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  • 11
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    Online Resource
    Haemodynamic resistance model of monochorionic twin pregnancies complicated by acardiac twinning
    IOP Publishing ; 2004
    In:  Physics in Medicine and Biology Vol. 49, No. 14 ( 2004-07-21), p. N205-N213
    Details
    In: Physics in Medicine and Biology, IOP Publishing, Vol. 49, No. 14 ( 2004-07-21), p. N205-N213
    Type of Medium: Online Resource
    ISSN: 0031-9155 , 1361-6560
    URL: Article
    DOI: 10.1088/0031-9155/49/14/N01
    RVK:
    WA 15000
    Language: Unknown
    Publisher: IOP Publishing
    Publication Date: 2004
    detail.hit.zdb_id: 1473501-5
    SSG: 12
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  • 12
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    Hypothesized pathogenesis of acardius acephalus, acormus, amorphus, anceps, acardiac edema, single umbilical artery, and pump twin risk prediction
    Wiley ; 2022
    In:  Birth Defects Research Vol. 114, No. 5-6 ( 2022-03), p. 149-164
    Details
    In: Birth Defects Research, Wiley, Vol. 114, No. 5-6 ( 2022-03), p. 149-164
    Abstract: Acardiac twinning complicates monochorionic twin pregnancies in ≈2.6%, in which arterioarterial (AA) and venovenous placental anastomoses cause a reverse circulation between prepump and preacardiac embryos and cessation of cardiac function in the preacardiac. Literature suggested four acardiac body morphologies in which select (groups of) organs fail to develop, deteriorate, or become abnormal: acephalus (≈64%, [almost] no head, part of body, legs), amorphus (≈22%, amorphous tissue lump), anceps (≈10%, cranial bones, well‐developed), and acormus (≈4%, head only). We sought to develop hypotheses that could explain acardiac pathogenesis, its progression, and develop methods for clinical testing. Methods We used qualitatively described pathophysiology during development, including twin‐specific AA and Hyrtl's anastomoses, the short umbilical cord syndrome, high capillary permeability, properties of spontaneous aborted embryos, and Pump/Acardiac umbilical venous diameter (UVD) ratios. Results We propose that each body morphology has a specific pathophysiologic pathway. An acephalus acardius may be larger than an anceps, verifiable from UVD ratio measurements. A single umbilical artery develops when one artery, unconnected to the AA, vanishes due to flow reduction by Hyrtl's anastomotic resistance. Acardiac edema may result from acardiac body hypoxemia combined with physiological high fetal capillary permeability, high interstitial compliance and low albumin synthesis. Morphological changes may occur after acardiac onset. Pump twin risk follows from UVD ratios. Conclusion Our suggested outcomes agree reasonably well with reported onset, incidence, and progression of acardiac morphologies. Guidance for clinical prediction and testing requires ultrasound anatomy/circulation study, from the first trimester onward.
    Type of Medium: Online Resource
    ISSN: 2472-1727 , 2472-1727
    URL: Issue
    URL: Article
    DOI: 10.1002/bdr2.v114.5-6
    DOI: 10.1002/bdr2.1976
    Language: English
    Publisher: Wiley
    Publication Date: 2022
    detail.hit.zdb_id: 2884154-2
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  • 13
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    Antenatal Allopurinol Reduces Hippocampal Brain Damage After Acute Birth Asphyxia in Late Gestation Fetal Sheep
    Springer Science and Business Media LLC ; 2014
    In:  Reproductive Sciences Vol. 21, No. 2 ( 2014-02), p. 251-259
    Details
    In: Reproductive Sciences, Springer Science and Business Media LLC, Vol. 21, No. 2 ( 2014-02), p. 251-259
    Type of Medium: Online Resource
    ISSN: 1933-7191 , 1933-7205
    URL: Article
    DOI: 10.1177/1933719113493516
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2014
    detail.hit.zdb_id: 2266096-3
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  • 14
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    Acardiac twin pregnancies part V: Why does an acardiac twin with renal tissue produce polyhydramnios?
    Wiley ; 2021
    In:  Birth Defects Research Vol. 113, No. 6 ( 2021-04), p. 500-510
    Details
    In: Birth Defects Research, Wiley, Vol. 113, No. 6 ( 2021-04), p. 500-510
    Abstract: Acardiac twinning is a complication of monochorionic twin pregnancies. From literature reports, 30 of 41 relatively large acardiac twins with renal tissue produced polyhydramnios within their amniotic compartment. We aim to investigate the underlying mechanisms that cause excess amniotic fluid using an established model of fetal fluid dynamics. Methods We assumed that acardiac onset is before 13 weeks, acardiacs with renal tissue have normal kidney function and produce urine flow from 11 weeks on, and acardiac urine production requires a pressure of half the pump twin's mean arterial pressure. We apply a resistance network with the pump twin's arterio‐venous pressure as source, pump umbilical arteries, placenta, placental arterio‐arterial (AA) anastomoses and acardiac resistances. Acardiac amniotic fluid dynamics excluded acardiac lung fluid secretion, swallowing and the relatively small intramembranous flow. Results In small acardiacs with sufficient urine production, polyhydramnios will occur due to the lack of amniotic fluid resorption. Urine production is dependent upon having sufficient mean arterial pressure, which requires nearly a two‐fold larger resistance within the acardiac as compared to the placental AA resistance. Subphysiologic arterial pressure may result in renal dysgenesis. Conclusion Our findings suggest the potential for prediction of which clinical acardiac cases may or may not develop polyhydramnios based upon noninvasive assessments of renal tissue, blood flow and urine production. This information would be of great value in determining early obstetric interventions as opposed to conservative management. These findings may also contribute to an improved knowledge of the fascinating pathophysiology that surrounds acardiac twinning.
    Type of Medium: Online Resource
    ISSN: 2472-1727 , 2472-1727
    URL: Issue
    URL: Article
    DOI: 10.1002/bdr2.v113.6
    DOI: 10.1002/bdr2.1874
    Language: English
    Publisher: Wiley
    Publication Date: 2021
    detail.hit.zdb_id: 2884154-2
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  • 15
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    Why does second trimester demise of a monochorionic twin not result in acardiac twinning?
    Wiley ; 2021
    In:  Birth Defects Research Vol. 113, No. 15 ( 2021-09), p. 1103-1111
    Details
    In: Birth Defects Research, Wiley, Vol. 113, No. 15 ( 2021-09), p. 1103-1111
    Abstract: We previously explained why acardiac twinning occurs in the first trimester. We raised the question why a sudden demised monochorionic twin beyond the first trimester does not lead to acardiac twinning. We argued that exsanguinated blood from the live twin would strongly increase the demised twins' vascular resistance, preventing its perfusion and acardiac onset. However, our current hypothesis is that perfusion of the demised twin does occur but that it is insufficient for onset of acardiac twinning. Methods We analyzed blood pressures and flows in a vascular resistance model of a monochorionic twin pregnancy where one of the fetuses demised. The resistance model consists of a demised twin with a (former) placenta, a live twin and its placenta, and arterioarterial (AA) and venovenous placental anastomoses. We assumed that only twins with a weight of at least 33% of normal survived the first trimester and that exsanguination of more than 50% of its blood volume is fatal for the live twin. Results At 20 weeks, only AA anastomoses with radii ≲1 mm keep the exsanguinated blood volume below 50%. Then, perfusion of the deceased body with arterial blood from the live fetus is about 5–40 times smaller than when that body was alive. Beyond 20 weeks, this factor is even smaller. At 14 weeks, this factor is at most 2. Conclusion We hypothesize that this small perfusion flow of arterial blood prevents further growth of the deceased body and hence precludes onset of acardiac twinning.
    Type of Medium: Online Resource
    ISSN: 2472-1727 , 2472-1727
    URL: Issue
    URL: Article
    DOI: 10.1002/bdr2.v113.15
    DOI: 10.1002/bdr2.1926
    Language: English
    Publisher: Wiley
    Publication Date: 2021
    detail.hit.zdb_id: 2884154-2
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  • 16
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    Table_1.DOCX
    Frontiers Media SA ; 2021
    In:  Frontiers in Medicine Vol. 8 ( 2021-2-22)
    Details
    In: Frontiers in Medicine, Frontiers Media SA, Vol. 8 ( 2021-2-22)
    Abstract: Perinatal inflammatory stress is strongly associated with adverse pulmonary outcomes after preterm birth. Antenatal infections are an essential perinatal stress factor and contribute to preterm delivery, induction of lung inflammation and injury, pre-disposing preterm infants to bronchopulmonary dysplasia. Considering the polymicrobial nature of antenatal infection, which was reported to result in diverse effects and outcomes in preterm lungs, the aim was to examine the consequences of sequential inflammatory stimuli on endogenous epithelial stem/progenitor cells and vascular maturation, which are crucial drivers of lung development. Therefore, a translational ovine model of antenatal infection/inflammation with consecutive exposures to chronic and acute stimuli was used. Ovine fetuses were exposed intra-amniotically to Ureaplasma parvum 42 days (chronic stimulus) and/or to lipopolysaccharide 2 or 7 days (acute stimulus) prior to preterm delivery at 125 days of gestation. Pulmonary inflammation, endogenous epithelial stem cell populations, vascular modulators and morphology were investigated in preterm lungs. Pre-exposure to UP attenuated neutrophil infiltration in 7d LPS-exposed lungs and prevented reduction of SOX-9 expression and increased SP-B expression, which could indicate protective responses induced by re-exposure. Sequential exposures did not markedly impact stem/progenitors of the proximal airways (P63+ basal cells) compared to single exposure to LPS. In contrast, the alveolar size was increased solely in the UP+7d LPS group. In line, the most pronounced reduction of AEC2 and proliferating cells (Ki67+) was detected in these sequentially UP + 7d LPS-exposed lambs. A similar sensitization effect of UP pre-exposure was reflected by the vessel density and expression of vascular markers VEGFR-2 and Ang-1 that were significantly reduced after UP exposure prior to 2d LPS, when compared to UP and LPS exposure alone. Strikingly, while morphological changes of alveoli and vessels were seen after sequential microbial exposure, improved lung function was observed in UP, 7d LPS, and UP+7d LPS-exposed lambs. In conclusion, although sequential exposures did not markedly further impact epithelial stem/progenitor cell populations, re-exposure to an inflammatory stimulus resulted in disturbed alveolarization and abnormal pulmonary vascular development. Whether these negative effects on lung development can be rescued by the potentially protective responses observed, should be examined at later time points.
    Type of Medium: Online Resource
    ISSN: 2296-858X
    URL: Article
    URL: Article
    DOI: 10.3389/fmed.2021.614239
    DOI: 10.3389/fmed.2021.614239.s001
    Language: Unknown
    Publisher: Frontiers Media SA
    Publication Date: 2021
    detail.hit.zdb_id: 2775999-4
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  • 17
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    Ribosomal protein gene RPL9 variants can differentially impair ribosome function and cellular metabolism
    Oxford University Press (OUP) ; 2020
    In:  Nucleic Acids Research Vol. 48, No. 2 ( 2020-01-24), p. 770-787
    Details
    In: Nucleic Acids Research, Oxford University Press (OUP), Vol. 48, No. 2 ( 2020-01-24), p. 770-787
    Abstract: Variants in ribosomal protein (RP) genes drive Diamond-Blackfan anemia (DBA), a bone marrow failure syndrome that can also predispose individuals to cancer. Inherited and sporadic RP gene variants are also linked to a variety of phenotypes, including malignancy, in individuals with no anemia. Here we report an individual diagnosed with DBA carrying a variant in the 5′UTR of RPL9 (uL6). Additionally, we report two individuals from a family with multiple cancer incidences carrying a RPL9 missense variant. Analysis of cells from these individuals reveals that despite the variants both driving pre-rRNA processing defects and 80S monosome reduction, the downstream effects are remarkably different. Cells carrying the 5′UTR variant stabilize TP53 and impair the growth and differentiation of erythroid cells. In contrast, ribosomes incorporating the missense variant erroneously read through UAG and UGA stop codons of mRNAs. Metabolic profiles of cells carrying the 5′UTR variant reveal an increased metabolism of amino acids and a switch from glycolysis to gluconeogenesis while those of cells carrying the missense variant reveal a depletion of nucleotide pools. These findings indicate that variants in the same RP gene can drive similar ribosome biogenesis defects yet still have markedly different downstream consequences and clinical impacts.
    Type of Medium: Online Resource
    ISSN: 0305-1048 , 1362-4962
    URL: Article
    DOI: 10.1093/nar/gkz1042
    RVK:
    WA 15000
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2020
    detail.hit.zdb_id: 1472175-2
    SSG: 12
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  • 18
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    Celiac disease is overrepresented in patients with constipation
    Fundacao Sociedade Brasileira de Pediatria ; 2012
    In:  Jornal de Pediatria Vol. , No. ( 2012-3-20)
    Details
    In: Jornal de Pediatria, Fundacao Sociedade Brasileira de Pediatria, Vol. , No. ( 2012-3-20)
    Type of Medium: Online Resource
    ISSN: 0021-7557
    URL: Article
    DOI: 10.2223/JPED.2155
    Language: English
    Publisher: Fundacao Sociedade Brasileira de Pediatria
    Publication Date: 2012
    detail.hit.zdb_id: 2105628-6
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  • 19
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    High Rate of Chronic Villitis in Placentas of Pregnancies Complicated by Influenza A/H1N1 Infection
    Hindawi Limited ; 2014
    In:  Infectious Diseases in Obstetrics and Gynecology Vol. 2014 ( 2014), p. 1-5
    Details
    In: Infectious Diseases in Obstetrics and Gynecology, Hindawi Limited, Vol. 2014 ( 2014), p. 1-5
    Abstract: Introduction . Pandemic influenza A/H1N1 infection during pregnancy has a negative impact on several aspects of pregnancy outcome. As yet, no elucidating mechanism has been revealed for these effects. We investigated whether placentas of pregnancies complicated by 2009 influenza A/H1N1 infection demonstrated an increased rate of chronic villitis and whether this villitis was caused by influenza virus. Methods . We performed a cohort study on 145 pregnant outpatients during the 2009-2010 influenza A H1N1 pandemic. The placentas of patients with influenza infection were examined for histologic signs of chronic villitis. In case of villitis, polymerase chain reaction (PCR) on influenza virus was performed on placental tissue. Results . 29 patients had influenza infection. Placentas of 15 of these patients were collected and examined. In 7 cases (47%) chronic villitis was detected. Placental weight and birth weight of the neonates did not differ between cases with and without chronic villitis. In all cases PCR was negative for influenza. Conclusion . In our series, chronic villitis was present in a high proportion of placentas of pregnancies complicated by 2009 influenza A/H1N1 infection. We could not demonstrate the presence of influenza virus in placental tissue.
    Type of Medium: Online Resource
    ISSN: 1064-7449 , 1098-0997
    URL: Article
    DOI: 10.1155/2014/768380
    Language: English
    Publisher: Hindawi Limited
    Publication Date: 2014
    detail.hit.zdb_id: 2001451-X
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  • 20
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    The value of postmortem computed tomography in paediatric natural cause of death: a Dutch observational study
    Springer Science and Business Media LLC ; 2017
    In:  Pediatric Radiology Vol. 47, No. 11 ( 2017-10), p. 1514-1522
    Details
    In: Pediatric Radiology, Springer Science and Business Media LLC, Vol. 47, No. 11 ( 2017-10), p. 1514-1522
    Abstract: Postmortem CT is a relatively new field of interest within paediatric radiology. This paper focusses on its value in cases of unexpected natural death. Objective We report on an observational Dutch study regarding the value of postmortem CT in children with an assumed natural unexpected death because postmortem CT is part of the Dutch NODO (additional investigations of cause of death) procedure. Materials and methods We included consecutive children who fulfilled criteria for the NODO procedure and were therefore referred to one of the centres for the procedure. Postmortem CT was performed in all cases and skeletal survey was performed in all children ages 〈 5 years. The cause of death was defined in a consensus meeting. Results We included a total of 54 children (30 boys, median age 1.1 years, and 24 girls, median age 0.8 years). A definitive cause of death was established in 38 cases. In 7 cases the cause of death could be identified on postmortem CT. In 7 cases imaging findings were clinically relevant but did not lead to a cause of death. In the remaining 40 cases postmortem CT did not add to the diagnostic workup. Conclusion Our study shows that in a group of children who unexpectedly died of an assumed natural cause of death and in whom a cause of death was found at autopsy, postmortem CT detected the cause of death in a minority of cases (12.9%). In the majority of cases (74.1%) postmortem CT did not add value in diagnosing the cause of death.
    Type of Medium: Online Resource
    ISSN: 0301-0449 , 1432-1998
    URL: Article
    DOI: 10.1007/s00247-017-3911-0
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2017
    detail.hit.zdb_id: 1463007-2
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