HSD10 disease, infantile type

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HSD10 disease, infantile type

ORPHA:391428Clinical subtype

Also called 2-methyl-3-hydroxybutyric aciduria, classic type · 2-methyl-3-hydroxybutyric aciduria, infantile type · 2-methyl-3-hydroxybutyryl-CoA dehydrogenase deficiency, classic type · 2-methyl-3-hydroxybutyryl-CoA dehydrogenase deficiency, infantile type · HSD10 deficiency, classic type · HSD10 deficiency, infantile type · HSD10 disease, classic type · MHBD deficiency, classic type · MHBD deficiency, infantile type

What it is

A clinical subtype of HSD10 disease, a rare neurometabolic disorder. Affected boys may show lethargy, poor feeding and evidence of mitochondrial dysfunction in the newborn period, with subsequent mild developmental delay and abnormal muscle tone. Hallmark of the disease is progressive neurodegeneration and cardiomyopathy, which usually manifests between ages 6 months and 2 years with developmental regression, progressive visual and hearing loss, epilepsy and other neurological symptoms, and severe cardiomyopathy. Laboratory investigations show signs of mitochondrial dysfunction, and increased urinary excretion of specific isoleucine metabolites. The disease is often fatal around 2-4 years of age.

Key facts

Age of onset
Infancy, Neonatal
Inheritance
X-linked dominant
Classified as
Clinical subtype

Recorded for the broader condition

Prevalence
<1 / 1 000 000HSD10 disease

Orphanet records these for the broader condition rather than for this specific form. They are a starting point, not a figure for this subtype — subtypes often differ, and some are defined by being rarer than the condition they sit under.

Signs and symptoms

These ranges are how often a sign appears across everyone diagnosed, not a prediction for one person — most people have some of these and not others. Orphanet also records findings that are specifically absent in this disease; those are in the data but not listed here.

Gene

HSD17B10Disease-causing germline mutation(s)

ICD-10 codes

E72.8filed under a broader ICD-10 category — shared with 24 other rare diseases

ICD-10 has no distinct code for most rare diseases, so many are filed under the nearest available one. A shared code identifies the group, not this disease — the ORPHAcode above is what identifies it.

Related health topics

Reached through the ICD-10 categories this disease is filed under, so these describe the broader group rather than this disease specifically.

Cross-references

MONDO 0018322OMIM 300438UMLS C5680025

Orphadata Science: Free access data from Orphanet. © INSERM 1999. Data version 2026-06-23. Available under CC BY 4.0. This is a reference, not medical advice.

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