Pelizaeus–Merzbacher disease: on the cusp of myelin medicine

2024

Matthew S. Elitt, Paul J. Tesar
 
 
 

Pervasive environmental chemicals impair oligodendrocyte development

2024

Nature Neuroscience

Erin F. Cohn, Benjamin L. L. Clayton, Mayur Madhavan, Kristin A. Lee, Sara Yacoub, Yuriy Fedorov, Marissa A. Scavuzzo, Katie Paul Friedman, Timothy J. Shafer & Paul J. Tesar 

 

A phenotypic screening platform for identifying chemical modulators of astrocyte reactivity

2024

Nature Neuroscience

Benjamin L. L. Clayton, James D. Kristell, Kevin C. Allan, Erin F. Cohn, Molly Karl, Andrew D. Jerome, Eric Garrison, Yuka Maeno-Hikichi, Annalise M. Sturno, Alexis Kerr, H. Elizabeth Shick, Jesse A. Sepeda, Eric C. Freundt, Andrew R. Sas, Benjamin M. Segal, Robert H. Miller & Paul J. Tesar 

Patient iPSC models reveal glia-intrinsic phenotypes in multiple sclerosis

2023

bioRxiv (pre-print)

Clayton BLL, Barbar L, Sapar M, Rusielewicz T, Kalpana K, Migliori B; NYSCF Global Stem Cell Array® Team; Paull D, Brenner K, Moroziewicz D, Sand IK, Casaccia P, Tesar PJ, Fossati V.

Enteric glial hub cells coordinate intestinal motility

2023

bioRxiv (pre-print)

Scavuzzo MA, Letai KC, Maeno-Hikichi Y, Wulftange WJ, Shah IK, Rameshbabu JS, Tomar A, Shick HE, Shah AK, Xiong Y, Cohn EF, Allan KC, Tesar PJ.

Pervasive environmental chemicals impair oligodendrocyte development

2023

bioRxiv (pre-print)

Cohn EF, Clayton BLL, Madhavan M, Yacoub S, Federov Y, Paul-Friedman K, Shafer TJ, Tesar PJ.

Cellular Maturation of Oligodendrocytes is Governed by Transient Gene Melting

2022

bioRxiv (pre-print)

Allan KC, Miller TE, Morton AR, Scavuzzo MA, Elitt ME, Clayton BLL, Hu LR, Vrabic JK, Olsen HE, Factor DC, Henninger JE, Young RA, Lin CY, Scacheri PC, Tesar PJ.

 

Oligodendrocyte differentiation alters tRNA modifications and codon optimality-mediated mRNA decay

2022

Nature Communications

Martin S, Allan KC, Pinkard O, Sweet T, Tesar PJ, Coller J.

Chemical inhibition of pathological reactive astrocytes promotes neural protection

2022

bioRxiv (pre-print)

Clayton BLL, Kristell JD, Allan KC, Karl M, Garrison E, Maeno-Hikichi Y, Sturno AM, Shick HE, Miller RH, Tesar PJ.

Oligodendrocyte progenitor cell fate and function in development and disease

2021

Current Opinion in Cell Biology

Clayton BLL, Tesar PJ.

Non-Canonical Targets of HIF1a Impair Oligodendrocyte Progenitor Cell Function

2021

Cell Stem Cell

Allan KC, Hu LR, Scavuzzo MA, Morton AR, Gevorgyan AS, Cohn, E., Clayton BLL, Bederman I, Hung S, Bartels CF, Madhavan M, Tesar PJ.

Non-canonical targets of HIF1a drive cell-type-specific dysfunction

2020

bioRxiv (pre-print)

Allan KC, Hu LR, Morton AR, Scavuzzo MA, Gevorgyan AS, Clayton BLL, Bederman I, Hung S, Bartels CF, Madhavan M, Tesar PJ.

Suppression of proteolipid protein rescues Pelizaeus-Merzbacher disease

2020

Nature

Elitt MS, Barbar L, Shick E, Powers BE, Maeno-Hikitchi Y, Madhavan M, Allan KC, Nawash BS, Gevorgyan AS, Hung S, Nevin ZS, Olsen HE, Hitomi M, Schlatzer DM, Zhao HT, Swayze A, LePage DF, Jiang W, Conlon RA, Rigo F, Tesar PJ.

Cell type-specific intralocus interactions reveal oligodendrocyte mechanisms in MS

2020

Cell

Factor DC, Barbeau AM, Allan KC, Hu LR, Madhavan M, Hoang AT, Hazel KEA, Hall PA, Nisraiyya S, Najm FJ, Miller TE, Nevin ZS, Karl RT, Lima BR, Song Y, Sibert AG, Dhillon GK, Volsko C, Bartels CF, Adams DJ, Dutta R, Gallagher MD, Phu W, Kozlenkov A, Dracheva S, Scacheri PC, Tesar PJ, Corradin O.

Pathogenic Prion Protein Isoforms Are Not Present in Cerebral Organoids Generated from Asymptomatic Donors Carrying the E200K Mutation Associated with Familial Prion Disease

2020

Pathogens

Foliaki ST, Groveman BR, Yuan J, Walters R, Zhang S, Tesar P, Zou W, Haigh CL.

Disorders of myelin (Chapter 13)

2020

Neurodevelopmental Disorders (Book)
Editors: Rubenstein J, Rakic P, Chen B, Kwan KY,

Clayton BL, Allan KC, Nevin ZS, Elitt MS, Tesar PJ.

Oligodendrocyte intrinsic miR-27a controls myelination and remyelination

2019

Cell Reports

Tripathi A, Volsko C, Garcia JP, Agirre E, Allan KC, Tesar PJ, Trapp BD, Castelo-Branco G, Sim FJ, Dutta R.

The chromatin environment around interneuron genes in oligodendrocyte precursor cells and their potential for interneuron reprograming

2019

Frontiers in Neuroscience

Boshans LL, Factor DC, Singh V, Liu J, Zhao C, Mandoiu I, Lu QR, Casaccia P, Tesar PJ, Nishiyama A.

Dysregulated glial differentiation in schizophrenia may be relieved by suppression of SMAD4- and REST-dependent signaling

2019

Cell Reports

Liu Z, Osipovitch M, Benraiss A, Huynh NPT, Foti R, Bates J, Chandler-Militello D, Findling RL, Tesar PJ, Nedergaard M, Windrem MS, Goldman SA.

Diverse chemical scaffolds enhance oligodendrocyte formation by inhibiting CYP51, TM7SF2, or EBP

2019

Cell Chemical Biology

Allimuthu D, Hubler Z, Najm FJ, Tang H, Bederman I, Seibel W, Tesar PJ, Adams DJ.

Developing therapeutic strategies to promote myelin repair in multiple sclerosis

2019

Expert Review of Neurotherapeutics

Baldassari LE, Feng J, Clayton BLL, Oh SH, Sakaie K, Tesar PJ, Wang Y, Cohen JA.

Therapeutic suppression of proteolipid protein rescues Pelizaeus-Merzbacher disease in mice

2018

bioRxiv (pre-print)

Elitt MS, Barbar L, Shick E, Powers BE, Maeno-Hikitchi Y, Madhavan M, Allan KC, Nawash BS, Nevin ZS, Olsen HE, Hitomi M, LePage DF, Jiang W, Conlon RA, Rigo F, Tesar PJ.

NG2 expression in NG2 glia is regulated by binding of SoxE and bHLH transcription factors to a Cspg4 intronic enhancer

2018

Glia

Gotoh H, Wood WM, Patel KD, Factor DC, Boshans LL, Nomura T, Tesar PJ, Ono K, Nishiyama A.

Rapid functional genetics of the oligodendrocyte lineage using pluripotent stem cells

2018

Nature Communications

Lager AM, Corradin OG, Cregg JM, Elitt MS, Shick HE, Clayton BLL, Allan KC, Olsen HE, Madhavan M, Tesar PJ.

Chemical screening identifies enhancers of mutant oligodendrocyte survival and unmasks a distinct pathological phase in Pelizaeus-Merzbacher disease

2018

Stem Cell Reports

Elitt MS, Shick HE, Madhavan M, Allan KC, Clayton BLL, Weng C, Miller TE, Factor DC, Barbar L, Nawash BS, Nevin ZS, Lager AM, Li Y, Jin F, Adams DJ, Tesar PJ.

Induction of myelinating oligodendrocytes in human cortical spheroids

2018

Nature Methods

Madhavan M, Nevin ZS, Shick HE, Garrison E, Clarkson-Paredes C, Karl M, Clayton BLL, Factor DC, Allan KC, Barbar L, Jain T, Douvaras P, Fossati V, Miller RH, Tesar PJ.

Physiological genomics identifies genetic modifiers of long QT syndrome type 2 severity

2018

Journal of Clinical Investigation

Chai S, Wan X, Ramirez-Navarro A, Tesar PJ, Kaufman ES, Ficker E, George AL Jr, Deschênes I.

Accumulation of 8,9-unsaturated sterols drives oligodendrocyte formation and remyelination

2018

Nature

Hubler Z, Allimuthu D, Bederman I, Elitt MS, Madhavan M, Allan KC, Shick HE, Garrison E, T Karl M, Factor DC, Nevin ZS, Sax JL, Thompson MA, Fedorov Y, Jin J, Wilson WK, Giera M, Bracher F, Miller RH, Tesar PJ, Adams DJ.

Drug screening for human genetic diseases using iPSC models

2018

Human Molecular Genetics

Elitt MS, Barbar L, Tesar PJ.

Human iPSC glial mouse chimeras reveal glial contributions to schizophrenia

2017

Cell Stem Cell

Windrem MS, Osipovitch M, Liu Z, Bates J, Chandler-Militello D, Zou L, Munir J, Schanz S, McCoy K, Miller RH, Wang S, Nedergaard M, Findling RL, Tesar PJ, Goldman SA.

Transcription elongation factors represent in vivo cancer dependencies in glioblastoma

2017

Nature

Miller TE, Liau BB, Wallace LC, Morton AR, Xie Q, Dixit D, Factor DC, Kim LJY, Morrow JJ, Wu Q, Mack SC, Hubert CG, Gillespie SM, Flavahan WA, Hoffmann T, Thummalapalli R, Hemann MT, Paddison PJ, Horbinski CM, Zuber J, Scacheri PC, Bernstein BE, Tesar PJ, Rich JN.

Modeling the mutational and phenotypic landscapes of Pelizaeus-Merzbacher disease with human iPSC-derived oligodendrocytes

2017

American Journal of Human Genetics

Nevin ZS, Factor DC, Karl RT, Douvaras P, Laukka J, Windrem MS, Goldman SA, Fossati V, Hobson GM, Tesar PJ.

Transcriptome-wide analyses of human neonatal articular cartilage and human mesenchymal stem cell-derived cartilage provide a new molecular target for evaluating engineered cartilage.

2017

Tissue Engineering Part A

Somoza RA, Correa D, Labat I, Sternberg H, Forrest ME, Khalil AM, West MD, Tesar P, Caplan AI.

iPSC reprogramming is not just an open and shut case

2017

Cell Stem Cell

Tesar PJ, Scacheri PC.

Clemastine fumarate for promotion of optic nerve remyelination

2017

Lancet

Cohen JA, Tesar PJ.

Cell-based therapeutic strategies for multiple sclerosis

2017

Brain

Scolding NJ, Pasquini M, Reingold SC, Cohen JA; International Conference on Cell-Based Therapies for Multiple Sclerosis.

Reprogramming of mouse fibroblasts to induced oligodendrocyte progenitor cells

2017

Stem Cell Technologies in Neuroscience
Neuromethods, Springer

Karl R, Lager A, Najm F, Tesar PJ.

Stem cell-based treatment of Pelizaeus-Merzbacher disease

2017

Stem Cells

Osorio MJ, Rowitch DH, Tesar PJ, Wernig M, Windrem MS, Goldman SA.

Depletion of Olig2 in oligodendrocyte progenitor cells infected by Theiler’s murine encephalomyelitis virus

2016

Journal of NeuroVirology

Benner B, Martorell AJ, Mahadevan P, Najm FJ, Tesar PJ, Freundt EC.

Using iPSC-derived human DA neurons from opioid-dependent subjects to study dopamine dynamics

2016

Brain and Behavior

Sheng Y, Filichia E, Shick E, Preston KL, Phillips KA, Cooperman L, Lin Z, Tesar PJ, Hoffer B, Luo Y.

Lower dopamine D2 receptor expression levels in human dopaminergic neurons derived from opioid-dependent iPSCs

2016

The American Journal of Psychiatry

Sheng Y, Filichia E, Shick E, Preston KL, Phillips KA, Cooperman L, Lin Z, Tesar PJ, Hoffer BJ, Luo Y.

Snapshots of pluripotency

2016

Stem Cell Reports

Tesar PJ.

Preferential iron trafficking characterizes glioblastoma stem-like cells

2015

Cancer Cell

Schonberg DL, Miller TE, Wu Q, Flavahan WA, Das NK, Hale JS, Hubert CG, Mack SC, Jarrar AM, Karl RT, Rosager AM, Nixon AM, Tesar PJ, Hamerlik P, Kristensen BW, Horbinski C, Connor JR, Fox PL, Lathia JD, Rich JN.

 

Drug-based modulation of endogenous stem cells promotes functional remyelination in vivo

2015

Nature

Najm FJ, Madhavan M, Zaremba A, Shick E, Karl RT, Factor DC, Miller TE, Nevin ZS, Kantor C, Sargent A, Quick KL, Schlatzer DM, Tang H, Papoian R, Brimacombe KR, Shen M, Boxer MB, Jadhav A, Robinson AP, Podojil JR, Miller SD, Miller RH, Tesar PJ.

Boosting our brains

2015

Science Translational Medicine

Tesar PJ.

Stem cell transplants may HALT multiple sclerosis

2015

Science Translational Medicine

Tesar PJ.

 
Epigenomic comparison reveals activation of ‘seed’ enhancers during transition from naive to primed pluripotency

2014

Cell Stem Cell

Factor DC, Corradin O, Zentner GE, Saiakhova A, Song L, Chenoweth JG, McKay RD, Crawford GE, Scacheri PC, Tesar

 
Derivation of naïve human embryonic stem cells

2014

Proceedings of the National Academy of Sciences

Ware CB, Nelson AM, Mecham B, Hesson J, Zhou W, Jonlin EC, Jimenez-Caliani AJ, Deng X, Cavanaugh C, Cook S, Tesar PJ, Okada J, Margaretha L, Sperber H, Choi M, Blau CA, Treuting PM, Hawkins RD, Cirulli V, Ruohola-Baker H.

NPTX1 regulates neural lineage specification from human pluripotent stem cells

2014

Cell Reports

Boles NC, Hirsch SE, Le S, Corneo B, Najm F, Minotti AP, Wang Q, Lotz S, Tesar PJ, Fasano CA.

Morphing cell identity to treat diabetes

2014

Science Translational Medicine

Tesar PJ.

Stem cell therapy for diabetes comes of age

2014

Science Translational Medicine

Tesar PJ.

Two paths diverged in a schwann cell

2014

Science Translational Medicine

Tesar PJ.

A pillar of hope for new multiple sclerosis therapeutics

2014

Science Translational Medicine

Tesar PJ.

Power of the young—New stem cell source for treating autoimmune disease

2014

Science Translational Medicine

Tesar PJ.

Somatic cell nuclear transfer is all grown up

2014

Science Translational Medicine

Tesar PJ.

Monkeying around with the genome

2014

Science Translational Medicine

Tesar PJ.

Retinal pigmented epithelial cells obtained from human induced pluripotent stem cells possess functional visual cycle enzymes in vitro and in vivo

2013

Journal of Biological Chemistry

Maeda T, Lee MJ, Palczewska G, Marsili S, Tesar PJ, Palczewski K, Takahashi M, Maeda A.

Contrasting effects of Deadend1 (Dnd1) gain and loss of function mutations on allelic inheritance, testicular cancer, and intestinal polyposis

2013

BMC Genetics

Zechel JL, Doerner SK, Lager A, Tesar PJ, Heaney JD, Nadeau JH.

Transcription factor-mediated reprogramming of fibroblasts to expandable, myelinogenic oligodendrocyte progenitor cells

2013

Nature Biotechnology

Najm FJ, Lager AM, Zaremba A, Wyatt K, Caprariello AV, Factor DC, Karl RT, Maeda T, Miller RH, Tesar PJ.

Transcription elongation factor Tcea3 regulates the pluripotent differentiation potential of mouse embryonic stem cells via the Lefty1-Nodal-Smad2 pathway

2013

Stem Cells

Park KS, Cha Y, Kim CH, Ahn HJ, Kim D, Ko S, Kim KH, Chang MY, Ko JH, Noh YS, Han YM, Kim J, Song J, Kim JY, Tesar PJ, Lanza R, Lee KA, Kim KS

Generation and characterization of epiblast stem cells from blastocyst-stage mouse embryos

2013

Methods in Molecular Biology

Factor DC, Najm FJ, Tesar PJ.

StemCellDB: the human pluripotent stem cell database at the National Institutes of Health

2013

Stem Cell Research

Mallon BS, Chenoweth JG, Johnson KR, Hamilton RS, Tesar PJ, Yavatkar AS, Tyson LJ, Park K, Chen KG, Fann YC, McKay RD.

Crowdsourcing science

2013

Cell Stem Cell

Tesar PJ.

Chromatin regulation by long non-coding RNAs

2013

Molecular Biology of Long Non-coding RNAs, Springer

Factor D, Tesar PJ, Khalil A.