Ophir Klein, PhD, MD

Professor of Orofacial Sciences and Pediatrics
Program Lead: Children's Precision Medicine
Committee Members

Ophir Klein is Professor of Orofacial Sciences and Pediatrics, the Larry L. Hillblom Distinguished Professor in Craniofacial Anomalies, and the Charles J. Epstein Professor of Human Genetics at the University of California, San Francisco (UCSF). He serves as Chief of the Division of Medical Genetics, Chair of the Division of Craniofacial Anomalies, Medical Director of the Craniofacial Center, and Director of the Program in Craniofacial Biology.
The Klein laboratory focuses on understanding how organs form in developing embryos and how they regenerate in adults. When developmental and regenerative processes go awry, then birth defects, cancer and other diseases can result. The group's research is centered on understanding how development and regeneration normally occur in the hope of one day treating diseases that result from abnormalities in these processes.
A central focus in the lab is craniofacial and dental development, as malformations in these organs are among the most common congenital abnormalities and have profound impacts on the lives of patients and their families. The maintenance, repair and growth of many adult organs, such as the bone marrow, skin, brain, and gastrointestinal tract, depend on tissue-specific populations of stem cells. The lab uses the rodent incisor, which grows continuously throughout the life of the animal, as a model system to understand adult stem cells. We intend to use the insights provided by our experiments in mice to guide us in the use of stem cells in regenerating dental and craniofacial tissues as a paradigm for developing replacement organs.
Another major area of study is regeneration of the gastrointestinal tract. The amazing ability of the mammalian gastrointestinal epithelium to renew has long fascinated biologists. Our research aims to address fundamental questions in this field, including the identity, regulation and plasticity of intestinal stem cells.
Finally, the laboratory is interested in how positive and negative modulators of signaling affect development, homeostasis and cancer. We are studying the roles of these molecules in the patterning and outgrowth of teeth, taste papillae, the oral mucosa, and other tissues and organs.

Publications: 

Early perturbation of Wnt signaling reveals patterning and invagination-evagination control points in molar tooth development.

Development (Cambridge, England)

Kim R, Yu T, Li J, Prochazka J, Sharir A, Green JBA, Klein OD

Large-scale open-source three-dimensional growth curves for clinical facial assessment and objective description of facial dysmorphism.

Scientific reports

Matthews HS, Palmer RL, Baynam GS, Quarrell OW, Klein OD, Spritz RA, Hennekam RC, Walsh S, Shriver M, Weinberg SM, Hallgrimsson B, Hammond P, Penington AJ, Peeters H, Claes PD

Transit-Amplifying Cells Coordinate Changes in Intestinal Epithelial Cell-Type Composition.

Developmental cell

Sanman LE, Chen IW, Bieber JM, Steri V, Trentesaux C, Hann B, Klein OD, Wu LF, Altschuler SJ

KrasP34R and KrasT58I mutations induce distinct RASopathy phenotypes in mice.

JCI insight

Wong JC, Perez-Mancera PA, Huang TQ, Kim J, Grego-Bessa J, Del Pilar Alzamora M, Kogan SC, Sharir A, Keefe SH, Morales CE, Schanze D, Castel P, Hirose K, Huang GN, Zenker M, Sheppard D, Klein OD, Tuveson DA, Braun BS, Shannon K

Engineering synthetic morphogen systems that can program multicellular patterning.

Science (New York, N.Y.)

Toda S, McKeithan WL, Hakkinen TJ, Lopez P, Klein OD, Lim WA

Dental cell type atlas reveals stem and differentiated cell types in mouse and human teeth.

Nature communications

Krivanek J, Soldatov RA, Kastriti ME, Chontorotzea T, Herdina AN, Petersen J, Szarowska B, Landova M, Matejova VK, Holla LI, Kuchler U, Zdrilic IV, Vijaykumar A, Balic A, Marangoni P, Klein OD, Neves VCM, Yianni V, Sharpe PT, Harkany T, Metscher BD, Bajénoff M, Mina M, Fried K, Kharchenko PV, Adameyko I

FaceBase 3: analytical tools and FAIR resources for craniofacial and dental research.

Development (Cambridge, England)

Samuels BD, Aho R, Brinkley JF, Bugacov A, Feingold E, Fisher S, Gonzalez-Reiche AS, Hacia JG, Hallgrimsson B, Hansen K, Harris MP, Ho TV, Holmes G, Hooper JE, Jabs EW, Jones KL, Kesselman C, Klein OD, Leslie EJ, Li H, Liao EC, Long H, Lu N, Maas RL, Marazita ML, Mohammed J, Prescott S, Schuler R, Selleri L, Spritz RA, Swigut T, van Bakel H, Visel A, Welsh I, Williams C, Williams TJ, Wysocka J, Yuan Y, Chai Y

Sox2 Controls Periderm and Rugae Development to Inhibit Oral Adhesions.

Journal of dental research

Sweat YY, Sweat M, Yu W, Sanz-Navarro M, Zhang L, Sun Z, Eliason S, Klein OD, Michon F, Chen Z, Amendt BA

FAM20B-catalyzed glycosaminoglycans control murine tooth number by restricting FGFR2b signaling.

BMC biology

Wu J, Tian Y, Han L, Liu C, Sun T, Li L, Yu Y, Lamichhane B, D'Souza RN, Millar SE, Krumlauf R, Ornitz DM, Feng JQ, Klein O, Zhao H, Zhang F, Linhardt RJ, Wang X

YAP/TAZ Regulate Elevation and Bone Formation of the Mouse Secondary Palate.

Journal of dental research

Goodwin AF, Chen CP, Vo NT, Bush JO, Klein OD

LGR5 in breast cancer and ductal carcinoma in situ: a diagnostic and prognostic biomarker and a therapeutic target.

BMC cancer

Hagerling C, Owyong M, Sitarama V, Wang CY, Lin C, van den Bijgaart RJE, Koopman CD, Brenot A, Nanjaraj A, Wärnberg F, Jirström K, Klein OD, Werb Z, Plaks V

Fully Automatic Landmarking of Syndromic 3D Facial Surface Scans Using 2D Images.

Sensors (Basel, Switzerland)

Bannister JJ, Crites SR, Aponte JD, Katz DC, Wilms M, Klein OD, Bernier FPJ, Spritz RA, Hallgrímsson B, Forkert ND

Automated syndrome diagnosis by three-dimensional facial imaging.

Genetics in medicine : official journal of the American College of Medical Genetics

Hallgrímsson B, Aponte JD, Katz DC, Bannister JJ, Riccardi SL, Mahasuwan N, McInnes BL, Ferrara TM, Lipman DM, Neves AB, Spitzmacher JAJ, Larson JR, Bellus GA, Pham AM, Aboujaoude E, Benke TA, Chatfield KC, Davis SM, Elias ER, Enzenauer RW, French BM, Pickler LL, Shieh JTC, Slavotinek A, Harrop AR, Innes AM, McCandless SE, McCourt EA, Meeks NJL, Tartaglia NR, Tsai AC, Wyse JPH, Bernstein JA, Sanchez-Lara PA, Forkert ND, Bernier FP, Spritz RA, Klein OD

Tools and Concepts for Interrogating and Defining Cellular Identity.

Cell stem cell

McKinley KL, Castillo-Azofeifa D, Klein OD

The UCSF Mouse Inventory Database Application, an Open Source Web App for Sharing Mutant Mice Within a Research Community.

G3 (Bethesda, Md.)

Wall E, Scoles J, Joo A, Klein O, Quinonez C, Bush JO, Martin GR, Laird DJ

Safety and immunogenicity of Fc-EDA, a recombinant ectodysplasin A1 replacement protein, in human subjects.

British journal of clinical pharmacology

Körber I, Klein OD, Morhart P, Faschingbauer F, Grange DK, Clarke A, Bodemer C, Maitz S, Huttner K, Kirby N, Durand C, Schneider H

Lgr5+ telocytes are a signaling source at the intestinal villus tip.

Nature communications

Bahar Halpern K, Massalha H, Zwick RK, Moor AE, Castillo-Azofeifa D, Rozenberg M, Farack L, Egozi A, Miller DR, Averbukh I, Harnik Y, Weinberg-Corem N, de Sauvage FJ, Amit I, Klein OD, Shoshkes-Carmel M, Itzkovitz S

From gut to glutes: The critical role of niche signals in the maintenance and renewal of adult stem cells.

Current opinion in cell biology

Trentesaux C, Striedinger K, Pomerantz JH, Klein OD

Generation of Knockout Gene-Edited Human Intestinal Organoids.

Methods in molecular biology (Clifton, N.J.)

Rajendra C, Wald T, Barber K, Spence JR, Fattahi F, Klein OD

Heterogeneity within Stratified Epithelial Stem Cell Populations Maintains the Oral Mucosa in Response to Physiological Stress.

Cell stem cell

Byrd KM, Piehl NC, Patel JH, Huh WJ, Sequeira I, Lough KJ, Wagner BL, Marangoni P, Watt FM, Klein OD, Coffey RJ, Williams SE

Use of organoids to study regenerative responses to intestinal damage.

American journal of physiology. Gastrointestinal and liver physiology

Blutt SE, Klein OD, Donowitz M, Shroyer N, Guha C, Estes MK

A large pool of actively cycling progenitors orchestrates self-renewal and injury repair of an ectodermal appendage.

Nature cell biology

Sharir A, Marangoni P, Zilionis R, Wan M, Wald T, Hu JK, Kawaguchi K, Castillo-Azofeifa D, Epstein L, Harrington K, Pagella P, Mitsiadis T, Siebel CW, Klein AM, Klein OD

Case Report of Floating-Harbor Syndrome With Bilateral Cleft Lip.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association

Ko J, Pomerantz JH, Perry H, Shieh JT, Slavotinek AM, Oberoi S, Klein OD

Sonic Hedgehog Signaling Is Required for Cyp26 Expression during Embryonic Development.

International journal of molecular sciences

El Shahawy M, Reibring CG, Hallberg K, Neben CL, Marangoni P, Harfe BD, Klein OD, Linde A, Gritli-Linde A

The Society for Craniofacial Genetics and Developmental Biology 41st Annual Meeting.

American journal of medical genetics. Part A

Taneyhill LA, Moody SA, Cox T, Klein OD, Marcucio R, Schneider RA, Trainor PA

Atoh1+ secretory progenitors possess renewal capacity independent of Lgr5+ cells during colonic regeneration.

The EMBO journal

Castillo-Azofeifa D, Fazio EN, Nattiv R, Good HJ, Wald T, Pest MA, de Sauvage FJ, Klein OD, Asfaha S

Introduction to themed series on intestinal stem cells and the NIDDK Intestinal Stem Cell Consortium.

American journal of physiology. Gastrointestinal and liver physiology

Wang TC, Martin MG, Kuo CJ, Klein OD, Niland J

Intestinal renewal across the animal kingdom: comparing stem cell activity in mouse and Drosophila.

American journal of physiology. Gastrointestinal and liver physiology

Zwick RK, Ohlstein B, Klein OD

Prominin-1 controls stem cell activation by orchestrating ciliary dynamics.

The EMBO journal

Singer D, Thamm K, Zhuang H, Karbanová J, Gao Y, Walker JV, Jin H, Wu X, Coveney CR, Marangoni P, Lu D, Grayson PRC, Gulsen T, Liu KJ, Ardu S, Wann AK, Luo S, Zambon AC, Jetten AM, Tredwin C, Klein OD, Attanasio M, Carmeliet P, Huttner WB, Corbeil D, Hu B

Quantitative Clonal Analysis and Single-Cell Transcriptomics Reveal Division Kinetics, Hierarchy, and Fate of Oral Epithelial Progenitor Cells.

Cell stem cell

Jones KB, Furukawa S, Marangoni P, Ma H, Pinkard H, D'Urso R, Zilionis R, Klein AM, Klein OD

Bones, Glands, Ears and More: The Multiple Roles of FGF10 in Craniofacial Development.

Frontiers in genetics

Prochazkova M, Prochazka J, Marangoni P, Klein OD

Diverse progenitor cells preserve salivary gland ductal architecture after radiation-induced damage.

Development (Cambridge, England)

May AJ, Cruz-Pacheco N, Emmerson E, Gaylord EA, Seidel K, Nathan S, Muench MO, Klein OD, Knox SM

Tissue Mechanical Forces and Evolutionary Developmental Changes Act Through Space and Time to Shape Tooth Morphology and Function.

BioEssays : news and reviews in molecular, cellular and developmental biology

Calamari ZT, Hu JK, Klein OD

Mechanoresponsive stem cells acquire neural crest fate in jaw regeneration.

Nature

Ransom RC, Carter AC, Salhotra A, Leavitt T, Marecic O, Murphy MP, Lopez ML, Wei Y, Marshall CD, Shen EZ, Jones RE, Sharir A, Klein OD, Chan CKF, Wan DC, Chang HY, Longaker MT

LRH-1 mitigates intestinal inflammatory disease by maintaining epithelial homeostasis and cell survival.

Nature communications

Bayrer JR, Wang H, Nattiv R, Suzawa M, Escusa HS, Fletterick RJ, Klein OD, Moore DD, Ingraham HA

Publisher Correction: Parasitic helminths induce fetal-like reversion in the intestinal stem cell niche.

Nature

Nusse YM, Savage AK, Marangoni P, Rosendahl-Huber AKM, Landman TA, de Sauvage FJ, Locksley RM, Klein OD

The intestinal epithelial response to damage.

Science China. Life sciences

Weichselbaum L, Klein OD

Bmi1-Progenitor Cell Ablation Impairs the Angiogenic Response to Myocardial Infarction.

Arteriosclerosis, thrombosis, and vascular biology

Herrero D, Cañón S, Pelacho B, Salvador-Bernáldez M, Aguilar S, Pogontke C, Carmona RM, Salvador JM, Perez-Pomares JM, Klein OD, Prósper F, Jimenez-Borreguero LJ, Bernad A

An integrated clinical program and crowdsourcing strategy for genomic sequencing and Mendelian disease gene discovery.

NPJ genomic medicine

Haghighi A, Krier JB, Toth-Petroczy A, Cassa CA, Frank NY, Carmichael N, Fieg E, Bjonnes A, Mohanty A, Briere LC, Lincoln S, Lucia S, Gupta VA, Söylemez O, Sutti S, Kooshesh K, Qiu H, Fay CJ, Perroni V, Valerius J, Hanna M, Frank A, Ouahed J, Snapper SB, Pantazi A, Chopra SS, Leshchiner I, Stitziel NO, Feldweg A, Mannstadt M, Loscalzo J, Sweetser DA, Liao E, Stoler JM, Nowak CB, Sanchez-Lara PA, Klein OD, Perry H, Patsopoulos NA, Raychaudhuri S, Goessling W, Green RC, Seidman CE, MacRae CA, Sunyaev SR, Maas RL, Vuzman D

Publisher Correction: FGF signalling controls the specification of hair placode-derived SOX9 positive progenitors to Merkel cells.

Nature communications

Nguyen MB, Cohen I, Kumar V, Xu Z, Bar C, Dauber-Decker KL, Tsai PC, Marangoni P, Klein OD, Hsu YC, Chen T, Mikkola ML, Ezhkova E

SOX2 regulation by hedgehog signaling controls adult lingual epithelium homeostasis.

Development (Cambridge, England)

Castillo-Azofeifa D, Seidel K, Gross L, Golden EJ, Jacquez B, Klein OD, Barlow LA

Parasitic helminths induce fetal-like reversion in the intestinal stem cell niche.

Nature

Nusse YM, Savage AK, Marangoni P, Rosendahl-Huber AKM, Landman TA, de Sauvage FJ, Locksley RM, Klein OD

Cellular aspect ratio and cell division mechanics underlie the patterning of cell progeny in diverse mammalian epithelia.

eLife

McKinley KL, Stuurman N, Royer LA, Schartner C, Castillo-Azofeifa D, Delling M, Klein OD, Vale RD

FGF signalling controls the specification of hair placode-derived SOX9 positive progenitors to Merkel cells.

Nature communications

Nguyen MB, Cohen I, Kumar V, Xu Z, Bar C, Dauber-Decker KL, Tsai PC, Marangoni P, Klein OD, Hsu YC, Chen T, Mikkola ML, Ezhkova E

Label reading, numeracy and food & nutrition involvement.

Appetite

Mulders MDGH, Corneille O, Klein O

Dental, Oral, and Craniofacial Regenerative Medicine: Transforming Biotechnologies for Innovating Patient Care.

Journal of dental research

Giannobile WV, Chai Y, Chen Y, Healy KE, Klein O, Lane N, Longaker MT, Lotz JC, Mooney DJ, Sfeir CS, Urata M, Wagner WR, Wu BM, Kohn DH

Epithelial WNT Ligands Are Essential Drivers of Intestinal Stem Cell Activation.

Cell reports

Zou WY, Blutt SE, Zeng XL, Chen MS, Lo YH, Castillo-Azofeifa D, Klein OD, Shroyer NF, Donowitz M, Estes MK

Bmi1+ Progenitor Cell Dynamics in Murine Cornea During Homeostasis and Wound Healing.

Stem cells (Dayton, Ohio)

Kalha S, Shrestha B, Sanz Navarro M, Jones KB, Klein OD, Michon F

Plasticity within the niche ensures the maintenance of a Sox2+ stem cell population in the mouse incisor.

Development (Cambridge, England)

Sanz-Navarro M, Seidel K, Sun Z, Bertonnier-Brouty L, Amendt BA, Klein OD, Michon F

Isl1 mediates mesenchymal expansion in the developing external genitalia via regulation of Bmp4, Fgf10 and Wnt5a.

Human molecular genetics

Ching ST, Infante CR, Du W, Sharir A, Park S, Menke DB, Klein OD

Engineered Tissue Folding by Mechanical Compaction of the Mesenchyme.

Developmental cell

Hughes AJ, Miyazaki H, Coyle MC, Zhang J, Laurie MT, Chu D, Vavrušová Z, Schneider RA, Klein OD, Gartner ZJ

Body size and allometric variation in facial shape in children.

American journal of physical anthropology

Larson JR, Manyama MF, Cole JB, Gonzalez PN, Percival CJ, Liberton DK, Ferrara TM, Riccardi SL, Kimwaga EA, Mathayo J, Spitzmacher JA, Rolian C, Jamniczky HA, Weinberg SM, Roseman CC, Klein O, Lukowiak K, Spritz RA, Hallgrimsson B

Meeting report: a hard look at the state of enamel research.

International journal of oral science

Klein OD, Duverger O, Shaw W, Lacruz RS, Joester D, Moradian-Oldak J, Pugach MK, Wright JT, Millar SE, Kulkarni AB, Bartlett JD, Diekwisch TG, DenBesten P, Simmer JP

Feedback regulation of RTK signaling in development.

Developmental biology

Neben CL, Lo M, Jura N, Klein OD

Isl1 Controls Patterning and Mineralization of Enamel in the Continuously Renewing Mouse Incisor.

Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research

Naveau A, Zhang B, Meng B, Sutherland MT, Prochazkova M, Wen T, Marangoni P, Jones KB, Cox TC, Ganss B, Jheon AH, Klein OD

IRF6 and SPRY4 Signaling Interact in Periderm Development.

Journal of dental research

Kousa YA, Roushangar R, Patel N, Walter A, Marangoni P, Krumlauf R, Klein OD, Schutte BC

Cell fate specification in the lingual epithelium is controlled by antagonistic activities of Sonic hedgehog and retinoic acid.

PLoS genetics

El Shahawy M, Reibring CG, Neben CL, Hallberg K, Marangoni P, Harfe BD, Klein OD, Linde A, Gritli-Linde A

Automatic recognition of the XLHED phenotype from facial images.

American journal of medical genetics. Part A

Hadj-Rabia S, Schneider H, Navarro E, Klein O, Kirby N, Huttner K, Wolf L, Orin M, Wohlfart S, Bodemer C, Grange DK

Ras Signaling Regulates Stem Cells and Amelogenesis in the Mouse Incisor.

Journal of dental research

Zheng X, Goodwin AF, Tian H, Jheon AH, Klein OD

A review of craniofacial and dental findings of the RASopathies.

Orthodontics & craniofacial research

Cao H, Alrejaye N, Klein OD, Goodwin AF, Oberoi S

FGF signaling refines Wnt gradients to regulate the patterning of taste papillae.

Development (Cambridge, England)

Prochazkova M, Häkkinen TJ, Prochazka J, Spoutil F, Jheon AH, Ahn Y, Krumlauf R, Jernvall J, Klein OD

Resolving stem and progenitor cells in the adult mouse incisor through gene co-expression analysis.

eLife

Seidel K, Marangoni P, Tang C, Houshmand B, Du W, Maas RL, Murray S, Oldham MC, Klein OD

The Interaction of Genetic Background and Mutational Effects in Regulation of Mouse Craniofacial Shape.

G3 (Bethesda, Md.)

Percival CJ, Marangoni P, Tapaltsyan V, Klein O, Hallgrímsson B

An FAK-YAP-mTOR Signaling Axis Regulates Stem Cell-Based Tissue Renewal in Mice.

Cell stem cell

Hu JK, Du W, Shelton SJ, Oldham MC, DiPersio CM, Klein OD

From snapshots to movies: Understanding early tooth development in four dimensions.

Developmental dynamics : an official publication of the American Association of Anatomists

Kim R, Green JBA, Klein OD

EPHRIN-B1 Mosaicism Drives Cell Segregation in Craniofrontonasal Syndrome hiPSC-Derived Neuroepithelial Cells.

Stem cell reports

Niethamer TK, Larson AR, O'Neill AK, Bershteyn M, Hsiao EC, Klein OD, Pomerantz JH, Bush JO

Intrinsically disordered proteins drive enamel formation via an evolutionarily conserved self-assembly motif.

Proceedings of the National Academy of Sciences of the United States of America

Wald T, Spoutil F, Osickova A, Prochazkova M, Benada O, Kasparek P, Bumba L, Klein OD, Sedlacek R, Sebo P, Prochazka J, Osicka R

Human Facial Shape and Size Heritability and Genetic Correlations.

Genetics

Cole JB, Manyama M, Larson JR, Liberton DK, Ferrara TM, Riccardi SL, Li M, Mio W, Klein OD, Santorico SA, Hallgrímsson B, Spritz RA

In vitro patterning of pluripotent stem cell-derived intestine recapitulates in vivo human development.

Development (Cambridge, England)

Tsai YH, Nattiv R, Dedhia PH, Nagy MS, Chin AM, Thomson M, Klein OD, Spence JR

Sox2 and Lef-1 interact with Pitx2 to regulate incisor development and stem cell renewal.

Development (Cambridge, England)

Sun Z, Yu W, Sanz Navarro M, Sweat M, Eliason S, Sharp T, Liu H, Seidel K, Zhang L, Moreno M, Lynch T, Holton NE, Rogers L, Neff T, Goodheart MJ, Michon F, Klein OD, Chai Y, Dupuy A, Engelhardt JF, Chen Z, Amendt BA

SPRY1 regulates mammary epithelial morphogenesis by modulating EGFR-dependent stromal paracrine signaling and ECM remodeling.

Proceedings of the National Academy of Sciences of the United States of America

Koledova Z, Zhang X, Streuli C, Clarke RB, Klein OD, Werb Z, Lu P

Genome-Wide Association Study Reveals Multiple Loci Influencing Normal Human Facial Morphology.

PLoS genetics

Shaffer JR, Orlova E, Lee MK, Leslie EJ, Raffensperger ZD, Heike CL, Cunningham ML, Hecht JT, Kau CH, Nidey NL, Moreno LM, Wehby GL, Murray JC, Laurie CA, Laurie CC, Cole J, Ferrara T, Santorico S, Klein O, Mio W, Feingold E, Hallgrimsson B, Spritz RA, Marazita ML, Weinberg SM

Genomewide Association Study of African Children Identifies Association of SCHIP1 and PDE8A with Facial Size and Shape.

PLoS genetics

Cole JB, Manyama M, Kimwaga E, Mathayo J, Larson JR, Liberton DK, Lukowiak K, Ferrara TM, Riccardi SL, Li M, Mio W, Prochazkova M, Williams T, Li H, Jones KL, Klein OD, Santorico SA, Hallgrimsson B, Spritz RA

aE-catenin inhibits YAP/TAZ activity to regulate signalling centre formation during tooth development.

Nature communications

Li CY, Hu J, Lu H, Lan J, Du W, Galicia N, Klein OD

Dual epithelial and immune cell function of Dvl1 regulates gut microbiota composition and intestinal homeostasis.

JCI insight

Belinson H, Savage AK, Fadrosh D, Kuo YM, Lin D, Valladares R, Nusse Y, Wynshaw-Boris A, Lynch SV, Locksley RM, Klein OD

JCI insight

Dual epithelial and immune cell function of Dvl1 regulates gut mircobiota composition and intestinal homeostasis.

Belinson H, Savage AK, Fadrosh D, Kuo YM, Lin D, Valladares R, Nusse Y, Wynshaw-Boris A, Lynch SV, Locksley RM, Klein OD.

Modeling craniofacial and skeletal congenital birth defects to advance therapies.

Human molecular genetics

Neben CL, Roberts RR, Dipple KM, Merrill AE, Klein OD

The FaceBase Consortium: a comprehensive resource for craniofacial researchers.

Development (Cambridge, England)

Brinkley JF, Fisher S, Harris MP, Holmes G, Hooper JE, Jabs EW, Jones KL, Kesselman C, Klein OD, Maas RL, Marazita ML, Selleri L, Spritz RA, van Bakel H, Visel A, Williams TJ, Wysocka J, Chai Y

Sprouty2 regulates endochondral bone formation by modulation of RTK and BMP signaling.

Bone

Joo A, Long R, Cheng Z, Alexander C, Chang W, Klein OD

Prenatal ß-catenin/Brn2/Tbr2 transcriptional cascade regulates adult social and stereotypic behaviors.

Molecular psychiatry

Belinson H, Nakatani J, Babineau BA, Birnbaum RY, Ellegood J, Bershteyn M, McEvilly RJ, Long JM, Willert K, Klein OD, Ahituv N, Lerch JP, Rosenfeld MG, Wynshaw-Boris A

Human iPS Cell-Derived Neurons Uncover the Impact of Increased Ras Signaling in Costello Syndrome.

The Journal of neuroscience : the official journal of the Society for Neuroscience

Rooney GE, Goodwin AF, Depeille P, Sharir A, Schofield CM, Yeh E, Roose JP, Klein OD, Rauen KA, Weiss LA, Ullian EM

Expression of FGFs during early mouse tongue development.

Gene expression patterns : GEP

Du W, Prochazka J, Prochazkova M, Klein OD

Migration of Founder Epithelial Cells Drives Proper Molar Tooth Positioning and Morphogenesis.

Developmental cell

Prochazka J, Prochazkova M, Du W, Spoutil F, Tureckova J, Hoch R, Shimogori T, Sedlacek R, Rubenstein JL, Wittmann T, Klein OD

Development of synovial membrane in the temporomandibular joint of the human fetus.

European journal of histochemistry : EJH

Carvalho de Moraes LO, Tedesco RC, Arraez-Aybar LA, Klein O, Mérida-Velasco JR, Alonso LG

Craniofacial Stem Cells in Health and Disease.

Journal of dental research

Klein OD, Nör JE

Inhibition of Notch Signaling During Mouse Incisor Renewal Leads to Enamel Defects.

Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research

Jheon AH, Prochazkova M, Meng B, Wen T, Lim YJ, Naveau A, Espinoza R, Cox TC, Sone ED, Ganss B, Siebel CW, Klein OD

Phenotypic and evolutionary implications of modulating the ERK-MAPK cascade using the dentition as a model.

Scientific reports

Marangoni P, Charles C, Tafforeau P, Laugel-Haushalter V, Joo A, Bloch-Zupan A, Klein OD, Viriot L

Identification of novel Fgf enhancers and their role in dental evolution.

Evolution & development

Tapaltsyan V, Charles C, Hu J, Mindell D, Ahituv N, Wilson GM, Black BL, Viriot L, Klein OD

Transcriptome-wide Analysis Reveals Hallmarks of Human Intestine Development and Maturation In Vitro and In Vivo.

Stem cell reports

Finkbeiner SR, Hill DR, Altheim CH, Dedhia PH, Taylor MJ, Tsai YH, Chin AM, Mahe MM, Watson CL, Freeman JJ, Nattiv R, Thomson M, Klein OD, Shroyer NF, Helmrath MA, Teitelbaum DH, Dempsey PJ, Spence JR

DLX4 is associated with orofacial clefting and abnormal jaw development.

Human molecular genetics

Wu D, Mandal S, Choi A, Anderson A, Prochazkova M, Perry H, Gil-Da-Silva-Lopes VL, Lao R, Wan E, Tang PL, Kwok PY, Klein O, Zhuan B, Slavotinek AM

Further supporting evidence for the SATB2-associated syndrome found through whole exome sequencing.

American journal of medical genetics. Part A

Zarate YA, Perry H, Ben-Omran T, Sellars EA, Stein Q, Almureikhi M, Simmons K, Klein O, Fish J, Feingold M, Douglas J, Kruer MC, Si Y, Mao R, McKnight D, Gibellini F, Retterer K, Slavotinek A

Hyperplasia of interstitial cells of cajal in sprouty homolog 4 deficient mice.

PloS one

Thys A, Vandenberghe P, Hague P, Klein OD, Erneux C, Vanderwinden JM

Continuously growing rodent molars result from a predictable quantitative evolutionary change over 50 million years.

Cell reports

Tapaltsyan V, Eronen JT, Lawing AM, Sharir A, Janis C, Jernvall J, Klein OD

Opposing activities of Notch and Wnt signaling regulate intestinal stem cells and gut homeostasis.

Cell reports

Tian H, Biehs B, Chiu C, Siebel CW, Wu Y, Costa M, de Sauvage FJ, Klein OD

In vitro generation of human pluripotent stem cell derived lung organoids.

eLife

Dye BR, Hill DR, Ferguson MA, Tsai YH, Nagy MS, Dyal R, Wells JM, Mayhew CN, Nattiv R, Klein OD, White ES, Deutsch GH, Spence JR

Spontaneous emergence of overgrown molar teeth in a colony of Prairie voles (Microtus ochrogaster).

International journal of oral science

Jheon AH, Prochazkova M, Sherman M, Manoli DS, Shah NM, Carbone L, Klein O

Developing and regenerating a sense of taste.

Current topics in developmental biology

Barlow LA, Klein OD

Replaying evolutionary transitions from the dental fossil record.

Nature

Harjunmaa E, Seidel K, Häkkinen T, Renvoisé E, Corfe IJ, Kallonen A, Zhang ZQ, Evans AR, Mikkola ML, Salazar-Ciudad I, Klein OD, Jernvall J

Induction of ectopic taste buds by SHH reveals the competency and plasticity of adult lingual epithelium.

Development (Cambridge, England)

Castillo D, Seidel K, Salcedo E, Ahn C, de Sauvage FJ, Klein OD, Barlow LA

Nuclear to cytoplasmic shuttling of ERK promotes differentiation of muscle stem/progenitor cells.

Development (Cambridge, England)

Michailovici I, Harrington HA, Azogui HH, Yahalom-Ronen Y, Plotnikov A, Ching S, Stumpf MP, Klein OD, Seger R, Tzahor E

Craniofacial morphometric analysis of individuals with X-linked hypohidrotic ectodermal dysplasia.

Molecular genetics & genomic medicine

Goodwin AF, Larson JR, Jones KB, Liberton DK, Landan M, Wang Z, Boekelheide A, Langham M, Mushegyan V, Oberoi S, Brao R, Wen T, Johnson R, Huttner K, Grange DK, Spritz RA, Hallgrímsson B, Jheon AH, Klein OD

Isolation and culture of dental epithelial stem cells from the adult mouse incisor.

Journal of visualized experiments : JoVE

Chavez MG, Hu J, Seidel K, Li C, Jheon A, Naveau A, Horst O, Klein OD

Crosstalk between miR-34a and Notch Signaling Promotes Differentiation in Apical Papilla Stem Cells (SCAPs).

Journal of dental research

Sun F, Wan M, Xu X, Gao B, Zhou Y, Sun J, Cheng L, Klein OD, Zhou X, Zheng L

The WNT10A gene in ectodermal dysplasias and selective tooth agenesis.

American journal of medical genetics. Part A

Mues G, Bonds J, Xiang L, Vieira AR, Seymen F, Klein O, D'Souza RN

Dental findings in 14q terminal deletion syndrome.

Clinical dysmorphology

Mendelsohn BA, Jeng LL, Oberoi S, Klein OD

Craniofacial and dental development in Costello syndrome.

American journal of medical genetics. Part A

Goodwin AF, Oberoi S, Landan M, Charles C, Massie JC, Fairley C, Rauen KA, Klein OD

Tooth, hair and claw: comparing epithelial stem cell niches of ectodermal appendages.

Experimental cell research

Naveau A, Seidel K, Klein OD

Coordinated activity of Spry1 and Spry2 is required for normal development of the external genitalia.

Developmental biology

Ching ST, Cunha GR, Baskin LS, Basson MA, Klein OD

Fibroblast growth factor signaling in mammalian tooth development.

Odontology

Li CY, Prochazka J, Goodwin AF, Klein OD

On the cutting edge of organ renewal: Identification, regulation, and evolution of incisor stem cells.

Genesis (New York, N.Y. : 2000)

Kuang-Hsien Hu J, Mushegyan V, Klein OD

Current trends in stem cell therapy for improvement of bone quality.

Histology and histopathology

Yamada Y, Nakamura S, Klein OD, Ito K

American Journal of Medical Genetics Part A

The society of craniofacial genetics and developmental biology 35th annual meeting

Klein OD, Bush JO, Knox SM, Jheon AH, Cordero DR, Richtsmeier JT

Developmental disorders of the dentition: an update.

American journal of medical genetics. Part C, Seminars in medical genetics

Klein OD, Oberoi S, Huysseune A, Hovorakova M, Peterka M, Peterkova R

Abnormal Ras signaling in Costello syndrome (CS) negatively regulates enamel formation.

Human molecular genetics

Goodwin AF, Tidyman WE, Jheon AH, Sharir A, Zheng X, Charles C, Fagin JA, McMahon M, Diekwisch TG, Ganss B, Rauen KA, Klein OD

Inductive ability of human developing and differentiated dental mesenchyme.

Cells, tissues, organs

Zheng L, Warotayanont R, Stahl J, Kunimatsu R, Klein O, DenBesten PK, Zhang Y

The Pitx2:miR-200c/141:noggin pathway regulates Bmp signaling and ameloblast differentiation.

Development (Cambridge, England)

Cao H, Jheon A, Li X, Sun Z, Wang J, Florez S, Zhang Z, McManus MT, Klein OD, Amendt BA

BMI1 represses Ink4a/Arf and Hox genes to regulate stem cells in the rodent incisor.

Nature cell biology

Biehs B, Hu JK, Strauli NB, Sangiorgi E, Jung H, Heber RP, Ho S, Goodwin AF, Dasen JS, Capecchi MR, Klein OD

Characterization of X-linked hypohidrotic ectodermal dysplasia (XL-HED) hair and sweat gland phenotypes using phototrichogram analysis and live confocal imaging.

American journal of medical genetics. Part A

Jones KB, Goodwin AF, Landan M, Seidel K, Tran DK, Hogue J, Chavez M, Fete M, Yu W, Hussein T, Johnson R, Huttner K, Jheon AH, Klein OD

The dynamics of supernumerary tooth development are differentially regulated by Sprouty genes.

Journal of experimental zoology. Part B, Molecular and developmental evolution

Lagronova-Churava S, Spoutil F, Vojtechova S, Lesot H, Peterka M, Klein OD, Peterkova R

Sox2 marks epithelial competence to generate teeth in mammals and reptiles.

Development (Cambridge, England)

Juuri E, Jussila M, Seidel K, Holmes S, Wu P, Richman J, Heikinheimo K, Chuong CM, Arnold K, Hochedlinger K, Klein O, Michon F, Thesleff I

Injectable bone tissue engineering using expanded mesenchymal stem cells.

Stem cells (Dayton, Ohio)

Yamada Y, Nakamura S, Ito K, Umemura E, Hara K, Nagasaka T, Abe A, Baba S, Furuichi Y, Izumi Y, Klein OD, Wakabayashi T

Lgr5-expressing cells are sufficient and necessary for postnatal mammary gland organogenesis.

Cell reports

Plaks V, Brenot A, Lawson DA, Linnemann JR, Van Kappel EC, Wong KC, de Sauvage F, Klein OD, Werb Z

microRNA miR-34a regulates cytodifferentiation and targets multi-signaling pathways in human dental papilla cells.

PloS one

Wan M, Gao B, Sun F, Tang Y, Ye L, Fan Y, Klein OD, Zhou X, Zheng L

Fgf8 dosage determines midfacial integration and polarity within the nasal and optic capsules.

Developmental biology

Griffin JN, Compagnucci C, Hu D, Fish J, Klein O, Marcucio R, Depew MJ

A genome-wide association study identifies susceptibility loci for nonsyndromic sagittal craniosynostosis near BMP2 and within BBS9.

Nature genetics

Justice CM, Yagnik G, Kim Y, Peter I, Jabs EW, Erazo M, Ye X, Ainehsazan E, Shi L, Cunningham ML, Kimonis V, Roscioli T, Wall SA, Wilkie AO, Stoler J, Richtsmeier JT, Heuzé Y, Sanchez-Lara PA, Buckley MF, Druschel CM, Mills JL, Caggana M, Romitti PA, Kay DM, Senders C, Taub PJ, Klein OD, Boggan J, Zwienenberg-Lee M, Naydenov C, Kim J, Wilson AF, Boyadjiev SA

Craniofacial and dental development in cardio-facio-cutaneous syndrome: the importance of Ras signaling homeostasis.

Clinical genetics

Goodwin AF, Oberoi S, Landan M, Charles C, Groth J, Martinez A, Fairley C, Weiss LA, Tidyman WE, Klein OD, Rauen KA

Characterization of dental epithelial stem cells from the mouse incisor with two-dimensional and three-dimensional platforms.

Tissue engineering. Part C, Methods

Chavez MG, Yu W, Biehs B, Harada H, Snead ML, Lee JS, Desai TA, Klein OD

Sox2+ stem cells contribute to all epithelial lineages of the tooth via Sfrp5+ progenitors.

Developmental cell

Juuri E, Saito K, Ahtiainen L, Seidel K, Tummers M, Hochedlinger K, Klein OD, Thesleff I, Michon F

Stem cell and biomaterials research in dental tissue engineering and regeneration.

Dental clinics of North America

Horst OV, Chavez MG, Jheon AH, Desai T, Klein OD

BCL11B regulates epithelial proliferation and asymmetric development of the mouse mandibular incisor.

PloS one

Kyrylkova K, Kyryachenko S, Biehs B, Klein O, Kioussi C, Leid M

From molecules to mastication: the development and evolution of teeth.

Wiley interdisciplinary reviews. Developmental biology

Jheon AH, Seidel K, Biehs B, Klein OD

E-cadherin regulates the behavior and fate of epithelial stem cells and their progeny in the mouse incisor.

Developmental biology

Li CY, Cha W, Luder HU, Charles RP, McMahon M, Mitsiadis TA, Klein OD

Spry1 and spry2 are essential for development of the temporomandibular joint.

Journal of dental research

Purcell P, Jheon A, Vivero MP, Rahimi H, Joo A, Klein OD

A reserve stem cell population in small intestine renders Lgr5-positive cells dispensable.

Nature

Tian H, Biehs B, Warming S, Leong KG, Rangell L, Klein OD, de Sauvage FJ

Expression of microRNAs in the stem cell niche of the adult mouse incisor.

PloS one

Jheon AH, Li CY, Wen T, Michon F, Klein OD

Regulation of tooth number by fine-tuning levels of receptor-tyrosine kinase signaling.

Development (Cambridge, England)

Charles C, Hovorakova M, Ahn Y, Lyons DB, Marangoni P, Churava S, Biehs B, Jheon A, Lesot H, Balooch G, Krumlauf R, Viriot L, Peterkova R, Klein OD

FGF signaling regulates the number of posterior taste papillae by controlling progenitor field size.

PLoS genetics

Petersen CI, Jheon AH, Mostowfi P, Charles C, Ching S, Thirumangalathu S, Barlow LA, Klein OD

Shh expression in a rudimentary tooth offers new insights into development of the mouse incisor.

Journal of experimental zoology. Part B, Molecular and developmental evolution

Hovorakova M, Prochazka J, Lesot H, Smrckova L, Churava S, Boran T, Kozmik Z, Klein O, Peterkova R, Peterka M

The FaceBase Consortium: a comprehensive program to facilitate craniofacial research.

Developmental biology

Hochheiser H, Aronow BJ, Artinger K, Beaty TH, Brinkley JF, Chai Y, Clouthier D, Cunningham ML, Dixon M, Donahue LR, Fraser SE, Hallgrimsson B, Iwata J, Klein O, Marazita ML, Murray JC, Murray S, de Villena FP, Postlethwait J, Potter S, Shapiro L, Spritz R, Visel A, Weinberg SM, Trainor PA

PERP regulates enamel formation via effects on cell-cell adhesion and gene expression.

Journal of cell science

Jheon AH, Mostowfi P, Snead ML, Ihrie RA, Sone E, Pramparo T, Attardi LD, Klein OD

Genotype-phenotype analysis of the branchio-oculo-facial syndrome.

American journal of medical genetics. Part A

Milunsky JM, Maher TM, Zhao G, Wang Z, Mulliken JB, Chitayat D, Clemens M, Stalker HJ, Bauer M, Burch M, Chénier S, Cunningham ML, Drack AV, Janssens S, Karlea A, Klatt R, Kini U, Klein O, Lachmeijer AM, Megarbane A, Mendelsohn NJ, Meschino WS, Mortier GR, Parkash S, Ray CR, Roberts A, Roberts A, Reardon W, Schnur RE, Smith R, Splitt M, Tezcan K, Whiteford ML, Wong DA, Zori R, Lin AE

Hedgehog signaling regulates the generation of ameloblast progenitors in the continuously growing mouse incisor.

Development (Cambridge, England)

Seidel K, Ahn CP, Lyons D, Nee A, Ting K, Brownell I, Cao T, Carano RA, Curran T, Schober M, Fuchs E, Joyner A, Martin GR, de Sauvage FJ, Klein OD

Signaling by FGFR2b controls the regenerative capacity of adult mouse incisors.

Development (Cambridge, England)

Parsa S, Kuremoto K, Seidel K, Tabatabai R, Mackenzie B, Yamaza T, Akiyama K, Branch J, Koh CJ, Al Alam D, Klein OD, Bellusci S

Clefting in trisomy 9p patients: genotype-phenotype correlation using microarray comparative genomic hybridization.

The Journal of craniofacial surgery

Jelin A, Perry H, Hogue J, Oberoi S, Cotter PD, Klein OD

Inhibition of Wnt signaling by Wise (Sostdc1) and negative feedback from Shh controls tooth number and patterning.

Development (Cambridge, England)

Ahn Y, Sanderson BW, Klein OD, Krumlauf R

Patterning by heritage in mouse molar row development.

Proceedings of the National Academy of Sciences of the United States of America

Prochazka J, Pantalacci S, Churava S, Rothova M, Lambert A, Lesot H, Klein O, Peterka M, Laudet V, Peterkova R

Dact1-3 mRNAs exhibit distinct expression domains during tooth development.

Gene expression patterns : GEP

Kettunen P, Kivimäe S, Keshari P, Klein OD, Cheyette BN, Luukko K

Modulation of Fgf3 dosage in mouse and men mirrors evolution of mammalian dentition.

Proceedings of the National Academy of Sciences of the United States of America

Charles C, Lazzari V, Tafforeau P, Schimmang T, Tekin M, Klein O, Viriot L

Temporal analysis of ectopic enamel production in incisors from sprouty mutant mice.

Journal of experimental zoology. Part B, Molecular and developmental evolution

Boran T, Peterkova R, Lesot H, Lyons DB, Peterka M, Klein OD

Revitalization of a diastemal tooth primordium in Spry2 null mice results from increased proliferation and decreased apoptosis.

Journal of experimental zoology. Part B, Molecular and developmental evolution

Peterkova R, Churava S, Lesot H, Rothova M, Prochazka J, Peterka M, Klein OD

Enamel-free teeth: Tbx1 deletion affects amelogenesis in rodent incisors.

Developmental biology

Catón J, Luder HU, Zoupa M, Bradman M, Bluteau G, Tucker AS, Klein O, Mitsiadis TA

Genomic variants of ATF3 in patients with hypospadias.

The Journal of urology

Kalfa N, Liu B, Klein O, Wang MH, Cao M, Baskin LS

Mutations of CXorf6 are associated with a range of severities of hypospadias.

European journal of endocrinology

Kalfa N, Liu B, Klein O, Ophir K, Audran F, Wang MH, Mei C, Sultan C, Baskin LS

Acute fatal presentation of ornithine transcarbamylase deficiency in a previously healthy male.

Hepatology international

Klein OD, Kostiner DR, Weisiger K, Moffatt E, Lindeman N, Goodman S, Tuchman M, Packman S

The branching programme of mouse lung development.

Nature

Metzger RJ, Klein OD, Martin GR, Krasnow MA

An FGF signaling loop sustains the generation of differentiated progeny from stem cells in mouse incisors.

Development (Cambridge, England)

Klein OD, Lyons DB, Balooch G, Marshall GW, Basson MA, Peterka M, Boran T, Peterkova R, Martin GR

Interstitial deletions of chromosome 6q: genotype-phenotype correlation utilizing array CGH.

Clinical genetics

Klein OD, Cotter PD, Moore MW, Zanko A, Gilats M, Epstein CJ, Conte F, Rauen KA

Sprouty genes control diastema tooth development via bidirectional antagonism of epithelial-mesenchymal FGF signaling.

Developmental cell

Klein OD, Minowada G, Peterkova R, Kangas A, Yu BD, Lesot H, Peterka M, Jernvall J, Martin GR

Interstitial deletion of chromosome 12q: genotype-phenotype correlation of two patients utilizing array comparative genomic hybridization.

American journal of medical genetics. Part A

Klein OD, Cotter PD, Schmidt AM, Bick DP, Tidyman WE, Albertson DG, Pinkel D, Rauen KA

Brachydactylic multiple delta phalanges plus syndrome.

American journal of medical genetics. Part A

Ahn CP, Lachman RS, Cox VA, Blumberg B, Klein OD

Case report: Y;6 translocation with deletion of 6p.

Clinical dysmorphology

Klein OD, Backstrand K, Cotter PD, Marco E, Sherr E, Slavotinek A

Prader-Willi syndrome resulting from an unbalanced translocation: characterization by array comparative genomic hybridization.

Clinical genetics

Klein OD, Cotter PD, Albertson DG, Pinkel D, Tidyman WE, Moore MW, Rauen KA

The Ultrasound Review of Obstetrics and Gynecology

Congenital Müllerian anomalies: A review of currently available imaging modalities

Raga F, Bonilla-Musoles F, Osborne NG, Casañ EM, Klein O, Bonilla F

Assessment of endometrial volume by three-dimensional ultrasound prior to embryo transfer: clues to endometrial receptivity.

Human reproduction (Oxford, England)

Raga F, Bonilla-Musoles F, Casañ EM, Klein O, Bonilla F

Structural models of the bovine papillomavirus E5 protein.

Proteins

Surti T, Klein O, Aschheim K, DiMaio D, Smith SO