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lysosomal multienzyme complex

Lysosomal enzymes sialidase (alpha-neuraminidase), beta-galactosidase, and N-acetylaminogalacto-6-sulfate sulfatase are involved in the catabolism of glycolipids, glycoproteins, and oligosaccharides.

Their functional activity in the cell depends on their association in a multienzyme complex with lysosomal carboxypeptidase, cathepsin A.

The integrity of the complex plays a crucial role at different stages of biogenesis of lysosomal enzymes, including intracellular sorting and proteolytic processing of their precursors.

The complex plays a protective role for all components, extending their half-life in the lysosome from several hours to several days; and for sialidase, the association with cathepsin A is also necessary for the expression of enzymatic activity.

Acidic glycosidases

Three acidic glycosidases: beta-galactosidase (beta-GAL, EC 3.2.1.23), alpha-neuraminidase (NEUR, sialidase, EC 3.2.1.18), N-acetylaminogalacto-6-sulfate sulfatase (GALNS, EC 3.1.6.4) and serine carboxypepidase cathepsin A (EC 3.4.16.1) form a functional high molecular weight complex in the lysosomes.

The major constituent of this complex is cathepsin A, the so-called "lysosomal protective protein" (PPCA).

By forming a multienzyme complex, it protects the glycosidases from rapid intralysosomal proteolysis, and it is also required for the intracellular sorting and proteolytic processing of their precursors. In man, a deficiency of cathepsin A leads to a combined deficiency of beta-GAL and NEUR activities, called "galactosialidosis".

Pathology

The disintegration of the complex due to genetic mutations in its components results in their functional deficiency and causes severe metabolic disorders:

- sialidosis (mutations in sialidase)
- GM1-gangliosidosis
- Morquio disease type B (mutations in beta-galactosidase)
- galactosialidosis (mutations in cathepsin A)
- Morquio disease type A (mutations in N-acetylaminogalacto-6-sulfate sulfatase)

Multiple mutations identified in the cathepsin A gene are the molecular basis of this lysosomal storage disease.

References

- Ostrowska H, Krukowska K, Kalinowska J, Orlowska M, Lengiewicz I. Lysosomal high molecular weight multienzyme complex. Cell Mol Biol Lett. 2003;8(1):19-24. PMID: 12655352

- Pshezhetsky AV, Ashmarina M. Lysosomal multienzyme complex: biochemistry, genetics, and molecular pathophysiology. Prog Nucleic Acid Res Mol Biol. 2001;69:81-114. PMID: 11550799