764 research outputs found
Recent Advances and Future Directions for Quality Engineering
- Author
- Publication venue
- 'Wiley'
- Publication date
- 01/01/2015
- Field of study
The origins of quality engineering are in manufacturing, where quality engineers apply basic statistical methodologies to improve the quality and productivity of products and processes. In the past decade, people have discovered that these methodologies are effective for improving almost any type of system or process, such as financial, health care, and supply chains.This paper begins with a review of key advances and trends within quality engineering over the past decade. The second part uses the first part as a foundation to outline new application areas for the field. It also discusses how quality engineering needs to evolve in order to make significant contributions to these new areas</p
Measurement of the hadronic photon structure function F_{2}^{γ} at LEP2
- Author
- Abbiendi G
- Ainsley C
- Alexander G
- Allison J
- Anagnostou G
- Anderson KJ
- Asai S
- Axen D
- Azuelos G
- Bailey I
- Barberio E
- Barlow RJ
- Batley RJ
- Bechtle P
- Behnke T
- Bell KW
- Bell PJ
- Bella G
- Bellerive A
- Benelli G
- Bethke S
- Biebel O
- Bloodworth IJ
- Bock P
- Boeriu O
- Bonacorsi D
- Boutemeur M
- Braibant S
- Brigliadori L
- Brown RM
- Buesser K
- Burckhart HJ
- Cammin J
- Campana S
- Carnegie RK
- Caron B
- Carter AA
- Carter JR
- Chang CY
- Charlton DG
- Cohen I
- Csilling A
- Cuffiani M
- Dado S
- Dallavalle GM
- Dallison S
- De Roeck A
- De Wolf EA
- Desch K
- Donkers M
- Dubbert J
- Duchovni E
- Duckeck G
- Duerdoth IP
- Etzion E
- Fabbri F
- Feld L
- Ferrari P
- Fiedler F
- Fleck I
- Ford M
- Frey A
- Fürtjes A
- Gagnon P
- Gary JW
- Gaycken G
- Geich-Gimbel C
- Giacomelli G
- Giacomelli P
- Giunta M
- Goldberg J
- Gross E
- Grunhaus J
- Gruwé M
- Gupta A
- Günther PO
- Hajdu C
- Hamann M
- Hanson GG
- Harder K
- Harel A
- Harin-Dirac M
- Hauschild M
- Hauschildt J
- Hawkes CM
- Hawkings R
- Åkesson PF
- Publication venue
- Publication date
- 09/05/2002
- Field of study
The hadronic structure function of the photon F_{2}^{γ} (x, Q²) is measured as a function of Bjorken x and of the photon virtuality Q² using deep-inelastic scattering data taken by the OPAL detector at LEP at e⁺e⁻ centre-of-mass energies from 183 to 209 GeV. Previous OPAL measurements of the x dependence of F_{2}^{γ} are extended to an average Q² of 〈Q²〉=780 GeV² using data in the kinematic range 0.15<x<0.98. The Q² evolution of F_{2}^{γ} is studied for 12.1<〈Q²〉<780 GeV² using three ranges of x. As predicted by QCD, the data show positive scaling violations in F_{2}^{γ} with F_{2}^{γ} (Q²)/α = (0.08±0.02⁺⁰·⁰⁵_₀.₀₃) + (0.13±0.01⁺⁰·⁰¹_₀.₀₁) lnQ², where Q² is in GeV², for the central x region 0.10–0.60. Several parameterisations of F_{2}^{γ} are in qualitative agreement with the measurements whereas the quark-parton model prediction fails to describe the data
Search for Second-Generation Scalar Leptoquarks in ppˉ Collisions at s=1.96 TeV
- Author
- Abulencia A
- Acosta D
- Adelman J
- Affolder T
- Akimoto T
- Albrow MG
- Ambrose D
- Amerio S
- Amidei D
- Anastassov A
- Anikeev K
- Annovi A
- Antos J
- Aoki M
- Apollinari G
- Arguin JF
- Arisawa T
- Artikov A
- Ashmanskas W
- Attal A
- Azfar F
- Azzi-Bacchetta P
- Azzurri P
- Bacchetta N
- Bachacou H
- Badgett W
- Barbaro-Galtieri A
- Barnes VE
- Barnett BA
- Baroiant S
- Bartsch V
- Bauer G
- Bedeschi F
- Behari S
- Belforte S
- Bellettini G
- Bellinger J
- Belloni A
- Ben Haim E
- Benjamin D
- Beretvas A
- Beringer J
- Berry T
- Bhatti A
- Binkley M
- Bisello D
- Bishai M
- Blair RE
- Blocker C
- Bloom K
- Blumenfeld B
- Bocci A
- Bodek A
- Boisvert V
- Bolla G
- Bolshov A
- Bortoletto D
- Boudreau J
- Bourov S
- Boveia A
- Brau B
- Bromberg C
- Brubaker E
- Budagov J
- Budd HS
- Budd S
- Burkett K
- Busetto G
- Bussey P
- Byrum KL
- Cabrera S
- Campanelli M
- Campbell M
- Canelli F
- Canepa A
- Carlsmith D
- Carosi R
- Carron S
- Casarsa M
- Castro A
- Catastini P
- Cauz D
- Cavalli-Sforza M
- Cerri A
- Cerrito L
- Chang SH
- Chapman J
- Chen YC
- Chertok M
- Chiarelli G
- Chlachidze G
- Chlebana F
- Cho I
- Cho K
- Chokheli D
- Chou JP
- Chu PH
- Chuang SH
- Chung K
- Chung WH
- Chung YS
- Ciljak M
- Ciobanu CI
- Ciocci MA
- Clark A
- Clark D
- Coca M
- Connolly A
- Convery ME
- Conway J
- Cooper B
- Copic K
- Cordelli M
- Cortiana G
- Cruz A
- Cuevas J
- Culbertson R
- Cyr D
- D'Auria S
- D'onofrio M
- da Costa JG
- Dagenhart D
- DaRonco S
- de Barbaro P
- De Cecco S
- De Lentdecker G
- De Pedis D
- Deisher A
- Dell'Orso M
- Demers S
- Demortier L
- Deng J
- Deninno M
- Derwent PF
- di Giovanni GP
- Dionisi C
- Dittmann JR
- DiTuro P
- Dominguez A
- Donati S
- Donega M
- Dong P
- Donini J
- Dorigo T
- Dorr C
- Dube S
- Ebina K
- Efron J
- Ehlers J
- Erbacher R
- Errede D
- Errede S
- Eusebi R
- Fang HC
- Farrington S
- Fedorko I
- Fedorko WT
- Feild RG
- Feindt M
- Fernandez JP
- Fernandez PM
- Field R
- Flanagan G
- Flores-Castillo LR
- Foland A
- Forrester S
- Foster GW
- Franklin M
- Freeman JC
- Fujii Y
- Furic I
- Gajjar A
- Gallinaro M
- Galyardt J
- Garcia JE
- Garfinkel AF
- Gay C
- Gerberich H
- Gerchtein E
- Gerdes D
- Giagu S
- Giannetti P
- Gibson A
- Gibson K
- Ginsburg C
- Giokaris N
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- Glenzinski D
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- Hatakeyama K
- Hauser J
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- Jun SY
- Junk TR
- Kamon T
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- Karagoz-Unel M
- Karchin PE
- Kato Y
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- Kerzel U
- Khotilovich V
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- Kirby M
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- Kotwal AV
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- Kuhlmann SE
- Kusakabe Y
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- Laasanen AT
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- Lander RL
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- Napier A
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- Oldeman R
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- Portell X
- Poukhov O
- Pounder N
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- Pronko A
- Proudfoot J
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- Richter S
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- Scuri F
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- Shapiro MD
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- Sinervo P
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- Snider FD
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- Tanaka M
- Tanaka R
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- Teng PK
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- Thompson AS
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- Thomson E.
- Tipton P
- Tipton P.
- Tiwari V
- Tiwari V.
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- Tkaczyk S.
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- Toback D.
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- Tokar S.
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- Tollefson K.
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- Tomura T.
- Tonelli D
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- Tonnesmann M
- Torre S
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- Tsuchiya R
- Tsuchiya R.
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- Turini N
- Turini N.
- Ukegawa F
- Ukegawa F.
- Unverhau T
- Unverhau T.
- Uozumi S
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- Usynin D.
- Vacavant L
- Vaiciulis A
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- Vallecorsa S.
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- van Remortel N.
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- Varganov A.
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- Vataga E.
- Vazquez F.
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- Veramendi G.
- Veszpremi V
- Veszpremi V.
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- Vidal R.
- Vila I
- Vila I.
- Vilar R
- Vilar R.
- Vine T.
- Vollrath I
- Vollrath I.
- Volobouev I
- Volobouev I.
- Volpi G.
- von der Mey M
- Wagner J.
- Wagner P
- Wagner P.
- Wagner R. G.
- Wagner R. L.
- Wagner RG
- Wagner RL
- Wagner W
- Wagner W.
- Wallny R
- Wallny R.
- Walter T
- Wan Z
- Wang MJ
- Wang S. M.
- Wang SM
- Warburton A
- Warburton A.
- Ward B
- Waschke S
- Waschke S.
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- Waters D.
- Watts T
- Weber M
- Weinberger M.
- Wester W. C. III
- Wester WC
- Whitehouse B
- Whitehouse B.
- Whiteson D
- Whiteson D.
- Wicklund A. B.
- Wicklund AB
- Wicklund E
- Wicklund E.
- Williams G.
- Williams H. H.
- Williams HH
- Wilson P
- Wilson P.
- Winer B. L.
- Winer BL
- Wittich P
- Wittich P.
- Wolbers S
- Wolbers S.
- Wolfe C
- Wolfe C.
- Worm S
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- Wright T.
- Wu X
- Wu X.
- Wurthwein F
- Wurthwein F.
- Wynne S. M.
- Wynne SM
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- Yagil A.
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- Yamamoto K.
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- Yamaoka J.
- Yamashita T.
- Yamashita Y
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- Yang C.
- Yang U. K.
- Yang UK
- Yang Y. C.
- Yao W. M.
- Yao WM
- Yeh G. P.
- Yeh GP
- Yoh J
- Yoh J.
- Yorita K
- Yorita K.
- Yoshida T
- Yoshida T.
- Yu G. B.
- Yu I
- Yu I.
- Yu S. S.
- Yu SS
- Yun J. C.
- Yun JC
- Zanello L
- Zanello L.
- Zanetti A
- Zanetti A.
- Zaw I
- Zaw I.
- Zetti F
- Zhang X
- Zhang X.
- Zhou J
- Zhou J.
- Zucchelli S.
- Zucchelli Slli Slli Selli Spson A. S.
- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 01/12/2005
- Field of study
Results on a search for pair production of second generation scalar
leptoquark in ppˉ collisions at s=1.96 TeV are reported. The
data analyzed were collected by the CDF detector during the 2002-2003 Tevatron
Run II and correspond to an integrated luminosity of 198 pb−1. Leptoquarks
(LQ) are sought through their decay into (charged) leptons and quarks, with
final state signatures represented by two muons and jets and one muon, large
transverse missing energy and jets. We observe no evidence for LQ production
and derive 95% C.L. upper limits on the LQ production cross sections as well
as lower limits on their mass as a function of β, where β is the
branching fraction for LQ→μq.Comment: 9 pages (3 author list) 5 figure
Observation of associated near-side and away-side long-range correlations in √sNN=5.02 TeV proton-lead collisions with the ATLAS detector
- Author
- Aad G
- Abajyan T
- Abbott B
- Abdallah J
- Abdel Khalek S
- Abdelalim AA
- Abdinov O
- Aben R
- Abi B
- Abolins M
- Abouzeid OS
- Abramowicz H
- Abreu H
- Acharya BS
- Adamczyk L
- Adams DL
- Addy TN
- Adelman J
- Adomeit S
- Adragna P
- Adye T
- Aefsky S
- Aguilar-Saavedra JA
- Agustoni M
- Ahlen SP
- Ahles F
- Ahmad A
- Ahsan M
- Aielli G
- Akesson TP
- Akimoto G
- Akimov AV
- Alam MA
- Albert J
- Albrand S
- Aleksa M
- Aleksandrov IN
- Alessandria F
- Alexa C
- Alexander G
- Alexandre G
- Alexopoulos T
- Alhroob M
- Aliev M
- Alimonti G
- Alison J
- Allbrooke BM
- Allison LJ
- Allport PP
- Allwood-Spiers SE
- Almond J
- Aloisio A
- Alon R
- Alonso A
- Alonso F
- Altheimer A
- Alvarez Gonzalez B
- Alviggi MG
- Amako K
- Amelung C
- Ammosov VV
- Amor Dos Santos SP
- Amorim A
- Amoroso S
- Amram N
- Anastopoulos C
- Ancu LS
- Andari N
- Andeen T
- Anders CF
- Anders G
- Anderson KJ
- Andreazza A
- Andrei V
- Anduaga XS
- Angelidakis S
- Anger P
- Angerami A
- Anghinolfi F
- Anisenkov A
- Anjos N
- Annovi A
- Antonaki A
- Antonelli M
- Antonov A
- Antos J
- Anulli F
- Aoki M
- Aperio Bella L
- Apolle R
- Arabidze G
- Aracena I
- Arai Y
- Arce AT
- Arfaoui S
- Arguin JF
- Argyropoulos S
- Arik E
- Arik M
- Armbruster AJ
- Arnaez O
- Arnal V
- Artamonov A
- Artoni G
- Arutinov D
- Asai S
- Ask S
- Asman B
- Asquith L
- Assamagan K
- Astalos R
- Astbury A
- Atkinson M
- ATLAS Collaboration The
- Auerbach B
- Auge E
- Augsten K
- Aurousseau M
- Avolio G
- Axen D
- Azuelos G
- Azuma Y
- Baak MA
- Baccaglioni G
- Bacci C
- Bach AM
- Bachacou H
- Bachas K
- Backes M
- Backhaus M
- Backus Mayes J
- Badescu E
- Bagnaia P
- Bai Y
- Bailey DC
- Bain T
- Baines JT
- Baker OK
- Baker S
- Balek P
- Balli F
- Banas E
- Banerjee P
- Banerjee S
- Banfi D
- Bangert A
- Bansal V
- Bansil HS
- Barak L
- Baranov SP
- Barber T
- Barberio EL
- Barberis D
- Barbero M
- Bardin DY
- Barillari T
- Barisonzi M
- Barklow T
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- Barreiro Guimarães da Costa J
- Barreiro F
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- Basye A
- Bates RL
- Batkova L
- Batley JR
- Battaglia A
- Battistin M
- Bauer F
- Bawa HS
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- Beauchemin PH
- Beccherle R
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- Becks KH
- Beddall A
- Beddall AJ
- Bedikian S
- Bednyakov VA
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- Beemster LJ
- Beermann TA
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- Benary O
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- Bittner B
- Black CW
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- Black KM
- Blair RE
- Blanchard JB
- Blazek T
- Bloch I
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- Blocki J
- Blum W
- Blumenschein U
- Bobbink GJ
- Bobrovnikov VS
- Bocchetta SS
- Bocci A
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- Bouhova-Thacker EV
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- Boutouil S
- Boveia A
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- Yurkewicz A
- Zabinski B
- Zaidan R
- Zaitsev AM
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- Zanzi D
- Zaytsev A
- Zeitnitz C
- Zeman M
- Zemla A
- Zenin O
- Zeniš T
- Zerwas D
- Zevi Della Porta G
- Zhang D
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- Zhao Z
- Zhemchugov A
- Zhong J
- Zhou B
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- Zhu CG
- Zhu H
- Zhu J
- Zhu Y
- Zhuang X
- Zhuravlov V
- Zibell A
- Zieminska D
- Zimin NI
- Zimmermann R
- Zimmermann S
- Zimmermann S
- Zinonos Z
- Ziolkowski M
- Zitoun R
- Zivković L
- Zmouchko VV
- Zobernig G
- Zoccoli A
- Zur Nedden M
- Zutshi V
- Zwalinski L
- Publication venue
- American Physical Society
- Publication date
- 01/01/2012
- Field of study
Two-particle correlations in relative azimuthal angle (Δϕ) and pseudorapidity (Δη) are measured in √sNN=5.02 TeV p+Pb collisions using the ATLAS detector at the LHC. The measurements are performed using approximately 1 μb-1 of data as a function of transverse momentum (pT) and the transverse energy (ΣETPb) summed over 3.1<η<4.9 in the direction of the Pb beam. The correlation function, constructed from charged particles, exhibits a long-range (2<|Δη|<5) “near-side” (Δϕ∼0) correlation that grows rapidly with increasing ΣETPb. A long-range “away-side” (Δϕ∼π) correlation, obtained by subtracting the expected contributions from recoiling dijets and other sources estimated using events with small ΣETPb, is found to match the near-side correlation in magnitude, shape (in Δη and Δϕ) and ΣETPb dependence. The resultant Δϕ correlation is approximately symmetric about π/2, and is consistent with a dominant cos2Δϕ modulation for all ΣETPb ranges and particle pT
Search for the standard model Higgs boson at LEP
- Author
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- Abbaneo D
- Abbiendi G
- Abbiendi G
- Abdallah J
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- Publication venue
- ELSEVIER SCIENCE BV
- Publication date
- 17/07/2003
- Field of study
Measurement of the charm structure function F_{2,c)^{γ} of the photon at LEP
- Author
- Abbiendi G
- Ainsley C
- Alexander G
- Allison J
- Amaral P
- Anagnostou G
- Anderson KJ
- Arcelli S
- Asai S
- Axen D
- Azuelos G
- Bailey I
- Barberio E
- Barlow RJ
- Batley RJ
- Bechtle P
- Behnke T
- Bell KW
- Bell PJ
- Bella G
- Bellerive A
- Benelli G
- Bethke S
- Biebel O
- Bloodworth IJ
- Bock P
- Boeriu O
- Bonacorsi D
- Boutemeur M
- Braibant S
- Brigliadori L
- Brown RM
- Buesser K
- Burckhart HJ
- Cammin J
- Campana S
- Carnegie RK
- Caron B
- Carter AA
- Carter JR
- Chang CY
- Charlton DG
- Cohen I
- Csilling A
- Cuffiani M
- Dado S
- Dallavalle GM
- Dallison S
- de Roeck A
- de Wolf EA
- Desch K
- Donkers M
- Dubbert J
- Duchovni E
- Duckeck G
- Duerdoth IP
- Elfgren E
- Etzion E
- Fabbri F
- Feld L
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- Gary JW
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- Geich-Gimbel C
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- Gruwé M
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- Günther PO
- Hajdu C
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- Hanson GG
- Harder K
- Harel A
- Harin-Dirac M
- Hauschild M
- Hauschildt J
- Hawkes CM
- Hawkings R
- Hemingway RJ
- Hensel C
- Herten G
- Heuer RD
- Hill JC
- Hoffman K
- Åkesson PF
- Publication venue
- Publication date
- 11/07/2002
- Field of study
The production of charm quarks is studied in deep-inelastic electron–photon scattering using data recorded by the OPAL detector at LEP at nominal e⁺e⁻ centre-of-mass energies from 183 to 209 GeV. The charm quarks have been identified by full reconstruction of charged D* mesons using their decays into D⁰π with the D⁰ observed in two decay modes with charged particle final states, Kπ and Kπππ. The cross-section σ^{D*} for production of charged D* in the reaction e⁺e⁻→e⁺e⁻D*Χ is measured in a restricted kinematical region using two bins in Bjorken x, 0.00140.1 the perturbative QCD calculation at next-to-leading order agrees perfectly with the measured cross-section. For x<0.1 the measured cross-section is 43.8±14.3±6.3±2.8 pb with a next-to-leading order prediction of 17.0⁺²·⁹_₂.₃ pb
Performance of the CMS Cathode Strip Chambers with Cosmic Rays
- Author
- Abadjiev K
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdullin S
- Abelev B
- Acosta D
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- Acosta MV
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- Adiguzel A
- Adler V
- Adolphi R
- Adzic P
- Afaq MA
- Agostino L
- Agram JL
- Aguilar-Benitez M
- Ahmad M
- Ahmad WH
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- Ahuja S
- Aisa D
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- Akgun B
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- Alberdi J
- Albergo S
- Albert E
- Albrow M
- Alexander J
- Alidra M
- Aliev T
- Allfrey P
- Almeida N
- Altenhofer G
- Altsybeev I
- Alver B
- Alverson G
- Alves GA
- Amaglobeli N
- Amapane N
- Ambroglini F
- Amsler C
- Anagnostou G
- Ananthan B
- Anastassov A
- Andelin D
- Anderson M
- Andrea J
- Andreev V
- Andreev Y
- Anghel IM
- Anguelov T
- Anh T. Vu
- Anisimov A
- Antillon E
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- Anttila E
- Antunes JR
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- Apollinari G
- Apresyan A
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- Auffray E
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- Publication venue
- 'IOP Publishing'
- Publication date
- 01/01/2009
- Field of study
The Cathode Strip Chambers (CSCs) constitute the primary muon tracking device
in the CMS endcaps. Their performance has been evaluated using data taken
during a cosmic ray run in fall 2008. Measured noise levels are low, with the
number of noisy channels well below 1%. Coordinate resolution was measured for
all types of chambers, and fall in the range 47 microns to 243 microns. The
efficiencies for local charged track triggers, for hit and for segments
reconstruction were measured, and are above 99%. The timing resolution per
layer is approximately 5 ns
Observation of the Baryonic Flavor-Changing Neutral Current Decay Lambda_b -> Lambda mu+ mu-
- Author
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- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 01/01/2011
- Field of study
We report the first observation of the baryonic flavor-changing neutral
current decay Lambda_b -> Lambda mu+ mu- with 24 signal events and a
statistical significance of 5.8 Gaussian standard deviations. This measurement
uses ppbar collisions data sample corresponding to 6.8fb-1 at sqrt{s}=1.96TeV
collected by the CDF II detector at the Tevatron collider. The total and
differential branching ratios for Lambda_b -> Lambda mu+ mu- are measured. We
find B(Lambda_b -> Lambda mu+ mu-) = [1.73+-0.42(stat)+-0.55(syst)] x 10^{-6}.
We also report the first measurement of the differential branching ratio of B_s
-> phi mu+ mu- using 49 signal events. In addition, we report branching ratios
for B+ -> K+ mu+ mu-, B0 -> K0 mu+ mu-, and B -> K*(892) mu+ mu- decays.Comment: 8 pages, 2 figures, 4 tables. Submitted to Phys. Rev. Let
Measurement of the Dipion Mass Spectrum in X(3872) -> J/Psi Pi+ Pi- Decays
- Author
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- Strologas J
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- Publication venue
- 'American Physical Society (APS)'
- Publication date
- 28/12/2005
- Field of study
We measure the dipion mass spectrum in X(3872)--> J/Psi Pi+ Pi- decays using
360 pb-1 of pbar-p collisions at 1.96 TeV collected with the CDF II detector.
The spectrum is fit with predictions for odd C-parity (3S1, 1P1, and 3DJ)
charmonia decaying to J/Psi Pi+ Pi-, as well as even C-parity states in which
the pions are from Rho0 decay. The latter case also encompasses exotic
interpretations, such as a D0-D*0Bar molecule. Only the 3S1 and J/Psi Rho
hypotheses are compatible with our data. Since 3S1 is untenable on other
grounds, decay via J/Psi Rho is favored, which implies C=+1 for the X(3872).
Models for different J/Psi-Rho angular momenta L are considered. Flexibility in
the models, especially the introduction of Rho-Omega interference, enable good
descriptions of our data for both L=0 and 1.Comment: 7 pages, 4 figures -- Submitted to Phys. Rev. Let
A cohort of 17 patients with kyphoscoliotic Ehlers-Danlos syndrome caused by biallelic mutations in FKBP14: expansion of the clinical and mutational spectrum and description of the natural history.
- Author
- A Karaa
- A Nadeau
- AA Aldeeri
- Adnan Y Manzur
- AK Lampe
- Angela F Brady
- Anthony Vandersteen
- AR Janecke
- Ariana Kariminejad
- B Steinmann
- Bernarda Lozic
- C Dordoni
- C Giunta
- C Nagashima
- Carsten G Bönnemann
- Cecilia Giunta
- CG Bönnemann
- Christine Fauth
- Diana S Johnson
- E Mercuri
- Ebtesam M Abdalla
- F Malfait
- F Michael Pope
- Fleur S van Dijk
- GJ Halko
- Glenda Sobey
- Gudrun Schreiber
- H Okamura
- J Kirschner
- Jacek Pilch
- Jahannaz Dastgir
- James Collins
- Jenny E V Morton
- Johannes Koch
- Johannes Zschocke
- L Van Laer
- M Baumann
- Marianne Rohrbach
- Marius Kraenzlin
- Martina Witsch-Baumgartner
- Matthias Baumann
- ME Kraenzlin
- ML Murray
- MP Wattjes
- Nayana Lahiri
- NC Voermans
- Neeti Ghali
- Nora L Shannon
- P Beighton
- PW Tan
- R Rizzo
- Rebecca C Pollitt
- Sandra Donkervoort
- SP Boudko
- TH Milhorat
- Uschi Lindert
- WI Schievink
- Y Ishikawa
- Y von Kodolitsch
- Y Zou
- Publication venue
- 'Springer Science and Business Media LLC'
- Publication date
- 15/06/2017
- Field of study
PurposeIn 2012 we reported in six individuals a clinical condition almost indistinguishable from PLOD1-kyphoscoliotic Ehlers-Danlos syndrome (PLOD1-kEDS), caused by biallelic mutations in FKBP14, and characterized by progressive kyphoscoliosis, myopathy, and hearing loss in addition to connective tissue abnormalities such as joint hypermobility and hyperelastic skin. FKBP14 is an ER-resident protein belonging to the family of FK506-binding peptidyl-prolyl cis-trans isomerases (PPIases); it catalyzes the folding of type III collagen and interacts with type III, type VI, and type X collagens. Only nine affected individuals have been reported to date.MethodsWe report on a cohort of 17 individuals with FKBP14-kEDS and the follow-up of three previously reported patients, and provide an extensive overview of the disorder and its natural history based on clinical, biochemical, and molecular genetics data.ResultsBased on the frequency of the clinical features of 23 patients from the present and previous cohorts, we define major and minor features of FKBP14-kEDS. We show that myopathy is confirmed by histology and muscle imaging only in some patients, and that hearing impairment is predominantly sensorineural and may not be present in all individuals.ConclusionOur data further support the extensive clinical overlap with PLOD1-kEDS and show that vascular complications are rare manifestations of FKBP14-kEDS
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