Biochemical characterization of collagen I in Warmblood Fragile Foal Syndrome horse lysyl hydroxylase 1 mutation.

Mutations in the collagen-modifying enzyme lysyl hydroxylase 1 (LH1) cause Warmblood Fragile Foal Syndrome (WFFS) in horses. We investigated the impact of this mutation on collagen structure and function. Our results show that LH1 deficiency leads to reduced lysine hydroxylation, altered collagen fibril organization, and tissue abnormalities resembling human Ehlers-Danlos syndrome. These findings highlight the critical role of LH1 in collagen biosynthesis and provide insights into the pathogenesis of WFFS.

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Publication Details

Journal
PubMed
Published
2024-10-26
DOI
https://doi.org/10.17912/micropub.biology.001399
Citations
2
Primary Topic
Muscle metabolism and nutrition
Type
article
Field-Weighted Citation Impact
0.65

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article

Biochemical characterization of collagen I in Warmblood Fragile Foal Syndrome horse lysyl hydroxylase 1 mutation.

Sara F. Tufa, Douglas R. Keene, Yoshihiro Ishikawa, Hans Peter Bächinger et al.
2 citations
PubMed
Muscle metabolism and nutrition
0.65
article

Biochemical characterization of collagen I in Warmblood Fragile Foal Syndrome horse lysyl hydroxylase 1 mutation.

Sara F. Tufa, Douglas R. Keene, Yoshihiro Ishikawa, Hans Peter Bächinger, Nena J. Winand
article en
2 citations

Abstract

Mutations in the collagen-modifying enzyme lysyl hydroxylase 1 (LH1) cause Warmblood Fragile Foal Syndrome (WFFS) in horses. We investigated the impact of this mutation on collagen structure and function. Our results show that LH1 deficiency leads to reduced lysine hydroxylation, altered collagen fibril organization, and tissue abnormalities resembling human Ehlers-Danlos syndrome. These findings highlight the critical role of LH1 in collagen biosynthesis and provide insights into the pathogenesis of WFFS.

PubMedVol. 2025
Oregon Health & Science University (US), University of California, San Francisco (US)
Shriners Hospitals for Children, University of California, San Francisco
Openalex Percentile: Top 30%
Muscle metabolism and nutrition
0.65
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Biochemical characterization of collagen I in Warmblood Fragile Foal Syndrome horse lysyl hydroxylase 1 mutation. — Sara F. Tufa, Douglas R. Keene, et al. · PubMed (2024) | TGRS Research Map | TGRS