What question did this study set out to answer?

To assess whether dietary mannose supplementation rescues embryonic lethality and restores normal development in a hypomorphic mouse model of phosphomannose isomerase deficiency.

January 13, 2014The FASEB Journal

Mannose supplements induce embryonic lethality and blindness in phosphomannose isomerase hypomorphic mice

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Authors

VSVandana SharmaSanford Burnham Prebys Medical Discovery Institute JNJonamani NayakDiscovery Institute CDCharles DeRossiIcahn School of Medicine at Mount Sinai

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Implication

Animal study reveals mannose supplementation triggers embryonic death and eye malformations in hypomorphic Mpi mice, highlighting potential prenatal risks of dietary therapy.

Key Points

To assess whether dietary mannose supplementation rescues embryonic lethality and restores normal development in a hypomorphic mouse model of phosphomannose isomerase deficiency.
Generated hypomorphic mice with phosphomannose isomerase (Mpi) residual enzymatic activity matching levels observed in human MPI-CDG patients.
Administered 1% to 2% mannose in drinking water to pregnant dams during gestation and to offspring either at birth or following complete eye development.
Tracked embryonic lethality, litter size, survival to weaning, ocular morphology, and accumulation of mannose-6-phosphate (Man-6-P) metabolites.
Maternal mannose supplementation exacerbated embryonic lethality, reducing litter size by 40% and survival to weaning by 66% compared to untreated mice.
Approximately 50% of surviving pups exposed midgestation or from birth developed severe eye defects, whereas exposure initiated after ocular maturation produced no defects.
Pathology was accompanied by abnormal accumulation of mannose-6-phosphate and related metabolites in adult eyes, placentas, and developing embryos.

Cite This Study

Sharma et al. (2014) studied this question.

synapsesocial.com/papers/6a85babb1f43de01ad1e5d47 https://doi.org/10.1096/fj.13-245514

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Implication

Animal study reveals mannose supplementation triggers embryonic death and eye malformations in hypomorphic Mpi mice, highlighting potential prenatal risks of dietary therapy.

Mannose supplements induce embryonic lethality and blindness in phosphomannose isomerase hypomorphic mice

Discussion

Member takes

Implication

Key Points

Cite This Study

Mannose supplements induce embryonic lethality and blindness in phosphomannose isomerase hypomorphic mice

Discussion

Member takes

Implication

Key Points

Cite This Study

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