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  • 1
    Online Resource
    Online Resource
    Cerebral Venous Thrombosis Mimicking a Discrete Brain Mass: A Case Report and Literature Review
    Hindawi Limited ; 2018
    In:  Case Reports in Rheumatology Vol. 2018 ( 2018), p. 1-5
    Details
    In: Case Reports in Rheumatology, Hindawi Limited, Vol. 2018 ( 2018), p. 1-5
    Abstract: The differential diagnosis for a focal brain lesion in a patient with systemic lupus erythematosus (SLE) is broad and includes infection, malignancy, and vascular and inflammatory etiologies. One rarely considered vascular pathology is cerebral venous thrombosis (CVT), which is often associated with a delay in diagnosis because of variable presentation and rare incidence. We present the case of a young woman with a new discrete brain lesion that appeared in the context of highly active SLE and was ultimately diagnosed with a CVT. We provide a literature review for diagnosis and management of cerebral venous thrombosis, a potentially serious complication of untreated systemic lupus erythematosus.
    Type of Medium: Online Resource
    ISSN: 2090-6889 , 2090-6897
    URL: Article
    DOI: 10.1155/2018/5862912
    Language: English
    Publisher: Hindawi Limited
    Publication Date: 2018
    detail.hit.zdb_id: 2666708-3
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  • 2
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    Proton pump inhibitors in systemic sclerosis: a reappraisal to optimise treatment of gastro-oesophageal reflux disease
    Elsevier BV ; 2022
    In:  The Lancet Rheumatology Vol. 4, No. 11 ( 2022-11), p. e795-e803
    Details
    In: The Lancet Rheumatology, Elsevier BV, Vol. 4, No. 11 ( 2022-11), p. e795-e803
    Type of Medium: Online Resource
    ISSN: 2665-9913
    URL: Article
    DOI: 10.1016/S2665-9913(22)00183-7
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2022
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  • 3
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    Risk of Digital Vascular Events in Scleroderma Patients Who Have Both Anticentromere and Anti–Interferon‐Inducible Protein 16 Antibodies
    Wiley ; 2017
    In:  Arthritis Care & Research Vol. 69, No. 6 ( 2017-06), p. 922-926
    Details
    In: Arthritis Care & Research, Wiley, Vol. 69, No. 6 ( 2017-06), p. 922-926
    Abstract: To evaluate whether scleroderma patients who are double‐positive for anti–interferon‐inducible protein 16 (anti–IFI‐16) antibodies and anticentromere (anti‐CENP) antibodies are at increased risk for significant digital vascular events relative to patients positive for anti‐CENP antibodies alone. Methods Sera from 165 scleroderma patients who tested positive for anti‐CENP antibodies upon clinical evaluation were reassayed for both anti‐CENP and anti–IFI‐16 antibodies using enzyme‐linked immunosorbent assay testing. Patients who were positive for anti‐CENP antibodies alone were then compared to patients who were double‐positive for both anti–IFI‐16 and anti‐CENP antibodies. The association between a history of significant digital vascular events (digital pits, ischemic digital ulcers, and/or gangrene) and double‐positive antibody status was examined using chi‐square tests. After completion of univariate analysis, multivariable analyses were done to adjust for clinically relevant covariates. Results Of the 165 anti–CENP antibody positive patients, 21 (12.7%) also had anti–IFI‐16 antibodies. Patients who were double‐positive for anti‐CENP and anti‐IFI‐16 antibodies were more likely to have had digital pits, ischemic digital ulcers, and/or gangrene ( P  = 0.03). After adjustment for clinically relevant covariates (age, cutaneous subtype, disease duration, and smoking), double‐positive patients remained at significantly higher odds of having severe Raynaud's phenomenon (odds ratio 3.5 [95% confidence interval 1.1–11.1]; P  = 0.03). Conclusion Scleroderma patients who are double‐positive for antibodies recognizing CENP and IFI‐16 are significantly more likely to have significant digital vascular events during the course of their disease. This study provides further evidence that anti‐CENP and anti–IFI‐16 antibodies are disease biomarkers that may be used for risk stratification of vascular events in scleroderma.
    Type of Medium: Online Resource
    ISSN: 2151-464X , 2151-4658
    URL: Issue
    URL: Article
    DOI: 10.1002/acr.v69.6
    DOI: 10.1002/acr.22978
    RVK:
    XA 30325
    Language: English
    Publisher: Wiley
    Publication Date: 2017
    detail.hit.zdb_id: 2016713-1
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  • 4
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    Gastrointestinal Tract Considerations Part I
    Elsevier BV ; 2023
    In:  Rheumatic Disease Clinics of North America Vol. 49, No. 2 ( 2023-05), p. 295-318
    Details
    In: Rheumatic Disease Clinics of North America, Elsevier BV, Vol. 49, No. 2 ( 2023-05), p. 295-318
    Type of Medium: Online Resource
    ISSN: 0889-857X
    URL: Article
    DOI: 10.1016/j.rdc.2023.01.006
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2023
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  • 5
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    Gastrointestinal Tract Considerations: Part II
    Elsevier BV ; 2023
    In:  Rheumatic Disease Clinics of North America Vol. 49, No. 2 ( 2023-05), p. 319-336
    Details
    In: Rheumatic Disease Clinics of North America, Elsevier BV, Vol. 49, No. 2 ( 2023-05), p. 319-336
    Type of Medium: Online Resource
    ISSN: 0889-857X
    URL: Article
    DOI: 10.1016/j.rdc.2023.01.007
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2023
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  • 6
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    Anti– RNPC ‐3 (U11/U12) Antibodies in Systemic Sclerosis in Patients With Moderate‐to‐Severe Gastrointestinal Dysmotility
    Wiley ; 2019
    In:  Arthritis Care & Research Vol. 71, No. 9 ( 2019-09), p. 1164-1170
    Details
    In: Arthritis Care & Research, Wiley, Vol. 71, No. 9 ( 2019-09), p. 1164-1170
    Abstract: To examine the association of anti– RNPC ‐3 antibodies in patients with systemic sclerosis (scleroderma or SS c) with selected gastrointestinal ( GI ) tract complications. Methods Sera from patients with SS c with or without severe GI dysfunction (total parenteral nutrition dependence) from the Johns Hopkins Scleroderma Center were screened for anti– RNPC ‐3 antibodies. We then examined anti– RNPC ‐3–positive cases and negative SS c controls from the University of Pittsburgh and the University of Pittsburgh Medical Center (UPMC) scleroderma cohort to confirm our findings and to examine whether specific GI features were associated with anti– RNPC ‐3 antibodies. Results In the discovery cohort, patients with SS c with severe GI dysfunction (n = 37) and without GI dysfunction (n = 38) were screened for anti– RNPC ‐3 antibodies. The former were more likely to have anti– RNPC ‐3 antibodies (14% versus 3%; P = 0.11). In the Pittsburgh cohort, moderate‐to‐severe GI dysfunction (Medsger GI score ≥2) was present in 36% of anti– RNPC ‐3–positive patients versus 15% of anti– RNPC ‐3–negative patients ( P ≤ 0.01). Anti– RNPC ‐3–positive patients were more likely to be male (31% versus 15%; P = 0.04), African American (18% versus 6%; P = 0.02), have esophageal dysmotility (93% versus 62%; P 〈 0.01), and interstitial lung disease ( ILD ) (77% versus 35%; P 〈 0.01). After adjusting for relevant covariates and potential confounders, moderate‐to‐severe GI disease was associated with anti– RNPC ‐3 antibodies (odds ratio [ OR ] 3.8 [95% confidence interval (95% CI ) 1.0–14.3]), and ILD trended toward significance ( OR 2.8 [95% CI 1.0–8.2]). Conclusion Patients with SS c and anti– RNPC ‐3 antibodies are more likely to be male and African American and to have moderate‐to‐severe GI disease and ILD . Further studies on larger patient cohorts may be helpful in further defining subsets of patients with SS c at risk for severe GI involvement.
    Type of Medium: Online Resource
    ISSN: 2151-464X , 2151-4658
    URL: Issue
    URL: Article
    DOI: 10.1002/acr.v71.9
    DOI: 10.1002/acr.23763
    RVK:
    XA 30325
    Language: English
    Publisher: Wiley
    Publication Date: 2019
    detail.hit.zdb_id: 2016713-1
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  • 7
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    Associations Between Patterns of Esophageal Dysmotility and Extra‐Intestinal Features in Patients With Systemic Sclerosis
    Wiley ; 2023
    In:  Arthritis Care & Research Vol. 75, No. 8 ( 2023-08), p. 1715-1724
    Details
    In: Arthritis Care & Research, Wiley, Vol. 75, No. 8 ( 2023-08), p. 1715-1724
    Abstract: The gastrointestinal tract is commonly involved in patients with systemic sclerosis (SSc) with varied manifestations. As our understanding of SSc gastrointestinal disease pathogenesis and risk stratification is limited, we sought to investigate whether patterns of esophageal dysfunction associate with specific clinical phenotypes in SSc. Methods Patients enrolled in the Johns Hopkins Scleroderma Center Research Registry who completed high‐resolution esophageal manometry (HREM) studies as part of their clinical care between 2011 and 2020 were identified. Associations between esophageal abnormalities on HREM (absent contractility [AC], ineffective esophageal motility [IEM] , hypotensive lower esophageal sphincter [hypoLES]) and patient demographic information, clinical characteristics, and autoantibody profiles were examined. Results Ninety‐five patients with SSc had HREM data. Sixty‐five patients (68.4%) had AC (37 patients with only AC, 28 patients with AC and a hypoLES), 9 patients (9.5%) had IEM, and 11 patients (11.6%) had normal studies. AC was significantly associated with diffuse cutaneous disease (38.5% versus 10.0%; P   〈  0.01), more severe Raynaud's phenomenon, including digital pits, ulcers, or gangrene (56.9% versus 30.0%; P  = 0.02), and reduced median diffusing capacity of lung for carbon monoxide (50.6% versus 72.2%; P  = 0.03). AC was observed in most of the patients who died (13 of 14; P  = 0.06). These findings were not seen in patients with IEM. Conclusion Among patients with SSc, AC is associated with a significantly more severe clinical phenotype. IEM may associate with a milder phenotype. Further studies are needed to evaluate AC, IEM, and their clinical impact relative to the timing of other end‐organ complications in SSc.
    Type of Medium: Online Resource
    ISSN: 2151-464X , 2151-4658
    URL: Issue
    URL: Article
    DOI: 10.1002/acr.v75.8
    DOI: 10.1002/acr.25080
    RVK:
    XA 30325
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2016713-1
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  • 8
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    Determining the Risk Factors and Clinical Features Associated With Severe Gastrointestinal Dysmotility in Systemic Sclerosis
    Wiley ; 2018
    In:  Arthritis Care & Research Vol. 70, No. 9 ( 2018-09), p. 1385-1392
    Details
    In: Arthritis Care & Research, Wiley, Vol. 70, No. 9 ( 2018-09), p. 1385-1392
    Abstract: A subset of patients with systemic sclerosis ( SS c) develop severe gastrointestinal ( GI ) dysmotility. We sought to determine predictors of severe SS c GI dysmotility and to identify distinct features associated with this phenotype. Methods Patients with SS c who required supplemental nutrition (enteral or parenteral tube feeding) were compared to SS c patients with mild GI symptoms in a cross‐sectional analysis. The association between severe GI dysmotility and clinical and serologic features was examined using logistic regression. Baseline data were examined to determine predictors of developing severe GI dysfunction using Cox regression. Results SS c patients with severe GI dysmotility (n = 66) were more likely than those patients with mild GI symptoms (n = 1,736) to be male (odds ratio [ OR ] 2.47 [95% confidence interval (95% CI ) 1.34–4.56]; P = 0.004), and to have myopathy ( OR 5.53 [95% CI 2.82–10.82]; P 〈 0.001), and sicca symptoms ( OR 2.40 [95% CI 1.30–4.42]; P = 0.005), even after adjustment for potential confounders. Baseline features that were associated with the future development of severe GI dysfunction included male sex (hazard ratio [ HR ] 2.99 [95% CI 1.53–5.84]; P = 0.001) and myopathy ( HR 5.08 [95% CI 2.21–11.67]; P 〈 0.001). Conclusion Distinct clinical features are present in SS c patients who are at risk of developing severe GI dysmotility. This finding is not only important clinically but also suggests that a unique pathologic process is at work in these patients.
    Type of Medium: Online Resource
    ISSN: 2151-464X , 2151-4658
    URL: Issue
    URL: Article
    DOI: 10.1002/acr.2018.70.issue-9
    DOI: 10.1002/acr.23479
    RVK:
    XA 30325
    Language: English
    Publisher: Wiley
    Publication Date: 2018
    detail.hit.zdb_id: 2016713-1
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  • 9
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    Heterogeneity of primary and secondary peristalsis in systemic sclerosis: A new model of “scleroderma esophagus”
    Wiley ; 2022
    In:  Neurogastroenterology & Motility Vol. 34, No. 7 ( 2022-07)
    Details
    In: Neurogastroenterology & Motility, Wiley, Vol. 34, No. 7 ( 2022-07)
    Abstract: Although esophageal dysmotility is common in systemic sclerosis (SSc)/scleroderma, little is known regarding the pathophysiology of motor abnormalities driving reflux severity and dysphagia. This study aimed to assess primary and secondary peristalsis in SSc using a comprehensive esophageal motility assessment applying high‐resolution manometry (HRM) and functional luminal imaging probe (FLIP) Panometry. Methods A total of 32 patients with scleroderma (28 female; ages 38–77; 20 limited SSc, 12 diffuse SSc) completed FLIP Panometry and HRM. Secondary peristalsis, i.e., contractile responses (CR), was classified on FLIP Panometry by pattern of contractility as normal (NCR), borderline (BCR), impaired/disordered (IDCR), or absent (ACR). Primary peristalsis on HRM was assessed according to the Chicago classification. Results The manometric diagnoses were 56% ( n  = 18) absent contractility, 22% ( n  = 7) ineffective esophageal motility (IEM), and 22% ( n  = 7) normal motility. Secondary peristalsis (CRs) included 38% ( n  = 12) ACR, 38% ( n  = 12) IDCR, 19% ( n =  6) BCR, and 15% ( n =  5) NCR. The median (IQR) esophagogastric junction (EGJ) distensibility index (DI) was 5.8 mm 2 /mmHg (4.8–10.1) mm 2 /mmHg; EGJ‐DI was 〉 8.0 mm 2 /mmHg in 31%, and 〉 2.0 mm 2 /mmHg in 100% of patients. Among 18 patients with absent contractility on HRM, 11 had ACR, 5 had IDCR, and 2 had BCR. Among 7 patients with IEM, 1 had ACR, 5 had IDCR, and 1 NCR. All of the patients with normal peristalsis had NCR or BCR. Conclusions This was the first study assessing combined HRM and FLIP Panometry in a cohort of SSc patients, which demonstrated heterogeneity in primary and secondary peristalsis. This complementary approach facilitates characterizing esophageal function in SSc, although future study to examine clinical outcomes remains necessary.
    Type of Medium: Online Resource
    ISSN: 1350-1925 , 1365-2982
    URL: Issue
    URL: Article
    DOI: 10.1111/nmo.v34.7
    DOI: 10.1111/nmo.14284
    Language: English
    Publisher: Wiley
    Publication Date: 2022
    detail.hit.zdb_id: 2008278-2
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  • 10
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    Anti‐Gephyrin antibodies: A novel specificity in patients with systemic sclerosis and lower bowel dysfunction
    Wiley ; 2023
    In:  Arthritis & Rheumatology
    Details
    In: Arthritis & Rheumatology, Wiley
    Abstract: Autoantibodies are clinically useful in phenotyping patients with systemic sclerosis (SSc). GI function is regulated by the enteric nervous system (ENS) and commonly impaired in SSc suggesting that the SSc autoimmune response may target ENS antigens. We sought to identify novel anti‐ENS autoantibodies with an aim to clinically phenotype SSc GI dysfunction. Methods Serum from a SSc patient with GI dysfunction but without defined SSc‐associated autoantibodies was used for autoantibody discovery. Immunoprecipitations performed with murine myenteric plexus lysates were on‐bead digested and autoantigens were identified by mass spectrometry. Prevalence was determined and clinical features associated with novel autoantibodies were evaluated in a SSc cohort using regression analyses. Expression of gephyrin in human GI tract tissue was examined by immunohistochemistry. Results We identified gephyrin as a novel SSc autoantigen. Anti‐gephyrin antibodies were present in 9% of SSc patients (16/188) and absent in healthy controls (0/46). Anti‐gephyrin antibody‐positive patients had higher constipation scores [1.00 vs. 0.50;p=0.02] and were more likely to have severe constipation and severe distention/bloating [46% vs. 15%;p=0.005; 54% vs. 25%;p=0.023, respectively] . Anti‐gephyrin antibody levels were significantly higher among patients with severe constipation [0.04 vs. 0.00;p=0.001] and severe distention and bloating [0.03 vs. 0.004;p=0.010] . Severe constipation was associated with anti‐gephyrin antibodies even in the adjusted model. Importantly, gephyrin was expressed in the ENS, which regulates gut motility. Conclusion Gephyrin is a novel ENS autoantigen that is expressed in human myenteric ganglia. Anti‐gephyrin autoantibodies are associated with the presence and severity of constipation in SSc patients.
    Type of Medium: Online Resource
    ISSN: 2326-5191 , 2326-5205
    URL: Article
    DOI: 10.1002/art.42667
    RVK:
    XA 10000
    RVK:
    XA 30325
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2754614-7
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