PKP1 (Plakophilin-1) variants and mutations
PKP1 (also known as Plakophilin-1) is a human protein-coding gene encoding a plakophilin-1 protein. It strengthens desmosomal adhesion by linking cadherins to intermediate filaments, particularly in stratified epithelia. Biallelic loss-of-function variants cause ectodermal dysplasia-skin fragility syndrome with trauma-induced blistering, palmoplantar keratoderma, and abnormal hair. This analysis covers 1,386 PKP1 variants and mutations. Of these, 58% have computational variant effect predictions. Disease context includes epidermolysis bullosa simplex due to plakophilin deficiency, seborrheic keratosis, and skin aging. Example PKP1 variants include M1?, N2S, and H3H.
Variant analysis overview
- Gene: PKP1
- Protein: Plakophilin-1
- UniProt accession: Q13835
- Organism: Homo sapiens
- Variants analyzed: 1386
- Variant scope: all variants
- Completed: 2026-08-22
Variant and mutation evidence
- Variant composition: 1,114 unspecified-consequence records; 116 synonymous variants; 138 missense variants; 8 stop-gained variants; 1 in-frame deletions; 8 frameshift variants; 2 splice-region variants
- Prediction scores: 800 variants have prediction scores (58% of the analyzed set).
Clinical, disease, and population context
- Disease context: 25 disease associations are represented. Top associations: epidermolysis bullosa simplex due to plakophilin deficiency, seborrheic keratosis, skin aging, skin disorder, knee fracture, congenital anomaly of cardiovascular system, hereditary disease, esophageal squamous cell carcinoma, ovarian carcinoma, ovarian cancer, erythrokeratodermia variabilis, lamellar ichthyosis.
Protein structure and variant hotspots
- Protein features: 5 post-translational modification sites.
- PTM context: 17 variants overlap post-translational modification sites.
Data sources
Evidence in this analysis draws on EBI Proteins Variation, UniProt, gnomAD v4, EuropePMC, 3D Hotspot Analysis, Interaction Network Analysis, Protein Data Bank, AlphaFold DB, gnomAD constraint, Open Targets, MaveDB, LitVar.
Notable PKP1 variants
Examples include M1?, N2S, H3H, S4*, S4L, S4W, S4T, P5L. Listed records include available protein-change notation, database identifiers, clinical classifications, computational predictions, population evidence, experimental measurements, and disease context.
- M1?, cosmic curated COSV10506
- N2S (p.Asn2Ser), gnomAD rs1249234066, REVEL 0.13, CADD 19.60
- H3H (p.His3His), gnomAD 1-201283711-C-T, CADD 14.70
- S4* (p.Ser4Ter), cosmic curated COSV99824, CADD 39.00
- S4L (p.Ser4Leu), cosmic curated COSV55823, TOPMed rs1475958808, gnomAD rs1475958808, REVEL 0.14, CADD 29.90
- S4W (p.Ser4Trp), TOPMed rs1475958808, gnomAD rs1475958808, REVEL 0.20, CADD 31.00
- S4T (p.Ser4Thr), gnomAD 1-201283712-T-A, REVEL 0.14, CADD 27.50
- P5L (p.Pro5Leu), rs1417514113, NCI-TCGA Cosmic COSV9982, cosmic curated COSV99825, gnomAD rs1417514113, REVEL 0.23, CADD 28.10, Variant assessed as somatic; moderate impact.
- P5S (p.Pro5Ser), rs1189119963, NCI-TCGA Cosmic COSV9982, cosmic curated COSV99825, gnomAD rs1189119963, AlphaMissense 0.64, MetaLR 0.23, Variant assessed as somatic; moderate impact.
- P5R (p.Pro5Arg), gnomAD 1-201283716-C-G, REVEL 0.25, CADD 27.10
- P5Q (p.Pro5Gln), gnomAD 1-201283716-C-A, REVEL 0.25, CADD 27.00
- P5P (p.Pro5Pro), rs760122260, gnomAD 1-201283717-G-C, CADD 14.90
- L6L (p.Leu6Leu), gnomAD 1-201283720-C-T, CADD 15.50
- T8T (p.Thr8Thr), gnomAD 1-201283726-C-A, CADD 13.70
- A9T (p.Ala9Thr), NCI-TCGA Cosmic COSV9982, cosmic curated COSV99825, Ensembl rs1655641389, Variant assessed as somatic; moderate impact.
- A9S (p.Ala9Ser), gnomAD 1-201283727-G-T, REVEL 0.33, CADD 28.80
- A9V (p.Ala9Val), gnomAD 1-201283728-C-T, REVEL 0.27, CADD 24.40
- L10L (p.Leu10Leu), gnomAD 1-201283730-T-C, CADD 11.10
- A11S (p.Ala11Ser), TOPMed rs986378706, gnomAD rs986378706, REVEL 0.21, CADD 23.20
- A11V (p.Ala11Val), TOPMed rs910380119, gnomAD rs910380119, REVEL 0.36, CADD 24.70
- A11A (p.Ala11Ala), rs1409504390, gnomAD 1-201283735-G-C, CADD 8.67
- Y12C (p.Tyr12Cys), gnomAD 1-201283737-A-G, REVEL 0.27, CADD 26.30
- Y12* (p.Tyr12Ter), gnomAD 1-201283738-C-A, CADD 36.00
- Y12Y (p.Tyr12Tyr), rs2268147, gnomAD 1-201283738-C-T, CADD 13.70
- E13K (p.Glu13Lys), NCI-TCGA TCGA novel, REVEL 0.24, CADD 27.90, Variant assessed as somatic; moderate impact.
- E13G (p.Glu13Gly), gnomAD 1-201283740-A-G, REVEL 0.23, CADD 26.80
- E13E (p.Glu13Glu), rs759929631, gnomAD 1-201283741-A-G, CADD 14.60
- C14F (p.Cys14Phe), cosmic curated COSV99825
- C14R (p.Cys14Arg), NCI-TCGA Cosmic COSV5582, cosmic curated COSV55820, Variant assessed as somatic; moderate impact.
- C14S (p.Cys14Ser), cosmic curated COSV10584
- C14Y (p.Cys14Tyr), gnomAD 1-201283743-G-A, REVEL 0.24, CADD 31.00
- F15L (p.Phe15Leu), TOPMed rs1655642084, cosmic curated COSV10456
- F15V (p.Phe15Val), ExAC rs765686962, gnomAD rs765686962, REVEL 0.27, CADD 25.30
- Q16* (p.Gln16Ter), TOPMed rs1655642159, gnomAD rs1655642159, CADD 39.00
- Q16E (p.Gln16Glu), TOPMed rs1655642159, gnomAD rs1655642159, REVEL 0.05, CADD 18.90
- Q16Q (p.Gln16Gln), rs1293694803, gnomAD 1-201283750-G-A, CADD 13.20
- D17N (p.Asp17Asn), gnomAD rs1339584228, REVEL 0.08, CADD 22.90
- D17D (p.Asp17Asp), rs1558180237, gnomAD 1-201283753-C-T, CADD 14.80
- Q18* (p.Gln18Ter), Ensembl rs2102402191
- Q18H (p.Gln18His), gnomAD rs1242339724, REVEL 0.24, CADD 26.00
- Q18R (p.Gln18Arg), gnomAD 1-201283755-A-G, REVEL 0.16, CADD 25.20
- D19G (p.Asp19Gly), gnomAD rs1339972669, REVEL 0.09, CADD 23.70
- D19N (p.Asp19Asn), gnomAD rs1271921485, REVEL 0.05, CADD 23.20
- D19E (p.Asp19Glu), gnomAD 1-201283759-C-A, REVEL 0.02, CADD 22.70
- N20S (p.Asn20Ser), cosmic curated COSV10506, ExAC rs199604952, TOPMed rs199604952, gnomAD rs199604952, REVEL 0.05, CADD 19.40, Uncertain significance, Inborn genetic diseases
- N20I (p.Asn20Ile), gnomAD 1-201283761-A-T, REVEL 0.10, CADD 25.10
- S21F (p.Ser21Phe), gnomAD 1-201283764-C-T, REVEL 0.18, CADD 25.10
- T22A (p.Thr22Ala), Ensembl rs1655642846
- T22M (p.Thr22Met), cosmic curated COSV55824
- T22T (p.Thr22Thr), rs528746460, gnomAD 1-201283768-G-T, CADD 11.20
- L23S (p.Leu23Ser), gnomAD rs1201125089, REVEL 0.29, CADD 31.00
- A24S (p.Ala24Ser), gnomAD rs1486454180, REVEL 0.16, CADD 28.80
- A24V (p.Ala24Val), gnomAD 1-201283773-C-T, REVEL 0.16, CADD 28.20
- L25S (p.Leu25Ser), NCI-TCGA TCGA novel, Variant assessed as somatic; moderate impact.
- L25W (p.Leu25Trp), gnomAD rs1211069504, REVEL 0.24, CADD 31.00
- P26L (p.Pro26Leu), ESP rs377481646, ExAC rs377481646, TOPMed rs377481646, gnomAD rs377481646, REVEL 0.40, CADD 29.60
- P26Q (p.Pro26Gln), ESP rs377481646, ExAC rs377481646, TOPMed rs377481646, gnomAD rs377481646, REVEL 0.38, CADD 28.40
- P26R (p.Pro26Arg), gnomAD 1-201283779-C-G, REVEL 0.45, CADD 28.80
- P26P (p.Pro26Pro), rs2102402211, gnomAD 1-201283780-G-A, CADD 14.40
- S27L (p.Ser27Leu), 1000Genomes rs2102402212, REVEL 0.22, CADD 29.00
- S27P (p.Ser27Pro), gnomAD 1-201283781-T-C, REVEL 0.20, CADD 31.00
- S27S (p.Ser27Ser), gnomAD 1-201283783-G-C, CADD 9.34
- D28E (p.Asp28Glu), 1000Genomes rs751815067, ExAC rs751815067, gnomAD rs751815067, REVEL 0.22, CADD 23.40
- D28H (p.Asp28His), cosmic curated COSV55821
- D28N (p.Asp28Asn), TOPMed rs1195751281, gnomAD rs1195751281
- D28Y (p.Asp28Tyr), gnomAD 1-201283784-G-T, REVEL 0.36, CADD 32.00
- D28G (p.Asp28Gly), gnomAD 1-201283785-A-G, REVEL 0.27, CADD 32.00
- Q29* (p.Gln29Ter), gnomAD rs1475350964
- Q29R (p.Gln29Arg), Ensembl rs975952428
- K30N (p.Lys30Asn), rs1322769789, TOPMed rs1322769789, gnomAD rs1322769789, REVEL 0.08, CADD 23.20, Variant assessed as somatic; moderate impact.
- K30R (p.Lys30Arg), NCI-TCGA TCGA novel, Variant assessed as somatic; moderate impact.
- K30T (p.Lys30Thr), ExAC rs757475801, gnomAD rs757475801
- M31T (p.Met31Thr), NCI-TCGA TCGA novel, Variant assessed as somatic; moderate impact.
- M31V (p.Met31Val), ESP rs370900946, ExAC rs370900946, TOPMed rs370900946, gnomAD rs370900946, REVEL 0.02, CADD 22.90
- T33A (p.Thr33Ala), NCI-TCGA TCGA novel, Variant assessed as somatic; moderate impact.
- T33I (p.Thr33Ile), gnomAD rs1435578626
- T33T (p.Thr33Thr), gnomAD 1-201283801-A-G, CADD 5.22
- G34D (p.Gly34Asp), ExAC rs749292645, gnomAD rs749292645, REVEL 0.05, CADD 24.10
- G34S (p.Gly34Ser), Ensembl rs1655644276
- G34V (p.Gly34Val), ExAC rs749292645, gnomAD rs749292645
- G34G (p.Gly34Gly), rs1243979384, gnomAD 1-201283804-C-T, CADD 18.90
- T35M (p.Thr35Met), TOPMed rs780267593, gnomAD rs780267593, REVEL 0.06, CADD 24.10
- T35S (p.Thr35Ser), gnomAD 1-201283805-A-T, REVEL 0.09, CADD 19.10
- T35T (p.Thr35Thr), gnomAD 1-201283807-G-T, CADD 11.90
- S36Y (p.Ser36Tyr), cosmic curated COSV99825
- S36S (p.Ser36Ser), gnomAD 1-201283810-T-A, CADD 14.10
- G37D (p.Gly37Asp), Ensembl rs113410388
- G37R (p.Gly37Arg), gnomAD rs1400323054
- G37V (p.Gly37Val), gnomAD 1-201283812-G-T, REVEL 0.22, CADD 27.50
- R38G (p.Arg38Gly), ExAC rs754822585, gnomAD rs754822585, REVEL 0.02, CADD 19.10
- R38K (p.Arg38Lys), cosmic curated COSV10506, TOPMed rs1320012247, gnomAD rs1320012247, REVEL 0.06, CADD 17.60
- R38S (p.Arg38Ser), ESP rs375084708, ExAC rs375084708, TOPMed rs375084708, gnomAD rs375084708, REVEL 0.02, CADD 22.70
- R38R (p.Arg38Arg), gnomAD 1-201283816-G-A, CADD 13.60
- Q39E (p.Gln39Glu), ExAC rs747869491, TOPMed rs747869491, gnomAD rs747869491, REVEL 0.12, CADD 25.50
- Q39H (p.Gln39His), Ensembl rs1571533976
- R40H (p.Arg40His), cosmic curated COSV10584, NCI-TCGA TCGA novel, REVEL 0.33, CADD 31.00, Variant assessed as somatic; moderate impact.
- R40R (p.Arg40Arg), rs1655645174, gnomAD 1-201283822-C-G, CADD 11.80
- V41L (p.Val41Leu), TOPMed rs1655645247, REVEL 0.17, CADD 24.60
- V41M (p.Val41Met), NCI-TCGA TCGA novel, Variant assessed as somatic; moderate impact.
- V41V (p.Val41Val), rs771727242, gnomAD 1-201283825-G-A, CADD 12.50
- Q42R (p.Gln42Arg), gnomAD 1-201283827-A-G, REVEL 0.04, CADD 21.50
- Q42Q (p.Gln42Gln), gnomAD 1-201283828-G-A, CADD 11.60
- E43K (p.Glu43Lys), rs1390932890, ClinGen CA344168644, ClinVar RCV004509265, TOPMed rs1390932890, REVEL 0.25, CADD 31.00, Uncertain significance, Inborn genetic diseases
- Q44H (p.Gln44His), cosmic curated COSV10802
- V45E (p.Val45Glu), gnomAD 1-201283836-T-A, REVEL 0.22, CADD 31.00
- V45V (p.Val45Val), rs1234362892, gnomAD 1-201283837-G-A, CADD 17.20
- M46L (p.Met46Leu), rs369806569, ClinGen CA1323975, ClinVar RCV000266980, ClinVar RCV004021410, REVEL 0.05, CADD 23.60, Uncertain significance, Inborn genetic diseases; Epidermolysis bullosa simplex due to plakophilin defici
- M46T (p.Met46Thr), cosmic curated COSV55825
- M46V (p.Met46Val), ExAC rs369806569, TOPMed rs369806569, gnomAD rs369806569, Uncertain significance
- M46* (p.Met46Ter), gnomAD 1-201283837-GA-G, CADD 29.00
- M47V (p.Met47Val), gnomAD rs1482053129, REVEL 0.10, CADD 25.40
- M47del (p.Met47del), rs1655645507, gnomAD 1-201283835-GTGA-, CADD 21.70
- T48N (p.Thr48Asn), Ensembl rs1655646237, REVEL 0.19, CADD 25.90
- T48T (p.Thr48Thr), rs1207292943, gnomAD 1-201283846-C-A, CADD 11.30
- V49I (p.Val49Ile), gnomAD 1-201283847-G-A, REVEL 0.09, CADD 22.30
- V49A (p.Val49Ala), gnomAD 1-201283848-T-C, REVEL 0.13, CADD 29.30
- V49V (p.Val49Val), gnomAD 1-201283849-C-T, CADD 14.70
- K50E (p.Lys50Glu), gnomAD 1-201283850-A-G, REVEL 0.02, CADD 24.40
- R51W (p.Arg51Trp), rs181972441, ClinGen CA36019373, ClinVar RCV003374414, 1000Genomes rs181972441, REVEL 0.20, CADD 27.10, Uncertain significance, Inborn genetic diseases
- R51R (p.Arg51Arg), rs181972441, gnomAD 1-201283853-C-A, CADD 14.20
- R51Q (p.Arg51Gln), gnomAD 1-201283854-G-A, REVEL 0.24, CADD 32.00
- Q52H (p.Gln52His), TOPMed rs1397427322, gnomAD rs1397427322, REVEL 0.03, CADD 22.60
- Q52K (p.Gln52Lys), rs200433412, ClinGen CA1323977, ClinVar RCV004509269, ESP rs200433412, REVEL 0.10, CADD 22.70, Uncertain significance, Inborn genetic diseases
- Q52R (p.Gln52Arg), cosmic curated COSV55821, gnomAD rs1187154159, REVEL 0.07, CADD 23.00
- Q52Q (p.Gln52Gln), gnomAD 1-201283858-G-A, CADD 13.80
- K53E (p.Lys53Glu), TOPMed rs1422306414
- K53N (p.Lys53Asn), NCI-TCGA Cosmic COSV5582, Variant assessed as somatic; moderate impact.
- K53R (p.Lys53Arg), 1000Genomes rs199797410, ExAC rs199797410, REVEL 0.12, CADD 31.00
- K53K (p.Lys53Lys), gnomAD 1-201283861-G-A, CADD 13.90
- S54A (p.Ser54Ala), Ensembl rs1571534023
- S54C (p.Ser54Cys), ExAC rs765625993, gnomAD rs765625993, REVEL 0.07, CADD 27.10
- S54F (p.Ser54Phe), gnomAD 1-201283863-C-T, REVEL 0.08, CADD 27.80
- S54S (p.Ser54Ser), gnomAD 1-201283864-C-A, CADD 15.30
- K55R (p.Lys55Arg), NCI-TCGA Cosmic COSV9982, cosmic curated COSV99825, Variant assessed as somatic; moderate impact.
- K55N (p.Lys55Asn), gnomAD 1-201283867-G-T, REVEL 0.11, CADD 26.00
- S56C (p.Ser56Cys), ExAC rs776004636, gnomAD rs776004636, REVEL 0.33, CADD 24.90
- S56F (p.Ser56Phe), gnomAD 1-201283869-C-T, REVEL 0.25, CADD 23.30
- S57F (p.Ser57Phe), cosmic curated COSV55826
- S57P (p.Ser57Pro), gnomAD rs1376177104, REVEL 0.15, CADD 25.80
- S57T (p.Ser57Thr), gnomAD 1-201283871-T-A, REVEL 0.06, CADD 26.40
- Q58H (p.Gln58His), 1000Genomes rs560818766, ExAC rs560818766, gnomAD rs560818766, REVEL 0.06, CADD 22.80
- Q58L (p.Gln58Leu), gnomAD 1-201283875-A-T, REVEL 0.08, CADD 23.30
- Q58R (p.Gln58Arg), gnomAD 1-201283875-A-G, REVEL 0.06, CADD 22.50
- S59L (p.Ser59Leu), NCI-TCGA TCGA novel, Variant assessed as somatic; moderate impact.
- S59W (p.Ser59Trp), gnomAD 1-201283878-C-G, REVEL 0.21, CADD 31.00
- S59S (p.Ser59Ser), gnomAD 1-201283879-G-C, CADD 14.60
- S60F (p.Ser60Phe), ExAC rs751867536, gnomAD rs751867536, REVEL 0.22, CADD 29.50
- S60P (p.Ser60Pro), ExAC rs764546449, gnomAD rs764546449
- S60S (p.Ser60Ser), gnomAD 1-201283882-C-T, CADD 15.30
- T61S (p.Thr61Ser), cosmic curated COSV10608
- T61T (p.Thr61Thr), rs757527243, gnomAD 1-201283885-C-T, CADD 7.60
- L62L (p.Leu62Leu), rs767657184, gnomAD 1-201283888-G-A, CADD 13.80
- S63R (p.Ser63Arg), gnomAD rs1441056275
- H64R (p.His64Arg), gnomAD rs1310097346, REVEL 0.08, CADD 22.60
- H64Y (p.His64Tyr), 1000Genomes rs529709320, ExAC rs529709320, TOPMed rs529709320, gnomAD rs529709320, REVEL 0.04, CADD 17.80
- H64L (p.His64Leu), gnomAD 1-201283893-A-T, REVEL 0.09, CADD 22.70
- S65S (p.Ser65Ser), rs754945554, gnomAD 1-201283897-C-A, CADD 14.90
- N66I (p.Asn66Ile), TOPMed rs1261682384, gnomAD rs1261682384, REVEL 0.07, CADD 23.10
- N66S (p.Asn66Ser), TOPMed rs1261682384, gnomAD rs1261682384, REVEL 0.05, CADD 19.00
- N66D (p.Asn66Asp), gnomAD 1-201283898-A-G, REVEL 0.08, CADD 23.40
- N66K (p.Asn66Lys), gnomAD 1-201283900-T-A, REVEL 0.05, CADD 18.80
- R67* (p.Arg67Ter), ExAC rs778655336, TOPMed rs778655336, gnomAD rs778655336, Likely benign
- R67Q (p.Arg67Gln), NCI-TCGA Cosmic COSV5581, cosmic curated COSV55819, Variant assessed as somatic; moderate impact.
- R67R (p.Arg67Arg), rs778655336, gnomAD 1-201283901-C-A, CADD 16.00
- G68D (p.Gly68Asp), cosmic curated COSV10584, TOPMed rs1407567667, gnomAD rs1407567667, REVEL 0.23, CADD 31.00
- G68S (p.Gly68Ser), ExAC rs748066233, gnomAD rs748066233, REVEL 0.16, CADD 34.00
- S69F (p.Ser69Phe), cosmic curated COSV10723
- S69T (p.Ser69Thr), rs2526690686, ClinGen CA344170837, ClinVar RCV004509270, REVEL 0.15, CADD 23.20, Uncertain significance, Inborn genetic diseases
- S69Y (p.Ser69Tyr), rs2526690691, ClinGen CA344170840, ClinVar RCV004509271, REVEL 0.21, CADD 23.70, Uncertain significance, Inborn genetic diseases
- M70R (p.Met70Arg), TOPMed rs1656000977
- M70V (p.Met70Val), ExAC rs777570553, TOPMed rs777570553, gnomAD rs777570553, REVEL 0.31, CADD 15.80, Uncertain significance, not provided; Epidermolysis bullosa simplex due to plakophilin deficiency
- M70T (p.Met70Thr), gnomAD 1-201293948-T-C, REVEL 0.24, CADD 20.10
- M70K (p.Met70Lys), gnomAD 1-201293948-T-A, REVEL 0.31, CADD 23.20
- Y71C (p.Tyr71Cys), ExAC rs751318467, gnomAD rs751318467, REVEL 0.49, CADD 27.60
- Y71F (p.Tyr71Phe), ExAC rs751318467, gnomAD rs751318467, REVEL 0.32, CADD 23.90
- Y71* (p.Tyr71Ter), gnomAD 1-201293952-T-G, CADD 28.70
- D72E (p.Asp72Glu), gnomAD rs1228107718, REVEL 0.36, CADD 13.80
- D72N (p.Asp72Asn), NCI-TCGA TCGA novel, REVEL 0.25, CADD 25.10, Variant assessed as somatic; moderate impact.
- G73G (p.Gly73Gly), rs756941010, gnomAD 1-201293958-C-T, CADD 10.30
- L74F (p.Leu74Phe), TOPMed rs1656001529
Public PKP1 analysis runs
- PKP1 analysis run — PKP1 (1,386 variants) — completed 2026-08-22