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Kyphosis-lateral tongue atrophy-myofibrillar myopathy syndrome

MedGen UID:
1798876
Concept ID:
C5567453
Disease or Syndrome
Synonyms: Kyphosis, lateral tongue atrophy, myofibrillar myopathy syndrome; kyphosis-lateral tongue atrophy-myofibrillar myopathy syndrome
SNOMED CT: Kyphosis, lateral tongue atrophy, myofibrillar myopathy syndrome (1172591008)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Monarch Initiative: MONDO:0044647
Orphanet: ORPHA496686

Definition

A rare genetic skeletal muscle disease with characteristics of neonatal to childhood onset of slowly progressive muscle weakness and atrophy primarily affecting the lower limbs, joint contractures, kyphosis or lordosis of the spine, lateral tongue atrophy, and pes equinus. In addition progression to upper limb involvement, facial weakness, language impairment, intellectual disability, and behavioural abnormalities has been reported. Muscle biopsy shows myopathic changes with increased fibre size variation, internalised nuclei, fibre atrophy, as well as rod structures and core targetoid defects. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVKyphosis-lateral tongue atrophy-myofibrillar myopathy syndrome

Professional guidelines

PubMed

Kozakovičová V, Onderka A, Res O, Stránský J, Kondé A, Štembírek J
Acta Chir Plast 2023;65(3-4):98-105. doi: 10.48095/ccachp202398. PMID: 38538296
Yanmaz MN, Atar S, Biçer M
J Back Musculoskelet Rehabil 2016 Apr 27;29(2):287-293. doi: 10.3233/BMR-150627. PMID: 26406206
Wan KY, McMillan AS, Wong MC
Community Dent Health 2012 Mar;29(1):110-6. PMID: 22482261

Recent clinical studies

Etiology

Kozakovičová V, Onderka A, Res O, Stránský J, Kondé A, Štembírek J
Acta Chir Plast 2023;65(3-4):98-105. doi: 10.48095/ccachp202398. PMID: 38538296
Farpour HR, Moosavi SA, Mohammadian Z, Farpour S
Dysphagia 2022 Aug;37(4):966-972. Epub 2021 Aug 25 doi: 10.1007/s00455-021-10359-4. PMID: 34432140
Zanin MC, Garcia DM, Rocha EM, de Felício CM
Arthritis Care Res (Hoboken) 2020 Aug;72(8):1057-1065. doi: 10.1002/acr.24001. PMID: 31207153
Gurwood AS, Drake J
Optometry 2006 Nov;77(11):540-6. doi: 10.1016/j.optm.2006.06.014. PMID: 17145565
Rosenow F, McCarthy V, Caruso AC
J Sleep Res 1998 Mar;7(1):3-11. doi: 10.1046/j.1365-2869.1998.00086.x. PMID: 9613423

Diagnosis

Kozakovičová V, Onderka A, Res O, Stránský J, Kondé A, Štembírek J
Acta Chir Plast 2023;65(3-4):98-105. doi: 10.48095/ccachp202398. PMID: 38538296
Dietrich E, Grimaux X, Martin L, Samimi M
Ann Dermatol Venereol 2022 Dec;149(4):228-237. Epub 2022 Oct 11 doi: 10.1016/j.annder.2022.03.011. PMID: 36229262
Elbarbary M, Oren A, Goldberg M, Freeman BV, Mock D, Tenenbaum HC, Azarpazhooh A
J Endod 2022 Jan;48(1):55-69. Epub 2021 Oct 25 doi: 10.1016/j.joen.2021.10.004. PMID: 34710470
Lauria G
Curr Opin Neurol 2005 Oct;18(5):591-7. doi: 10.1097/01.wco.0000177330.35147.70. PMID: 16155446
Abdollahi M, Radfar M
J Contemp Dent Pract 2003 Feb 15;4(1):10-31. PMID: 12595930

Therapy

Shimizu M, Kaneda T, Sekino N, Nishizawa T
BMJ Case Rep 2021 Apr 13;14(4) doi: 10.1136/bcr-2021-242283. PMID: 33849884Free PMC Article
Fernandez-Nieto D, Burgos-Blasco P, Jimenez-Cauhe J, Ortega-Quijano D, Taboada-Illan J, Bea-Ardebol S
Am J Med 2020 Nov;133(11):1283-1286. Epub 2020 Apr 8 doi: 10.1016/j.amjmed.2020.02.050. PMID: 32277887
de Silva NL, Weeratunga P, Umapathi T, Malavige N, Chang T
J Med Case Rep 2019 May 2;13(1):120. doi: 10.1186/s13256-019-2066-z. PMID: 31043165Free PMC Article
Morgan AT, Dodrill P, Ward EC
Cochrane Database Syst Rev 2012 Oct 17;10:CD009456. doi: 10.1002/14651858.CD009456.pub2. PMID: 23076958
Orlikowski D, Terzi N, Blumen M, Sharshar T, Raphael JC, Annane D, Lofaso F
Acta Neurol Scand 2009 Jun;119(6):364-70. Epub 2008 Oct 22 doi: 10.1111/j.1600-0404.2008.01107.x. PMID: 18976323

Prognosis

Motoda A, Takahashi T, Watanabe C, Tachiyama Y, Ochi K, Saito Y, Iida A, Nishino I, Maruyama H
Neuromuscul Disord 2021 Nov;31(11):1220-1225. Epub 2021 Jul 20 doi: 10.1016/j.nmd.2021.07.011. PMID: 34635388
Pinto WBVR, Naylor FGM, Chieia MAT, de Souza PVS, Oliveira ASB
Rev Neurol (Paris) 2019 Apr;175(4):238-246. Epub 2018 Oct 5 doi: 10.1016/j.neurol.2018.04.010. PMID: 30293881
Orlikowski D, Terzi N, Blumen M, Sharshar T, Raphael JC, Annane D, Lofaso F
Acta Neurol Scand 2009 Jun;119(6):364-70. Epub 2008 Oct 22 doi: 10.1111/j.1600-0404.2008.01107.x. PMID: 18976323
Gurwood AS, Drake J
Optometry 2006 Nov;77(11):540-6. doi: 10.1016/j.optm.2006.06.014. PMID: 17145565
Mao CC, Coull BM, Golper LA, Rau MT
Neurology 1989 Sep;39(9):1169-72. doi: 10.1212/wnl.39.9.1169. PMID: 2771066

Clinical prediction guides

O'Connor-Reina C, Rodriguez-Alcala L, Ignacio JM, Baptista P, Garcia-Iriarte MT, Plaza G
Otolaryngol Head Neck Surg 2023 Sep;169(3):725-733. Epub 2023 Feb 7 doi: 10.1002/ohn.283. PMID: 36939539
Zanin MC, Garcia DM, Rocha EM, de Felício CM
Arthritis Care Res (Hoboken) 2020 Aug;72(8):1057-1065. doi: 10.1002/acr.24001. PMID: 31207153
Pinto WBVR, Naylor FGM, Chieia MAT, de Souza PVS, Oliveira ASB
Rev Neurol (Paris) 2019 Apr;175(4):238-246. Epub 2018 Oct 5 doi: 10.1016/j.neurol.2018.04.010. PMID: 30293881
McGuigan M
Homeopathy 2014 Jul;103(3):198-202. doi: 10.1016/j.homp.2014.01.002. PMID: 24931752
Ruiz-Sandoval JL, García-Navarro V, Chiquete E, Dobson-Stone C, Monaco AP, Alvarez-Palazuelos LE, Padilla-Martínez JJ, Barrera-Chairez E, Rodríguez-Figueroa EI, Pérez-García G
Arch Neurol 2007 Nov;64(11):1661-4. doi: 10.1001/archneur.64.11.1661. PMID: 17998451

Recent systematic reviews

Dietrich E, Grimaux X, Martin L, Samimi M
Ann Dermatol Venereol 2022 Dec;149(4):228-237. Epub 2022 Oct 11 doi: 10.1016/j.annder.2022.03.011. PMID: 36229262
Morgan AT, Dodrill P, Ward EC
Cochrane Database Syst Rev 2012 Oct 17;10:CD009456. doi: 10.1002/14651858.CD009456.pub2. PMID: 23076958

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