Frequency
It is estimated that 1 in 30,000 to 1 in 100,000 people in the world have Silver-Russell syndrome.
Causes
The genetic causes of Silver-Russell syndrome are complex. The disorder is often the result of the abnormal regulation of genes that control growth. These genes are located in particular regions of chromosome 7 and chromosome 11.
Most cases of Silver-Russell syndrome are caused by changes in chromosome 11 that affect the regulation of specific genes. The genes that are thought to play the most significant role in Silver-Russell syndrome are the H19 and IGF2 genes. Disruption of the activity of these genes leads to slow growth and the other characteristic features of Silver-Russell syndrome.
When Silver-Russell syndrome is caused by problems that involve chromosome 7, it is primarily due to issues with the inheritance of the chromosome. Specifically, Silver-Russell syndrome can occur when both copies of chromosome 7 are derived from the egg cell. Normally, this does not affect gene activity, but some genes only work when they are derived from the egg cell or from the sperm cell. When both copies of chromosome 7 are derived from the egg cell, affected individuals may have two active copies of some genes and no active copies of other genes. This dysregulation of gene activity results in the signs and symptoms of Silver-Russell syndrome.
Rarely, variants (also called mutations) in a single gene cause Silver-Russell syndrome. These genes include the CDKN1C, IGF2, HMGA2, and PLAG-1 genes.
In 30 to 40 percent of people with Silver-Russell syndrome, the cause of the condition is unknown. Researchers are working to identify additional genetic changes that cause this disorder.
Inheritance
Most cases of Silver-Russell syndrome result from a new (de novo) event that occurs during the formation of reproductive cells (eggs or sperm) in an affected individual's parent or during early embryonic development. These affected individuals typically have no history of the disorder in their family.
Rarely, Silver-Russell syndrome can run in families. In some affected families, the condition is inherited in an autosomal dominant pattern, which means one copy of the altered gene in each cell is sufficient to cause the disorder. However, the pattern of inheritance in a specific family depends on the exact genetic change and whether the change came from the egg cell or the sperm cell.
Other Names for This Condition
- RSS
- Russell-Silver syndrome
- Silver-Russell dwarfism
- SRS
Additional Information & Resources
Genetic Testing Information
- Genetic Testing Registry: Silver-Russell syndrome 1
- Genetic Testing Registry: Silver-russell syndrome 2
- Genetic Testing Registry: Silver-Russell syndrome 3
- Genetic Testing Registry: Silver-russell syndrome 4
- Genetic Testing Registry: Silver-Russell syndrome 5
- Genetic Testing Registry: Silver-Russell syndrome due to 11p15 microduplication
- Genetic Testing Registry: Silver-Russell syndrome due to 7p11.2p13 microduplication
- Genetic Testing Registry: Silver-Russell syndrome due to a point mutation
- Genetic Testing Registry: Silver-Russell syndrome due to an imprinting defect of 11p15
- Genetic Testing Registry: Silver-Russell syndrome due to maternal uniparental disomy of chromosome 11
- Genetic Testing Registry: Silver-Russell syndrome due to maternal uniparental disomy of chromosome 7
Genetic and Rare Diseases Information Center
Patient Support and Advocacy Resources
Clinical Trials
Catalog of Genes and Diseases from OMIM
Scientific Articles on PubMed
References
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- Wakeling EL, Brioude F, Lokulo-Sodipe O, O'Connell SM, Salem J, Bliek J, Canton AP, Chrzanowska KH, Davies JH, Dias RP, Dubern B, Elbracht M, Giabicani E, Grimberg A, Gronskov K, Hokken-Koelega AC, Jorge AA, Kagami M, Linglart A, Maghnie M, Mohnike K, Monk D, Moore GE, Murray PG, Ogata T, Petit IO, Russo S, Said E, Toumba M, Tumer Z, Binder G, Eggermann T, Harbison MD, Temple IK, Mackay DJ, Netchine I. Diagnosis and management of Silver-Russell syndrome: first international consensus statement. Nat Rev Endocrinol. 2017 Feb;13(2):105-124. doi: 10.1038/nrendo.2016.138. Epub 2016 Sep 2. Citation on PubMed
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