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#lysosomal storage disorders

2 public questions tagged with this topic.

Which of the following is a characteristic of lysosomal storage disorders?

Lysosomal storage disorders form a group of about seventy rare inherited metabolic diseases sharing unifying pathophysiology despite diverse enzyme defects. Mutations in genes encoding lysosomal acid hydrolases, accessory activator proteins GM2 activator and saposins, sulfatases requiring formylglycine modification by SUMF1, or lysosomal integral membrane transporters such as cystinosin and sialin impair specific catabolic steps. Because residual enzyme activity falls below threshold, typically less than ten percent of normal, undegraded macromolecular substrates such as sphingolipids, mucopolysaccharides, glycogen, oligosaccharides, ceroid lipofuscin or free amino acids accumulate inside endolysosomal compartments, physically swelling lysosomes to microns, engorging cytoplasm, disrupting trafficking, autophagy, mTOR signaling and calcium homeostasis via TRPML1 inhibition. Neurons, macrophages, hepatocytes and skeletal muscle heavily loaded with storage material display vacuolation, impaired function and apoptosis. Examples include Tay-Sachs with GM2 ganglioside accumulation from hexosaminidase A deficiency, Gaucher with glucosylceramide, Pompe with glycogen, and cystinosis with cystine crystals due to defective cystine exporter. Cells show material overload rather than overexpression of enzymes; often mutant enzymes are unstable, misfolded and degraded by ER-associated degradation. Lysosomal biogenesis via TFEB compensates initially but fails as storage progresses, causing multi-systemic pathology often beginning in infancy.

Ref: Parenti et al., Nature Reviews Drug Discovery 2015: Lysosomal Storage Disorders – Substrate Accumulation.

Gaucher’s disease is caused by a deficiency of:

Gaucher disease, the most common lysosomal storage disorder with incidence about 1 in 40,000 to 60,000 in general population and 1 in 800 among Ashkenazi Jews, arises from autosomal recessive mutations in GBA1 gene on chromosome 1q21 encoding lysosomal glucocerebrosidase, also called acid beta-glucosidase or glucosylceramide beta-glucosidase. This membrane-associated enzyme normally cleaves glucosylceramide, a major membrane glycosphingolipid intermediate, into ceramide and glucose within lysosomes of macrophages that recycle membranes from phagocytosed senescent erythrocytes and leukocytes. Deficiency leads to accumulation of undegraded glucosylceramide and glucosylsphingosine in lysosomes, producing engorged macrophages, Gaucher cells, with fibrillar wrinkled tissue paper cytoplasm due to lipid-laden lysosomes in spleen, liver, bone marrow and sometimes brain. Three clinical subtypes exist: Type 1 non-neuronopathic adult chronic with hepatosplenomegaly, anemia, thrombocytopenia and bone crises; Type 2 acute neuronopathic infantile rapidly lethal; and Type 3 chronic neuronopathic with horizontal gaze palsy and progressive neurological decline. Diagnosis measures leukocyte enzyme activity and GBA sequencing. Therapies include enzyme replacement with recombinant glucocerebrosidase imiglucerase and velaglucerase and substrate reduction with miglustat and eliglustat inhibiting glucosylceramide synthase.

Ref: Scriver et al., The Metabolic and Molecular Bases of Inherited Disease, Chapter 146: Gaucher Disease – Glucocerebrosidase.