Affinage

WDR87

WD repeat-containing protein 87 · UniProt Q6ZQQ6

Length
2873 aa
Mass
333.2 kDa
Annotated
2026-06-11
6 papers in source corpus 3 papers cited in narrative 3 extracted findings
Cross-family judge faithfulness: 1/2 claims corpus-supported (50%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

WDR87 is a WD40-repeat protein implicated in flagellar (sperm tail) assembly during spermiogenesis (PMID:36571501). It localizes to the middle piece of the spermatozoa tail and physically interacts with the cilia- and flagella-associated protein CFAP47 to form a complex required for sperm tail assembly; during spermiogenesis WDR87 migrates from perinuclear cytoplasm through the manchette to the tail middle piece, and CFAP47 is required for its proper transport, since patient CFAP47 loss-of-function reduces and mislocalizes WDR87 (PMID:36571501). Beyond this flagellar role, no further mechanistic detail on WDR87's biochemical activity has been characterized in the available corpus.

Mechanistic history

Synthesis pass · year-by-year structured walk · 3 steps
  1. 2016 Low

    An initial association linked WDR87 loss-of-function to a human developmental phenotype, raising the question of where WDR87 acts in vivo.

    Evidence Whole-exome sequencing identifying a biallelic truncating mutation in a single non-syndromic pediatric cataract family, with iSyTE lens expression corroboration

    PMID:27878435

    Open questions at the time
    • Single family with no functional or rescue experiment on WDR87
    • No mechanism connecting WDR87 to lens biology established
    • Genetic association only, causality not demonstrated
  2. 2016 Low

    A proteomic screen placed WDR87 at a chromatin cis-regulatory element, hinting at a possible nuclear/regulatory association, though without WDR87-specific validation.

    Evidence DNA affinity purification followed by mass spectrometry at the PRE-PIK3C2B element across three cell lines

    PMID:27932267

    Open questions at the time
    • No functional validation specific to WDR87
    • WDR87 was a minor finding in a paper centered on MLL/EZH2 biology
    • DNA binding not demonstrated to be direct
  3. 2022 Medium

    Establishing WDR87's molecular partner and subcellular itinerary defined its function in sperm tail assembly, answering what cellular process WDR87 contributes to.

    Evidence Immunofluorescence localization across spermatogenesis, co-immunoprecipitation of WDR87 with CFAP47, and immunoblotting in CFAP47-mutant patient spermatozoa

    PMID:36571501

    Open questions at the time
    • Single lab; interaction not yet reconstituted with purified components
    • Catalytic or structural role of WDR87 within the complex undefined
    • Connection between the flagellar role and the earlier cataract/chromatin observations unexplained

Open questions

Synthesis pass · forward-looking unresolved questions
  • The biochemical activity of WDR87 and how it reconciles its sperm flagellar role with the reported lens phenotype remain unresolved.
  • No defined enzymatic or scaffolding activity
  • No structural model of the WDR87-CFAP47 complex
  • No mechanism linking WDR87 to lens development

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Localization
GO:0005856 cytoskeleton 1 GO:0005929 cilium 1
Partners
CFAP47

Evidence

Reading pass · 3 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2022 WDR87 localizes to the middle piece of the spermatozoa tail and physically interacts with CFAP47 (a cilia- and flagella-associated protein) in the flagellar midpiece, forming a complex involved in spermatozoa tail assembly. During spermiogenesis, WDR87 migrates from perinuclear cytoplasm through the manchette to the sperm tail middle piece. Loss-of-function of CFAP47 (due to patient mutations) dramatically reduces CFAP47 protein and also significantly decreases WDR87 expression and mislocalization, indicating CFAP47 is required for WDR87 transport during flagella biogenesis. Immunofluorescence staining (subcellular localization during spermatogenesis), co-immunoprecipitation / protein interaction assay (WDR87–CFAP47 interaction), immunoblotting in patient spermatozoa with CFAP47 mutations Molecular human reproduction Medium 36571501
2016 WDR87 was identified as a protein that interacts with the human PRE-PIK3C2B cis-regulatory DNA element, detected by DNA affinity purification followed by mass spectrometry in three cell lines (HEK, U87, HeLa), suggesting a role in chromatin regulatory complex assembly at Polycomb/Trithorax responsive elements. DNA affinity purification followed by mass spectrometry (three biological replicates in three cell lines) Biochimica et biophysica acta. Gene regulatory mechanisms Low 27932267
2016 Biallelic inactivating mutation in WDR87 was identified in a family with non-syndromic pediatric cataract, implicating WDR87 loss-of-function in lens development; iSyTE developmental lens expression analysis corroborated its expression in the lens. Whole-exome sequencing with homozygous truncating mutation identification; iSyTE lens expression database corroboration Human genetics Low 27878435

Source papers

Stage 0 corpus · 6 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2016 Novel phenotypes and loci identified through clinical genomics approaches to pediatric cataract. Human genetics 75 27878435
2015 Phenotypes of Recessive Pediatric Cataract in a Cohort of Children with Identified Homozygous Gene Mutations (An American Ophthalmological Society Thesis). Transactions of the American Ophthalmological Society 40 26622071
2021 Genomic alterations associated with mutational signatures, DNA damage repair and chromatin remodeling pathways in cervical carcinoma. NPJ genomic medicine 16 34620846
2022 WDR87 interacts with CFAP47 protein in the middle piece of spermatozoa flagella to participate in sperm tail assembly. Molecular human reproduction 12 36571501
2016 Human PRE-PIK3C2B, an intronic cis-element with dual function of activation and repression. Biochimica et biophysica acta. Gene regulatory mechanisms 3 27932267
2026 Integrative Multiomics Approaches Identify Biomarkers Associated With Progression From Arthralgia to Rheumatoid Arthritis. Arthritis & rheumatology (Hoboken, N.J.) 0 42010917

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