Abstract
ATP8A2 can translocate phospholipids from exoplasmic to the cytoplasmic leaflets to establish and maintain the lipid asymmetry between the two leaflets. ATP8A2 has been recently reported due to the patients possessing severe neurological disorders with the mutated gene of ATP8A2, including cerebellar ataxia, mental retardation and dysequilibrium. In vivo experiments have identified the critical role of ATP8A2 in the nerve system of mice. In vitro experiments have shown the function of ATP8A2 in promoting the development of hippocampal neurons in rats together with CDC50A. Our previous research has focused on the presence and expression level of ATP8A2 in the prefrontal cortex of mice in neuroinflammation conditions and found that neonatal LPS exposure reduces ATP8A2 level in the prefrontal cortex in mice via increasing IFN-γ level. We write the review to facilitate understanding ATP8A2. This review focuses on the structure, function and distribution of ATP8A2 in humans and rodents and the human diseases related to ATP8A2.
Similar content being viewed by others
REFERENCES
Vestergaard, A.L., Coleman, J.A., Lemmin, T., Mikkelsen, S.A., Molday, L.L., Vilsen, B., Molday, R.S., Dal Peraro, M. and Andersen, J.P., Proc. Natl. Acad. Sci. USA, 2014, vol. 111, no. 14, pp. E1334–1343.
Coleman, J.A., Vestergaard, A.L., Molday, R.S., Vilsen, B. and Andersen, J.P., Proc. Natl. Acad. Sci. USA, 2012, vol. 109, no. 5, pp. 1449–1454.
Toyoshima, C., Biochim. Biophys. Acta., 2009, vol. 1793, no. 6, pp. 941–946.
Moller, J.V., Olesen, C., Winther, A.M. and Nissen, P., Q. Rev. Biophys., 2010, vol. 43, no. 4, pp. 501–566
Morth, J.P., Pedersen, B.P., Toustrup-Jensen, M.S., Sorensen, T.L., Petersen, J., Andersen, J.P., Vilsen, B. and Nissen, P., Nature, 2007, vol. 450, no. 7172, pp. 1043–1049.
Coleman, J.A., Kwok, M.C. and Molday, R.S., J. Biol. Chem., 2009, vol. 284, no. 47, pp. 32670–32679.
Mikkelsen, S.A., Mogensen, L.S., Vilsen, B., Molday, R.S., Vestergaard, A.L. and Andersen, J.P., J. Biol. Chem., 2019, vol. 294, no. 15, pp. 5970–5979.
Jacquot, A., Montigny, C., Hennrich, H., Barry, R., le Maire, M., Jaxel, C., Holthuis, J., Champeil, P. and Lenoir, G., J. Biol. Chem., 2012, vol. 287, no. 16, pp. 13249–13261.
Palmgren, M.G. and Nissen, P., Annu. Rev. Biophys., 2011, vol. 40, pp. 243–266.
Durr, K.L., Tavraz, N.N., Dempski, R.E., Bamberg, E. and Friedrich, T., J. Biol. Chem., 2009, vol. 284, no. 6, pp. 3842–3854.
Coleman, J.A. and Molday, R.S., J. Biol Chem, 2011, vol. 286, no. 19, pp. 17205–17216.
Post, R.L., Hegyvary, C. and Kume, S., J. Biol. Chem., 1972, vol. 247, no. 20, pp. 6530–6540.
van der Mark, V., Elferink, R. and Paulusma, C., Int. J. Mol. Sci., 2013, vol. 14, no. 4, pp. 7897–7922
Blostein, R., Wilczynska, A., Karlish, S.J., Arguello, J.M. and Lingrel, J.B., J. Biol. Chem., 1997, vol. 272, no. 40, pp. 24987–24993.
Enyedi, A., Vorherr, T., James, P., McCormick, D.J., Filoteo, A.G., Carafoli, E. and Penniston, J.T., J. Biol. Chem., 1989, vol. 264, no. 21, pp. 12313–12321.
Fuglsang, A.T., Visconti, S., Drumm, K., Jahn, T., Stensballe, A., Mattei, B., Jensen, O.N., Aducci, P. and Palmgren, M.G., J. Biol. Chem., 1999, vol. 274, no. 51, pp. 36774–36780.
Zhou, X., Sebastian, T.T. and Graham, T.R., J. Biol. Chem., 2013, vol. 288, no. 44, pp. 31807–31815.
Chalat, M., Moleschi, K. and Molday, R. S., Mol. Biol. Cell., 2017, vol. 28, no. 3, pp. 452–462.
Natarajan, P., Liu, K., Patil, D.V., Sciorra, V.A., Jackson, C.L. and Graham, T.R., Nat. Cell. Biol., 2009, vol. 11, no. 12, pp. 1421–1426.
Puts, C.F. and Holthuis, J.C., Biochim. Biophys. Acta., 2009, vol. 1791, no. 7, pp. 603–611.
Baldridge, R.D. and Graham, T.R., Proc. Natl. Acad. Sci. USA, 2012, vol. 109, no. 6, pp. E290-298.
Baldridge, R.D. and Graham, T.R., Proc. Natl. Acad. Sci. USA, 2013, vol. 110, no. 5, pp. E358-367.
Andersen, J.P., Vestergaard, A.L., Mikkelsen, S.A., Mogensen, L.S., Chalat, M. and Molday, R.S., Front. Physiol., 2016, vol. 7, pp. 275.
Onat, O.E., Gulsuner, S., Bilguvar, K., Nazli Basak, A., Topaloglu, H., Tan, M., Tan, U., Gunel, M. and Ozcelik, T., Eur. J. Hum. Genet., 2013, vol. 21, no. 3, pp. 281–285.
Coleman, J.A., Quazi, F. and Molday, R.S., Biochim. Biophys. Acta., 2013, vol. 1831, no. 3, pp. 555–574.
Vestergaard, A.L., Mikkelsen, S.A., Coleman, J.A., Molday, R.S., Vilsen, B. and Andersen, J.P., FEBS Lett., 2015, vol. 589, no. 24 Pt B, pp. 3908–3914.
Folmer, D.E., Mok, K.S., de Wee, S.W., Duijst, S., Hiralall, J.K., Seppen, J., Oude Elferink, R.P. and Paulusma, C.C., J. Histochem. Cytochem., 2012, vol. 60, no. 3, pp. 205–218.
van der Velden, L.M., Wichers, C.G., van Breevoort, A.E., Coleman, J.A., Molday, R.S., Berger, R., Klomp, L.W. and van de Graaf, S.F., J. Biol. Chem., 2010, vol. 285, no. 51, pp. 40088–40096.
Coleman, J.A., Zhu, X., Djajadi, H R., Molday, L.L., Smith, R.S., Libby, R.T., John, S.W. and Molday, R.S., J. Cell. Sci., 2014, vol. 127, no. Pt 5, pp. 1138–1149.
Yang, Y., Sun, K., Liu, W., Zhang, L., Peng, K., Zhang, S., Li, S., Yang, M., Jiang, Z., Lu, F. and Zhu, X., Cell Death Dis., 2018, vol. 9, no. 9, pp. 899.
Tadini-Buoninsegni, F., Mikkelsen, S.A., Mogensen, L.S., Molday, R.S. and Andersen, J.P., Proc. Natl. Acad. Sci. USA, 2019, vol. 116, no. 33, pp. 16332–16337.
Tadini-Buoninsegni, F., Mikkelsen, S.A., Mogensen, L.S., Holm, R., Molday, R.S. and Andersen, J.P., FEBS Lett., 2022.
Maruoka, M. and Suzuki, J., Neurosci. Res., 2021, vol. 167, pp. 30–37.
DeVries, G.H. and Zmachinski, C.J., J. Neurochem. 1980, vol. 34, no. 2, pp. 424–430.
Kalin, N., Fernandes, J., Hrafnsdottir, S. and van Meer, G., J. Biol. Chem., 2004, vol. 279, no. 32, pp. 33228–33236.
Xu, Q., Yang, G.Y., Liu, N., Xu, P., Chen, Y.L., Zhou, Z., Luo, Z.G. and Ding, X., FEBS Lett., 2012, vol. 586, no. 13, pp. 1803–1812.
Zhu, X., Libby, R.T., de Vries, W.N., Smith, R.S., Wright, D.L., Bronson, R.T., Seburn, K.L. and John, S.W., PLoS. Genet., 2012, vol. 8, no. 8, pp. e1002853.
Deng, J., Song, L., Yang, Z., Zheng, S., Du, Z., Luo, L., Liu, J., Jin, X. and Yang, J., Brain Res. Bull., 2021, vol. 171, pp. 103–112.
Zhang, L., Yang, Y., Li, S., Zhang, S., Zhu, X., Tai, Z., Yang, M., Liu, Y., Guo, X., Chen, B., Jiang, Z., Lu, F. and Zhu, X., Sci. Rep., 2017, vol. 7, no. 1.
Wu, Y., Tibrewal, N. and Birge, R.B., Trends Cell. Biol., 2006, vol. 16, no. 4, pp. 189–197.
Almasieh, M., Faris, H. and Levin, L.A., Int. J. Biochem. Cell Biol., 2022, vol. 150, pp. 106264.
Ruggiero, L., Connor, M.P., Chen, J., Langen, R. and Finnemann, S.C., Proc. Natl. Acad. Sci. USA, 2012, vol. 109, no. 21, pp. 8145–8148.
Cacciagli, P., Haddad, M.R., Mignon-Ravix, C., El-Waly, B., Moncla, A., Missirian, C., Chabrol, B. and Villard, L., Eur. J. Hum. Genet., 2010, vol. 18, no. 12, pp. 1360–1363.
Martin-Hernandez, E., Rodriguez-Garcia, M.E., Camacho, A., Matilla-Duenas, A., Garcia-Silva, M.T., Quijada-Fraile, P., Corral-Juan, M., Tejada-Palacios, P., de Las Heras, R.S., Arenas, J., Martin, M.A. and Martinez-Azorin, F., Neurogenetics, 2016, vol. 17, no. 4, pp. 259–263.
Alsahli, S., Alrifai, M.T., Al Tala, S., Mutairi, F.A. and Alfadhel, M., J. Cent. Nerv. Syst. Dis., 2018, vol. 10.
McMillan, H.J., Telegrafi, A., Singleton, A., Cho, M., Lelli, D., Lynn, F.C., Griffin, J., Asamoah, A., Rinne, T., Erasmus, C.E., Koolen, D.A., Haaxma, C.A., Keren, B., Doummar, D., Mignot, C., Thompson, I., Velsher, L., Dehghani, M., Vahidi Mehrjardi, M., Maroofian, R., Tchan, M., Simons, C., Christodoulou, J., Martín-Hernández, E., Guillen Sacoto, M.J., Henderson, L.B., McLaughlin, H., Molday, L.L., Molday, R.S. and Yoon, G., Can. J. Neurol. Sci., 2018, vol. 45, no. s2, pp. S12–S12.
Guissart, C., Harrison, A.N., Benkirane, M., Oncel, I., Arslan, E.A., Chassevent, A.K., Baraano, K., Larrieu, L., Iascone, M., Tenconi, R., Claustres, M., Eroglu-Ertugrul, N., Calvas, P., Topaloglu, H., Molday, R.S. and Koenig, M., J. Neurol., 2020, vol. 267, no. 1, pp. 203–213.
Mohamadian, M., Ghandil, P., Naseri, M., Bahrami, A. and Momen, A.A., J. Clin. Lab. Anal., 2020, vol. 34, no. 11, pp. e23484.
Damasio, J., Santos, D., Morais, S., Bras, J., Guerreiro, R., Sardoeira, A., Cavaco, S., Carrilho, I., Barbot, C., Barros, J. and Sequeiros, J., Clin. Genet., 2021, vol. 100, no. 1, pp. 79–83.
Heidari, E., Harrison, A.N., Jafarinia, E., Tavasoli, A.R., Almadani, N., Molday, R.S. and Garshasbi, M., Hum. Mutat., 2021, vol. 42, no. 5, pp. 491–497.
Narishige, Y., Yaoita, H., Shibuya, M., Ikeda, M., Kodama, K., Kawashima, A., Okubo, Y., Endo, W., Inui, T., Togashi, N., Tanaka, S., Kobayashi, Y., Onuma, A., Takayama, J., Tamiya, G., Kikuchi, A., Kure, S. and Haginoya, K., Tohoku J. Exp. Med., 2022, vol. 256, no. 4, pp. 321–326.
Quintas, S., Moldovan, O., dos Santos, T.P. and Levy, A., Eur. J. Paediatr. Neuro., 2017, vol. 21, pp. e65.
Choi, H., Andersen, J.P. and Molday, R.S., Hum. Mutat., 2019, vol. 40, no. 12, pp. 2353–2364.
McMillan, H.J., Telegrafi, A., Singleton, A., Cho, M.T., Lelli, D., Lynn, F.C., Griffin, J., Asamoah, A., Rinne, T., Erasmus, C.E., Koolen, D.A., Haaxma, C.A., Keren, B., Doummar, D., Mignot, C., Thompson, I., Velsher, L., Dehghani, M., Vahidi Mehrjardi, M.Y., Maroofian, R., Tchan, M., Simons, C., Christodoulou, J., Martin-Hernandez, E., Guillen Sacoto, M.J., Henderson, L.B., McLaughlin, H., Molday, L.L., Molday, R.S. and Yoon, G., Orphanet J. Rare Dis., 2018, vol. 13, no. 1, pp. 86.
Funding
This work was supported by the starting fund for high-level talent introduction into Guangdong Pharmaceutical University (51355093).
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interests. The authors declare no conflict of interest.
Ethical approval. This article does not contain any studies with human participants or experimental animals performed by any of the authors.
Additional information
Correspondence author; address: Room 510#, Building of Basic Medical Sciences, School of Life Sciences & Biopharmaceutics, Guangdong Pharmaceutical University, No. 280, East Waihuan Street, Higher Education Mega Center, Guangzhou City, Guangdong Province, 510006 P. R. China; e-mail: jhyang2018@gdpu.edu.cn.
Supplementary Information
Rights and permissions
About this article
Cite this article
Junyuan Zeng, Junhua Yang Structure, Function and Distribution of ATP8A2 in Human and Mice. Neurochem. J. 17, 443–451 (2023). https://doi.org/10.1134/S1819712423030200
Received:
Revised:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1134/S1819712423030200