Dragony Fu

Dragony Fu

Mercer Brugler Distinguished Teaching Professor

Professor of Biology

Director, Graduate Affairs and Admissions in Biology

Research Active

Now accepting:

PhD students

Please email with inquiries.

Office Location
333 Hutchison
Telephone
(585) 276-3346
Web Address
Website

Office Hours: By appointment

Research Overview

  • Biological functions of RNA modifications
  • Human disorders linked to RNA modifications
  • Regulation of protein synthesis

Our laboratory investigates the emerging roles of RNA modification in biological processes ranging from protein synthesis to neurodevelopment. We use an integrated biochemical, molecular, and genetic approach that employs mammalian tissue culture cells, human brain organoids, and animal models such as Drosophila and mice. Through this approach, we have discovered novel targets and functions for RNA modification. The pathways and mechanisms identified through our studies have provided critical insight into multiple aspects of human health and disease, including anti-cancer chemotherapy, neurological disorders, and aging.


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Research Interests

  • Biological functions of RNA modifications
  • Human disorders linked to RNA modifications
  • Regulation of protein synthesis

Selected Publications

  • Efthymiou S, Leo CP, Deng C, Lin SJ, Maroofian R, Lin R, Karagoz I, Zhang K, Kaiyrzhanov R, Scardamaglia A, Owrang D, Turchetti V, Jahnke F, Huang K, Petree C, Derrick AV, Rees MI, Alvi JR, Sultan T, Li C, Jacquemont ML, Tran-Mau-Them F, Valenzuela-Palafoll M, Sidlow R, Yoon G, Morrow MM, Carere DA, O'Connor M, Fleischer J, Gerkes EH, Phornphutkul C, Isidor B, Rivier-Ringenbach C, Philippe C, Kurul SH, Soydemir D, Kara B, Sunnetci-Akkoyunlu D, Bothe V, Platzer K, Wieczorek D, Koch-Hogrebe M, Rahner N, Thuresson AC, Matsson H, Frykholm C, Bozdoğan ST, Bisgin A, Chatron N, Lesca G, Cabet S, Tümer Z, Hjortshøj TD, Rønde G, Marquardt T, Reunert J, Afzal E, Zamani M, Azizimalamiri R, Galehdari H, Nourbakhsh P, Chamanrou N, Chung SK, Suri M, Benke PJ, Zaki MS, Gleeson JG, Calame DG, Pehlivan D, Yilmaz HI, Gezdirici A, Rad A, Abumansour IS, Oprea G, Bereketoğlu MB, Banneau G, Julia S, Zeighami J, Ashoori S, Shariati G, Sedaghat A, Sabri A, Hamid M, Parvas S, Tajudin TA, Abdullah U, Baig SM, Chung WK, Glazunova OO, Sabine S, Cheema HA, Zifarelli G, Bauer P, Sidpra J, Mankad K, Vona B, Fry AE, Varshney GK, Houlden H, Fu D*.  Bi-allelic pathogenic variants in TRMT1 disrupt tRNA modification and induce a neurodevelopmental disorder.Am J Hum Genet. 2025 May 1;112(5):1117-1138. doi: 10.1016/j.ajhg.2025.03.015. Epub 2025 Apr 16. PMID: 40245862.
  • Berger KD, Puthenpeedikakkal AMK, Mathews DH, Fu D*. Structural Impact of 3-methylcytosine Modification on the Anticodon Stem-loop of a Neuronally-enriched Arginine tRNA. Journal of Molecular Biology. 2025 Mar 29;:169096. doi: 10.1016/j.jmb.2025.169096. [Epub ahead of print] PubMed PMID: 40158946; NIHMSID:NIHMS2067644.
  • Zhang K, Manning AC, Lentini JM, Howard J, Dalwigk F, Maroofian R, Efthymiou S, Chan P, Eliseev SI, Yang Z, Chang H, Karimiani EG, Bakhshoodeh B, Houlden H, Kaiser SM, Lowe TM, Fu D*.Human TRMT1 and TRMT1L paralogs ensure the proper modification state, stability, and function of tRNAs. Cell Reports. 2025 Jan 28;44(1):115092. doi: 10.1016/j.celrep.2024.115092. Epub 2025 Jan 8. PubMed PMID: 39786990; PubMed Central PMCID: PMC11831618.
  • Zhang K, Löhner K, Lemmink HH, Boon M, Lentini JM, de Silva N, Fu D*. Epileptic encephalopathy linked to a DALRD3 missense variant that impairs tRNA modification. Human Genetics and Genomics Advances. 2025 Jan 9;6(1):100377. doi: 10.1016/j.xhgg.2024.100377. Epub 2024 Oct 31. PubMed PMID: 39482881; PubMed Central PMCID: PMC11615593.
  • Madhwani KR, Sayied S, Ogata CH, Hogan CA, Lentini JM, Mallik M, Dumouchel JL, Storkebaum E, Fu D, O'Connor-Giles KM. tRNA modification enzyme-dependent redox homeostasis regulates synapse formation and memory. Proc Natl Acad Sci U S A. 2024 Nov 12;121(46):e2317864121. doi: 10.1073/pnas.2317864121. Epub 2024 Nov 4. PubMed PMID: 39495910; PubMed Central PMCID: PMC11572970.
  • Zhang K, Eldin P, Ciesla JH, Briant L, Lentini JM, Ramos J, Cobb J, Munger J, Fu D*. Proteolytic cleavage and inactivation of the TRMT1 tRNA modification enzyme by SARS-CoV-2 main protease. Elife. 2024 May 30;12. doi: 10.7554/eLife.90316. PubMed PMID: 38814682; PubMed Central PMCID: PMC11139479.
  • Lentini JM, Bargabos R, Chen C, Fu D*. Methyltransferase METTL8 is required for 3-methylcytosine modification in human mitochondrial tRNAs. J Biol Chem. 2022 Mar 3;298(4):101788. doi: 10.1016/j.jbc.2022.101788. [Epub ahead of print] PubMed PMID: 35247384; PubMed Central PMCID: PMC8980813.
  • Ramos J, Proven M, Halvardson J, Hagelskamp F, Kuchinskaya E, Phelan B, Bell R, Kellner S, Feuk L, Thuresson AC, Fu D*. Identification and rescue of a tRNA wobble inosine deficiency causing intellectual disability disorder. RNA. 2020 Aug 6;. doi: 10.1261/rna.076380.120. [Epub ahead of print] PubMed PMID: 32763916.
  • Lentini JM, Alsaif HS, Faqeih E, Alkuraya FS, Fu D*. DALRD3 encodes a protein mutated in epileptic encephalopathy that targets arginine tRNAs for 3-methylcytosine modification. Nature Communications. 2020 May 19;11(1):2510. doi: 10.1038/s41467-020-16321-6. PubMed PMID: 32427860; PubMed Central PMCID: PMC7237682.
  • Zhang K, Lentini JM, Prevost CT, Hashem MO, Alkuraya FS, Fu D*. An intellectual disability-associated missense variant in TRMT1 impairs tRNA modification and reconstitution of enzymatic activity. Human Mutation. 2020 Mar;41(3):600-607. doi: 10.1002/humu.23976. Epub 2020 Jan 16. PubMed PMID: 31898845.
  • Ramos J, Han L, Li Y, Hagelskamp F, Kellner SM, Alkuraya FS, Phizicky EM, Fu D*. Formation of tRNA wobble inosine in humans is disrupted by a millennia-old mutation causing intellectual disability. Mol Cell Biol. 2019 Jul 1;. doi: 10.1128/MCB.00203-19. [Epub ahead of print] PubMed PMID: 31263000.
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