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Sialidosis type 1

MedGen UID:
44174
Concept ID:
C0023806
Disease or Syndrome
Synonyms: Cherry red spot myoclonus syndrome; Sialidosis type I
SNOMED CT: Sialidosis type 1 (723675006); Cherry red spot myoclonus syndrome (723675006); Lipomucopolysaccharidosis (723675006); Normomorphic sialidosis (723675006)
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:0019346
OMIM®: 256550
Orphanet: ORPHA812

Definition

Sialidosis is a severe inherited disorder that affects many organs and tissues, including the nervous system. This disorder is divided into two types, which are distinguished by the age at which symptoms appear and the severity of features.

Sialidosis type I, also referred to as cherry-red spot myoclonus syndrome, is the less severe form of this condition. People with type I develop signs and symptoms of sialidosis in their teens or twenties. Initially, affected individuals experience problems walking (gait disturbance) and/or a loss of sharp vision (reduced visual acuity). Individuals with sialidosis type I also experience muscle twitches (myoclonus), difficulty coordinating movements (ataxia), leg tremors, and seizures. The myoclonus worsens over time, causing difficulty sitting, standing, or walking. People with sialidosis type I eventually require wheelchair assistance. Affected individuals have progressive vision problems, including impaired color vision or night blindness. An eye abnormality called a cherry-red spot, which can be identified with an eye examination, is characteristic of this disorder. Sialidosis type I does not affect intelligence or life expectancy.

Sialidosis type II, the more severe type of the disorder, is further divided into congenital, infantile, and juvenile forms. The features of congenital sialidosis type II can develop before birth. This form of sialidosis is associated with an abnormal buildup of fluid in the abdominal cavity (ascites) or widespread swelling before birth caused by fluid accumulation (hydrops fetalis). Affected infants may also have an enlarged liver and spleen (hepatosplenomegaly), abnormal bone development (dysostosis multiplex), and distinctive facial features that are often described as "coarse." As a result of these serious health problems, individuals with congenital sialidosis type II usually are stillborn or die soon after birth.

Infantile sialidosis type II shares some features with the congenital form, although the signs and symptoms are slightly less severe and begin within the first year of life. Features of the infantile form include hepatosplenomegaly, dysostosis multiplex, "coarse" facial features, short stature, and intellectual disability. As children with infantile sialidosis type II get older, they may develop myoclonus and cherry-red spots. Other signs and symptoms include hearing loss, overgrowth of the gums (gingival hyperplasia), and widely spaced teeth. Affected individuals may survive into childhood or adolescence.

The juvenile form has the least severe signs and symptoms of the different forms of sialidosis type II. Features of this condition usually appear in late childhood and may include mildly "coarse" facial features, mild bone abnormalities, cherry-red spots, myoclonus, intellectual disability, and dark red spots on the skin (angiokeratomas). The life expectancy of individuals with juvenile sialidosis type II varies depending on the severity of symptoms. [from MedlinePlus Genetics]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
Follow this link to review classifications for Sialidosis type 1 in Orphanet.

Professional guidelines

PubMed

Du YC, Ma LH, Li QF, Ma Y, Dong Y, Wu ZY
Orphanet J Rare Dis 2024 Sep 30;19(1):362. doi: 10.1186/s13023-024-03378-5. PMID: 39350194Free PMC Article
Riboldi GM, Martone J, Rizzo JR, Hudson TE, Rucker JC, Frucht SJ
Tremor Other Hyperkinet Mov (N Y) 2021;11:53. Epub 2021 Dec 9 doi: 10.5334/tohm.652. PMID: 34992946Free PMC Article

Recent clinical studies

Etiology

Gultekin M, Bayramov R, Karaca C, Acer N
Neurosciences (Riyadh) 2018 Jan;23(1):57-61. doi: 10.17712/nsj.2018.1.20170328. PMID: 29455223Free PMC Article
Lai SC, Chen RS, Wu Chou YH, Chang HC, Kao LY, Huang YZ, Weng YH, Chen JK, Hwu WL, Lu CS
Eur J Neurol 2009 Aug;16(8):912-9. Epub 2009 Apr 14 doi: 10.1111/j.1468-1331.2009.02622.x. PMID: 19473359

Diagnosis

Wang F, Lin L, Hu J, Zhang J, Wang K
Int J Neurosci 2022 Jun;132(6):589-592. Epub 2020 Oct 13 doi: 10.1080/00207454.2020.1829615. PMID: 32988250
Riboldi GM, Martone J, Rizzo JR, Hudson TE, Rucker JC, Frucht SJ
Tremor Other Hyperkinet Mov (N Y) 2021;11:53. Epub 2021 Dec 9 doi: 10.5334/tohm.652. PMID: 34992946Free PMC Article
Han X, Wu S, Wang M, Li H, Huang Y, Sui R
Mol Genet Genomic Med 2020 Aug;8(8):e1316. Epub 2020 May 26 doi: 10.1002/mgg3.1316. PMID: 32453490Free PMC Article
Bou Ghannam AS, Mehner LC, Pelak VS
J Neuroophthalmol 2019 Sep;39(3):388-390. doi: 10.1097/WNO.0000000000000773. PMID: 31107347
Gascon GG, Youssef NG, Subramanyam SB, Ozand PT
J Child Neurol 1992 Apr;7 Suppl:S73-8. doi: 10.1177/08830738920070011111. PMID: 1588018

Prognosis

Naganawa Y, Itoh K, Shimmoto M, Takiguchi K, Doi H, Nishizawa Y, Kobayashi T, Kamei S, Lukong KE, Pshezhetsky AV, Sakuraba H
J Hum Genet 2000;45(4):241-9. doi: 10.1007/s100380070034. PMID: 10944856

Clinical prediction guides

Du YC, Ma LH, Li QF, Ma Y, Dong Y, Wu ZY
Orphanet J Rare Dis 2024 Sep 30;19(1):362. doi: 10.1186/s13023-024-03378-5. PMID: 39350194Free PMC Article
Han X, Wu S, Wang M, Li H, Huang Y, Sui R
Mol Genet Genomic Med 2020 Aug;8(8):e1316. Epub 2020 May 26 doi: 10.1002/mgg3.1316. PMID: 32453490Free PMC Article
Lu CS, Ng SH, Lai SC, Kao LY, Liu L, Lin WY, Wu YM, Chen YL, Wang JJ
Brain Imaging Behav 2017 Feb;11(1):214-223. doi: 10.1007/s11682-016-9517-6. PMID: 26843009
Naganawa Y, Itoh K, Shimmoto M, Takiguchi K, Doi H, Nishizawa Y, Kobayashi T, Kamei S, Lukong KE, Pshezhetsky AV, Sakuraba H
J Hum Genet 2000;45(4):241-9. doi: 10.1007/s100380070034. PMID: 10944856
Swallow DM, Evans L, Stewart G, Thomas PK, Abrams JD
Ann Hum Genet 1979 Jul;43(1):27-35. PMID: 496393

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