Abstract
The physicochemical properties of hair from a new recessive syndrome associated with brittle hair, intellectual impairment, decreased fertility, and short stature have been studied. Electrophoresis of the SCM-structural proteins showed that the alpha polypeptides appeared normal, but the matrix component was markedly reduced. This was confirmed by finding a normal alpha X-ray diffraction pattern but a reduced 1/2 cystine content of hair and an abnormal stress-strain curve. Electron-microscopic studies revealed extreme disorganization of the filaments which most likely resulted from the absence of normal cross-linking. Nails, which contain structural proteins similar to hair, also showed the abnormality. Since the matrix component seen by electrophoresis consists of more than one component the defect cannot be explained as a single structural gene abnormality.
MeSH Terms
Amino Acids/analysis
Female
Genes, Recessive
Growth Disorders
Hair/abnormalities,analysis,ultrastructure
Humans
Infertility
Intellectual Disability
Male
Microscopy, Electron
Nails/analysis,ultrastructure
Proteins/analysis
Syndrome
Chemicals
Amino Acids
Proteins
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Baden H P
Jackson C E
Weiss L
Jimbow K
Lee L
Kubilus J
Gold R J
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9 references, click to expand
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