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Journal ArticleDOI: 10.1093/RHEUMATOLOGY/KEAA441

Ultrasound-guided core needle biopsy compared with open biopsy: a new diagnostic approach to salivary gland enlargement in Sjögren's syndrome?

02 Mar 2021-Rheumatology (Oxford Academic)-Vol. 60, Iss: 3, pp 1282-1290
Abstract: Objective Persistent (≥2 months) major salivary gland enlargement in primary SS (pSS) patients is a well-known sign of possible involvement by B cell lymphoma. The study aimed to evaluate the diagnostic accuracy and safety of US-guided core needle biopsy (CNB) of major salivary glands compared with open surgical biopsy. Methods Prospective pSS patients (cases) with clinically persistent salivary gland enlargement underwent US-guided CNB and were compared with retrospective pSS patients (controls) submitted to open surgical biopsy. The features analysed were pre-biopsy clinical and laboratory findings, adequacy of the material for histology and diagnostic-rendered and biopsy-related complications (reported by the patient with a questionnaire and clinically verified). Results Thirteen cases underwent US-guided CNB: in nine, biopsy was performed on the parotid gland and in four it was performed on the submandibular gland. Sufficient material was obtained for pathological diagnosis in all samples. The final diagnoses were 5 (38.5%) B cell lymphoma, 1 (7.7%) lymphoepithelial sialadenitis, 4 (30.7%) other sialadenitis (granulomatous consistent with sarcoidosis, IgG4-related disease, chronic sclerosing, diffuse chronic) and 3/13 (23.1%) miscellaneous lesions. Thirteen controls underwent open surgical biopsy of the parotid. In one, inadequate material was obtained, while in 12 (92.3%) the pathologic diagnoses were 4 (33.3%) B cell lymphoma, 2 (16.7%) lymphoepithelial sialadenitis, 4 (33.3%) uncertain lymphoproliferative lesions and 2 (16.7%) miscellaneous lesions. Six cases (46.1%) reported six transient complications and 12/13 (92.3%) controls had 2 persistent and 14 transient complications. Conclusion US-guided CNB represents a novel, clinically relevant and safe approach for the management of pSS patients with parotid or submandibular persistent enlargement.

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Topics: Open biopsy (59%), Sialadenitis (59%), Parotid gland (57%) ... show more
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5 results found


Open accessJournal ArticleDOI: 10.3389/FMED.2020.602354
Abstract: Objective: Salivary gland ultrasound (SGUS) is emerging as a valid tool in the management of primary Sjogren's syndrome (pSS). This study aimed to investigate whether pSS patients with normal-appearing or pathological SGUS findings showed different clinical, laboratory, and pathologic pSS-related features, and to compare the results by using two different SGUS scores. Methods: Consecutive pSS patients, according to the ACR-EULAR classification criteria, were evaluated. Salivary glands were scored using the early 1992 score by De Vita et al. and the latest 2019 OMERACT score, both being semiquantitative 0-3 scoring systems focused on ultrasonographic parenchymal inhomogeneity (grades 0 and 1, normal-appearing; grades 2 and 3, pathological). The patients were then divided into two groups: "SGUS normal-appearing" if all the salivary glands had normal-appearing parenchyma (grade 0 or 1), or "SGUS pathological" if the grade was 2 or 3 in at least one salivary gland. The associations between SGUS and pSS-related clinical, laboratory, and pathological features were then investigated in the two groups. Results: One hundred pSS patients were evaluated, the mean age (±SD) was 60.9 ± 12.0 years, and mean disease duration was 11.7 ± 7.2 years. Twenty-nine out of 100 (29%) patients were in the "SGUS normal-appearing" group and 71/100 (71%) were in the "SGUS pathological" group. A normal-appearing SGUS was significantly associated with the absence of anti-La/SSB antibodies (p < 0.001) and normal unstimulated salivary flow rate (p = 0.02) by both univariate and multivariate analyses. By univariate analysis, a normal-appearing SGUS was significantly associated also with the absence of rheumatoid factor (p = 0.002) and of serum monoclonal component (p = 0.003), ESSDAI < 5 (p = 0.03), and with a negative lip biopsy (p = 0.029). No associations were found with other items, including anti-Ro/SSA (p = 0.145), Schirmer's test (p = 0.793), ESSPRI (p = 0.47), and demographic data. No differences in these results were observed by using the two SGUS scoring systems. Conclusion: The SGUS allowed the identification of different phenotypes of pSS, and different SGUS scores focused on salivary gland inhomogeneity may be effective to this end.

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1 Citations


Open accessJournal ArticleDOI: 10.1136/ANNRHEUMDIS-2021-EULAR.1775
Abstract: Fatigue is among the top complaints of patients with systemic lupus erythematosus (SLE), but only in part associated with SLE disease activity. Physical activity can help to reduce fatigue and should therefore be recommended to SLE patients. Vice versa, fatigue may arguably lead to reduced physical activity.To investigate the extent of physical activity and the perception of fatigue and sleep quality in patients with SLE.Starting in February 2019, SLE patients were invited to participate in a cross-sectional survey study of fatigue and physical exercise during their routine outpatient clinic visits. Participants filled out a ten-page paper questionnaire focused on physical activity. To evaluate fatigue, we primarily used a 10 cm visual analogue scale (0-100 mm, with 100 meaning most fatigued), but also the FACIT fatigue score (range 0-52). Sleep quality was estimated using grades from 1 (excellent) to 6 (extremely poor).93 SLE patients took part in the study. All patients fulfilled the European League Against Rheumatism/ American College of Rheumatology (EULAR/ACR) 2019 classification criteria for SLE. 91% of the patients were female. Their mean (SD) age was 45.5 (14.3) years and their mean disease duration 12.1 (9.4) years. The mean BMI was 25.2 (5.6). Of all patients, 7.5% had a diagnosis of (secondary) fibromyalgia. The mean fatigue VAS was 32 (27) mm and the mean FACIT fatigue score 35.7 (10.3). As expected, fatigue by VAS and FACIT was correlated (Spearman r=-0.61, p<0.0001). The mean SLEDAI was 1 (1) with a range of 0 to 6. Median glucocorticoid doses were 2 mg prednisolone equivalent, with a range from 0 to 10 mg.Out of 66 patients in payed jobs, 64 (97%) reported details on their working space. One person (2%) worked in a predominanty standing position, 37 (58%) worked in essentially sedentary jobs and 26 (40%) were in positions where they were mildly physically active in part. The mean fatigue VAS was 31 (24) mm for patients with partly active jobs and 27 (30) mm for those in sedentary jobs. Sleep was graded 2.9 (0.9) by those with active and 3.1 (1.3) by those with sedentary jobs.Half of the patients (51%) reported more than one physical recreational activity. 44 (47%) were walking and for five persons (5%) this was the only form of activity. Cycling was reported by 19 patients (20%), 18 of whom also practiced other activities. For transport, 52 (56%) in part chose active modes, such as walking and cycling. Patients who reported any of the above activities showed a mean fatigue VAS of 28 (25) mm, compared to 36 (28) mm in the patient group without a reported activity. Sleep quality was very similar: 3.1 (1.2) and 3.2 (1.1) for more active and more passive patients, respectively.65 (70%) patients regularly practiced sports. Of these, 39 (60%) practiced one kind of sport, 15 (23%) two, 7 (11%) three, and 2 (3%) each four and five kinds of sports. Fatigue VAS of patients practicing sports was 27 (25) mm versus 43 (28) in those who did not (p=0.0075). Sleep quality was 2.9 (1.1) in the sports cohort and 3.5 (1.1) in the no-sports cohort (p=0.0244).A majority of SLE patients in remission or low to moderate disease activity regularly practiced sports, and those doing so reported lesser fatigue and better sleep quality. The absolute values on the fatigue VAS were in a moderate range that made fatigue as the main cause of not performing sports rather unlikely for most patients.Helena Wohland: None declared, Nicolai Leuchten Speakers bureau: AbbVie, Janssen, Novartis, Roche, UCB, Consultant of: AbbVie, Janssen, Novartis, Roche, Martin Aringer Speakers bureau: AbbVie, Astra Zeneca, BMS, Boehringer Ingelheim, Chugai, Gilead, GSK, HEXAL, Lilly, MSD, Novartis, Pfizer, Roche, Sanofi, UCB, Consultant of: AbbVie, Astra Zeneca, BMS, Boehringer Ingelheim, GSK, Lilly, MSD, Roche, Sanofi, UCB

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Open accessJournal ArticleDOI: 10.1177/1759720X211040696
Abstract: Lung involvement in systemic lupus erythematosus (SLE) and primary Sjogren's syndrome (pSS) has extensively been outlined with a multiplicity of different manifestations. In SLE, the most frequent finding is pleural effusion, while in pSS, airway disease and parenchymal disorders prevail. In both cases, there is an increased risk of pre-capillary and post-capillary pulmonary arterial hypertension (PAH) and pulmonary venous thromboembolism (VTE). The risk of VTE is in part due to an increased thrombophilic status secondary to systemic inflammation or to the well-established association with antiphospholipid antibody syndrome (APS). The lung can also be the site of an organ-specific complication due to the aberrant pathologic immune-hyperactivation as occurs in the development of lymphoma or amyloidosis in pSS. Respiratory infections are a major issue to be addressed when approaching the differential diagnosis, and their exclusion is required to safely start an immunosuppressive therapy. Treatment strategy is mainly based on glucocorticoids (GCs) and immunosuppressants, with a variable response according to the primary pathologic process. Anticoagulation is recommended in case of VTE and multi-targeted treatment regimens including different drugs are the mainstay for PAH management. Antibiotics and respiratory physiotherapy can be considered relevant complement therapeutic measures. In this article, we reviewed lung manifestations in SLE and pSS with the aim to provide a comprehensive overview of their diagnosis and management to physicians taking care of patients with connective tissue diseases.

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Topics: Pleural effusion (50%)

Open accessJournal ArticleDOI: 10.1186/S13075-021-02432-Y
Yanying Liu1, Fei Yang1, Xiying Chi1, Yuxin Zhang1  +4 moreInstitutions (1)
Abstract: The growing utilization of needle biopsy has challenged the current pathology consensus of IgG4-related disease (IgG4-RD). The aims of this study were to identify the histological characteristics of needle biopsy and surgical specimens and evaluate the ability of needle biopsy in histological diagnosis of IgG4-RD. Biopsies from patients who were referred to as IgG4-RD by the 2019 ACR/EULAR IgG4-RD classification criteria in Peking University People’s Hospital from 2012 to 2019 were re-evaluated. Typical histological features and diagnostic categories were compared between needle biopsy and surgical biopsy. In total, 69 patients met the 2019 ACR/EULAR classification criteria and 72 biopsies of them were re-evaluated. All cases showed lymphoplasmacytic infiltrate, while storiform fibrosis and obliterative phlebitis were only present in 35 (48.6%) and 23 (31.9%) specimens, respectively. Storiform fibrosis was more likely to be seen in retroperitoneum lesion (P = 0.033). Surgical biopsy showed significantly higher IgG4+ plasma cells/high-power field (IgG4/HPF) count (P 10 (P 100, which was significantly less than 15/23 (65.2%) of surgical ones (P < 0.01). Needle biopsy shows an inferiority in detecting IgG4/HPF count but not in IgG4/IgG ratio, storiform fibrosis, and obliterative phlebitis. Compared with surgical samples, needle biopsy is less likely to obtain a histological diagnosis of IgG4-RD. A different IgG4/HPF threshold for needle biopsy of the salivary glands may be considered.

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Topics: Biopsy (64%)

Open accessJournal ArticleDOI: 10.3390/JCM10061171
Abstract: Primary Sjogren's syndrome (pSS) is a chronic and heterogeneous disorder characterized by a wide spectrum of glandular and extra-glandular features. The hallmark of pSS is considered to be the immune-mediated involvement of the exocrine glands and B-cell hyperactivation. This leads pSS patients to an increased risk of developing lymphoproliferative diseases, and persistent (>2 months) major salivary gland enlargement is a well-known clinical sign of possible involvement by B cell lymphoma. Better stratification of the patients may improve understanding of the mechanism underlying the risk of lymphoproliferative disorder. Here, we summarize the role of different imaging techniques and a bioptic approach in pSS patients, focusing mainly on the role of salivary gland ultrasonography (SGUS) and a US-guided core needle biopsy (Us-guided CNB) as diagnostic and prognostic tools in pSS patients with persistent parotid swelling.

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Topics: Major Salivary Gland (50%)
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30 results found


Open accessJournal ArticleDOI: 10.1089/THY.2015.0020
01 Nov 2009-Thyroid
Abstract: Background: Thyroid nodules are a common clinical problem, and differentiated thyroid cancer is becoming increasingly prevalent. Since the American Thyroid Association's (ATA's) guidelines for the management of these disorders were revised in 2009, significant scientific advances have occurred in the field. The aim of these guidelines is to inform clinicians, patients, researchers, and health policy makers on published evidence relating to the diagnosis and management of thyroid nodules and differentiated thyroid cancer. Methods: The specific clinical questions addressed in these guidelines were based on prior versions of the guidelines, stakeholder input, and input of task force members. Task force panel members were educated on knowledge synthesis methods, including electronic database searching, review and selection of relevant citations, and critical appraisal of selected studies. Published English language articles on adults were eligible for inclusion. The American College of Physicians Guideline Gr...

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Topics: Thyroid neoplasm (73%), Thyroid nodules (72%), Thyroid cancer (69%) ... show more

8,016 Citations


Journal ArticleDOI: 10.1136/ANNRHEUMDIS-2016-210571
Abstract: Objectives To develop and validate an international set of classification criteria for primary Sjogren's syndrome (SS) using guidelines from the American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR) These criteria were developed for use in individuals with signs and/or symptoms suggestive of SS Methods We assigned preliminary importance weights to a consensus list of candidate criteria items, using multi-criteria decision analysis We tested and adapted the resulting draft criteria using existing cohort data on primary SS cases and non-SS controls, with case/non-case status derived from expert clinical judgement We then validated the performance of the classification criteria in a separate cohort of patients Results The final classification criteria are based on the weighted sum of five items: anti-SSA/Ro antibody positivity and focal lymphocytic sialadenitis with a focus score of ≥1 foci/4 mm2, each scoring 3; an abnormal Ocular Staining Score of ≥5 (or van Bijsterveld score of ≥4), a Schirmer's test result of ≤5 mm/5 min and an unstimulated salivary flow rate of ≤01 mL/min, each scoring 1 Individuals with signs and/or symptoms suggestive of SS who have a total score of ≥4 for the above items meet the criteria for primary SS Sensitivity and specificity against clinician-expert—derived case/non-case status in the final validation cohort were high, that is, 96% (95% CI92% to 98%) and 95% (95% CI 92% to 97%), respectively Conclusion Using methodology consistent with other recent ACR/EULAR-approved classification criteria, we developed a single set of data-driven consensus classification criteria for primary SS, which performed well in validation analyses and are well suited as criteria for enrolment in clinical trials

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553 Citations


Journal ArticleDOI: 10.1136/ANNRHEUMDIS-2019-216561
Zachary S. Wallace1, Ray Naden2, Suresh T. Chari3, Hyon K. Choi1  +16 moreInstitutions (10)
Abstract: IgG4-related disease (IgG4-RD) can cause fibroinflammatory lesions in nearly any organ. Correlation among clinical, serological, radiological and pathological data is required for diagnosis. This work was undertaken to develop and validate an international set of classification criteria for IgG4-RD. An international multispecialty group of 86 physicians was assembled by the American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR). Investigators used consensus exercises; existing literature; derivation and validation cohorts of 1879 subjects (1086 cases, 793 mimickers); and multicriterion decision analysis to identify, weight and test potential classification criteria. Two independent validation cohorts were included. A three-step classification process was developed. First, it must be demonstrated that a potential IgG4-RD case has involvement of at least one of 11 possible organs in a manner consistent with IgG4-RD. Second, exclusion criteria consisting of a total of 32 clinical, serological, radiological and pathological items must be applied; the presence of any of these criteria eliminates the patient from IgG4-RD classification. Third, eight weighted inclusion criteria domains, addressing clinical findings, serological results, radiological assessments and pathological interpretations, are applied. In the first validation cohort, a threshold of 20 points had a specificity of 99.2% (95% CI 97.2% to 99.8%) and a sensitivity of 85.5% (95% CI 81.9% to 88.5%). In the second, the specificity was 97.8% (95% CI 93.7% to 99.2%) and the sensitivity was 82.0% (95% CI 77.0% to 86.1%). The criteria were shown to have robust test characteristics over a wide range of thresholds. ACR/EULAR classification criteria for IgG4-RD have been developed and validated in a large cohort of patients. These criteria demonstrate excellent test performance and should contribute substantially to future clinical, epidemiological and basic science investigations.

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187 Citations


Open accessJournal ArticleDOI: 10.1093/RHEUMATOLOGY/KEL266
J. Pijpe1, Warner Kalk2, J.E. van der Wal1, Arjan Vissink  +5 moreInstitutions (3)
01 Oct 2007-Rheumatology
Abstract: Objective. To assess the value of the parotid biopsy as a diagnostic tool for primary Sjogren's syndrome ( pSS), and to compare the parotid biopsy and the labial biopsy with regard to diagnostic value and biopsy-related morbidity. Methods. In 15 consecutive patients with pSS and 20 controls, the parotid biopsy was assessed as a diagnostic tool based on the presence of lymphocytic foci, benign lymphoepithelial lesions and lymphoid follicles. These new histological criteria were compared with established diagnostic criteria for the labial biopsy in 35 consecutive patients suspected for pSS who underwent simultaneous biopsies from both sites. In addition, both biopsies were compared for morbidity. Results. The first analysis revealed a focus score of >= 1 or lymphocytic infiltrates ( not fulfilling the criterion of a focus score of 1) combined with benign lymphoepithelial lesions as diagnostic criteria for pSS. When comparing the parotid biopsy with the labial biopsy sensitivity and specificity were comparable (sensitivity 78%, specificity 86%). Level of pain was comparable and no loss of motor function was observed. No permanent sensory loss was observed after parotid biopsy, while labial biopsy led to permanent sensory loss in 6% of the patients. Malignant lymphoma was detected in one parotid biopsy by chance, without involvement of the labial salivary gland. Conclusion. A parotid biopsy has a diagnostic potential comparable with that of a labial biopsy in the diagnosis of pSS, and may be associated with less morbidity.

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Topics: Biopsy (60%), Parotid gland (59%), Histopathology (51%)

131 Citations


Open accessJournal Article
De Vita S1, Lorenzon G, Giuseppe Rossi, Sabella M  +1 moreInstitutions (1)
Abstract: A series of different ultrasonographic abnormalities detected by salivary gland echography (SGE) were investigated for their discriminant power for Sjogren's syndrome (SS) in 53 patients with either primary SS (n = 27) or secondary SS (n = 26), as well as in 90 controls. Among the controls, 26 suffered from dry mouth and/or recurrent or persistent swelling of at least one parotid or submandibular gland due to other selected disorders, while 64 were healthy, asymptomatic subjects. Mild, evident or gross inhomogeneous parenchymal patterns were the only variables selected by stepwise discriminant analysis, when comparing patients to controls. However, a mild submandibular inhomogeneity did not prove useful for such a discrimination. Based on these data, a simplified evaluation and standardised quantification of salivary involvement, as detected by SGE, is proposed using an echographic score (range 0 to 6) which assigns points to the different degrees of glandular inhomogeneity. Score values above 0 showed a sensitivity of 88.8% in primary SS and of 53.8% in secondary SS, as well as a specificity of 84.6% and of 92.2% with respect to either symptomatic or healthy controls. The lower sensitivity of SGE for patients with secondary SS presumably was a result of their milder salivary involvement.

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127 Citations


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