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    Criteria sets for primary Sjogren's syndrome are not adequate for those presenting with extraglandular organ involvements as their dominant clinical features
    (SPRINGER HEIDELBERG, 2017) Kabasakal, Yasemin; Kitapcioglu, Gul; Karabulut, Gonca; Tezcan, Mehmet; Balkarli, Ayse; Aksoy, Adem; Yavuz, Aule; Yilmaz, Sema; Kasifoglu, Timucin; Kalyoncu, Umut; Dalkilic, Ediz; Tufan, Abdurrahman; Mercan, Ridvan; Yildiz, Fatih; Senturk, Taskin; Onen, Fatos; Bes, Cemal; Erken, Eren; Tunc, Ercan; Kamali, Sevil; Tarhan, Emine; Yazici, Ayten; Duzgun, Nursen; Bicakcigil, Muge; Yilmaz, Sedat; Ozmen, Mustafa; Ocal, Lale; Alibaz-Oner, Fatma; Solmaz, Dilek; Cobankara, Veli; Nalbant, Selim; Gunal, Esen Kasapoglu; Kaskari, Derya; Goker, Berna
    Patients with primary Sjogren's syndrome (pSS) may go undiagnosed or be misclassified due to the insidious nature and wide spectrum of the disease. The available several classification criteria emphasize glandular findings. We aimed to analyze the efficiency of various classification criteria sets in patients diagnosed on the clinical basis by expert opinion and to compare those pSS patients who fulfilled these criteria with those who did not. This is a multicenter study in which 834 patients from 22 university-based rheumatology clinics are included. Diagnosis of pSS was made on the clinical basis by the expert opinion. In this study, we only interviewed patients once and collected available data from the medical records. The European criteria, American-European Consensus Group (AECG) and American College of Rheumatology (ACR) Sjogren's criteria were applied. Majority of the patients were women (F/M was 20/1). The median duration from the first pSS-related symptom to diagnosis was significantly shorter in men (2.5 +/- 2.3 vs 4.3 +/- 5.9 years) (p = 0 < 0.016). When the European, AECG and ACR Sjogren's criteria were applied, 666 patients (79.9%) satisfied at least one of them. In total, 539 patients (64.4%) satisfied the European, 439 (52.6%) satisfied the AECG, and 359 (43%) satisfied the ACR criteria. Among the entire group, 250 patients (29.9%) satisfied all and 168 (20.1%) met none of the criteria. The rates of extraglandular organ involvements were not different between patients who met at least one of the criteria sets and those who met none. There is an urgent need for the modification of the pSS criteria sets to prevent exclusion of patients with extraglandular involvements as the dominant clinical features.
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    Low frequency of HLA-B27 in ankylosing spondylitis patients from Turkey
    (ELSEVIER FRANCE-EDITIONS SCIENTIFIQUES MEDICALES ELSEVIER, 2008) Gunal, Esen Kasapoglu; Sarvan, Fatma Oguz; Karnali, Sevil; Gul, Ahmet; Inanc, Murat; Carin, Mahmut; Konice, Meral; Aral, Orhan; Ocal, Lale
    Objectives: Ankylosing spondylitis is strongly associated with HLA-B27. However, the strength of the association with HLA-B27 and the clinical features may vary in different parts of the world. The aim of this study is to compare the clinical features of AS and the frequencies of HLA-B27 and its alleles in patients from Turkey with other series. Methods: One hundred and twelve patients (72 male/40 female) fulfilling the modified New York criteria for the classification of AS and 55 (27 male/28 female) healthy controls were typed for HLA-B27 and allele frequencies by sequence specific primer (PCR/SSP) method and assessed for clinical manifestations. Results: Male to female ratio was 1.8, mean age at disease onset was 23.5 and 24.1% of patients reported juvenile onset of symptoms. Peripheral arthritis was seen in 52.7% of patients. Family history (p = 0.01) and peripheral arthritis (p = 0.02) were more frequent in females and spinal involvement in males. HLA-B27 was found to be positive in 70% of patients and associated with younger mean age, uveitis and shorter time elapsed from symptom to diagnosis. The frequency of HLA-B27 alleles associated with SpA was not different between ankylosing spondylitis patients and healthy controls. Conclusion: Low frequency of HLA-B27 and clinical variations in ankylosing spondylitis may be due to different genetic and/or environmental factors in Turkey. (C) 2008 Elsevier Masson SAS. All rights reserved.
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    Rosai-Dorfman disease with factor XII deficiency
    (SPRINGER LONDON LTD, 2009) Gunal, Esen Kasapoglu; Kamali, Sevil; Akdogan, Mehmet Fatih; Cimen, Arif Oguz; Ocal, Lale; Agan, Mehmet; Gul, Ahmet; Inanc, Murat; Konice, Meral; Aral, Orhan
    A 17-year-old female patient presented with chronic symmetrical oligoarthritis of both knees and ankles, xerostomia, xerophthalmia, multiple bilateral lymphadenopathies in the cervical region, and bilateral parotid enlargement with the histological finding of chronic sialoadenitis. She had been already given methotrexate, chloroquine, and corticosteroids with the diagnosis of rheumatoid arthritis (RA) before referral to our outpatient clinic. Because her complaints and the lumps did not remit and she could be classified as neither RA nor primary Sjogren's syndrome (SS) according to 1987 ACR RA criteria or European preliminary criteria for SS, lymph node biopsy was repeated and revealed the diagnosis of Rosai-Dorfman disease (RDD) with the histological findings of histiocytes, phagocyting lymphocytes in enlarged sinuses, and mature plasma cells infiltrating the pulpa. All the medications were stopped after the pathological diagnosis of RDD and consulting with the Division of Hematology. She was reevaluated with magnetic resonance imaging, which showed dense infiltrative areas around knee and ankle joints, and computed tomography that showed a soft tissue mass surrounding the descending aorta and upper part of the abdominal aorta. Activated partial thromboplastin time was found to be prolonged in prebiopsy examinations, and factor XII deficiency was detected after detailed hematological evaluation. The symptoms of joint involvement were relieved with nonsteroidal antiinflammatory drugs. She has been followed-up without medication without obvious clinical or laboratory change. We herein report a patient with RDD mimicking RA and SS. We consider that RDD should be kept in mind especially in patients with resistant symptoms to conventional therapies, younger disease onset, and predominant parotid and lymph node enlargement.