CoChat
Sign Up
Cited by 103melanin and skin pigmentationZebrafish Biomedical Research ApplicationsDevelopmental Biology and Gene Regulation

transparent, a gene affecting stripe formation in Zebrafish, encodes the mitochondrial protein Mpv17 that is required for iridophore survival

Open access
Krauss, Jana, Astrinides, Pantilis, Frohnhöfer, Hans Georg, Walderich, Brigitte, Nüsslein-Volhard, Christiane·2013·Biology Open
DOI 10.1242/bio.20135132PMC PMC3711038
Open this paper in CoChat — free accountOpen PDF

Abstract

Summary: In the skin of adult zebrafish, three pigment cell types arrange into alternating horizontal stripes, melanophores in dark stripes, xanthophores in light interstripes and iridophores in both stripes and interstripes. The analysis of mutants and regeneration studies revealed that this pattern depends on interactions between melanophores and xanthophores; however, the role of iridophores in this process is less understood. We describe the adult viable and fertile mutant transparent (tra), which shows a loss or strong reduction of iridophores throughout larval and adult stages. In addition, in adults only the number of melanophores is strongly reduced, and stripes break up into spots. Stripes in the fins are normal. By cell transplantations we show that tra acts cell-autonomously in iridophores, whereas the reduction in melanophores in the body occurs secondarily as a consequence of iridophore loss. We conclude that differentiated iridophores are required for the accumulation and maintenance of melanophores during pigment pattern formation. The tra mutant phenotype is caused by a small deletion in mpv17, an ubiquituously expressed gene whose protein product, like its mammalian and yeast homologs, localizes to mitochondria. Iridophore death might be the result of mitochondrial dysfunction, consistent with the mitochondrial DNA depletion syndrome observed in mammalian mpv17 mutants. The specificity of the tra phenotype is most likely due to redundancy after gene multiplication, making this mutant a valuable model to understand the molecular function of Mpv17 in mitochondria.

The claims and statistics of this paper haven’t been checked yet.

Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.

A few free checks a day · papers already verified open instantly · results are public

Integrity signals

Registry

Crossref · 10.1242/bio.20135132

Cites

Statistics

Claims the paper makes

The claims and statistics of this paper haven’t been checked yet.

Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.

A few free checks a day · papers already verified open instantly · results are public

Claims other papers make about this one

Each is checked against this paper’s own text: does it say what the citing paper says it does?

  • The roy orbison mutation is phenotypically and molecularly remarkably similar to the zebrafish iridophore mutant transparent.

    “The roy orbison mutation is phenotypically and molecularly remarkably similar to another zebrafish iridophore mutant called transparent.”

    From A defect in the mitochondrial protein Mpv17 underlies the transparent casper zebrafish (2017) · cites this paper as Krauss et al., 2013

    The Introduction describes transparent as a zebrafish mutant with strongly reduced iridophore pigmentation, and the Results identify a deletion in mpv17. Our copy does not describe the roy orbison mutation or establish that the two mutants are phenotypically and molecularly similar (searched: “roy orbison” and “roy mpv17 iridophore mutant”). (LLM, conf=0.81)

    Experimental — accuracy not yet measured

  • Both roy orbison and transparent affect the mpv17 locus by a similar if not identical genetic lesion.

    “Our work provides genetic evidence that both roy orbison and transparent affect the mpv17 locus by a similar if not identical genetic lesion.”

    From A defect in the mitochondrial protein Mpv17 underlies the transparent casper zebrafish (2017) · cites this paper as Krauss et al., 2013

    The Results section (‘transparent encodes the mitochondrial protein Mpv17’; page unavailable) identifies a deletion in mpv17 that causes the transparent phenotype. Our copy does not identify a genetic lesion in roy orbison or compare it with transparent’s, so the claim that the two lesions are similar or identical is unsupported by this source. (LLM, conf=0.86)

    Experimental — accuracy not yet measured

  • Zebrafish mpv17 localizes to the mitochondria, similar to the human and mouse protein.

    “Krauss, et al, also recently noted that zebrafish mpv17 also localizes to the mitochondria, similar the the human and mouse protein.”

    From A defect in the mitochondrial protein Mpv17 underlies the transparent casper zebrafish (2017) · cites this paper as Krauss et al., 2013

  • The transparent mutant was linked to the identical defect in the mpv17 mRNA.

    “Importantly, a recent publication linked the highly similar mutant tra to the identical defect in the mpv17 mRNA (Krauss et al., 2013), suggesting these two mutants are caused by similar or even the same lesion.”

    From A defect in the mitochondrial protein Mpv17 underlies the transparent casper zebrafish (2017) · cites this paper as Krauss et al., 2013

Self-check

Percentages, sums and reported differences re-computed from the paper’s own numbers.

The self-check needs the paper’s full text.

Cites

No references could be resolved yet.

Research works printed but not linked: 48

  1. AlSaman A. Tomoum H. Invernizzi F. Zeviani M. (2012). Hepatocerebral form of mitochondrial DNA depletion syndrome due to mutation in MPV17 gene. Saudi J. Gastroenterol. 18, 285– 289 10.4103/1319-3767.98439 22824774 PMC3409892 · DOI
  2. Brand M. Granato M. Nüsslein-Volhard C. (2002). Keeping and raising zebrafish. Zebrafish: A Practical Approach Nüsslein-Volhard C Dahm R, ed 7– 37 Oxford; New York, NY: Oxford Universtiy Press.
  3. Budi E. H. Patterson L. B. Parichy D. M. (2008). Embryonic requirements for ErbB signaling in neural crest development and adult pigment pattern formation. Development 135, 2603– 2614 10.1242/dev.019299 18508863 PMC2704560 · DOI · Paper page
  4. Budi E. H. Patterson L. B. Parichy D. M. (2011). Post-embryonic nerve-associated precursors to adult pigment cells: genetic requirements and dynamics of morphogenesis and differentiation. PLoS Genet. 7, e1002044 10.1371/journal.pgen.1002044 21625562 PMC3098192 · DOI · Paper page
  5. Dooley C. M. Mongera A. Walderich B. Nüsslein-Volhard C. (2013). On the embryonic origin of adult melanophores: the role of ErbB and Kit signalling in establishing melanophore stem cells in zebrafish. Development 140, 1003– 1013 10.1242/dev.087007 23364329 · DOI
  6. Eisen J. S. Weston J. A. (1993). Development of the neural crest in the zebrafish. Dev. Biol. 159, 50– 59 10.1006/dbio.1993.1220 8365574 · DOI
  7. El-Hattab A. W. Li F. Y. Schmitt E. Zhang S. Craigen W. J. Wong L. J. (2010). MPV17-associated hepatocerebral mitochondrial DNA depletion syndrome: new patients and novel mutations. Mol. Genet. Metab. 99, 300– 308 10.1016/j.ymgme.2009.10.003 20074988 · DOI
  8. Frohnhöfer H. G. Krauss J. Maischein H.-M. Nüsslein-Volhard C. (2013). Iridophores and their interaction with other chromatophores are required for stripe formation in the zebrafish. Development 140, (in press). 10.1242/dev.096719 PMC3912879 23821036 · DOI · Paper page
  9. Furutani-Seiki M. Jiang Y. J. Brand M. Heisenberg C. P. Houart C. Beuchle D. van Eeden F. J. Granato M. Haffter P. Hammerschmidt M. (1996). Neural degeneration mutants in the zebrafish, Danio rerio. Development 123, 229– 239. 9007243
  10. Geisler R. Rauch G. J. Geiger-Rudolph S. Albrecht A. van Bebber F. Berger A. Busch-Nentwich E. Dahm R. Dekens M. P. Dooley C. (2007). Large-scale mapping of mutations affecting zebrafish development. BMC Genomics 8, 11 10.1186/1471-2164-8-11 17212827 PMC1781435 · DOI
  11. Haffter P. Odenthal J. Mullins M. C. Lin S. Farrell M. J. Vogelsang E. Haas F. Brand M. van Eeden F. J. M. Furutani-Seiki M. (1996). Mutations affecting pigmentation and shape of the adult zebrafish. Dev. Genes Evol. 206, 260– 276 10.1007/s004270050051 24173565 · DOI · Paper page
  12. Hirata M. Nakamura K. Kanemaru T. Shibata Y. Kondo S. (2003). Pigment cell organization in the hypodermis of zebrafish. Dev. Dyn. 227, 497– 503 10.1002/dvdy.10334 12889058 · DOI
  13. Hirata M. Nakamura K. Kondo S. (2005). Pigment cell distributions in different tissues of the zebrafish, with special reference to the striped pigment pattern. Dev. Dyn. 234, 293– 300 10.1002/dvdy.20513 16110504 · DOI
  14. Hultman K. A. Johnson S. L. (2010). Differential contribution of direct-developing and stem cell-derived melanocytes to the zebrafish larval pigment pattern. Dev. Biol. 337, 425– 431 10.1016/j.ydbio.2009.11.019 19931238 PMC2812685 · DOI
  15. Hultman K. A. Budi E. H. Teasley D. C. Gottlieb A. Y. Parichy D. M. Johnson S. L. (2009). Defects in ErbB-dependent establishment of adult melanocyte stem cells reveal independent origins for embryonic and regeneration melanocytes. PLoS Genet. 5, e1000544 10.1371/journal.pgen.1000544 19578401 PMC2699538 · DOI
  16. Johnson S. L. Africa D. Walker C. Weston J. A. (1995). Genetic control of adult pigment stripe development in zebrafish. Dev. Biol. 167, 27– 33 10.1006/dbio.1995.1004 7851648 · DOI
  17. Kane D. A. Kishimoto T. (2002). Cell labeling and transplantation techniques. Zebrafish: A Practical Approach Nüsslein-Volhard C Dahm R, ed 95– 119 Oxford; New York, NY: Oxford Universtiy Press.
  18. Kelsh R. N. (2004). Genetics and evolution of pigment patterns in fish. Pigment Cell Res. 17, 326– 336 10.1111/j.1600-0749.2004.00174.x 15250934 · DOI
  19. Kelsh R. N. Brand M. Jiang Y. J. Heisenberg C. P. Lin S. Haffter P. Odenthal J. Mullins M. C. van Eeden F. J. Furutani-Seiki M. (1996). Zebrafish pigmentation mutations and the processes of neural crest development. Development 123, 369– 389. 9007256
  20. Kimmel C. B. Ballard W. W. Kimmel S. R. Ullmann B. Schilling T. F. (1995). Stages of embryonic development of the zebrafish. Dev. Dyn. 203, 253– 310 10.1002/aja.1002030302 8589427 · DOI · Paper page
  21. Kirschbaum F. (1975). Untersuchungen über das Farbmuster der Zebrabarbe Brachydanio rerio (Cyprinidae, Teleostei). Roux's Arch. Dev. Biol. 177, 129– 152 10.1007/BF00848526 28304951 · DOI
  22. Lang M. R. Patterson L. B. Gordon T. N. Johnson S. L. Parichy D. M. (2009). Basonuclin-2 requirements for zebrafish adult pigment pattern development and female fertility. PLoS Genet. 5, e1000744 10.1371/journal.pgen.1000744 19956727 PMC2776513 · DOI · Paper page
  23. Le Douarin N. M. Dupin E. (2003). Multipotentiality of the neural crest. Curr. Opin. Genet. Dev. 13, 529– 536 10.1016/j.gde.2003.08.002 14550420 · DOI
  24. Lister J. A. Robertson C. P. Lepage T. Johnson S. L. Raible D. W. (1999). nacre encodes a zebrafish microphthalmia-related protein that regulates neural-crest-derived pigment cell fate. Development 126, 3757– 3767. 10433906
  25. Lister J. A. Cooper C. Nguyen K. Modrell M. Grant K. Raible D. W. (2006). Zebrafish Foxd3 is required for development of a subset of neural crest derivatives. Dev. Biol. 290, 92– 104 10.1016/j.ydbio.2005.11.014 16364284 · DOI · Paper page
  26. Lopes S. S. Yang X. Müller J. Carney T. J. McAdow A. R. Rauch G. J. Jacoby A. S. Hurst L. D. Delfino-Machín M. Haffter P. (2008). Leukocyte tyrosine kinase functions in pigment cell development. PLoS Genet. 4, e1000026 10.1371/journal.pgen.1000026 18369445 PMC2265441 · DOI · Paper page
  27. Maderspacher F. Nüsslein-Volhard C. (2003). Formation of the adult pigment pattern in zebrafish requires leopard and obelix dependent cell interactions. Development 130, 3447– 3457 10.1242/dev.00519 12810592 · DOI · Paper page
  28. Mandel H. Szargel R. Labay V. Elpeleg O. Saada A. Shalata A. Anbinder Y. Berkowitz D. Hartman C. Barak M. (2001). The deoxyguanosine kinase gene is mutated in individuals with depleted hepatocerebral mitochondrial DNA. Nat. Genet. 29, 337– 341 10.1038/ng746 11687800 · DOI
  29. Meyer zum Gottesberge A. M. Massing T. Hansen S. (2012). Missing mitochondrial Mpv17 gene function induces tissue-specific cell-death pathway in the degenerating inner ear. Cell Tissue Res. 347, 343– 356 10.1007/s00441-012-1326-7 22322422 · DOI
  30. Müller M. Smolders J. W. Meyer zum Gottesberge A. M. Reuter A. Zwacka R. M. Weiher H. Klinke R. (1997). Loss of auditory function in transgenic Mpv17-deficient mice. Hear. Res. 114, 259– 263 10.16/s0378-5955(97)00175-5 9447939
  31. Nakamasu A. Takahashi G. Kanbe A. Kondo S. (2009). Interactions between zebrafish pigment cells responsible for the generation of Turing patterns. Proc. Natl. Acad. Sci. USA 106, 8429– 8434 10.1073/pnas.0808622106 19433782 PMC2689028 · DOI · Paper page
  32. Navarro-Sastre A. García-Silva M. T. Martín-Hernández E. Lluch M. Briones P. Ribes A. (2010). Functional splicing assay supporting that c.70 + 5G > A mutation in the MPV17 gene is disease causing. J. Inherit. Metab. Dis. [Epub ahead of print] 10.1007/s10545-010-9155-x 20614188 · DOI
  33. Ng A. Uribe R. A. Yieh L. Nuckels R. Gross J. M. (2009). Zebrafish mutations in gart and paics identify crucial roles for de novo purine synthesis in vertebrate pigmentation and ocular development. Development 136, 2601– 2611 10.1242/dev.038315 19570845 PMC2709066 · DOI · Paper page
  34. Odenthal J. Rossnagel K. Haffter P. Kelsh R. N. Vogelsang E. Brand M. van Eeden F. J. Furutani-Seiki M. Granato M. Hammerschmidt M. (1996). Mutations affecting xanthophore pigmentation in the zebrafish, Danio rerio. Development 123, 391– 398. 9007257
  35. Parichy D. M. (2003). Pigment patterns: fish in stripes and spots. Curr. Biol. 13, R947– R950 10.1016/j.cub.2003.11.038 14680649 · DOI
  36. Parichy D. M. (2007). Homology and the evolution of novelty during Danio adult pigment pattern development. J. Exp. Zool. B 308, 578– 590 10.1002/jez.b.21141 17094081 · DOI
  37. Parichy D. M. Mellgren E. M. Rawls J. F. Lopes S. S. Kelsh R. N. Johnson S. L. (2000a). Mutational analysis of endothelin receptor b1 (rose) during neural crest and pigment pattern development in the zebrafish Danio rerio. Dev. Biol. 227, 294– 306 10.1006/dbio.2000.9899 11071756 · DOI · Paper page
  38. Parichy D. M. Ransom D. G. Paw B. Zon L. I. Johnson S. L. (2000b). An orthologue of the kit-related gene fms is required for development of neural crest-derived xanthophores and a subpopulation of adult melanocytes in the zebrafish, Danio rerio. Development 127, 3031– 3044. 10862741
  39. Parichy D. M. Elizondo M. R. Mills M. G. Gordon T. N. Engeszer R. E. (2009). Normal table of postembryonic zebrafish development: staging by externally visible anatomy of the living fish. Dev. Dyn. 238, 2975– 3015 10.1002/dvdy.22113 19891001 PMC3030279 · DOI · Paper page
  40. Raible D. W. Eisen J. S. (1994). Restriction of neural crest cell fate in the trunk of the embryonic zebrafish. Development 120, 495– 503. 8162850
  41. Rawls J. F. Mellgren E. M. Johnson S. L. (2001). How the zebrafish gets its stripes. Dev. Biol. 240, 301– 314 10.1006/dbio.2001.0418 11784065 · DOI · Paper page
  42. Spinazzola A. Viscomi C. Fernandez-Vizarra E. Carrara F. D'Adamo P. Calvo S. Marsano R. M. Donnini C. Weiher H. Strisciuglio P. (2006). MPV17 encodes an inner mitochondrial membrane protein and is mutated in infantile hepatic mitochondrial DNA depletion. Nat. Genet. 38, 570– 575 10.1038/ng1765 16582910 · DOI · Paper page
  43. Spinazzola A. Santer R. Akman O. H. Tsiakas K. Schaefer H. Ding X. Karadimas C. L. Shanske S. Ganesh J. Di Mauro S. (2008). Hepatocerebral form of mitochondrial DNA depletion syndrome: novel MPV17 mutations. Arch. Neurol. 65, 1108– 1113 10.1001/archneur.65.8.1108 18695062 · DOI
  44. Trott A. Morano K. A. (2004). SYM1 is the stress-induced Saccharomyces cerevisiae ortholog of the mammalian kidney disease gene Mpv17 and is required for ethanol metabolism and tolerance during heat shock. Eukaryot. Cell 3, 620– 631 10.1128/EC.3.3.620-631.2004 15189984 PMC420134 · DOI
  45. Viscomi C. Spinazzola A. Maggioni M. Fernandez-Vizarra E. Massa V. Pagano C. Vettor R. Mora M. Zeviani M. (2009). Early-onset liver mtDNA depletion and late-onset proteinuric nephropathy in Mpv17 knockout mice. Hum. Mol. Genet. 18, 12– 26 10.1093/hmg/ddn309 18818194 PMC2644642 · DOI · Paper page
  46. Walker C. Streisinger G. (1983). Induction of mutations by gamma-rays in pregonial germ cells of zebrafish embryos. Genetics 103, 125– 136. 17246099 PMC1202017
  47. Weiher H. Noda T. Gray D. A. Sharpe A. H. Jaenisch R. (1990). Transgenic mouse model of kidney disease: insertional inactivation of ubiquitously expressed gene leads to nephrotic syndrome. Cell 62, 425– 434 10.1016/0092-8674(90)90008-3 1696177 · DOI
  48. Wong L. J. Brunetti-Pierri N. Zhang Q. Yazigi N. Bove K. E. Dahms B. B. Puchowicz M. A. Gonzalez-Gomez I. Schmitt E. S. Truong C. K. (2007). Mutations in the MPV17 gene are responsible for rapidly progressive liver failure in infancy. Hepatology 46, 1218– 1227 10.1002/hep.21799 17694548 · DOI

Cited by

Sorted by citations

Authors
Krauss, J., Astrinides, P., Frohnhöfer, H. G., Walderich, B., & Nüsslein-Volhard, C.
Affiliations
  • University of Tübingen (DE)
  • Max Planck Institute for Developmental Biology (DE)
  • Max-Planck-Institut für Entwicklungsbiologie, Spemannstrasse 35, 72076 Tübingen, Germany
Corresponding author
Krauss, Jana
ORCID
Frohnhöfer, Hans Georg
Venue
Biology Open
Publisher
The Company of Biologists
Published
2013-06-03
Volume
2
Issue
7
Pages
703-710
ISSN
2046-6390 (ISSN-L)
DOI
10.1242/bio.20135132
PMC
PMC3711038
OpenAlex
W2100856195
Cited by
103
References
0
Citations by year
2026: 6 · 2025: 4 · 2024: 1 · 2023: 6 · 2022: 5 · 2021: 17 · 2020: 10 · 2019: 13 · 2018: 5 · 2017: 9 · 2016: 9 · 2015: 6 · 2014: 10 · 2013: 2
FWCI
4.15
Type
journal article
Language
en
Open access
Licence
Locations
  • Biology Open (publishedVersion, cc-by)
  • Europe PMC (PubMed Central) (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed Central (publishedVersion)
  • PubMed (publishedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Funders
Max-Planck-Gesellschaft
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › melanin and skin pigmentation
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Zebrafish Biomedical Research Applications
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
Keywords
MPV17, iridophores, melanophores, xanthophores, pigment pattern formation, stripe formation, mitochondrial dysfunction, mitochondrial DNA depletion syndrome, cell transplantation, gene duplication

About these checks

Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.

Statistics and arithmetic are recomputed, not judged.

Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.

What counts as a claim ›How we measure confidence ›How we check this ›

Are you an author, or do you see a problem with this page? Request a review