Mutation in human CLPX elevates levels of δ-aminolevulinate synthase and protoporphyrin IX to promote erythropoietic protoporphyria.

PubWeight™: 0.75‹?›

🔗 View Article (PMID 28874591)

Published in Proc Natl Acad Sci U S A on September 05, 2017

Authors

Yvette Y Yien1, Sarah Ducamp2,3,4, Lisa N van der Vorm1, Julia R Kardon5,6, Hana Manceau2,3,4, Caroline Kannengiesser2,4,7, Hector A Bergonia8, Martin D Kafina1, Zoubida Karim2,3,4, Laurent Gouya2,3,4, Tania A Baker9,6, Hervé Puy10,3,4, John D Phillips11, Gaël Nicolas12, Barry H Paw13,14,15

Author Affiliations

1: Division of Hematology, Brigham & Women's Hospital, Harvard Medical School, Boston, MA 02115.
2: INSERM U1149, CNRS ERL 8252, Centre de Recherche sur l'inflammation, Université Paris Diderot, Site Bichat, Sorbonne Paris Cité, 75018 Paris, France.
3: Assistance Publique-Hôpitaux de Paris, Centre Français des Porphyries, Hôpital Louis Mourier, 92701 Colombes Cedex, France.
4: Laboratory of Excellence, GR-Ex, 75015 Paris, France.
5: Department of Biology, Massachusetts Institute of Technology, Cambridge, MA 02139.
6: Howard Hughes Medical Institute, Massachusetts Institute of Technology, Cambridge, MA 02139.
7: Département de Génétique, Assistance Publique-Hôpitaux de Paris, HUPNVS, Hôpital Bichat, 75877 Paris Cedex, France.
8: Division of Hematology, University of Utah School of Medicine, Salt Lake City, UT 84112.
9: Department of Biology, Massachusetts Institute of Technology, Cambridge, MA 02139; tabaker@mit.edu herve.puy@aphp.fr john.phillips@hsc.utah.edu bpaw@rics.bwh.harvard.edu.
10: INSERM U1149, CNRS ERL 8252, Centre de Recherche sur l'inflammation, Université Paris Diderot, Site Bichat, Sorbonne Paris Cité, 75018 Paris, France; tabaker@mit.edu herve.puy@aphp.fr john.phillips@hsc.utah.edu bpaw@rics.bwh.harvard.edu.
11: Division of Hematology, University of Utah School of Medicine, Salt Lake City, UT 84112; tabaker@mit.edu herve.puy@aphp.fr john.phillips@hsc.utah.edu bpaw@rics.bwh.harvard.edu.
12: INSERM, U1016, Institut Cochin, Paris, France; Centre National de la Recherche Scientifique, Unité Mixte de Recherche 8104, Paris, France; Université Paris Descartes, Sorbonne Paris Cité, Paris, France; Laboratory of Excellence GR-Ex; and.
13: Division of Hematology, Brigham & Women's Hospital, Harvard Medical School, Boston, MA 02115; tabaker@mit.edu herve.puy@aphp.fr john.phillips@hsc.utah.edu bpaw@rics.bwh.harvard.edu.
14: Division of Hematology-Oncology, Boston Children's Hospital, Harvard Medical School, Boston, MA 02115.
15: Department of Pediatric Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, MA 02115.

Articles cited by this

Inhibition of eukaryotic translation elongation by cycloheximide and lactimidomycin. Nat Chem Biol (2010) 3.44

Structures of asymmetric ClpX hexamers reveal nucleotide-dependent motions in a AAA+ protein-unfolding machine. Cell (2009) 2.87

Porphyrias. Lancet (2010) 2.76

Human erythroid 5-aminolevulinate synthase: promoter analysis and identification of an iron-responsive element in the mRNA. EMBO J (1991) 2.18

ClpXP, an ATP-powered unfolding and protein-degradation machine. Biochim Biophys Acta (2011) 2.17

Polyhydramnios, Transient Antenatal Bartter's Syndrome, and MAGED2 Mutations. N Engl J Med (2016) 2.08

Distinct static and dynamic interactions control ATPase-peptidase communication in a AAA+ protease. Mol Cell (2007) 1.91

Communication between ClpX and ClpP during substrate processing and degradation. Nat Struct Mol Biol (2004) 1.85

C-terminal deletions in the ALAS2 gene lead to gain of function and cause X-linked dominant protoporphyria without anemia or iron overload. Am J Hum Genet (2008) 1.64

ACP Best Practice No 165: front line tests for the investigation of suspected porphyria. J Clin Pathol (2001) 1.50

Enzymatic defect in "X-linked" sideroblastic anemia: molecular evidence for erythroid delta-aminolevulinate synthase deficiency. Proc Natl Acad Sci U S A (1992) 1.46

Inducing iron deficiency improves erythropoiesis and photosensitivity in congenital erythropoietic porphyria. Blood (2015) 1.40

Perrault syndrome is caused by recessive mutations in CLPP, encoding a mitochondrial ATP-dependent chambered protease. Am J Hum Genet (2013) 1.37

The porphyrias: advances in diagnosis and treatment. Blood (2012) 1.34

Functional proteolytic complexes of the human mitochondrial ATP-dependent protease, hClpXP. J Biol Chem (2002) 1.24

Rapid procedure for fecal porphyrin assay. Clin Chem (1985) 1.07

Snx3 regulates recycling of the transferrin receptor and iron assimilation. Cell Metab (2013) 1.06

Modulation of the phenotype in dominant erythropoietic protoporphyria by a low expression of the normal ferrochelatase allele. Am J Hum Genet (1996) 1.05

Mechanistic insights into bacterial AAA+ proteases and protein-remodelling machines. Nat Rev Microbiol (2015) 1.01

Mitochondrial ClpX Activates a Key Enzyme for Heme Biosynthesis and Erythropoiesis. Cell (2015) 1.00

Increased plasma transferrin, altered body iron distribution, and microcytic hypochromic anemia in ferrochelatase-deficient mice. Blood (2006) 0.99

Lon peptidase 1 (LONP1)-dependent breakdown of mitochondrial 5-aminolevulinic acid synthase protein by heme in human liver cells. J Biol Chem (2011) 0.99

TMEM14C is required for erythroid mitochondrial heme metabolism. J Clin Invest (2014) 0.96

Pyridoxine refractory X-linked sideroblastic anemia caused by a point mutation in the erythroid 5-aminolevulinate synthase gene. Blood (1997) 0.96

EKLF/KLF1 is ubiquitinated in vivo and its stability is regulated by activation domain sequences through the 26S proteasome. FEBS Lett (2006) 0.93

High-performance liquid chromatography of porphyrins. J Chromatogr (1988) 0.91

Excessive erythrocyte PPIX influences the hematologic status and iron metabolism in patients with dominant erythropoietic protoporphyria. Cell Mol Biol (Noisy-le-grand) (2009) 0.90

Gene dosage analysis identifies large deletions of the FECH gene in 10% of families with erythropoietic protoporphyria. J Invest Dermatol (2007) 0.89

Molecular and functional analysis of the C-terminal region of human erythroid-specific 5-aminolevulinic synthase associated with X-linked dominant protoporphyria (XLDPP). Hum Mol Genet (2012) 0.87

Functional interactions between erythroid Krüppel-like factor (EKLF/KLF1) and protein phosphatase PPM1B/PP2Cβ. J Biol Chem (2012) 0.83

The Mitochondrial Unfoldase-Peptidase Complex ClpXP Controls Bioenergetics Stress and Metastasis. PLoS Biol (2016) 0.82

Barrel-shaped ClpP Proteases Display Attenuated Cleavage Specificities. ACS Chem Biol (2015) 0.82

A new mutation of the ALAS2 gene in a large family with X-linked sideroblastic anemia. Hum Genet (1995) 0.81

Down-regulation of the mitochondrial matrix peptidase ClpP in muscle cells causes mitochondrial dysfunction and decreases cell proliferation. Free Radic Biol Med (2015) 0.80

X-linked protoporphyria: Iron supplementation improves protoporphyrin overload, liver damage and anaemia. Br J Haematol (2015) 0.80

Role of genetic testing in the management of patients with inherited porphyria and their families. Ann Clin Biochem (2013) 0.80

In ferrochelatase-deficient protoporphyria patients, ALAS2 expression is enhanced and erythrocytic protoporphyrin concentration correlates with iron availability. Blood Cells Mol Dis (2014) 0.80

Novel Mechanisms for Heme-dependent Degradation of ALAS1 Protein as a Component of Negative Feedback Regulation of Heme Biosynthesis. J Biol Chem (2016) 0.78