Literature DB >> 9727700

Aceruloplasminemia.

J D Gitlin1.   

Abstract

Aceruloplasminemia is an autosomal recessive disorder of iron metabolism characterized by diabetes, retinal degeneration, and neurologic symptoms. Affected patients evidence marked parenchymal iron accumulation in conjunction with an absence of circulating serum ceruloplasmin and molecular genetic analysis reveals inherited mutations in the ceruloplasmin gene. Taken together with earlier studies that characterized ceruloplasmin as a ferroxidase and recent work indicating an essential role for a homologous multicopper oxidase in iron metabolism in Saccharomyces cerevisiae, these findings reveal an essential role for ceruloplasmin in human iron metabolism. The presence of neurologic symptoms in patients with aceruloplasminemia is unique among the characterized disorders of iron metabolism, and recent findings indicate that astrocyte-specific ceruloplasmin gene expression is critical for iron metabolism and neuronal survival in the retina and basal ganglia. The discovery of this disease provides new insights into the pathways of CNS iron metabolism of direct relevance to a variety of nutritional and genetic disorders of childhood.

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Year:  1998        PMID: 9727700     DOI: 10.1203/00006450-199809000-00001

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  36 in total

1.  Ferroportin mutation in autosomal dominant hemochromatosis: loss of function, gain in understanding.

Authors:  R E Fleming; W S Sly
Journal:  J Clin Invest       Date:  2001-08       Impact factor: 14.808

Review 2.  Molecular pathogenesis of iron overload.

Authors:  D Trinder; C Fox; G Vautier; J K Olynyk
Journal:  Gut       Date:  2002-08       Impact factor: 23.059

Review 3.  Neurodegeneration with brain iron accumulation.

Authors:  Allison Gregory; Susan J Hayflick
Journal:  Folia Neuropathol       Date:  2005       Impact factor: 2.038

4.  Silencing the Menkes copper-transporting ATPase (Atp7a) gene in rat intestinal epithelial (IEC-6) cells increases iron flux via transcriptional induction of ferroportin 1 (Fpn1).

Authors:  Sukru Gulec; James F Collins
Journal:  J Nutr       Date:  2013-10-30       Impact factor: 4.798

5.  Serum ceruloplasmin protein expression and activity increases in iron-deficient rats and is further enhanced by higher dietary copper intake.

Authors:  Perungavur N Ranganathan; Yan Lu; Lingli Jiang; Changae Kim; James F Collins
Journal:  Blood       Date:  2011-07-18       Impact factor: 22.113

Review 6.  Aceruloplasminaemia: a rare but important cause of iron overload.

Authors:  Adam Doyle; Ferry Rusli; Prithi Bhathal
Journal:  BMJ Case Rep       Date:  2015-05-14

7.  Microarray analysis of murine retinal light damage reveals changes in iron regulatory, complement, and antioxidant genes in the neurosensory retina and isolated RPE.

Authors:  Majda Hadziahmetovic; Usha Kumar; Ying Song; Steven Grieco; Delu Song; Yafeng Li; John W Tobias; Joshua L Dunaief
Journal:  Invest Ophthalmol Vis Sci       Date:  2012-08-07       Impact factor: 4.799

8.  Bmp6 regulates retinal iron homeostasis and has altered expression in age-related macular degeneration.

Authors:  Majda Hadziahmetovic; Ying Song; Natalie Wolkow; Jared Iacovelli; Leon Kautz; Marie-Paule Roth; Joshua L Dunaief
Journal:  Am J Pathol       Date:  2011-05-03       Impact factor: 4.307

9.  Age-dependent retinal iron accumulation and degeneration in hepcidin knockout mice.

Authors:  Majda Hadziahmetovic; Ying Song; Padmavathi Ponnuru; Jared Iacovelli; Allan Hunter; Nadine Haddad; John Beard; James R Connor; Sophie Vaulont; Joshua L Dunaief
Journal:  Invest Ophthalmol Vis Sci       Date:  2011-01-05       Impact factor: 4.799

10.  Normal iron metabolism and the pathophysiology of iron overload disorders.

Authors:  Chiang W Siah; John Ombiga; Leon A Adams; Debbie Trinder; John K Olynyk
Journal:  Clin Biochem Rev       Date:  2006-02
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