:NBK1305]. Only CAG expansions are considered pathogenic, but interruptions impact pathogenicity (e.g. CAA) [@pmid:35245110]. Only fathers with premutations are considered at risk of transmitting pathogenic alleles [@pmid:19507258]. CAG repeats in the non-HD range (>= 21 repeats) may modulate psychiatric disease risk in an age-dependent manner [@pmid:39572770]",
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"details": "27-35 motifs are unstable/premutations, while 36-39 motifs are associated with reduced penetrance and mild phenotypes [@pmid:39572770], and alleles over 40 repeats are typically fully penetrant [@genereviews:NBK1305]. >60 motifs associated with onset age <20 years [@genereviews:NBK1305]. Only CAG expansions are considered pathogenic, but interruptions impact pathogenicity (e.g. CAA) [@pmid:35245110]. Only fathers with premutations are considered at risk of transmitting pathogenic alleles [@pmid:19507258]. CAG repeats in the non-HD range (>= 21 repeats) may modulate psychiatric disease risk in an age-dependent manner [@pmid:39572770]",
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"omim": ["143100"],
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"prevalence": "1/10000",
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"prevalence_details": "6.5-15/100,000 [@pmid:29100084]. 9.71-17:100,000 (European) vs. 0.1-2/100,000 (African), as many as 1 in 400 have reduced penetrance (0.2-2% for 36-38 CAG) HTT alleles [@genereviews:NBK1305]. Found across ethnicities/ancestries, with population-dependent prevalence [@genereviews:NBK1305].",
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"prevalence": null,
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"prevalence_details": "Single proband found in Japanese cohort [@pmid:39455596].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["NAXE"],
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"genereviews": [],
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"mondo": ["0020781"],
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"year": "2024 [@pmid:39455596]",
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"prevalence": null,
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"prevalence_details": "Population dependent: 21.6% of one OPDM cohort [@pmid:35148830]. Typically found in individuals of East Asian ancestry [@pmid:38876750].",
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"stripy": ["RILPL1"],
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"gnomad": [],
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"gnomad": ["RILPL1"],
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"genereviews": [],
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"mondo": ["0030712"],
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"year": "2022 [@pmid:35148830]",
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"prevalence": null,
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"prevalence_details": "Has been observed in Chinese families [@pmid:37148549; @pmid:24677642]. Interruptions predisposing to expansion/disease appear to vary by ancestry [@pmid:39651830].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["THAP11"],
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"genereviews": [],
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"mondo": [],
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"year": "2023 [@pmid:37148549]",
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"prevalence": null,
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"prevalence_details": "Observed in Swedish individuals: 2 original kindreds and 3 additional families [@omim:600223]; common ancestral allele has been identified [@pmid:39635987].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["ZFHX3"],
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"genereviews": [],
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"mondo": ["0010847"],
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"year": "2023 [@pmid:38035881]",
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"prevalence": null,
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"prevalence_details": "1 proband reported alongside 3 individuals with premutations, found in 2 families without discussion of ancestry/ethnicity [@pmid:25196122].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["ZNF713"],
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"genereviews": [],
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"mondo": [],
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"year": "2014 [@pmid:25196122]",
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"prevalence": null,
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"prevalence_details": "Found in 12 families, with ancestry including: Irish, White European, French (Normandie), Ashkenazi, French Canadian, Danish/White European, Welsh, Scottish, Italian, British/White European, Italian/Turkish/Lebanese, German/Palestinian, and South Chinese [@pmid:38714868].",
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"stripy": [],
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"gnomad": [],
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"gnomad": ["PRE-MIR7-2"],
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"genereviews": [],
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"mondo": ["0012360"],
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"year": "2024 [@pmid:38714869]",

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