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  • 1
    Online Resource
    Online Resource
    Anti-FHL1 autoantibodies in juvenile myositis are associated with anti-Ro52 autoantibodies but not with severe disease features
    Oxford University Press (OUP) ; 2023
    In:  Rheumatology Vol. 62, No. SI2 ( 2023-02-23), p. SI226-SI234
    Details
    In: Rheumatology, Oxford University Press (OUP), Vol. 62, No. SI2 ( 2023-02-23), p. SI226-SI234
    Abstract: Four-and-a-half LIM domains 1 (FHL1) is a muscle-specific protein. Autoantibodies against FHL1 were recently discovered in adults with idiopathic inflammatory myopathies (IIMs) and were found to be associated with clinical features and outcomes indicative of increased disease severity. Anti-FHL1 autoantibodies have not been described in children. Here, the prevalence and clinical features associated with anti-FHL1 autoantibodies were examined in a large North American cohort of juvenile patients with IIM. Methods Sera from 338 juvenile IIM patients and 91 juvenile healthy controls were screened for anti-FHL1 autoantibodies by ELISA. Clinical characteristics and HLA alleles of those with and without anti-FHL1 autoantibodies were compared among those with juvenile IIM. Results Anti-FHL1 autoantibodies were present in 10.9% of juvenile IIM patients and 1.1% of controls. The frequency of anti-FHL1 autoantibodies among clinical and serologic subgroups did not differ. A higher percentage of Asian patients had anti-FHL1 autoantibodies (11% vs 0.7%; P = 0.002). Myositis-associated autoantibodies (MAAs) [odds ratio (OR) 2.09 (CI 1.03, 4.32)], anti-Ro52 autoantibodies specifically [OR 4.17 (CI 1.83, 9.37)] and V-sign rash [OR 2.59 (CI 1.22, 5.40)] were associated with anti-FHL1 autoantibodies. There were no differences in other features or markers of disease severity. No HLA associations with anti-FHL1 autoantibodies in Caucasian myositis patients were identified. Conclusion Anti-FHL1 autoantibodies are present in ∼11% of juvenile IIM patients and commonly co-occur with MAAs, including anti-Ro52 autoantibodies. In contrast to adult IIM, anti-FHL1 autoantibodies in juvenile myositis are associated with V-sign rash but not with other distinctive clinical features or worse outcomes.
    Type of Medium: Online Resource
    ISSN: 1462-0324 , 1462-0332
    URL: Article
    DOI: 10.1093/rheumatology/keac428
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2023
    detail.hit.zdb_id: 1474143-X
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  • 2
    Online Resource
    Online Resource
    Role of Suppressive Oral Antibiotics in Orthopedic Hardware Infections for Those Not Undergoing Two-Stage Replacement Surgery
    Oxford University Press (OUP) ; 2016
    In:  Open Forum Infectious Diseases Vol. 3, No. 4 ( 2016-10-01)
    Details
    In: Open Forum Infectious Diseases, Oxford University Press (OUP), Vol. 3, No. 4 ( 2016-10-01)
    Abstract: Background.  The use of suppressive antibiotics in treatment of orthopedic hardware infections (OHIs), including spinal hardware infections, prosthetic joint infections, and infections of internal fixation devices, is controversial. Methods.  Over a 4-year period at 2 academic medical centers, patients with OHI who were treated with debridement and retention of hardware components, with single-stage exchange, or without surgery were studied to determine whether use of oral antibiotics for at least 6 months after diagnosis impacts successful treatment of the infection at 1 year after diagnosis. Results.  Of 89 patients in the study, 42 (47.2%) were free of clinical infection 1 year after initial diagnosis. Suppressive antibiotics used for at least 6 months after diagnosis was not associated with being free of clinical infection (adjusted odds ratio [aOR], 5.29; 95% confidence interval [CI] , .74–37.80), but being on suppressive antibiotics at least 3 months after diagnosis was associated with being free of clinical infection (OR, 3.50; 95% CI, 1.30–9.43). Causative organisms impacted the likelihood of success; patients with methicillin-resistant Staphylococcus aureus as well as with Gram-negative rods were both less likely to have achieved clinical success at 1 year after surgery (aOR = 0.018, 95% CI = .0017–.19 and aOR = 0.20, 95% CI = .039–.99, respectively). Conclusions.  Oral suppressive antibiotic therapy in treatment of OHI with retention of hardware for 3 months, but not 6 months, postdiagnosis increases the likelihood of treatment success. The organisms implicated in the infection directly impact the likelihood of treatment success.
    Type of Medium: Online Resource
    ISSN: 2328-8957
    URL: Article
    DOI: 10.1093/ofid/ofw176
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2016
    detail.hit.zdb_id: 2757767-3
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  • 3
    Online Resource
    Online Resource
    Genome-wide screening reveals the genetic basis of mammalian embryonic eye development
    Springer Science and Business Media LLC ; 2023
    In:  BMC Biology Vol. 21, No. 1 ( 2023-02-03)
    Details
    In: BMC Biology, Springer Science and Business Media LLC, Vol. 21, No. 1 ( 2023-02-03)
    Abstract: Microphthalmia, anophthalmia, and coloboma (MAC) spectrum disease encompasses a group of eye malformations which play a role in childhood visual impairment. Although the predominant cause of eye malformations is known to be heritable in nature, with 80% of cases displaying loss-of-function mutations in the ocular developmental genes OTX2 or SOX2, the genetic abnormalities underlying the remaining cases of MAC are incompletely understood. This study intended to identify the novel genes and pathways required for early eye development. Additionally, pathways involved in eye formation during embryogenesis are also incompletely understood. This study aims to identify the novel genes and pathways required for early eye development through systematic forward screening of the mammalian genome. Results Query of the International Mouse Phenotyping Consortium (IMPC) database (data release 17.0, August 01, 2022) identified 74 unique knockout lines (genes) with genetically associated eye defects in mouse embryos. The vast majority of eye abnormalities were small or absent eyes, findings most relevant to MAC spectrum disease in humans. A literature search showed that 27 of the 74 lines had previously published knockout mouse models, of which only 15 had ocular defects identified in the original publications. These 12 previously published gene knockouts with no reported ocular abnormalities and the 47 unpublished knockouts with ocular abnormalities identified by the IMPC represent 59 genes not previously associated with early eye development in mice. Of these 59, we identified 19 genes with a reported human eye phenotype. Overall, mining of the IMPC data yielded 40 previously unimplicated genes linked to mammalian eye development. Bioinformatic analysis showed that several of the IMPC genes colocalized to several protein anabolic and pluripotency pathways in early eye development. Of note, our analysis suggests that the serine-glycine pathway producing glycine, a mitochondrial one-carbon donator to folate one-carbon metabolism (FOCM), is essential for eye formation. Conclusions Using genome-wide phenotype screening of single-gene knockout mouse lines, STRING analysis, and bioinformatic methods, this study identified genes heretofore unassociated with MAC phenotypes providing models to research novel molecular and cellular mechanisms involved in eye development. These findings have the potential to hasten the diagnosis and treatment of this congenital blinding disease.
    Type of Medium: Online Resource
    ISSN: 1741-7007
    URL: Article
    DOI: 10.1186/s12915-022-01475-0
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2023
    detail.hit.zdb_id: 2133020-7
    SSG: 12
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  • 4
    Online Resource
    Online Resource
    Pilot-scale expanded assessment of inorganic and organic tapwater exposures and predicted effects in Puerto Rico, USA
    Elsevier BV ; 2021
    In:  Science of The Total Environment Vol. 788 ( 2021-09), p. 147721-
    Details
    In: Science of The Total Environment, Elsevier BV, Vol. 788 ( 2021-09), p. 147721-
    Type of Medium: Online Resource
    ISSN: 0048-9697
    URL: Article
    DOI: 10.1016/j.scitotenv.2021.147721
    RVK:
    AR 10100
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2021
    detail.hit.zdb_id: 1498726-0
    detail.hit.zdb_id: 121506-1
    SSG: 12
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  • 5
    Online Resource
    Online Resource
    Cellular extrusion bioprinting improves kidney organoid reproducibility and conformation
    Springer Science and Business Media LLC ; 2021
    In:  Nature Materials Vol. 20, No. 2 ( 2021-02), p. 260-271
    Details
    In: Nature Materials, Springer Science and Business Media LLC, Vol. 20, No. 2 ( 2021-02), p. 260-271
    Type of Medium: Online Resource
    ISSN: 1476-1122 , 1476-4660
    URL: Article
    DOI: 10.1038/s41563-020-00853-9
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2021
    detail.hit.zdb_id: 2088679-2
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  • 6
    Online Resource
    Online Resource
    A potential diagnostic pitfall for hobnail variant of papillary thyroid carcinoma
    Wiley ; 2020
    In:  Histopathology Vol. 76, No. 5 ( 2020-04), p. 707-713
    Details
    In: Histopathology, Wiley, Vol. 76, No. 5 ( 2020-04), p. 707-713
    Abstract: Hobnail variant of papillary thyroid carcinoma (PTC) is an aggressive PTC subtype characterised by a hobnail cytomorphology. However, some classic PTC have a ‘hobnail‐like’ cytomorphology associated with thick, hyalinised, variably oedematous fibrovascular cores that appears to be a form of ischaemic/degenerative atypia. Methods and results We studied three cohorts to compare the histopathological characteristics and clinical outcome of ‘hobnail‐like’ classic PTC and true hobnail variant of PTC: cohort 1, PTC consecutively resected between 2016 and 2017 (to assess frequency of ‘hobnail‐like’ cytomorphology); cohort 2, 20 ‘hobnail‐like’ classic PTC resected between 2005 and 2007 (to assess clinical outcome); and cohort 3, seven true hobnail variant of PTC. A ‘hobnail‐like’ cytomorphology was identified in 16% of consecutively resected PTC. Compared with true hobnail variant, ‘hobnail‐like’ classic PTC occurred in younger patients (mean age 40 years versus 68 years, P   〈  0.001), were smaller tumours (mean tumour size 2.1 cm versus 4.4 cm, P   〈  0.001), had a lower rate of gross extrathyroidal extension (0% versus 71%, P   〈  0.001), had a lower proliferative rate (≥3 mitoses per 10 high‐power fields seen in 0% versus 71%, P   〈  0.001; Ki67 index ≥5% in 0% versus 86%, P   〈  0.001), a lower rate of secondary pathogenic mutations (for cases with molecular data, 0% versus 100%, P  = 0.0061) and improved survival (for cases with sufficient clinical outcome data, 10‐year disease‐free survival of 93% versus 0%, P  = 0.0016). Conclusion Classic PTC can show ischaemic/degenerative atypia that mimics the hobnail cytomorphology of true hobnail variant; however, these tumours lack aggressive histopathological features and pursue an indolent clinical course.
    Type of Medium: Online Resource
    ISSN: 0309-0167 , 1365-2559
    URL: Issue
    URL: Article
    DOI: 10.1111/his.v76.5
    DOI: 10.1111/his.14042
    RVK:
    XA 10000
    Language: English
    Publisher: Wiley
    Publication Date: 2020
    detail.hit.zdb_id: 2006447-0
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  • 7
    Online Resource
    Online Resource
    Applications of Immunohistochemistry to Endocrine Pathology
    Ovid Technologies (Wolters Kluwer Health) ; 2018
    In:  Advances in Anatomic Pathology Vol. 25, No. 6 ( 2018-11), p. 413-429
    Details
    In: Advances in Anatomic Pathology, Ovid Technologies (Wolters Kluwer Health), Vol. 25, No. 6 ( 2018-11), p. 413-429
    Abstract: The role of immunohistochemistry (IHC) in endocrine pathology is similar to that in other organ systems in that it can aid in the subclassification of tumors within an organ, confirm site of primary in metastatic disease, provide prognostic information, identify underlying genetic alterations, and predict response to treatment. Although most endocrine tumors do not require IHC to render a diagnosis, there are certain scenarios in which IHC can be extremely helpful. For example, in thyroid, IHC can be used to support tumor dedifferentiation, in the adrenal it can aid in the diagnosis of low-grade adrenocortical carcinomas, and in paragangliomas it can help identify tumors arising as part of an inherited tumor syndrome. This review will focus on the applications of IHC in tumors of the thyroid, parathyroids, adrenals, and paraganglia in adults.
    Type of Medium: Online Resource
    ISSN: 1072-4109
    URL: Article
    DOI: 10.1097/PAP.0000000000000209
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2018
    detail.hit.zdb_id: 2062617-4
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  • 8
    Online Resource
    Online Resource
    Tall Cell Variant of Papillary Thyroid Carcinoma: Impact of Change in WHO Definition and Molecular Analysis
    Springer Science and Business Media LLC ; 2019
    In:  Endocrine Pathology Vol. 30, No. 1 ( 2019-3), p. 43-48
    Details
    In: Endocrine Pathology, Springer Science and Business Media LLC, Vol. 30, No. 1 ( 2019-3), p. 43-48
    Type of Medium: Online Resource
    ISSN: 1046-3976 , 1559-0097
    URL: Article
    DOI: 10.1007/s12022-018-9561-4
    Language: English
    Publisher: Springer Science and Business Media LLC
    Publication Date: 2019
    detail.hit.zdb_id: 2091856-2
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  • 9
    Online Resource
    Online Resource
    Pulmonary Adenocarcinomas of Low Malignant Potential : Proposed Criteria to Expand the Spectrum Beyond Adenocarcinoma In Situ and Minimally Invasive Adenocarcinoma
    Ovid Technologies (Wolters Kluwer Health) ; 2021
    In:  American Journal of Surgical Pathology Vol. 45, No. 4 ( 2021-04), p. 567-576
    Details
    In: American Journal of Surgical Pathology, Ovid Technologies (Wolters Kluwer Health), Vol. 45, No. 4 ( 2021-04), p. 567-576
    Abstract: Lung cancer screening has improved mortality among high-risk smokers but has coincidentally detected a fraction of nonprogressive adenocarcinoma historically classified as bronchoalveolar carcinoma (BAC). In the National Lung Screening Trial (NLST) the majority of BAC—comprising 29% of computed tomography–detected stage I lung adenocarcinoma—were considered overdiagnosis after extended follow-up comparison with the control arm. In the current classification, adenocarcinoma in situ and minimally invasive adenocarcinoma have replaced BAC but together comprise only ∼5% of stage I lung adenocarcinoma. Lepidic and subsets of papillary and acinar adenocarcinoma also infrequently recur. We, therefore, propose criteria for low malignant potential (LMP) adenocarcinoma among nonmucinous adenocarcinoma measuring ≤3 cm in total, exhibiting ≥15% lepidic growth, and lacking nonpredominant high-grade patterns (≥10% cribriform, ≥5% micropapillary, ≥5% solid), 〉 1 mitosis per 2 mm 2 , angiolymphatic or visceral pleural invasion, spread through air spaces or necrosis. We tested these criteria in a multi-institutional cohort of 328 invasive stage I (eighth edition) and in situ adenocarcinomas and observed 16% LMP and 7% adenocarcinoma in situ/minimally invasive adenocarcinoma which together (23%) approximated the frequency of overdiagnosed stage I BAC in the NLST. The LMP group had 100% disease-specific survival. The proposed LMP criteria, incorporating multiple histologic parameters, may be a clinically useful “low-grade” prognostic group. Validation of these criteria in additional retrospective cohorts and prospective screen-detected cohorts should be considered.
    Type of Medium: Online Resource
    ISSN: 0147-5185
    URL: Article
    DOI: 10.1097/PAS.0000000000001618
    RVK:
    XA 10000
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2021
    detail.hit.zdb_id: 2029143-7
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  • 10
    Online Resource
    Online Resource
    Comparison of soil phosphorus index systems for grassland in the cross‐border region of Ireland #
    Wiley ; 2022
    In:  Journal of Plant Nutrition and Soil Science Vol. 185, No. 1 ( 2022-02), p. 110-119
    Details
    In: Journal of Plant Nutrition and Soil Science, Wiley, Vol. 185, No. 1 ( 2022-02), p. 110-119
    Abstract: The use of soil phosphorus (P) tests and index systems provides a guide for agronomic nutrient requirements and is frequently also used to estimate risk of P losses to watercourses. Use of soil testing and management based on the results thereof is mandated in some regions. Several P extraction methods are available which evaluate different P pools and are designed for particular soil types. Further to this, index systems categorising specific ranges of plant‐available P, differ. Hence, translation between different tests and index systems is not straightforward. In cross‐border regions, where hydrologic basins encompass more than one political jurisdiction, different tests and rules are implemented in adjacent lands. This can create disparities in land management, confusion as to what legislation applies and obscures the impacts of best management practices at catchment scale. Aims The aim of this research was to compare the Morgan's and Olsen soil tests used to quantify plant‐available P and the respective index systems, in a border region of the Republic of Ireland (ROI)–Northern Ireland (NI). Methods Olsen, Morgans and water extractable P (WEP) were evaluated ( n = 1,038). Statistical analysis was conducted to derive conversion equations to translate between the statutory test methods and comparison of the respective index categories was performed. Results The conversion equations compared favourably with previous attempts. A stronger relationship was observed between Morgan P and WEP ( R 2 = 0.60) than between Olsen P and WEP ( R 2 = 0.45) (including pH and site as interaction factors). The ROI index system was found to indicate lower levels of plant available P in the soil compared to the NI system, for the same soils. Conclusions The differences in categorisation of P availability using either index system creates differences in fertiliser recommendations and also perceived aquatic risks even within small cross‐border catchments. This study points to a wider implication for international cross‐border catchments, suggesting that evaluation of the relationships between adjacent national soil index systems is required to achieve harmonised management of shared waterbodies. Neither index system is preferred, but a combination of soil P tests incorporating both agronomic and environmentally oriented analyses may have utility in future decision support tools.
    Type of Medium: Online Resource
    ISSN: 1436-8730 , 1522-2624
    URL: Issue
    URL: Article
    DOI: 10.1002/jpln.v185.1
    DOI: 10.1002/jpln.202100194
    RVK:
    RA 5430
    Language: English
    Publisher: Wiley
    Publication Date: 2022
    detail.hit.zdb_id: 1481142-X
    detail.hit.zdb_id: 1470765-2
    detail.hit.zdb_id: 200063-5
    SSG: 12
    SSG: 13
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