Renal manifestation of tuberous sclerosis complex

Am J Med Genet C Semin Med Genet. 2018 Sep;178(3):338-347. doi: 10.1002/ajmg.c.31654. Epub 2018 Oct 11.

Abstract

Tuberous sclerosis complex (TSC) is a tumor predisposition syndrome with significant renal cystic and solid tumor disease. It commonly causes several types of cystic disease and benign tumors (angiomyolipomata) in the kidneys that can both lead to significant premature loss of glomerular filtration rate. The main risks of angiomyolipomata, severe bleeding, loss of renal function, and pulmonary lymphangioleiomyomatosis, can be ameliorated by active surveillance and preemptive therapy with mTOR inhibitors. The cystogenic mechanism may involve primary cilia, but also appears to also involve a majority of normal tubular cells and may be driven by a minority of cells with mutations inactivating both their TSC1 or TSC2 genes. Malignant tumors are rare.

Keywords: angiomyolipoma; chronic; kidney disease; renal cystic disease; tuberous sclerosis complex.

Publication types

  • Review

MeSH terms

  • Angiomyolipoma / etiology
  • Animals
  • Humans
  • Hypertension / drug therapy
  • Hypertension / etiology
  • Kidney Diseases, Cystic / etiology
  • Kidney Neoplasms / etiology*
  • Lymphangioleiomyomatosis / etiology
  • Mutation
  • Renal Insufficiency, Chronic / etiology
  • Renal Insufficiency, Chronic / therapy*
  • Tuberous Sclerosis / etiology*
  • Tuberous Sclerosis Complex 1 Protein / genetics
  • Tuberous Sclerosis Complex 2 Protein / genetics

Substances

  • TSC1 protein, human
  • TSC2 protein, human
  • Tuberous Sclerosis Complex 1 Protein
  • Tuberous Sclerosis Complex 2 Protein