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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

Ali NE, Alyono JC, Kumar AR, Cheng H, Koltai PJ
Am J Otolaryngol 2020 Jul-Aug;41(4):102566. Epub 2020 May 27 doi: 10.1016/j.amjoto.2020.102566. PMID: 32504854
Arakawa M, Yamazaki M, Toda Y, Ozawa A, Kimura K
Intern Med 2020 May 1;59(9):1215-1217. Epub 2020 Feb 5 doi: 10.2169/internalmedicine.3395-19. PMID: 32023583Free PMC Article
Batifol D, Huart A, Finiels PJ, Nagot N, Jammet P
J Stomatol Oral Maxillofac Surg 2018 Sep;119(4):319-324. Epub 2018 Jun 8 doi: 10.1016/j.jormas.2018.06.002. PMID: 29885911
Simões WA
Cranio 1999 Apr;17(2):109-25. doi: 10.1080/08869634.1999.11746085. PMID: 10425938
Sonies BC, Dalakas MC
N Engl J Med 1991 Apr 25;324(17):1162-7. doi: 10.1056/NEJM199104253241703. PMID: 2011159

Diagnosis

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
Arakawa M, Yamazaki M, Toda Y, Ozawa A, Kimura K
Intern Med 2020 May 1;59(9):1215-1217. Epub 2020 Feb 5 doi: 10.2169/internalmedicine.3395-19. PMID: 32023583Free PMC Article
Garibaldi M, Pennisi EM, Bruttini M, Bizzarri V, Bucci E, Morino S, Talerico C, Stoppacciaro A, Renieri A, Antonini G
Neuromuscul Disord 2015 Nov;25(11):869-72. Epub 2015 Sep 7 doi: 10.1016/j.nmd.2015.08.011. PMID: 26494409
Simões WA
Cranio 1999 Apr;17(2):109-25. doi: 10.1080/08869634.1999.11746085. PMID: 10425938
Friedlander AH, Runyon C
Oral Surg Oral Med Oral Pathol 1990 Mar;69(3):317-21. doi: 10.1016/0030-4220(90)90292-z. PMID: 2138275

Therapy

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
Arakawa M, Yamazaki M, Toda Y, Ozawa A, Kimura K
Intern Med 2020 May 1;59(9):1215-1217. Epub 2020 Feb 5 doi: 10.2169/internalmedicine.3395-19. PMID: 32023583Free PMC Article
Batifol D, Huart A, Finiels PJ, Nagot N, Jammet P
J Stomatol Oral Maxillofac Surg 2018 Sep;119(4):319-324. Epub 2018 Jun 8 doi: 10.1016/j.jormas.2018.06.002. PMID: 29885911
McGuigan M
Homeopathy 2014 Jul;103(3):198-202. doi: 10.1016/j.homp.2014.01.002. PMID: 24931752
Cummings NA
Rheumatology 1973;4(0):60-97. PMID: 4600423

Prognosis

Rousseff RT, Khuraibet AJ, Neubauer D
Neuropediatrics 2008 Dec;39(6):354-6. Epub 2009 Jun 30 doi: 10.1055/s-0029-1202768. PMID: 19569002
Gurwood AS, Drake J
Optometry 2006 Nov;77(11):540-6. doi: 10.1016/j.optm.2006.06.014. PMID: 17145565
Finsterer J, Hess B
Int J Neurosci 2004 Dec;114(12):1573-80. doi: 10.1080/00207450490509311. PMID: 15512840
Sonies BC, Dalakas MC
N Engl J Med 1991 Apr 25;324(17):1162-7. doi: 10.1056/NEJM199104253241703. PMID: 2011159
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

Ali NE, Alyono JC, Kumar AR, Cheng H, Koltai PJ
Am J Otolaryngol 2020 Jul-Aug;41(4):102566. Epub 2020 May 27 doi: 10.1016/j.amjoto.2020.102566. PMID: 32504854
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
McGuigan M
Homeopathy 2014 Jul;103(3):198-202. doi: 10.1016/j.homp.2014.01.002. PMID: 24931752
Rousseff RT, Khuraibet AJ, Neubauer D
Neuropediatrics 2008 Dec;39(6):354-6. Epub 2009 Jun 30 doi: 10.1055/s-0029-1202768. PMID: 19569002
Corbett CE, El Khouri M, Costa AN, Gyuricza JV, Corbett JF, Frizzarini R, de Araújo Andrade DC, Cordeiro Q, Stravogiannis A, Chassot CA, Vieira JL, Pinheiro Mda C
Arch Environ Occup Health 2007 Fall;62(3):121-8. doi: 10.3200/AEOH.62.3.121-128. PMID: 18400651

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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