260 research outputs found
Pyruvate Dehydrogenase Complex Deficiency: Updating the Clinical, Metabolic and Mutational Landscapes in a Cohort of Portuguese Patients
- Author
- Publication venue
- 'Springer Science and Business Media LLC'
- Publication date
- 01/01/2020
- Field of study
Background: The pyruvate dehydrogenase complex (PDC) catalyzes the irreversible decarboxylation of pyruvate into acetyl-CoA. PDC deficiency can be caused by alterations in any of the genes encoding its several subunits. The resulting phenotype, though very heterogeneous, mainly affects the central nervous system. The aim of this study is to describe and discuss the clinical, biochemical and genotypic information from thirteen PDC deficient patients, thus seeking to establish possible genotype-phenotype correlations.
Results: The mutational spectrum showed that seven patients carry mutations in the PDHA1 gene encoding the E1α subunit, five patients carry mutations in the PDHX gene encoding the E3 binding protein, and the remaining patient carries mutations in the DLD gene encoding the E3 subunit. These data corroborate earlier reports describing PDHA1 mutations as the predominant cause of PDC deficiency but also reveal a notable prevalence of PDHX mutations among Portuguese patients, most of them carrying what seems to be a private mutation (p.R284X). The biochemical analyses revealed high lactate and pyruvate plasma levels whereas the lactate/pyruvate ratio was below 16; enzymatic activities, when compared to control values, indicated to be independent from the genotype and ranged from 8.5% to 30%, the latter being considered a cut-off value for primary PDC deficiency. Concerning the clinical features, all patients displayed psychomotor retardation/developmental delay, the severity of which seems to correlate with the type and localization of the mutation carried by the patient. The therapeutic options essentially include the administration of a ketogenic diet and supplementation with thiamine, although arginine aspartate intake revealed to be beneficial in some patients. Moreover, in silico analysis of the missense mutations present in this PDC deficient population allowed to envisage the molecular mechanism underlying these pathogenic variants.
Conclusion: The identification of the disease-causing mutations, together with the functional and structural characterization of the mutant protein variants, allow to obtain an insight on the severity of the clinical phenotype and the selection of the most appropriate therapy.info:eu-repo/semantics/publishedVersio
Effects of orofacial myofunctional therapy on masticatory function in individuals submitted to orthognathic surgery: a randomized trial
- Author
- Aléssio CV
- Berretin-Felix G
- Castroflorio T
- Daniela Galvão de Almeida Prado
- Di Palma E
- Disselhorst-Klug C
- Engelen L
- Felicio CM
- Felício CM
- Frongia G
- Gadotti IC
- Gavião MB
- Giédre Berretin-Felix
- Hanzelka T
- Isadora Ferraz Teixeira
- Iwase M
- Kasai RC
- Ko EW
- Kobayashi T
- Marcela Polizel
- Maria Beatriz Duarte Gavião
- Mariana da Rocha Salles Bueno
- Pereira AC
- Pereira JB
- Piancino MG
- Raquel Rodrigues Rosa
- Renata Resina Migliorucci
- Shimazaki K
- Takeshita N
- Trawitsky LV
- Trawitzki LV
- Trench JA
- Trulsson M
- Ueki K
- Van den Braber W
- Youssef RE
- Publication venue
- 'FapUNIFESP (SciELO)'
- Publication date
- Field of study
Functional study of DAND5 variant in patients with Congenital Heart Disease and laterality defects
- Author
- A Cecchetto
- A Kawasumi
- A Luca De
- A Megarbane
- AC Araujo
- AF Ramsdell
- B Gelb
- B Mohapatra
- B Wang
- B Wang
- B Wang
- C Wren
- D Srivastava
- Duarte Saraiva Martins
- E Goldmuntz
- E Roessler
- E Roessler
- F Yuan
- Fernando Cristo
- H Shiratori
- H Shiratori
- I Shiraishi
- J Wang
- JA Belo
- JA Goodship
- JB Huang
- JM Icardo
- JM Inacio
- José A. Belo
- José M. Inácio
- José Maio
- K Koefoed
- K Kosaki
- L Ma
- M Levin
- Patrícia Mendes
- R Kosaki
- RN Bamford
- Rui Anjos
- S Marques
- S Nowotschin
- S Oki
- S Richards
- Salomé de Almeida
- SM Channabasappa
- T Nakamura
- TA Andersen
- U Gaio
- X Deng
- Publication venue
- 'Springer Science and Business Media LLC'
- Publication date
- Field of study
Compressed representation of a partially defined integer function over multiple arguments
- Author
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdelalim AA
- Abdullin S
- Abdulsalam A
- Acosta D
- Acosta MV
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- Adams MR
- Adams T
- Adiguzel A
- Adler V
- Adzic P
- Agram J-L
- Aguilar-Benitez M
- 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
- Alderweireldt S
- Alexander J
- Aliev T
- Alimena J
- Almeida N
- Alverson G
- Alves GA
- Amapane N
- Amsler C
- Anagnostou G
- Anastassov A
- Anderson J
- Andrea J
- Andreev V
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- Andreev Y
- Andrews W
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- Antonelli L
- Antunovic Z
- Apanasevich L
- Apollinari G
- Appelt E
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- Arcidiacono R
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- Asghar MI
- Asin I
- Askew A
- Aslanoglou X
- Assran Y
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- Publication venue
- Publication date
- 01/01/2013
- Field of study
In OLAP (OnLine Analitical Processing) data are analysed in an n-dimensional cube. The cube may be represented as a partially defined function over n arguments. Considering that often the function is not defined everywhere, we ask: is there a known way of representing the function or the points in which it is defined, in a more compact manner than the trivial one
Difficulties of familes in caring for children and adolescents with mental disorders: an integrative review
- Author
- Athay MM
- Bee H
- Borba LO
- Cardoso L
- Damião EBC
- Duarte CS
- Geraghty K
- Gerkensmeyer JE
- Goés FGB
- Kilmer RP
- Knock J
- Lany Leide de Castro Rocha Campelo
- Lecavalier L
- Lin LY
- Luciana de Almeida Colvero
- Machineski GG
- McDonald TP
- Mendes KDS
- Moreno V
- Okewole A
- Oruche UM
- Sant’Ana MM
- Sarah Maria Esequiel Costa
- Schein S
- Schein S
- Schutsky K
- Sung V
- Sá DGF
- Telles HPRS
- Vicente JB
- Publication venue
- 'FapUNIFESP (SciELO)'
- Publication date
- Field of study
Association between serum selenium level and conversion of bacteriological tests during antituberculosis treatment
- Author
- Afrânio Lineu Kritski
- Antônio Ruffino-Netto
- Ciftci TU
- Corrêa CR
- Daniela Maria de Paula Ramalho
- Eliene Denites Duarte Mesquita
- Ewing JA
- Frisancho AR
- Gibson RS
- Hedi Marinho de Melo Guedes de Oliveira
- Heymsfield SB
- Karina Neves Delogo
- Kassu A
- Lönnroth K
- Lönnroth K
- Macallan DC
- Martha Maria de Oliveira
- Mehta JB
- Milena Lima de Moraes
- Mugusi FM
- Papathakis PC
- Paulo César de Almeida
- Peresi E
- Pryscila Fernandes Campino Miranda
- Rachel Ann Hauser-Davis
- Reddy YN
- Rehm J
- Reinaldo Calixto Campos
- Schwenk A
- Seyedrezazadeh E
- Shor-Posner G
- van Lettow M
- van Lettow M
- Villamor E
- Wallis RS
- Wiid I
- Wilson D
- Wilson D
- Publication venue
- 'FapUNIFESP (SciELO)'
- Publication date
- Field of study
Multi-messenger observations of a binary neutron star merger
- Author
- Aab A
- Aartsen MG
- Abbott BP
- Abbott R
- Abbott TD
- Abbott TMC
- Abdalla H
- Abe F
- Abeysekara AU
- Abramo LR
- Abramowski A
- Abreu P
- Acernese F
- Acero F
- Ackermann M
- Ackley K
- Ackley K
- Adams C
- Adams J
- Adams SM
- Adams T
- Addesso P
- Adhikari RX
- Adya VB
- Affeldt C
- Afrough M
- Agarwal B
- Agathos M
- Agatsuma K
- Aggarwal N
- Aglietta M
- Agliozzo C
- Agudo I
- Aguiar OD
- Aguilar JA
- Aharonian F
- Ahlers M
- Ahrens M
- Aiello L
- Ain A
- Ajith P
- Akras S
- Al Samarai I
- Albert A
- Albert A
- Albuquerque IFM
- Albury JM
- Alcaniz JS
- Alexander KD
- Alfaro R
- Alighieri S Di Serego
- Allam S
- Allekotte I
- Allen B
- Allen G
- Allison J
- Allison JR
- Allocca A
- Almela A
- Altin PA
- Altmann D
- Alvarez C
- Alvarez JD
- Alvarez M Dovale
- Alvarez-Muiz J
- Amati L
- Amato A
- An T
- Ananyeva A
- Anastasi GA
- Anchordoqui L
- Andeen K
- Anderson JP
- Anderson SB
- Anderson T
- Anderson WG
- Andrada B
- Andre M
- Andreoni I
- Andreoni I
- Andringa S
- Angelova SV
- Anghinolfi M
- Angner EO
- Angus CR
- Annis J
- Ansseau I
- Antier S
- Anton G
- Antonelli LA
- Antonelli LA
- Anupama GC
- Aoki K
- Aoki W
- Appert S
- Aptekar RL
- Arai K
- Arakawa M
- Aramo C
- Araya MC
- Arcavi I
- Arceo R
- Ardid M
- Areeda JS
- Aresu G
- Argan A
- Argelles C
- Arnaud N
- Array LWA Long Wavelength
- Array MWA Murchison Widefield
- Arrieta M
- Arsene N
- Arteaga-Velzquez JC
- Artola R
- Arun KG
- Asakura Y
- Asaoka Y
- Ascenzi S
- Ashall C
- Ashley MCB
- Ashton G
- Asorey H
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- Astone P
- Atallah DV
- ATLAS
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- Balasubramanian A
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- Ballardin G
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- Botticella MT
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- Zhu Z
- Ziegler A
- Zimmerman AB
- Zimmermann B
- Zimnukhov DS
- Ziolkowski M
- Zoll M
- Zong Z
- Zorn J
- Zornoza JD
- Zou CL
- Zuccarello F
- Zucker ME
- Zuniga J
- Zweizig J
- Zywucka N
- Publication venue
- 'American Astronomical Society'
- Publication date
- 01/01/2017
- Field of study
On 2017 August 17 a binary neutron star coalescence candidate (later designated GW170817) with merger time 12:41:04 UTC was observed through gravitational waves by the Advanced LIGO and Advanced Virgo detectors. The Fermi Gamma-ray Burst Monitor independently detected a gamma-ray burst (GRB 170817A) with a time delay of ~1.7 s with respect to the merger time. From the gravitational-wave signal, the source was initially localized to a sky region of 31 deg2 at a luminosity distance of 40+8-8 Mpc and with component masses consistent with neutron stars. The component masses were later measured to be in the range 0.86 to 2.26 Mo. An extensive observing campaign was launched across the electromagnetic spectrum leading to the discovery of a bright optical transient (SSS17a, now with the IAU identification of AT 2017gfo) in NGC 4993 (at ~40 Mpc) less than 11 hours after the merger by the One- Meter, Two Hemisphere (1M2H) team using the 1 m Swope Telescope. The optical transient was independently detected by multiple teams within an hour. Subsequent observations targeted the object and its environment. Early ultraviolet observations revealed a blue transient that faded within 48 hours. Optical and infrared observations showed a redward evolution over ~10 days. Following early non-detections, X-ray and radio emission were discovered at the transient’s position ~9 and ~16 days, respectively, after the merger. Both the X-ray and radio emission likely arise from a physical process that is distinct from the one that generates the UV/optical/near-infrared emission. No ultra-high-energy gamma-rays and no neutrino candidates consistent with the source were found in follow-up searches. These observations support the hypothesis that GW170817 was produced by the merger of two neutron stars in NGC4993 followed by a short gamma-ray burst (GRB 170817A) and a kilonova/macronova powered by the radioactive decay of r-process nuclei synthesized in the ejecta
Search for the pair production of light top squarks in the e(+/-)mu(-/+) final state in proton-proton collisions at root s=13 TeV
- Author
- Aarrestad TK
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdullah WAT Wan
- Abdullin S
- Abdulsalam A
- Abercrombie D
- Abu Zeid S
- Ackert A
- Acosta D
- Acosta G
- Adam W
- Adams MR
- Adams T
- Adzic P
- Afanasiev S
- Agapitos A
- Agaras MN
- Aggleton R
- Agram J-L
- Ahmad A
- Ahmad M
- Ahmad M
- Ahmed I
- Ahuja S
- Akbiyik M
- Akchurin N
- Akgun B
- Al-Bataineh A
- Albajar C
- Albergo S
- Albert A
- Albrow M
- Alcaraz Maestre J
- Aleksandrov A
- Alexakhin V
- Alexander J
- Alhusseini M
- Ali MAB Md
- Alimena J
- Allen B
- Almond J
- Alvarez Fernandez A
- Alvarez JD Ruiz
- Alverson G
- Alves FL
- Alves GA
- Alyari M
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- Ambrogi F
- Amendola C
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- Amsler C
- Anagnostou G
- Anampa K Hurtado
- Anderson D
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- Publication venue
- Publication date
- 01/01/2019
- Field of study
A search for the production of a pair of top squarks at the LHC is presented. This search targets a region of parameter space where the kinematics of top squark pair production and top quark pair production are very similar, because of the mass difference between the top squark and the neutralino being close to the top quark mass. The search is performed with 35.9 fb(-1) of proton-proton collisions at a centre-of-mass energy of root s = 13 TeV, collected by the CMS detector in 2016, using events containing one electron-muon pair with opposite charge. The search is based on a precise estimate of the top quark pair background, and the use of the M-T2 variable, which combines the transverse mass of each lepton and the missing transverse momentum. No excess of events is found over the standard model predictions. Exclusion limits are placed at 95% confidence level on the production of top squarks up to masses of 208 GeV for models with a mass difference between the top squark and the lightest neutralino close to that of the top quark.Peer reviewe
Search for Higgs boson pair production in the gamma gamma b(b)over-bar final state in pp collisions at root s=13 TeV
- Author
- Aarrestad TK
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdullah WAT Wan
- Abdullin S
- Abdulsalam A
- Abercrombie D
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- Publication venue
- Publication date
- 01/01/2018
- Field of study
A search is presented for the production of a pair of Higgs bosons, where one decays into two photons and the other one into a bottom quark-antiquark pair. The analysis is performed using proton-proton collision data at root s = 13 TeV recorded in 2016 by the CMS detector at the LHC, corresponding to an integrated luminosity of 35.9 fb(-1) . The results are in agreement with standard model (SM) predictions. In a search for resonant production, upper limits are set on the cross section for new spin-0 or spin-2 particles. For the SM-like nonresonant production hypothesis, the data exclude a product of cross section and branching fraction larger than 2.0 fb at 95% confidence level (CL), corresponding to about 24 times the SM prediction. Values of the effective Higgs boson self-coupling K X are constrained to be within the range -11 < K-lambda < 17 at 95% CL, assuming all other Higgs boson couplings are at their SM value. The constraints on K-lambda, are the most restrictive to date. (C) 2018 The Author(s). Published by Elsevier B.V.Peer reviewe
Combination of CMS searches for heavy resonances decaying to pairs of bosons or leptons
- Author
- Aarrestad TK
- Abbaneo D
- Abbiendi G
- Abbrescia M
- Abdullah WAT Wan
- Abdullin S
- Abercrombie D
- Acharya H
- Acosta D
- Acosta JG
- Adam W
- Adams MR
- Adams T
- Adan D Perez
- Adzic P
- Afanasiev S
- Agapitos A
- Aggleton R
- Agram J-L
- Aguilar-Benitez M
- Ahmad A
- Ahmad M
- Ahmad W Haj
- Ahuja S
- Akbiyik M
- Akchurin N
- Akgun B
- Albajar C
- Albergo S
- Albert A
- Albrow M
- Alcaraz Maestre J
- Aleksandrov A
- Alexander J
- Alhusseini M
- Alimena J
- Allen B
- Almond J
- Alvarez Fernandez A
- Alverson G
- Alves FL
- Alves GA
- Alyari M
- Amapane N
- Ambrogi F
- Amendola C
- Amin N
- Amsler C
- Anagnostou G
- Anampa K Hurtado
- Anderson D
- Andrea J
- Andreev V
- Andreev Yu
- Andrews MB
- Androsov K
- Angioni GL Pinna
- Antchev G
- Antunovic Z
- Apanasevich L
- Apollinari G
- Apresyan A
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- Araujo M
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- Arcidiacono R
- Arenton MW
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- Asavapibhop B
- Asawatangtrakuldee C
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- Assran Y
- Atakisi IO
- Ather MW
- Attikis A
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- Aushev T
- Autermann C
- Auzinger G
- Avati V
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- Ayala E
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- Azhgirey I
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- Azzurri P
- Baarmand MM
- Babaev A
- Babounikau I
- Bacchetta N
- Bachiller I
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- Backhaus M
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- Baechler J
- Bagliesi G
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- Yagil A
- Yalvac M
- Yang UK
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- Yetkin EA
- Yohay R
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- Yoo J
- Yoon I
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- Yumiceva F
- Yusli MN
- Zabi A
- Zaganidis N
- Zahid S
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- Zalewski P
- Zanetti A
- Zanetti M
- Zarubin A
- Zarucki M
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- Zenaiev O
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- Publication venue
- Elsevier
- Publication date
- 01/01/2019
- Field of study
CMS Collaboration: et al.A statistical combination of searches for heavy resonances decaying to pairs of bosons or leptons is presented. The data correspond to an integrated luminosity of 35.9 fb collected during 2016 by the CMS experiment at the LHC in proton-proton collisions at a center-of-mass energy of 13 TeV. The data are found to be consistent with expectations from the standard model background. Exclusion limits are set in the context of models of spin-1 heavy vector triplets and of spin-2 bulk gravitons. For mass-degenerate W′ and Z′ resonances that predominantly couple to the standard model gauge bosons, the mass exclusion at 95% confidence level of heavy vector bosons is extended to 4.5 TeV as compared to 3.8 TeV determined from the best individual channel. This excluded mass increases to 5.0 TeV if the resonances couple predominantly to fermions.Individuals have received support from the Marie-Curie program and the European Research Council and Horizon 2020 Grant, contract Nos. 675440, 752730, and 765710 (European Union); the Programa Estatal de Fomento de la Investigación Científica y Técnica de Excelencia María de Maeztu, grant MDM-2015-0509 and the Programa Severo Ochoa del Principado de Asturias
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