ArticleFASEB journal : official publication of the Federation of American Societies for Experimental Biology2014
Mannose supplements induce embryonic lethality and blindness in phosphomannose isomerase hypomorphic mice.
Article in FASEB journal : official publication of the Federation of American Societies for Experimental Biology, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed, 34 citations in OpenAlex.
- Mpi-driven N-glycosylation orchestrates mucin O-glycosylation and intestinal homeostasis.Nature communications · 2026Article
- A hypomorphic Mpi mutation unlocks an in vivo tool for studying global N-glycosylation deficiency.JCI insight · 2025Article
- Aldolase B Deficient Mice Are Characterized by Hepatic Nucleotide Sugar Abnormalities.Journal of inherited metabolic disease · 2025Article
- Mannose phosphate isomerase-congenital disorder of glycosylation leads to asymptomatic hypoglycemia.Molecular genetics and metabolism reports · 2024Article
- Consequences of GMPPB deficiency for neuromuscular development and maintenance.Frontiers in molecular neuroscience · 2024Article
- Mannose metabolism inhibition sensitizes acute myeloid leukaemia cells to therapy by driving ferroptotic cell death.Nature communications · 2023Article
- Role of D-mannose in urinary tract infections - a narrative review.Nutrition journal · 2022Review
- Variation of the serumJIMD reports · 2021Article
- A patient-based medaka alg2 mutant as a model for hypo-N-glycosylation.Development (Cambridge, England) · 2021Article
- Rescuing lethal phenotypes induced by disruption of genes in mice: a review of novel strategies.Physiological research · 2021Review
- Evolutionary conservation of human ketodeoxynonulosonic acid production is independent of sialoglycan biosynthesis.The Journal of clinical investigation · 2021Article
- Placental Impact of Dietary Supplements: More Than Micronutrients.Clinical therapeutics · 2021Review
- Synthesis and photoinitiated thiol-ene reactions ofRSC advances · 2020Article
- Therapeutic Monosaccharides: Looking Back, Moving Forward.Biochemistry · 2020Review
- Consensus guideline for the diagnosis and management of mannose phosphate isomerase-congenital disorder of glycosylation.Journal of inherited metabolic disease · 2020Review
- Congenital disorders of glycosylation.Annals of translational medicine · 2018Review
- CDG Therapies: From Bench to Bedside.International journal of molecular sciences · 2018Review
- Article
- UroPathogenicFrontiers in microbiology · 2017Review
- A mouse model of a human congenital disorder of glycosylation caused by loss of PMM2.Human molecular genetics · 2016Article
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Authors and funding
17 authors at 2 institutions in 1 country.
Funding
Abstract
Patients with congenital disorder of glycosylation (CDG), type Ib (MPI-CDG or CDG-Ib) have mutations in phosphomannose isomerase (MPI) that impair glycosylation and lead to stunted growth, liver dysfunction, coagulopathy, hypoglycemia, and intestinal abnormalities. Mannose supplements correct hypoglycosylation and most symptoms by providing mannose-6-P (Man-6-P) via hexokinase. We generated viable Mpi hypomorphic mice with residual enzymatic activity comparable to that of patients, but surprisingly, these mice appeared completely normal except for modest (~15%) embryonic lethality. To overcome this lethality, pregnant dams were provided 1-2% mannose in their drinking water. However, mannose further reduced litter size and survival to weaning by 40 and 66%, respectively. Moreover, ~50% of survivors developed eye defects beginning around midgestation. Mannose started at birth also led to eye defects but had no effect when started after eye development was complete. Man-6-P and related metabolites accumulated in the affected adult eye and in developing embryos and placentas. Our results demonstrate that disturbing mannose metabolic flux in mice, especially during embryonic development, induces a highly specific, unanticipated pathological state. It is unknown whether mannose is harmful to human fetuses during gestation; however, mothers who are at risk for having MPI-CDG children and who consume mannose during pregnancy hoping to benefit an affected fetus in utero should be cautious.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.