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Sjogren's syndrome;biopsy

Nicole C Droz, Stephanie D Mathew
After presenting with nonspecific symptoms, a 52-year-old female was found to have a 3.4 × 2.6 cm retroperitoneal mass resulting in hydronephrosis on abdominal computed tomography scan. After extensive workup including surgical debulking, excisional biopsy, and autoimmune panel, she was found to have idiopathic retroperitoneal fibrosis (IRF) with negative staining for immunoglogulin G4 (IgG4). Two years after presentation, the patient developed dyspnea, cough, and xerostomia. Repeat anti-Sjögren Syndrome A and anti-Sjögren Syndrome B titers and labial gland biopsy confirmed the diagnosis of concomitant Sjögren's syndrome (SS)...
October 2016: Military Medicine
Melis Palamar, Nazan Ozsan, Fahri Sahin
A 31-year-old female with Primary Sjögren Syndrome (pSS) presented with bilateral puffiness around the eye for 3 years. The lacrimal glands were hypertrophic and edematous bilaterally. Schirmer 1 score was 2 and 1 mm and tear-film break-up time was 3 and 4 seconds, in the right and the left eyes, respectively. An incisional biopsy from the left lacrimal gland revealed diffuse and intense CD20, CD5, and bcl-2 positivity with negative cyclin D1 and CD23 which supported lymphoma. Upon haematology consultation extranodal marginal zone lymphoma diagnosis was made...
2016: Case Reports in Ophthalmological Medicine
Mehmet Taşdemir, Chiar Hasan, Ayşe Ağbaş, Özgür Kasapçopur, Nur Canpolat, Lale Sever, Salim Çalışkan
Systemic lupus erythematosus and Sjögren's syndrome are chronic auto- inflammatory disorders which can lead to serious organ damage. Although systemic lupus erythematosus and Sjögren's syndrome were previously considered two forms of the same disease because of presence of clinical coexistence of these two conditions, the view that they are two different conditions with mutual characteristics has become prominent in recent years. In this paper, we reported a 16 year-old girl who was followed up with a diagnosis of Sjögren's syndrome for six years and then was observed to have overlap of systemic lupus erythematosus...
September 2016: Türk Pediatri Arşivi
Prajwal Boddu, Abdul S Mohammed, Sonali Khandelwal
Hypergammaglobulinemic purpura (HGP) is a syndrome constellating recurrent purpura, hypergammaglobulinemia, positive rheumatoid factor (RF), anti-Ro/La antibodies, and elevated erythrocyte sedimentation rate (ESR). We present a case of a 29-year-old female who was diagnosed with Sjogren's syndrome four years prior to presenting with rash on her lower extremities for a period of 6 months. Skin biopsy at the initial visit was consistent with leukocytoclastic vasculitis and was initiated on treatment for it. Her rash evolved into 2-5 mm scattered purpurae while she was on the treatment and a repeat biopsy showed extravasation of RBCs, a sparse mononuclear infiltrate with deposition of plasma cells, and no evidence of leukocytoclastic vasculitis, thus showing a transition from neutrophilic to mononuclear inflammatory vascular disease which is a rare occurrence...
2016: Case Reports in Rheumatology
Divi Cornec, Sandrine Jousse-Joulin, Sebastian Costa, Thierry Marhadour, Pascale Marcorelles, Jean-Marie Berthelot, Eric Hachulla, Pierre-Yves Hatron, Vincent Goeb, Olivier Vittecoq, Emmanuel Nowak, Jacques-Olivier Pers, Valérie Devauchelle-Pensec, Alain Saraux
PURPOSE: To determine whether the severity of salivary-gland involvement, assessed using salivary gland ultrasonography [SGUS], histological focus score, or the unstimulated whole salivary flow [UWSF], was associated with the response to rituximab in patients with primary Sjögren's syndrome [pSS]. MATERIALS AND METHODS: Among the 120 patients with pSS enrolled in the randomised TEARS trial of rituximab versus placebo, 35 underwent either centralised minor salivary-gland biopsy or SGUS at inclusion...
2016: PloS One
Vatinee Y Bunya, Satasuk Joy Bhosai, Ana Maria Heidenreich, Kazuko Kitagawa, Genevieve B Larkin, Thomas M Lietman, Bruce D Gaynor, Esen K Akpek, Mina Massaro-Giordano, M Srinivasan, Travis C Porco, John P Whitcher, Stephen C Shiboski, Lindsey A Criswell, Caroline H Shiboski
PURPOSE: To identify a screening strategy for dry eye patients with a high likelihood of having Sjogren's syndrome (SS) through the evaluation of the association of ocular surface tests with the extraocular signs used for the diagnosis of SS. DESIGN: Multi-center cross-sectional study. METHODS: The Sjogren's International Clinical Collaborative Alliance (SICCA) registry enrolled 3,514 participants with SS or possible SS from 9 international academic sites...
September 16, 2016: American Journal of Ophthalmology
Kenichiro Tanaka, Hiroyuki Nakayasu, Yutaka Suto, Shotaro Takahashi, Yoshihiro Konishi, Hirotake Nishimura, Rino Ueno, Susumu Kusunoki, Kenji Nakashima
A patient with xerostomia and xerophthalmia due to Sjögren's syndrome presented with acute motor-dominant polyneuropathy and multiple mononeuropathy with antiganglioside antibodies. Nerve conduction studies and a sural nerve biopsy revealed the neuropathy as a mixture of segmental demyelination and axonal degeneration. Positive results were obtained for several antiganglioside antibodies. Corticosteroid treatment proved effective. The neuropathy was considered to represent a mixture of polyneuropathy as Guillain-Barré syndrome and multiple mononeuropathy via Sjögren's syndrome...
2016: Internal Medicine
Janine Amos, Anthony Baron, Adam D Rubin
PURPOSE OF REVIEW: Dysphagia can be the presenting symptom of autoimmune disease. The otolaryngologist should be familiar with and consider these disorders when assessing the patient with swallowing difficulties. RECENT FINDINGS: The present review gives a brief overview of Sjogren's syndrome, granulomatosis with polyangiitis, pemphigus and pemphigoid, rheumatoid arthritis, systemic lupus erythematosus, scleroderma and inflammatory myopathies, and how they affect swallowing...
August 31, 2016: Current Opinion in Otolaryngology & Head and Neck Surgery
Margherita Sisto, Loredana Lorusso, Sabrina Lisi
IL-15 is a key regulatory cytokine that shares many biological properties with IL-2. Recently, it has been shown that IL-15 could be up-regulated in T cell-mediated inflammatory disorders, such as rheumatoid arthritis and inflammatory bowel diseases. However, the role and expression of IL-15 in the inflammatory autoimmune disease Sjögren's syndrome (SS) has not been investigated. In the present study we evaluated the expression of IL-15 mRNA and protein in minor salivary gland (MSG) biopsy specimens and in human salivary gland epithelial cell (SGEC) cultures obtained from patients with primary SS (pSS) and compared their expression with that seen in normal healthy control subjects...
October 2016: Pathology
Serena Vettori, Rosaria Irace, Antonella Riccardi, Daniela Iacono, Luciana Pellecchia, Lucia Vicedomini, Gabriele Valentini
CXCL4 is an antiangiogenic and immunomodulatory chemokine. We aimed to investigate serum levels of CXCL4 in primary Sjögren's syndrome (pSS), looking for associations with disease features. Thirty-nine consecutive pSS patients underwent clinical-serological assessment and nailfold videocapillaroscopy (NVC). Thirty-six patients and 30 controls affected by osteoarthritis were also investigated for serum levels of CXCL4 and soluble E-selectin (sE-selectin). CXCL4 was higher in pSS patients than in controls (1...
October 2016: Clinical Rheumatology
Radjiv Goulabchand, Assia Hafidi, Ingrid Millet, Jacques Morel, Cédric Lukas, Sébastien Humbert, Sophie Rivière, Christian Gény, Christian Jorgensen, Alain Le Quellec, Hélène Perrochia, Philippe Guilpain
Sjögren's syndrome is well known to target exocrine glands, especially lacrimal and salivary glands, which share with mammary glands anatomical, histological, and immunological features. Herein, we investigated the mammary involvement in patients with Sjögren's syndrome and compared the histological findings with minor salivary gland involvement. We reviewed the charts of patients with Sjögren's syndrome (followed in Montpellier University Hospital, between January 2000 and January 2015), in whom minor salivary gland and mammary tissues were available...
August 25, 2016: Immunologic Research
Katarzyna Romanowska-Próchnicka, Marzena Olesińska, Agnieszka Paradowska-Gorycka, Małgorzata Mańczak, Anna Felis-Giemza, Piotr Wojdasiewicz, Dariusz Szukiewicz
OBJECTIVES: To investigate whether a difference exists between DAS28 from CRP and DAS28 from ESR in patients with rheumatoid arthritis (RA) and secondary Sjögren's syndrome (sSS). MATERIAL AND METHODS: One group comprised patients with RA and sSS, the control group comprised patients with RA. The inclusion criteria for the RA and sSS group have been specified as follows: presence of at least one symptom of dryness, and also presence of anti-SS-A and anti-SS-B or at least focus score of one in biopsy...
2016: Central-European Journal of Immunology
Susanna Maddali Bongi, Martina Orlandi, Angelina De Magnis, Daniela Moncini, Angela Del Rosso, Felice Galluccio, Alessandro Franchi
The goal of this study was to evaluate the characteristics and the prevalence of histopathologic and immunohistochemical changes in vulvar biopsies, in women with Primary Sjögren Syndrome (pSS) and non-Sjögren Sicca Syndrome (nS-SS). Twenty-one women with pSS and 11 with nS-SS (investigated by xerophtalmia and xerostomia tests, biopsy of minor salivary glands, gynecological history, and gynecologic symptoms score) underwent vulvar biopsies, evaluated for histopathologic and immunohistochemicalchanges, and compared with those obtained from 26 patients with lichen sclerosus...
November 2016: International Journal of Gynecological Pathology
Sülen Sarioğlu, Ülkü Küçük, Pınar Çetin, İsmail Sari, Ahmet Merih Birlik
BACKGROUND/AIM:  We aimed to analyze the value of 3 serial sections, spaced 200 µm apart, for quantification of lymphocyte and plasma cell foci in minor salivary gland biopsy (MSGB). MATERIALS AND METHODS:  Labial MSGBs from 69 patients with Sjögren's syndrome (SS) and scleroderma were used for this study. Each sample was prepared as 3 serial sections spaced 200 µm apart. Lymphocytic and plasma cell focus score (LFS, PFS) were determined for each section, and the diagnostic results were compared to those obtained from a single section...
2016: Turkish Journal of Medical Sciences
Supraja Yeturi, Mary Cronin, Adam Robin, Campbell Lorna, Ann K Rosenthal
Pauci-immune crescentic glomerulonephritis is commonly seen in ANCA-associated vasculitis but it is rarely seen during the course of other connective tissue diseases like lupus or Sjogren's syndrome or MCTD. We report 3 cases of pauci-immune crescentic glomerulonephritis in patients with connective tissue disease other than vasculitis. We reviewed literature and made summary of previously reported cases of this rare entity. Clinical and laboratory features of these patients varied widely, but most of patients have met criteria for lupus...
2016: Case Reports in Rheumatology
Masahiro Oishi, Yasuhiko Hayashi, Issei Fukui, Daisuke Kita, Tadao Miyamori, Mitsutoshi Nakada
BACKGROUND: Xanthomatous hypophysitis (XH) is an extremely rare form of primary hypophysitis characterized by infiltration of the pituitary gland by mixed types of inflammatory cells, including foamy cells, plasma cells, and small mature lymphocytes. XH manifests as varying degrees of hypopituitarism. Although several previous reports have denied a possible contribution of autoimmune mechanism, the exact pathogenesis of XH remains unclear. CASE DESCRIPTION: We describe the case of a 72-year-old woman with a history of rheumatoid arthritis and Sjögren's syndrome who presented with panhypopituitarism and diabetes insipidus...
2016: Surgical Neurology International
Masato Kono, Yutaro Nakamura, Katsuhiro Yoshimura, Yasunori Enomoto, Yoshiyuki Oyama, Hironao Hozumi, Noriyuki Enomoto, Tomoyuki Fujisawa, Naoki Inui, Etsuko Hamada, Thomas V Colby, Masato Maekawa, Takafumi Suda
BACKGROUND: The aim of this study was to evaluate the incidence and clinical features of patients who developed collagen vascular disease (CVD) after an initial diagnosis of idiopathic nonspecific interstitial pneumonia (NSIP). METHODS: We conducted a retrospective review of 72 consecutive patients with NSIP who were diagnosed by surgical lung biopsy in our institution (idiopathic NSIP, n = 35; CVD-NSIP, n = 37 at initial diagnosis). No patients fulfilled the American College of Rheumatology criteria for a diagnosis with CVD within six months after the diagnosis of idiopathic NSIP...
August 2016: Respiratory Medicine
María-José Barrera, Sergio Aguilera, Isabel Castro, Juan Cortés, Verónica Bahamondes, Andrew F G Quest, Claudio Molina, Sergio González, Marcela Hermoso, Ulises Urzúa, Cecilia Leyton, María-Julieta González
Salivary gland (SG) acinar-cells are susceptible to endoplasmic reticulum (ER) stress related to their secretory activity and the complexity of synthesized secretory products. SGs of Sjögren's syndrome patients (SS)-patients show signs of inflammation and altered proteostasis, associated with low IRE1α/XBP-1 pathway activity without avert increases in apoptosis. Acinar-cells may avoid apoptosis by activation of the ATF6α pathway and ER-associated protein degradation (ERAD). The aim of this study was to evaluate the role of pro-inflammatory cytokines in ATF6α pathway/ERAD activation and cell viability in labial salivary glands (LSG) of SS-patients...
July 23, 2016: Journal of Autoimmunity
Hiroto Tsuboi, Isao Matsumoto, Shinya Hagiwara, Tomoya Hirota, Hiroyuki Takahashi, Hiroshi Ebe, Masahiro Yokosawa, Mizuki Yagishita, Hidenori Takahashi, Izumi Kurata, Ayako Ohyama, Fumika Honda, Hiromitsu Asashima, Haruka Miki, Naoto Umeda, Yuya Kondo, Shintaro Hirata, Kazuyoshi Saito, Yoshiya Tanaka, Yoshiro Horai, Hideki Nakamura, Atsushi Kawakami, Takayuki Sumida
OBJECTIVE: To clarify the efficacy and safety of abatacept for secondary Sjögren's syndrome (SS) associated with rheumatoid arthritis (RA). METHODS: The primary endpoint of this open-labeled, prospective, observational multicenter study for secondary SS with RA was the remission rate of Simplified Disease Activity Index (SDAI) at 52 weeks after initiation of abatacept. The secondary endpoints included Saxon's test and Schirmer's test. Adverse events and adherence rate during the study period were also analyzed...
July 26, 2016: Modern Rheumatology
Piotr Zajkowski, Aleksandra Ochal-Choińska
The paper is an update of 2011 Standards for Ultrasound Assessment of Salivary Glands, which were developed by the Polish Ultrasound Society. We have described current ultrasound technical requirements, assessment and measurement techniques as well as guidelines for ultrasound description. We have also discussed an ultrasound image of normal salivary glands as well as the most important pathologies, such as inflammation, sialosis, collagenosis, injuries and proliferative processes, with particular emphasis on lesions indicating high risk of malignancy...
June 2016: Journal of Ultrasonography
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