58 research outputs found
Systematic genetic testing for recessively inherited monogenic diabetes: a cross-sectional study in paediatric diabetes clinics
- Author
- Publication venue
- 'Springer Science and Business Media LLC'
- Publication date
- 23/10/2021
- Field of study
Data availability:
The datasets supporting the current study have not been deposited in a public repository due to institutional ethics restrictions but are available from the corresponding author on request.This is the final version. Available from Springer via the DOI in this record.âŻAIMS/HYPOTHESIS: Current clinical guidelines for childhood-onset monogenic diabetes outside infancy are mainly focused on identifying and testing for dominantly inherited, predominantly MODY genes. There are no systematic studies of the recessively inherited causes of monogenic diabetes that are likely to be more common in populations with high rates of consanguinity. We aimed to determine the contribution of recessive causes of monogenic diabetes in paediatric diabetes clinics and to identify clinical criteria by which to select individuals for recessive monogenic diabetes testing. METHODS: We conducted a cross-sectional study of 1093 children from seven paediatric diabetes clinics across Turkey (a population with high rates of consanguinity). We undertook genetic testing of 50 known dominant and recessive causes of monogenic diabetes for 236 children at low risk of type 1 diabetes. As a comparison, we used monogenic diabetes cases from UK paediatric diabetes clinics (a population with low rates of consanguinity). RESULTS: Thirty-four children in the Turkish cohort had monogenic diabetes, equating to a minimal prevalence of 3.1%, similar to that in the UK cohort (pâ=â0.40). Forty-one per cent (14/34) had autosomal recessive causes in contrast to 1.6% (2/122) in the UK monogenic diabetes cohort (pâ10%) assisted the identification of the dominant (all pââ¤â0.0003) but not recessive cases (all pââĽÂ 0.2) in Turkey. The presence of certain non-autoimmune extra-pancreatic features greatly assisted the identification of recessive (pâ<â0.0001, OR 66.9) but not dominant cases. CONCLUSIONS/INTERPRETATION: Recessively inherited mutations are a common cause of monogenic diabetes in populations with high rates of consanguinity. Present MODY-focused genetic testing strategies do not identify affected individuals. To detect all cases of monogenic paediatric diabetes, it is crucial that recessive genes are included in genetic panels and that children are selected for testing if they have certain non-autoimmune extra-pancreatic features in addition to current criteria.Wellcome TrustRoyal SocietyNational Institute for Health Researc
Wolcott-Rallison syndrome
- Author
- AF Amos
- B Bin-Abbas
- CD Wolcott
- CM Thornton
- CĂŠcile Julier
- D Feng
- D Iafusco
- DA Cunha
- DA Stoffers
- DL Eizirik
- EL Edghill
- GS Hotamisligil
- GS Sellick
- H Inoue
- H Stoss
- HP Harding
- HP Harding
- I Kim
- J Stoy
- J Wei
- K Iida
- LI Al-Gazali
- M Akiyama
- M Delepine
- M Eiken
- M Nicolino
- Marc Nicolino
- MC de Wit
- MN Ozbek
- O Rubio-Cabezas
- P Castelnau
- P Goumy
- P Goumy
- P Zhang
- PM Nicolino
- RC Wek
- RS Wildin
- S Brickwood
- S Duchatelet
- S Gupta
- S Iyer
- SG Fonseca
- SM Adler
- V Senee
- V Senee
- W Zhang
- Y Li
- Publication venue
- BioMed Central
- Publication date
- 01/01/2010
- Field of study
Wolcott-Rallison syndrome (WRS) is a rare autosomal recessive disease, characterized by neonatal/early-onset non-autoimmune insulin-requiring diabetes associated with skeletal dysplasia and growth retardation. Fewer than 60 cases have been described in the literature, although WRS is now recognised as the most frequent cause of neonatal/early-onset diabetes in patients with consanguineous parents. Typically, diabetes occurs before six months of age, and skeletal dysplasia is diagnosed within the first year or two of life. Other manifestations vary between patients in their nature and severity and include frequent episodes of acute liver failure, renal dysfunction, exocrine pancreas insufficiency, intellectual deficit, hypothyroidism, neutropenia and recurrent infections. Bone fractures may be frequent. WRS is caused by mutations in the gene encoding eukaryotic translation initiation factor 2Îą kinase 3 (EIF2AK3), also known as PKR-like endoplasmic reticulum kinase (PERK). PERK is an endoplasmic reticulum (ER) transmembrane protein, which plays a key role in translation control during the unfolded protein response. ER dysfunction is central to the disease processes. The disease variability appears to be independent of the nature of the EIF2AK3 mutations, with the possible exception of an older age at onset; other factors may include other genes, exposure to environmental factors and disease management. WRS should be suspected in any infant who presents with permanent neonatal diabetes associated with skeletal dysplasia and/or episodes of acute liver failure. Molecular genetic testing confirms the diagnosis. Early diagnosis is recommended, in order to ensure rapid intervention for episodes of hepatic failure, which is the most life threatening complication. WRS should be differentiated from other forms of neonatal/early-onset insulin-dependent diabetes based on clinical presentation and genetic testing. Genetic counselling and antenatal diagnosis is recommended for parents of a WRS patient with confirmed EIF2AK3 mutation. Close therapeutic monitoring of diabetes and treatment with an insulin pump are recommended because of the risk of acute episodes of hypoglycaemia and ketoacidosis. Interventions under general anaesthesia increase the risk of acute aggravation, because of the toxicity of anaesthetics, and should be avoided. Prognosis is poor and most patients die at a young age. Intervention strategies targeting ER dysfunction provide hope for future therapy and prevention
Spatial and temporal population genetic variation and structure of Nothotsuga longibracteata (Pinaceae), a relic conifer species endemic to subtropical China
- Author
- Achere V
- Ally DG
- Alsos I
- Austerlitz F
- Burban C
- Chung MY
- Ciofi C
- Dieringer D
- Doi H
- Doyle JJ
- Epperson BK
- Erickson DL
- EspĂndola A
- Evanno G
- Excoffier L
- Gao LM
- Geng YP
- Gilbert KJ
- Godbout J
- Guanmei Yi
- Hampe A
- Hamrick JL
- Hamrick JL
- Hewitt G
- Hongwen Huang
- Jump AS
- Jump AS
- Kalisz S
- Karavaev MN
- Kelly BA
- Kitamura K
- Kunzmann L
- Leberg PL
- Ledig FT
- Leigh FJ
- Linhart YB
- Lu SY
- Mariette S
- Marquardt PE
- Meloni M
- Michalakis Y
- Miki S
- Ming Kang
- Nei M
- Oddou-Muratorio S
- Ortego J
- Ozbek O
- Parchman TL
- Parmesan C
- Peakall R
- Peakall R
- Petit RJ
- Petit RJ
- Petit RJ
- Pons Q
- Pritchard JK
- Provan J
- Provan J
- Qiu YJ
- Qiu YJ
- Raymond M
- Shen L
- Soldati MC
- Taylor SS
- Vendramin GG
- Vendramin GG
- Walter R
- Wang HS
- Wang HW
- Wang J
- Wang J
- Weir BS
- Wu CZ
- Yifei Liu
- Ying TS
- Yingjun Qiu
- Zhang DQ
- Zhao YJ
- Zou JB
- Publication venue
- 'FapUNIFESP (SciELO)'
- Publication date
- 01/01/2013
- Field of study
Measurement of the rapidity and transverse momentum distributions of Z bosons in pp collisions at â(s)=7ââTeV
- Author
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdullin S
- Acosta D
- Acosta JG
- Acosta MV
- Adair A
- Adam N
- Adam W
- Adams MR
- Adams T
- Adiguzel A
- Adler V
- Adzic P
- Afanasiev S
- Agostino L
- Agram JL
- Aguilar-Benitez M
- Aguilo E
- Ahmad M
- Ahmad WH
- Ahuja S
- Akchurin N
- Akgun B
- Akgun U
- Akin IV
- Alagoz E
- Albajar C
- Albayrak EA
- Albergo S
- Albrow M
- Alcaraz Maestre J
- Alda Junior WL
- Alexander J
- Aliev T
- Almeida N
- Alver B
- Alverson G
- Alves GA
- Amapane N
- Amsler C
- Anagnostou G
- Anastassov A
- Anderson J
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- Andreev V
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- Auzinger G
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- Azarkin M
- Azhgirey I
- Aziz T
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- Azzolini V
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- Baarmand MM
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- Wickramage N
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- Wyslouch B
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- Yang F
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- Yang ZC
- Yazgan E
- Yelton J
- Yetkin T
- Yi K
- Yildirim E
- Yilmaz Y
- Yohay R
- Yoo HD
- Yoo J
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- Yu I
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- Yumiceva F
- Yun JC
- Zabel J
- Zabi A
- Zablocki J
- Zaganidis N
- Zakaria M
- Zalewski P
- Zanetti M
- Zang J
- Zang SL
- Zarubin A
- Zatserklyaniy A
- Zeinali M
- Zeise M
- Zeuner WD
- Zeyrek M
- Zhang J
- Zhang Z
- Zheng Y
- Zhokin A
- Zhu B
- Zhu RY
- Zhukov V
- Zhukova V
- Ziebarth EB
- Ziegler J
- Zielinski M
- Zito G
- Zoeller MH
- Zotto P
- Zou W
- Zumerle G
- Zuranski A
- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 01/10/2011
- Field of study
Measurements of the normalized rapidity (y) and transverse-momentum (qT) distributions of DrellâYan muon and electron pairs in the Z-boson mass region (60<Mââ<120ââGeV) are reported. The results are obtained using a data sample of proton-proton collisions at a center-of-mass energy of 7 TeV, collected by the CMS experiment at the Large Hadron Collider (LHC), corresponding to an integrated luminosity of 36ââpb-1. The distributions are measured over the ranges |y|<3.5 and qT<600ââGeV and compared with quantum chromodynamics (QCD) calculations using recent parton distribution functions to model the momenta of the quarks and gluons in the protons. Overall agreement is observed between the models and data for the rapidity distribution, while no single model describes the Z transverse-momentum distribution over the full range
Search for dijet resonances in 7 TeV pp collisions at CMS
- Author
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdel-Basit A
- Abdullin S
- Acosta D
- Acosta JG
- Acosta MV
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- Contardo D
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- D'Agnolo RT
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- D'Enterria D
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- Da Costa EM
- Da Silva WLP
- Daeuwel D
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- Dahms T
- Dallavalle GM
- Damgov J
- Damiao DD
- Dammann D
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- Darmenov N
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- Daubie E
- David A
- Davids M
- Davies G
- De Araujo FTD
- de Barbaro P
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- de Cassagnac RG
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- De Guio F
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- de Monchenault GH
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- Yilmaz Y
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- Zeinali M
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- Zeuner WD
- Zeyrek M
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- Zhang Z
- Zheng Y
- Zhokin A
- Zhu B
- Zhu RY
- Zhukov V
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- Ziebarth EB
- Ziegler J
- Zielinski M
- Zito G
- Zoeller MH
- Zorbilmez C
- Zotto P
- Zuranski A
- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 01/01/2010
- Field of study
This is the Pre-print version of the Article. The official published version of the paper can be accessed from the link below - Copyright @ 2010 APSA search for narrow resonances in the dijet mass spectrum is performed using data corresponding to an integrated luminosity of 2.9ââpb-1 collected by the CMS experiment at the Large Hadron Collider. Upper limits at the 95% confidence level are presented on the product of the resonance cross section, branching fraction into dijets, and acceptance, separately for decays into quark-quark, quark-gluon, or gluon-gluon pairs. The data exclude new particles predicted in the following models at the 95% confidence level: string resonances, with mass less than 2.50 TeV, excited quarks, with mass less than 1.58 TeV, and axigluons, colorons, and E6 diquarks, in specific mass intervals. This extends previously published limits on these models.This work is supported by FMSR (Austria); FNRS and FWO (Belgium); CNPq, CAPES, FAPERJ, and FAPESP (Brazil); MES (Bulgaria); CERN; CAS, MoST, and NSFC (China); COLCIENCIAS (Colombia); MSES (Croatia); RPF (Cyprus); Academy of Sciences and NICPB (Estonia); Academy of Finland, ME, and HIP (Finland); CEA and CNRS/IN2P3 (France); BMBF, DFG, and HGF (Germany); GSRT (Greece); OTKA and NKTH (Hungary); DAE and DST (India); IPM (Iran); SFI (Ireland); INFN (Italy); NRF and WCU (Korea); LAS (Lithuania); CINVESTAV, CONACYT, SEP, and UASLP-FAI (Mexico); PAEC (Pakistan); SCSR (Poland); FCT (Portugal); JINR (Armenia, Belarus, Georgia, Ukraine, Uzbekistan); MST and MAE (Russia); MSTD (Serbia); MICINN and CPAN (Spain); Swiss Funding Agencies (Switzerland); NSC (Taipei); TUBITAK and TAEK (Turkey); STFC (United Kingdom); DOE and NSF (USA)
Search for supersymmetry in events with b jets and missing transverse momentum at the LHC
- Author
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdullin S
- Acosta D
- Acosta JG
- Acosta MV
- Adam N
- Adam W
- Adams MR
- Adams T
- Adiguzel A
- Adler V
- Adzic P
- Afanasiev S
- Agostino L
- Agram JL
- Aguilar-Benitez M
- Aguilo E
- Ahmad M
- Ahmad WH
- Ahmed I
- Ahuja S
- Akchurin N
- Akgun B
- Akgun U
- Akin IV
- Alagoz E
- Albajar C
- Albayrak EA
- Albergo S
- Albrow M
- Alexander J
- Aliev T
- Almeida N
- Alver B
- Alverson G
- Alves GA
- Amapane N
- Amsler C
- Anagnostou G
- Anastassov A
- Anderson J
- Anderson M
- Andrea J
- Andreev V
- Andreev V
- Andreev Y
- Andrews W
- Anghel IM
- Antonelli L
- Antunes JR
- Antunovic Z
- Apanasevich L
- Apollinari G
- Appelt E
- Apresyan A
- Aranyi A
- Arce P
- Arcidiacono R
- Arenton MW
- Arfaei H
- Argiro S
- Arisaka K
- Arneodo M
- Asghar MI
- Askew A
- Assran Y
- Ata M
- Atac M
- Atramentov O
- Attikis A
- Auffray E
- Autermann C
- Auzinger G
- Avery P
- Avetisyan A
- Azarkin M
- Azhgirey I
- Aziz T
- Azzi P
- Azzolini V
- Azzurri P
- Baarmand MM
- Babb J
- Bacchetta N
- Bachtis M
- Baden A
- Baesso P
- Baffioni S
- Bagliesi G
- Bai Y
- Baillon P
- Bainbridge R
- Bakhshiansohi H
- Bakirci MN
- Bakken JA
- Balazs M
- Ball AH
- Ball G
- Ballin J
- Ban Y
- Banerjee S
- Banerjee S
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- Publication venue
- 'Springer Science and Business Media LLC'
- Publication date
- 01/01/2011
- Field of study
A search for supersymmetry is presented using a sample of events with b jets and missing transverse momentum. The search uses a data sample of proton-proton collisions at a centre-of-mass energy of 7TeV, corresponding to an integrated luminosity of 35 pb(-1), collected with the CMS detector. A total of 0.33(-0.33)(+0.43) (stat.) +/- 0.13 (syst.) events is predicted, using control samples in the data, to arise from standard model processes, and one event is observed in the data. Upper limits are set at the 95% confidence level on the cross sections of benchmark supersymmetric models
Indications of suppression of excited ÎĽ states in Pb-Pb collisions at âsNN = 2.76TeV
- Author
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- Zang J
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- Zeinali M
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- Zennamo J
- Zeuner WD
- Zeyrek M
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- Zhu B
- Zhu RY
- Zhukov V
- Zhukova V
- Ziebarth EB
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- Zito G
- Zoeller MH
- Zotto P
- Zou W
- Zumerle G
- Zuranski A
- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 01/01/2011
- Field of study
This is the pre-print version of the Published Article which can be accessed from the link below.A comparison of the relative yields of ÎĽ resonances in the Îź+Îź- decay channel in Pb-Pb and pp collisions at a center-of-mass energy per nucleon pair of 2.76 TeV is performed with data collected with the CMS detector at the LHC. Using muons of transverse momentum above 4ââGeV/c and pseudorapidity below 2.4, the double ratio of the ÎĽ(2S) and ÎĽ(3S) excited states to the ÎĽ(1S) ground state in Pb-Pb and pp collisions, [ÎĽ(2S+3S)/ÎĽ(1S)]Pb-Pb/[ÎĽ(2S+3S)/ÎĽ(1S)]pp, is found to be 0.31-0.15+0.19(stat)Âą0.03(syst). The probability to obtain the measured value, or lower, if the true double ratio is unity, is calculated to be less than 1%
Measurement of the t-channel single top quark production cross section in pp collisions at âs = 7TeV
- Author
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- Zoeller MH
- Zotto P
- Zou W
- Zumerle G
- Zuranski A
- Publication venue
- American Physical Society
- Publication date
- 01/01/2011
- Field of study
This is the pre-print version of the Published Article which can be accessed from the link below.Electroweak production of the top quark is measured for the first time in pp collisions at âs=7ââTeV, using a data set collected with the CMS detector at the LHC and corresponding to an integrated luminosity of 36ââpb-1. With an event selection optimized for t-channel production, two complementary analyses are performed. The first one exploits the special angular properties of the signal, together with background estimates from the data. The second approach uses a multivariate analysis technique to probe the compatibility with signal topology expected from electroweak top-quark production. The combined measurement of the cross section is 83.6Âą29.8(stat+syst)Âą3.3(lumi)ââpb, consistent with the standard model expectation
Search for same-sign top-quark pair production at root s=7 TeV and limits on flavour changing neutral currents in the top sector
- Author
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- Abbiendi G
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- Yumiceva F
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- Zhukov V
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- Zoeller MH
- Zotto P
- Zou W
- Zumerle G
- Zuranski A
- Publication venue
- 'Springer Science and Business Media LLC'
- Publication date
- 01/01/2011
- Field of study
An inclusive search for same-sign top-quark pair production in pp collisions at root s = 7 TeV is performed using a data sample recorded with the CMS detector in 2010, corresponding to an integrated luminosity of 35 pb(-1). This analysis is motivated by recent studies of p (p) over bar -> t (t) over bar reporting mass-dependent forward-backward asymmetries larger than expected from the standard model. These asymmetries could be due to Flavor Changing Neutral Currents (FCNC) in the top sector induced by t -channel exchange of a massive neutral vector boson (Z'). Models with such a Z' also predict enhancement of same-sign top-pair production in pp or pp collisions. Limits are set as a function of the Z' mass and its couplings to u and t quarks. These limits disfavour the FCNC interpretation of the Tevatron results
Search for new physics with a monojet and missing transverse energy in pp collisions at âs=7 TeV
- Author
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- Abbiendi G
- Abbrescia M
- Abdullin S
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- Villella I
- Vinogradov A
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- Weinberg M
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- Wendland L
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- Wenger EA
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- Whyntie T
- Wickramage N
- Widl E
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- Wissing C
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- Won S
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- Wood JS
- Worm SD
- Wright D
- Wrochna G
- Wu JH
- Wu W
- Wulz CE
- Wyslouch B
- WĂźrthwein F
- Xiao H
- Xie S
- Xu M
- Yagil A
- Yan M
- Yang F
- Yang M
- Yang Y
- Yang ZC
- Yazgan E
- Yelton J
- Yetkin T
- Yi K
- Yildirim E
- Yilmaz Y
- Yohay R
- Yoo HD
- Yoo J
- Yoon AS
- York A
- Yu I
- Yu SS
- Yumiceva F
- Yun JC
- Zabel J
- Zabi A
- Zablocki J
- Zaganidis N
- Zakaria M
- Zalewski P
- Zanetti M
- Zang J
- Zang SL
- Zarubin A
- Zatserklyaniy A
- Zeinali M
- Zeise M
- Zennamo J
- Zeuner WD
- Zeyrek M
- Zhang J
- Zhang Z
- Zheng Y
- Zhokin A
- Zhu B
- Zhu RY
- Zhukov V
- Zhukova V
- Ziebarth EB
- Ziegler J
- Zielinski M
- Zito G
- Zoeller MH
- Zotto P
- Zou W
- Zumerle G
- Zuranski A
- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 01/01/2011
- Field of study
This is the pre-print version of the Published Article which can be accessed at the link below.A study of events with missing transverse energy and an energetic jet is performed using pp collision data at a center-of-mass energy of 7 TeV. The data were collected by the CMS detector at the LHC, and correspond to an integrated luminosity of 36ââpb-1. An excess of these events over standard model contributions is a signature of new physics such as large extra dimensions and unparticles. The number of observed events is in good agreement with the prediction of the standard model, and significant extension of the current limits on parameters of new physics benchmark models is achieved
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