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Peroxisome Morphology in Pathologies

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Book cover Molecular Machines Involved in Peroxisome Biogenesis and Maintenance

Abstract

Peroxisomes are ubiquitous and heterogeneous multi-purpose organelles, which are indispensable for human health and development. The invention of specific cytochemical staining methods for peroxisomes revealed their high plasticity and ability to alter their morphology in response to environmental cues. Peroxisome dynamics depend on peroxisomal morphology proteins such as Pex11p, DLP1/Drp1, Fis1, Mff, and GDAP1 which are partially shared with mitochondria. Here, we address variations of peroxisome morphology in the healthy organism and summarize findings on altered organelle morphology in peroxisomal disorders. We highlight recent insights in novel disorders with defects in peroxisome morphology proteins and alterations of peroxisomes during stress and signaling, as well as secondary alterations in liver disease and cancer.

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Abbreviations

AOX:

Acyl-CoA oxidase

CMT:

Charcot-Marie-Tooth disease

D-BP:

D-bifunctional protein

DAB:

Diaminobenzidine

DHA:

Docosahexaenoic acid

DLP1/Drp1:

Dynamin-like/related protein 1

ER:

Endoplasmic reticulum

GDAP1:

Ganglioside-induced differentiation associated protein 1

Mff:

Mitochondrial fission factor

PBD:

Peroxisome biogenesis disorder

PEX:

Peroxin

PMP:

Peroxisomal membrane protein

PPAR:

Peroxisome proliferator activated receptor

PTS:

Peroxisomal targeting signal

ROS:

Reactive oxygen species

SED:

Single enzyme deficiency

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Acknowledgments

We would like to thank M. Ebberink and H. Waterham (Amsterdam Medical Center), H. Shamseddin and F.S. Alkuraya (King Faisal Specialist Hospital and Research Center, Riyadh) for kindly providing images of patient fibroblasts and members of the laboratory for stimulating discussions and comments on the manuscript. We apologize to those whose work has not been cited owing to space limitations. This work was generously supported by a Wellcome Trust Institutional Strategic Support Award (WT097835MF), BBSRC (BB/K006231/1), the Portuguese Foundation for Science and Technology (FCT) and FEDER/COMPETE (PTDC/SAU/OSM/103647/2008; PTDC/BIA-BCM/099613/2008; PTDC/BIA-BCM/118605/2010; SFRH/BPD/74428/2010 to M.I.). I.C. is supported by a fellowship from the University of Exeter, UK.

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Authors and Affiliations

  1. College of Life and Environmental Sciences, Biosciences, University of Exeter, Geoffrey Pope Building, Stocker Road, Exeter, EX4 4QD, UK

    Michael Schrader & Inês Castro

  2. Department of Biology, Centre for Cell Biology, University of Aveiro, Campus Universitário de Santiago, 3810-193, Aveiro, Portugal

    Michael Schrader

  3. Division of Medical Cell Biology, Department of Anatomy and Cell Biology, University of Heidelberg, 69120, Heidelberg, Germany

    H. Dariush Fahimi

  4. Department of Neuroanatomy, Center for Biomedicine and Medical Technology Mannheim, University of Heidelberg, 68167, Mannheim, Germany

    Markus Islinger

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  1. Michael Schrader
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  2. Inês Castro
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  3. H. Dariush Fahimi
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  4. Markus Islinger
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Correspondence to Michael Schrader .

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  1. Mauerbach, Austria

    Cecile Brocard

  2. Max F. Perutz Laboratories University of Vienna, Vienna, Austria

    Andreas Hartig

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Schrader, M., Castro, I., Fahimi, H.D., Islinger, M. (2014). Peroxisome Morphology in Pathologies. In: Brocard, C., Hartig, A. (eds) Molecular Machines Involved in Peroxisome Biogenesis and Maintenance. Springer, Vienna. https://doi.org/10.1007/978-3-7091-1788-0_7

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