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RYR1-related myopathy

MedGen UID:
987079
Concept ID:
CN305348
Disease or Syndrome
Monarch Initiative: MONDO:0100150

Definition

A disorder of the musculoskeletal system caused by pathogenic variants in the RYR1 gene, which encodes the ryanodine receptor type 1 protein. These variants are associated with a variety of overlapping features characterized by symmetric proximal muscle weakness, often with pronounced facial weakness with or without dysmorphism and ophthalmoparesis/ophthalmoplegia with ptosis, bulbar weakness, significant respiratory involvement, severe neonatal hypotonia, scoliosis, orthopedic deformities including arthrogryposis, hip dislocation, club feet, and King Denborough syndrome (pectus carinatum or excavatum, short stature, joint contractures, facial and skeletal deformities), malignant hyperthermia susceptibility, anesthesia-induced rhabdomyolysis, fatigue, exercise-induced hyperthermia/exertional heat stroke, and exertional myalgia. Histologic findings on skeletal muscle biopsy reveal a wide range of structural abnormalities and can include central core disease, multiminicore disease, cone-rod myopathy, centronuclear myopathy, and congenital fiber-type disproportion. [from MONDO]

Professional guidelines

PubMed

Dosi C, Rubegni A, Baldacci J, Galatolo D, Doccini S, Astrea G, Berardinelli A, Bruno C, Bruno G, Comi GP, Donati MA, Dotti MT, Filosto M, Fiorillo C, Giannini F, Gigli GL, Grandis M, Lopergolo D, Magri F, Maioli MA, Malandrini A, Massa R, Matà S, Melani F, Messina S, Mignarri A, Moggio M, Pennisi EM, Pegoraro E, Ricci G, Sacchini M, Schenone A, Sampaolo S, Sciacco M, Siciliano G, Tasca G, Tonin P, Tupler R, Valente M, Volpi N, Cassandrini D, Santorelli FM
Genes (Basel) 2023 Jan 23;14(2) doi: 10.3390/genes14020298. PMID: 36833224Free PMC Article
Dowling JJ, Arbogast S, Hur J, Nelson DD, McEvoy A, Waugh T, Marty I, Lunardi J, Brooks SV, Kuwada JY, Ferreiro A
Brain 2012 Apr;135(Pt 4):1115-27. Epub 2012 Mar 14 doi: 10.1093/brain/aws036. PMID: 22418739Free PMC Article

Recent clinical studies

Etiology

Ogasawara M, Nishino I
Curr Opin Neurol 2024 Oct 1;37(5):504-508. Epub 2024 Jul 12 doi: 10.1097/WCO.0000000000001296. PMID: 38994695
Sarkozy A, Sa M, Ridout D, Fernandez-Garcia MA, Distefano MG, Main M, Sheehan J, Manzur AY, Munot P, Robb S, Wraige E, Quinlivan R, Scoto M, Baranello G, Gowda V, Mein R, Phadke R, Jungbluth H, Muntoni F
Neurology 2023 Oct 10;101(15):e1495-e1508. Epub 2023 Aug 29 doi: 10.1212/WNL.0000000000207723. PMID: 37643885Free PMC Article
Dosi C, Rubegni A, Baldacci J, Galatolo D, Doccini S, Astrea G, Berardinelli A, Bruno C, Bruno G, Comi GP, Donati MA, Dotti MT, Filosto M, Fiorillo C, Giannini F, Gigli GL, Grandis M, Lopergolo D, Magri F, Maioli MA, Malandrini A, Massa R, Matà S, Melani F, Messina S, Mignarri A, Moggio M, Pennisi EM, Pegoraro E, Ricci G, Sacchini M, Schenone A, Sampaolo S, Sciacco M, Siciliano G, Tasca G, Tonin P, Tupler R, Valente M, Volpi N, Cassandrini D, Santorelli FM
Genes (Basel) 2023 Jan 23;14(2) doi: 10.3390/genes14020298. PMID: 36833224Free PMC Article
van den Bersselaar LR, Hellblom A, Gashi M, Kamsteeg EJ, Voermans NC, Jungbluth H, de Puydt J, Heytens L, Riazi S, Snoeck MMJ
Anesthesiology 2022 Jun 1;136(6):940-953. doi: 10.1097/ALN.0000000000004199. PMID: 35285867
Hayakawa I, Abe Y, Ono H, Kubota M
Ital J Pediatr 2019 Dec 19;45(1):165. doi: 10.1186/s13052-019-0756-1. PMID: 31856875Free PMC Article

Diagnosis

Dosi C, Rubegni A, Baldacci J, Galatolo D, Doccini S, Astrea G, Berardinelli A, Bruno C, Bruno G, Comi GP, Donati MA, Dotti MT, Filosto M, Fiorillo C, Giannini F, Gigli GL, Grandis M, Lopergolo D, Magri F, Maioli MA, Malandrini A, Massa R, Matà S, Melani F, Messina S, Mignarri A, Moggio M, Pennisi EM, Pegoraro E, Ricci G, Sacchini M, Schenone A, Sampaolo S, Sciacco M, Siciliano G, Tasca G, Tonin P, Tupler R, Valente M, Volpi N, Cassandrini D, Santorelli FM
Genes (Basel) 2023 Jan 23;14(2) doi: 10.3390/genes14020298. PMID: 36833224Free PMC Article
Ogasawara M, Nishino I
Neuromuscul Disord 2021 Oct;31(10):968-977. Epub 2021 Sep 17 doi: 10.1016/j.nmd.2021.08.015. PMID: 34627702
Hayakawa I, Abe Y, Ono H, Kubota M
Ital J Pediatr 2019 Dec 19;45(1):165. doi: 10.1186/s13052-019-0756-1. PMID: 31856875Free PMC Article
Matthews E, Neuwirth C, Jaffer F, Scalco RS, Fialho D, Parton M, Raja Rayan D, Suetterlin K, Sud R, Spiegel R, Mein R, Houlden H, Schaefer A, Healy E, Palace J, Quinlivan R, Treves S, Holton JL, Jungbluth H, Hanna MG
Neurology 2018 Jan 30;90(5):e412-e418. Epub 2018 Jan 3 doi: 10.1212/WNL.0000000000004894. PMID: 29298851Free PMC Article
Laughlin RS, Niu Z, Wieben E, Milone M
Mol Genet Genomic Med 2017 Nov;5(6):800-804. Epub 2017 Oct 4 doi: 10.1002/mgg3.338. PMID: 29178655Free PMC Article

Therapy

Kushnir A, Todd JJ, Witherspoon JW, Yuan Q, Reiken S, Lin H, Munce RH, Wajsberg B, Melville Z, Clarke OB, Wedderburn-Pugh K, Wronska A, Razaqyar MS, Chrismer IC, Shelton MO, Mankodi A, Grunseich C, Tarnopolsky MA, Tanji K, Hirano M, Riazi S, Kraeva N, Voermans NC, Gruber A, Allen C, Meilleur KG, Marks AR
Acta Neuropathol 2020 Jun;139(6):1089-1104. Epub 2020 Mar 31 doi: 10.1007/s00401-020-02150-w. PMID: 32236737Free PMC Article
Hayakawa I, Abe Y, Ono H, Kubota M
Ital J Pediatr 2019 Dec 19;45(1):165. doi: 10.1186/s13052-019-0756-1. PMID: 31856875Free PMC Article
Illingworth MA, Main M, Pitt M, Feng L, Sewry CA, Gunny R, Vorstman E, Beeson D, Manzur A, Muntoni F, Robb SA
Neuromuscul Disord 2014 Aug;24(8):707-12. Epub 2014 May 23 doi: 10.1016/j.nmd.2014.05.003. PMID: 24951453
Dowling JJ, Arbogast S, Hur J, Nelson DD, McEvoy A, Waugh T, Marty I, Lunardi J, Brooks SV, Kuwada JY, Ferreiro A
Brain 2012 Apr;135(Pt 4):1115-27. Epub 2012 Mar 14 doi: 10.1093/brain/aws036. PMID: 22418739Free PMC Article

Prognosis

Janßen S, Erbe LS, Kneifel M, Vorgerd M, Döring K, Lubieniecki KP, Lubieniecka JM, Gerding WM, Casadei N, Güttsches AK, Heyer C, Lücke T, Nguyen HHP, Köhler C, Hoffjan S
Int J Mol Sci 2024 Oct 9;25(19) doi: 10.3390/ijms251910867. PMID: 39409197Free PMC Article
Sarkozy A, Sa M, Ridout D, Fernandez-Garcia MA, Distefano MG, Main M, Sheehan J, Manzur AY, Munot P, Robb S, Wraige E, Quinlivan R, Scoto M, Baranello G, Gowda V, Mein R, Phadke R, Jungbluth H, Muntoni F
Neurology 2023 Oct 10;101(15):e1495-e1508. Epub 2023 Aug 29 doi: 10.1212/WNL.0000000000207723. PMID: 37643885Free PMC Article
Kushnir A, Todd JJ, Witherspoon JW, Yuan Q, Reiken S, Lin H, Munce RH, Wajsberg B, Melville Z, Clarke OB, Wedderburn-Pugh K, Wronska A, Razaqyar MS, Chrismer IC, Shelton MO, Mankodi A, Grunseich C, Tarnopolsky MA, Tanji K, Hirano M, Riazi S, Kraeva N, Voermans NC, Gruber A, Allen C, Meilleur KG, Marks AR
Acta Neuropathol 2020 Jun;139(6):1089-1104. Epub 2020 Mar 31 doi: 10.1007/s00401-020-02150-w. PMID: 32236737Free PMC Article
Hayakawa I, Abe Y, Ono H, Kubota M
Ital J Pediatr 2019 Dec 19;45(1):165. doi: 10.1186/s13052-019-0756-1. PMID: 31856875Free PMC Article
Attali R, Aharoni S, Treves S, Rokach O, Becker Cohen M, Fellig Y, Straussberg R, Dor T, Daana M, Mitrani-Rosenbaum S, Nevo Y
PLoS One 2013;8(7):e69296. Epub 2013 Jul 24 doi: 10.1371/journal.pone.0069296. PMID: 23894444Free PMC Article

Clinical prediction guides

Sarkozy A, Sa M, Ridout D, Fernandez-Garcia MA, Distefano MG, Main M, Sheehan J, Manzur AY, Munot P, Robb S, Wraige E, Quinlivan R, Scoto M, Baranello G, Gowda V, Mein R, Phadke R, Jungbluth H, Muntoni F
Neurology 2023 Oct 10;101(15):e1495-e1508. Epub 2023 Aug 29 doi: 10.1212/WNL.0000000000207723. PMID: 37643885Free PMC Article
van den Bersselaar LR, Hellblom A, Gashi M, Kamsteeg EJ, Voermans NC, Jungbluth H, de Puydt J, Heytens L, Riazi S, Snoeck MMJ
Anesthesiology 2022 Jun 1;136(6):940-953. doi: 10.1097/ALN.0000000000004199. PMID: 35285867
Kushnir A, Todd JJ, Witherspoon JW, Yuan Q, Reiken S, Lin H, Munce RH, Wajsberg B, Melville Z, Clarke OB, Wedderburn-Pugh K, Wronska A, Razaqyar MS, Chrismer IC, Shelton MO, Mankodi A, Grunseich C, Tarnopolsky MA, Tanji K, Hirano M, Riazi S, Kraeva N, Voermans NC, Gruber A, Allen C, Meilleur KG, Marks AR
Acta Neuropathol 2020 Jun;139(6):1089-1104. Epub 2020 Mar 31 doi: 10.1007/s00401-020-02150-w. PMID: 32236737Free PMC Article
Witherspoon JW, Vuillerot C, Vasavada RP, Waite MR, Shelton M, Chrismer IC, Jain MS, Meilleur KG
Muscle Nerve 2019 Jul;60(1):80-87. doi: 10.1002/mus.26491. PMID: 31004442Free PMC Article

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