Table 1.

The causes of lysosomal storage diseases, the organelles affected, and major sites of pathology

Mechanism of lysosomal storage Disease examples Lysosomal protein defect (gene symbol) Substrate(s) stored Major peripheral organ systems affected CNS pathology 
Lysosomal enzyme deficiencies Aspartylglucosaminuria Aspartylglucosaminidase (glycosylasparaginase, AGA) aspartylglucosamine (N-acetylglucosaminyl-asparagine) Skeleton, connective tissue 
 Fabry α-Galactosidase (GLA) (Lyso-)Globotriaosylceramide Kidney, heart − 
 Gaucher types 1, 2, and 3 β-Glucocerebrosidase (GBA) Glucosylceramide, glucosylsphingosine Spleen/liver, bone marrow +a 
 GM1-gangliosidosis β-Galactosidase (GLB1) GM1-ganglioside, oligosaccharides Skeleton, heart 
 Krabbe (globoid cell leukodystrophy) Galactocerebrosidase (GALC) Galactosylceramide Heart 
 Metachromatic leukodystrophy Arylsulfatase A (ARSA) Sulfogalactosylceramide  
 Mucopolysaccharidoses Enzymes involve in mucopolysaccharide catabolism Mucopolysaccharides Cartilage, bone, heart, lungs +b 
 Multiple sulfatase deficiency SUMF1 (Formylglycine-generating enzyme needed to activate sulfatases) Multiple, including sulfated glycosaminoglycans Spleen/liver, bone, skin 
 Pompe α-Glucosidase (GAA) Glycogen Skeletal muscle − 
 Sandhoff β-hexosaminidase A and B (HEXB) GM2-ganglioside  
Trafficking defect of lysososomal enzymes Mucolipidosis type II (I-cell disease) N-acetyl glucosamine phosphoryl transferase α/β (GNPTAB) Carbohydrates, lipids, proteins Skeleton, heart 
 Mucolipidosis type IIIA (pseudo-Hurler polydystrophy) N-acetyl glucosamine phosphoryl transferase α/β (GNPTAB) Carbohydrates, lipids, proteins Skeleton, heart +/− 
Defects in soluble non-enzymatic lysosomal proteins Niemann-Pick disease type C2 NPC2 (soluble cholesterol binding protein) Cholesterol and sphingolipids Liver 
Defects in lysosomal membrane proteins Cystinosis Cystinosin (cysteine transporter, CTNS) Cystine Kidney, eye − 
 Danon disease Lysosomal-associated membrane protein 2, splicing variant A (LAMP2) Glycogen and other autophagic components Cardiac and skeletal muscle 
 Free sialic acid storage disorder Sialin (sialic acid transporter, SLC17A5) Free sialic acid Liver/spleen, skeleton 
 Mucolipidosis IV Mucolipin-I (MCOLN1) Mucopolysaccharides and lipids Eye 
 Niemann-Pick disease type C1 NPC1 (membrane protein involved in lipid transport) Cholesterol and sphingolipids Liver 
Enigmatic lysosomal disorders Neuronal ceroid lipofuscinoses (NCLs, including Batten disease) Disparate group of diseases with genetic defects in apparently unrelated genes, not all of which are associated with the lysosomal system. Not known if these genes cooperate in common cellular pathways. Autofluorescent lipofuscin is a common feature, with convergent clinical signs, e.g., visual system defects/blindness  
Mechanism of lysosomal storage Disease examples Lysosomal protein defect (gene symbol) Substrate(s) stored Major peripheral organ systems affected CNS pathology 
Lysosomal enzyme deficiencies Aspartylglucosaminuria Aspartylglucosaminidase (glycosylasparaginase, AGA) aspartylglucosamine (N-acetylglucosaminyl-asparagine) Skeleton, connective tissue 
 Fabry α-Galactosidase (GLA) (Lyso-)Globotriaosylceramide Kidney, heart − 
 Gaucher types 1, 2, and 3 β-Glucocerebrosidase (GBA) Glucosylceramide, glucosylsphingosine Spleen/liver, bone marrow +a 
 GM1-gangliosidosis β-Galactosidase (GLB1) GM1-ganglioside, oligosaccharides Skeleton, heart 
 Krabbe (globoid cell leukodystrophy) Galactocerebrosidase (GALC) Galactosylceramide Heart 
 Metachromatic leukodystrophy Arylsulfatase A (ARSA) Sulfogalactosylceramide  
 Mucopolysaccharidoses Enzymes involve in mucopolysaccharide catabolism Mucopolysaccharides Cartilage, bone, heart, lungs +b 
 Multiple sulfatase deficiency SUMF1 (Formylglycine-generating enzyme needed to activate sulfatases) Multiple, including sulfated glycosaminoglycans Spleen/liver, bone, skin 
 Pompe α-Glucosidase (GAA) Glycogen Skeletal muscle − 
 Sandhoff β-hexosaminidase A and B (HEXB) GM2-ganglioside  
Trafficking defect of lysososomal enzymes Mucolipidosis type II (I-cell disease) N-acetyl glucosamine phosphoryl transferase α/β (GNPTAB) Carbohydrates, lipids, proteins Skeleton, heart 
 Mucolipidosis type IIIA (pseudo-Hurler polydystrophy) N-acetyl glucosamine phosphoryl transferase α/β (GNPTAB) Carbohydrates, lipids, proteins Skeleton, heart +/− 
Defects in soluble non-enzymatic lysosomal proteins Niemann-Pick disease type C2 NPC2 (soluble cholesterol binding protein) Cholesterol and sphingolipids Liver 
Defects in lysosomal membrane proteins Cystinosis Cystinosin (cysteine transporter, CTNS) Cystine Kidney, eye − 
 Danon disease Lysosomal-associated membrane protein 2, splicing variant A (LAMP2) Glycogen and other autophagic components Cardiac and skeletal muscle 
 Free sialic acid storage disorder Sialin (sialic acid transporter, SLC17A5) Free sialic acid Liver/spleen, skeleton 
 Mucolipidosis IV Mucolipin-I (MCOLN1) Mucopolysaccharides and lipids Eye 
 Niemann-Pick disease type C1 NPC1 (membrane protein involved in lipid transport) Cholesterol and sphingolipids Liver 
Enigmatic lysosomal disorders Neuronal ceroid lipofuscinoses (NCLs, including Batten disease) Disparate group of diseases with genetic defects in apparently unrelated genes, not all of which are associated with the lysosomal system. Not known if these genes cooperate in common cellular pathways. Autofluorescent lipofuscin is a common feature, with convergent clinical signs, e.g., visual system defects/blindness  

Listed are the diseases discussed in the main text. Mucopolysaccharidoses and neuronal ceroid lipofuscinoses refer to collections of related disorders.

a

Types 2 and 3.

b

Most mucopolysaccharidosis disorders.

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