The DOI matches its Crossref record; 9 of the 9 references it cites were found in a registry or an index.
FAM 134B oligomerization drives endoplasmic reticulum membrane scission for ER ‐phagy
Open accessAbstract
Degradation of endoplasmic reticulum (ER) by selective autophagy (ER‐phagy) is crucial for ER homeostasis. However, it remains unclear how ER scission is regulated for subsequent autophagosomal sequestration and lysosomal degradation. Here, we show that oligomerization of ER‐phagy receptor FAM134B (also referred to as reticulophagy regulator 1 or RETREG1) through its reticulon‐homology domain is required for membrane fragmentation in vitro and ER‐phagy in vivo. Under ER‐stress conditions, activated CAMK2B phosphorylates the reticulon‐homology domain of FAM134B, which enhances FAM134B oligomerization and activity in membrane fragmentation to accommodate high demand for ER‐phagy. Unexpectedly, FAM134B G216R, a variant derived from a type II hereditary sensory and autonomic neuropathy (HSAN) patient, exhibits gain‐of‐function defects, such as hyperactive self‐association and membrane scission, which results in excessive ER‐phagy and sensory neuron death. Therefore, this study reveals a mechanism of ER membrane fragmentation in ER‐phagy, along with a signaling pathway in regulating ER turnover, and suggests a potential implication of excessive selective autophagy in human diseases.
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Cites
Guidelines for the use and interpretation of assays for monitoring autophagy (3rd edition).
Klionsky, D. J., Abdelmohsen, K., Abe, A., Abedin, M. J., Abeliovich, H., Acevedo Arozena, A., Adachi, H., Adams, C. M., Adams, P. D., Adeli, K., et al. (2016) · Autophagy · doi:10.1080/15548627.2015.1100356 · Cited by 5,894
Self-citationThe BioPlex network: a systematic exploration of the human interactome
Huttlin, E. L., Ting, L., Bruckner, R. J., Gebreab, F., Gygi, M. P., Szpyt, J., Tam, S., Zarraga, G., Colby, G., Baltier, K., et al. (2015) · Cell · doi:10.1016/j.cell.2015.06.043 · Cited by 1,425 · cited 1×
“Another ER-phagy receptor, SEC62, also locates on ER sheets (Fumagalli et al , 2016), and interestingly, SEC62 associates with FAM134C (Huttlin et al , 2015), a FAM134 family member that has broader tissue distribution than FAM134B (Kurth et al , 2009).”
Cargo recognition and trafficking in selective autophagy
Stolz, A., Ernst, A., & Dikic, I. (2014) · Nature Cell Biology · doi:10.1038/ncb2979 · Cited by 1,207
Cargo recognition and degradation by selective autophagy
Gatica, D., Lahiri, V., & Klionsky, D. J. (2018) · Nature Cell Biology · doi:10.1038/s41556-018-0037-z · Cited by 1,067
Regulation of endoplasmic reticulum turnover by selective autophagy
Khaminets, A., Heinrich, T., Mari, M., Grumati, P., Huebner, A. K., Akutsu, M., Liebmann, L., Stolz, A., Nietzsche, S., Koch, N., et al. (2015) · Nature · doi:10.1038/nature14498 · Cited by 956
Receptor-mediated selective autophagy degrades the endoplasmic reticulum and the nucleus
Mochida, K., Oikawa, Y., Kimura, Y., Kirisako, H., Hirano, H., Ohsumi, Y., & Nakatogawa, H. (2015) · Nature · doi:10.1038/nature14506 · Cited by 611
Kurth, I., Pamminger, T., Hennings, J. C., Soehendra, D., Huebner, A. K., Rotthier, A., Baets, J., Senderek, J., Topaloglu, H., Farrell, S. A., et al. (2009) · Nature Genetics · doi:10.1038/ng.464 · Cited by 260 · cited 3×
“…a good model system to address above questions, because FAM134B induces liposome fragmentation in vitro and mediates ER-phagy in vivo (Khaminets et al , 2015), and more importantly, the dysfunction of FAM134B causes hereditary sensory and autonomic neuropathy type 2 (HSAN II; Kurth et al , 2009).”
Rose, A. J., Kiens, B., & Richter, E. A. (2006) · The Journal of Physiology · doi:10.1113/jphysiol.2006.111757 · Cited by 247
Mutation in FAM134B causing severe hereditary sensory neuropathy: Figure 1
Murphy, S. M., Davidson, G. L., Brandner, S., Houlden, H., & Reilly, M. M. (2012) · Journal of Neurology, Neurosurgery & Psychiatry · doi:10.1136/jnnp.2010.228965 · Cited by 62
Cited by
Autophagy and organelle homeostasis in cancer
Miller, D. R., & Thorburn, A. (2021) · Developmental Cell · doi:10.1016/j.devcel.2021.02.010 · Cited by 258
Metadata
- Authors
- Jiang, X., Wang, X., Ding, X., Du, M., Li, B., Weng, X., Zhang, J., Li, L., Tian, R., Zhu, Q., Chen, S., Wang, L., Liu, W., Fang, L., Neculai, D., & Sun, Q.
- Venue
- The EMBO Journal
- Published
- 2020-01-13
- Volume
- 39
- Issue
- 5
- Pages
- e102608-e102608
- ISSN
- 0261-4189 (ISSN-L)
- 1460-2075
- OpenAlex
- W2998839719
- Cited by
- 161
- References
- 9
- FWCI
- 12.13
- Type
- journal article
- Language
- en
- Open access
- bronze
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- Jiang, X., Wang, X., Ding, X., Du, M., Li, B., Weng, X., Zhang, J., Li, L., Tian, R., Zhu, Q., Chen, S., Wang, L., Liu, W., Fang, L., Neculai, D., & Sun, Q. (2020). FAM 134B oligomerization drives endoplasmic reticulum membrane scission for ER ‐phagy. The EMBO Journal. https://doi.org/10.15252/embj.2019102608
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