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Infantile bilateral striatal necrosis(SNDI; IBSN)

MedGen UID:
167090
Concept ID:
C0795996
Disease or Syndrome
Synonym: Striatonigral degeneration infantile
SNOMED CT: Infantile striatonigral degeneration (718174008); Infantile bilateral striatal necrosis (718174008)
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).
Autosomal dominant inheritance
MedGen UID:
141047
Concept ID:
C0443147
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 heterozygotes. In the context of medical genetics, an autosomal dominant disorder is caused when a single copy of the mutant allele is present. Males and females are affected equally, and can both transmit the disorder with a risk of 50% for each child of inheriting the mutant allele.
Mitochondrial inheritance
MedGen UID:
165802
Concept ID:
C0887941
Genetic Function
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on the mitochondrial genome. Because the mitochondrial genome is essentially always maternally inherited, a mitochondrial condition can only be transmitted by females, although the condition can affect both sexes. The proportion of mutant mitochondria can vary (heteroplasmy).
Not genetically inherited
MedGen UID:
988794
Concept ID:
CN307044
Finding
Source: Orphanet
clinical entity without genetic inheritance.
 
Related gene: NUP62
 
Monarch Initiative: MONDO:0015518
OMIM®: 271930; 605815
Orphanet: ORPHA1576

Definition

Comprises of several syndromes of bilateral symmetric spongy degeneration of the caudate nucleus, putamen and globus pallidus with characteristics of developmental regression, choreoathetosis and dystonia progressing to spastic quadriparesis. Can be familial or sporadic. The familial form has an insidious onset and a slowly progressive downhill course, while the sporadic form is associated with abrupt neurologic dysfunction following an acute systemic febrile illness such as a mycoplasma, measles or streptococcus infection. Familial disease can be inherited as an autosomal recessive or mitochondrial disorder. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  

Recent clinical studies

Etiology

Ganelin Cohen E, Bessler H, Djaldetti M, Straussberg R
Neuroimmunomodulation 2017;24(4-5):276-281. Epub 2018 Mar 6 doi: 10.1159/000486861. PMID: 29510394
Basel-Vanagaite L, Muncher L, Straussberg R, Pasmanik-Chor M, Yahav M, Rainshtein L, Walsh CA, Magal N, Taub E, Drasinover V, Shalev H, Attia R, Rechavi G, Simon AJ, Shohat M
Ann Neurol 2006 Aug;60(2):214-22. doi: 10.1002/ana.20902. PMID: 16786527
Leuzzi V, Bertini E, De Negri AM, Gallucci M, Garavaglia B
J Neurol Neurosurg Psychiatry 1992 Jan;55(1):16-9. doi: 10.1136/jnnp.55.1.16. PMID: 1548491Free PMC Article
Aoki N
Neurol Res 1992 Jun;14(3):252-4. doi: 10.1080/01616412.1992.11740064. PMID: 1355279
Mito T, Tanaka T, Becker LE, Takashima S, Tanaka J
Arch Neurol 1986 Jul;43(7):677-80. doi: 10.1001/archneur.1986.00520070035014. PMID: 3729745

Diagnosis

Ganelin Cohen E, Bessler H, Djaldetti M, Straussberg R
Neuroimmunomodulation 2017;24(4-5):276-281. Epub 2018 Mar 6 doi: 10.1159/000486861. PMID: 29510394
La Piana R, Uggetti C, Olivieri I, Tonduti D, Balottin U, Fazzi E, Orcesi S
Am J Med Genet A 2014 Mar;164A(3):815-9. Epub 2013 Dec 20 doi: 10.1002/ajmg.a.36360. PMID: 24376015
Basel-Vanagaite L, Muncher L, Straussberg R, Pasmanik-Chor M, Yahav M, Rainshtein L, Walsh CA, Magal N, Taub E, Drasinover V, Shalev H, Attia R, Rechavi G, Simon AJ, Shohat M
Ann Neurol 2006 Aug;60(2):214-22. doi: 10.1002/ana.20902. PMID: 16786527
Fujita K, Takeuchi Y, Nishimura A, Takada H, Sawada T
Pediatr Neurol 1994 Mar;10(2):157-60. doi: 10.1016/0887-8994(94)90050-7. PMID: 8024666
Röyttä M, Olsson I, Sourander P, Svendsen P
Acta Neuropathol 1981;55(2):97-103. doi: 10.1007/BF00699234. PMID: 6118994

Therapy

Tonduti D, Zorzi G, Ghezzi D, Zibordi F, Garavaglia B, Nardocci N
J Child Neurol 2015 Nov;30(13):1800-5. Epub 2015 Apr 23 doi: 10.1177/0883073815581608. PMID: 25907776
Straussberg R, Shorer Z, Weitz R, Basel L, Kornreich L, Corie CI, Harel L, Djaldetti R, Amir J
Neurology 2002 Oct 8;59(7):983-9. doi: 10.1212/wnl.59.7.983. PMID: 12374138
Fujita K, Takeuchi Y, Nishimura A, Takada H, Sawada T
Pediatr Neurol 1994 Mar;10(2):157-60. doi: 10.1016/0887-8994(94)90050-7. PMID: 8024666
Aoki N
Neurol Res 1992 Jun;14(3):252-4. doi: 10.1080/01616412.1992.11740064. PMID: 1355279

Prognosis

Cambonie G, Houdon L, Rivier F, Bongrand AF, Echenne B
Brain Dev 2000 Jun;22(4):221-3. doi: 10.1016/s0387-7604(00)00105-4. PMID: 10838107
Leuzzi V, Bertini E, De Negri AM, Gallucci M, Garavaglia B
J Neurol Neurosurg Psychiatry 1992 Jan;55(1):16-9. doi: 10.1136/jnnp.55.1.16. PMID: 1548491Free PMC Article
Aoki N
Neurol Res 1992 Jun;14(3):252-4. doi: 10.1080/01616412.1992.11740064. PMID: 1355279

Clinical prediction guides

Tonduti D, Zorzi G, Ghezzi D, Zibordi F, Garavaglia B, Nardocci N
J Child Neurol 2015 Nov;30(13):1800-5. Epub 2015 Apr 23 doi: 10.1177/0883073815581608. PMID: 25907776
Basel-Vanagaite L, Straussberg R, Ovadia H, Kaplan A, Magal N, Shorer Z, Shalev H, Walsh C, Shohat M
Neurology 2004 Jan 13;62(1):87-90. doi: 10.1212/01.wnl.0000101680.49036.69. PMID: 14718703
Cambonie G, Houdon L, Rivier F, Bongrand AF, Echenne B
Brain Dev 2000 Jun;22(4):221-3. doi: 10.1016/s0387-7604(00)00105-4. PMID: 10838107

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