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IEMbase 0317: MSMO1-related sterol C4-methyloxidase deficiency

Scope

Field Value
IEMbase ID 317
Nosology 14.7.07.01
Gene MSMO1
External IDs OMIM:607545; ORPHA:488168
Generated mapping UNMAPPED
Candidate DisMech targets Fuzzy candidate Cerebrotendinous_Xanthomatosis.yaml rejected
Review date 2026-07-07

IEMbase phenotype signal

IEMbase represents MSMO1-related sterol C4-methyloxidase deficiency, also labeled SC4MOL, as an autosomal recessive sterol-biosynthesis disorder. Characteristic rows include developmental delay, hypotonia, microcephaly, and short stature.

Additional clinical rows include high-arched palate, cataract, psoriasiform dermatitis, failure to thrive, frontal bossing, and osteoporosis. The biochemical signal is sterol-focused: increased 4,4-dimethyl sterols and 4-methyl sterols in fibroblasts, lymphoblasts, and plasma. No treatment rows are present in the cached record.

DisMech phenotype coverage

The generated fuzzy candidate is Cerebrotendinous_Xanthomatosis.yaml, but that is a pathway-neighbor false positive. The local CTX entry is CYP27A1 disease with cholestanol and bile-acid abnormalities, tendon xanthomas, neuro-ophthalmic features, and chenodeoxycholic acid treatment.

There is no valid local MSMO1/SC4MOL disease target. CTX overlaps only at a very broad sterol-metabolism level and should not be used for this record.

Concordance and completeness

Judgement: true local disease gap.

IEMbase provides a compact but distinctive clinical and biochemical profile for future curation: developmental delay, hypotonia, microcephaly, short stature, cataract, psoriasiform dermatitis, failure to thrive, frontal bossing, osteoporosis, and elevated methylated sterol intermediates across multiple sample types.

Curation actions

  • Do not map this record to cerebrotendinous xanthomatosis.
  • Add a standalone MSMO1/SC4MOL sterol C4-methyloxidase deficiency target if this disease is prioritized.
  • Preserve the 4,4-dimethyl sterol and 4-methyl sterol biochemical rows as key differentiators from CYP27A1/CTX.