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Mitochondrial DNA Depletion Syndrome 3

OMIM ID:

autosomal recessive

Mitochondrial DNA Depletion Syndrome 3

Alternate Names

MTDPS3
hepatocerebral type DNA depletion syndrome

Defective Genes

DGUOK

Clinical Characteristics

Ocular Features

Nystagmus, disconjugate eye movements, and “optic dysplasia” have been noted.

Systemic Features

Infants feed poorly which is frequently associated with vomiting, failure to thrive, and growth delay.  They are hypothermic, hypoglycemic, and often jaundiced with signs of liver failure noted between birth and 6 months of age and death by approximately 1 year of age.  Hepatosplenomegaly is present early with abnormal liver enzymes, cholestasis, steatosis, and hepatocellular loss followed by cirrhosis with portal hypertension.  Metabolic acidosis, hyperbilirubinemia, hypoalbuminemia, and hypoglycemia are often present.  Mitochondrial DNA depletion in the liver approaches 84-90%.

All patients have encephalopathic signs with evidence of cerebral atrophy, microcephaly, hypotonia.  Hyperreflexia may be present and some infants have seizures.  Muscle tissue, however, has normal histology and respiratory chain activity.

Genetics

Inheritance

This disorder results from homozygous or compound heterozygous mutations in the DGUOK gene (2p13).

The same gene is mutated in PEOB4 (617070).

Pedigree

Autosomal recessive

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent.  Carriers with only one mutation, such as the parents, do not have clinical disease.  Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Image
Sample pedigree of autosomal recessive inheritance

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent. Carriers with only one mutation, such as the parents, do not have clinical disease. Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Treatment & Management

There is no effective treatment.  Liver transplantation in one infant was unsuccessful.  

Publications

Displaying 1 - 2 of 2

New DGK Gene Mutations in the Hepatocerebral Form of Mitochondrial DNA Depletion Syndrome

PubMedID: 15883261

The deoxyguanosine kinase gene is mutated in individuals with depleted hepatocerebral mitochondrial DNA

PubMedID: 11687800