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      Parotiditis Crónica Recurrente o Síndrome de SjÖgren Primario Juvenil?: Reporte de un Caso Translated title: Chronic Recurrent Parotiditis or Juvenile Primary SjÖgren?: A Case Report

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          Abstract

          Introducción: La parotiditis crónica recurrente es una inflamación recidivante de la glándula, producida por diversos factores, sin embargo, ésta en algunos casos puede presentarse como primera manifestación del síndrome de SjÖgren. Objetivo: Describir la relación existente entre los signos y síntomas de ambas patologías, evaluando los hallazgos clínicos, radiográficos, histológicos y de laboratorio encontrados en un paciente con antecedentes de parotiditis crónica recurrente y criterios positivos para Síndrome de SjÖgren. Presentación del caso: Paciente femenino de 14 años de edad, atendida en la consulta de Medicina Bucal del Hospital Universitario de Maracaibo por presentar múltiples aumentos de volumen parotídeo de 1 año de evolución. Clínicamente se confirmó los cambios glandulares, observándose además caries y salivación espesa, sin sequedad bucal ni molestias oculares, llegando al diagnóstico de Parotiditis Crónica Recurrente. Ante las múltiples caries avanzadas, la marcada recurrencia de los episodios inflamatorios y respuesta inadecuada al tratamiento, se solicitó biopsia de glándula salival menor de labio inferior, anticuerpos específicos para el síndrome de SjÖgren, sialometría y test de Schirmer, encontrando criterios positivos suficientes para diagnosticar este Síndrome. Conclusión: En pacientes niños y adolescentes con PCR en los cuales a pesar del tratamiento indicado no se logre prolongar el tiempo entre las recurrencias ni disminuir el aumento de tamaño de la glándula satisfactoriamente, debe ser evaluada la presencia del Síndrome de SjÖgren, aún en ausencia de síntomas oculares y bucales.

          Translated abstract

          Introduction: Chronic Recurrent Parotiditis is a recurrent swelling of the parotid gland, of multiple etiology. In some cases it may be an early manifestation of SjÖgren's syndrome. Objective: A comparison of both diseases, in light of the clinical, radiological, histological and laboratory findings in a patient suffering from a chronic recurrent parotiditis suggestive of SjÖgren's syndrome. Case Report: A 14 years old girl, seen in Oral Maxillary Medicine consultation at "Hospital Universitario de Maracaibo", for a year's history of multiple episodes of parotid swelling. Glandular involvement was confirmed clinically. While dental cavities and thick saliva were observed, the absence of oral dryness and ocular manifestations resulted in a diagnosis of Chronic Recurrent Parotitis at the time. Due to multiple recurrent dental cavities, high recurrence of the swelling episodes, and poor response to treatment, SjÖgren's syndrome was suspected. Lower lip minor salivary gland biopsy, specific antibodies, sialometry and Schirmer's test were requested, finding enough positive criteria for the diagnosis of SjÖgren's syndrome. Conclusions: Among children and teenagers with CRT with treatment failure, the presence of Sjógren's syndrome must be evaluated, even in the absence of oral and ocular symptoms.

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          Medical research activities, funding, and creativity in Europe: comparison with research in the United States.

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            Primary Sjögren syndrome in the paediatric age: a multicentre survey.

            Primary Sjögren syndrome (SS) is very rare in childhood. We collected a series of primary paediatric SS cases from different centres. A data collection form was prepared and sent to rheumatologists who were willing to participate. Data on 40 cases of primary SS with onset before the 16th birthday were collected. Almost all patients (35/40) were females, age at onset varied from 9.3 to 12.4 years (mean 10.7 years). Signs and symptoms at disease onset were mainly recurrent parotid swelling followed by sicca symptoms. Abnormal laboratory tests were found in the majority of cases. Regarding treatment, 22 patients were treated at some time with oral corticosteroids, seven with non-steroidal anti-inflammatory drugs, and five with hydroxychloroquine; two patients needed cyclosporine and one cyclophosphamide. Follow-up varied from 0 to 7.5 years from onset, without major complications in the majority of patients. recurrent parotid swelling is a common feature of primary Sjögren syndrome in childhood and often occurs as a presenting feature. Sicca symptoms may be rarer.
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              Anti-Ro/SSA and anti-La/SSB autoantibodies in the tear fluid of patients with Sjögren's syndrome.

              To investigate the presence of anti-Ro/SSA and anti-La/SSB antibodies in the tear fluid and serum of patients with Sjögren's syndrome and to evaluate the association of these autoantibodies with the severity of keratoconjunctivitis sicca. Tear fluid and serum were obtained from 28 patients with Sjögren's syndrome and 17 age matched normal control subjects. Evaluation of tear fluid and sera anti-Ro/SSA and anti-La/SSB levels was done by using a quantitative enzyme linked immunosorbent assay kit designed for the quantitative measurement of IgG class autoantibodies directed against highly purified SSA and SSB antigens. Tear function and ocular surface were evaluated by Schirmer I test, tear break up time, and rose bengal staining. Dry eye symptom scores were recorded. Increased levels of anti-Ro/SSA and anti-La/SSB antibodies were detected in sera of 57.1% and 50% of SS patients, respectively. Six patients had increased levels of anti-Ro/SSA in the tear fluid, in one case anti-Ro/SSA being detected in tear fluid when it was negative in serum. Ten patients had positive anti-La/SSB titres in tear fluid and in four of these patients, anti-La/SSB titres were not elevated in serum. A positive correlation was observed between serum and tear fluid titres of anti-Ro/SSA (r = 0.43, p = 0.02), but not of anti-La/SSB. Serum anti-Ro/SSA and anti-La/SSB concentrations correlated positively with dry eye symptom scores (r = 0.42, p = 0.02 and r = 0.48, p = 0.01, respectively) and negatively correlated with Schirmer I test scores (r = -0.39, p = 0.04 and r = -0.40, p = 0.03, respectively). Significant correlations were found between tear anti-La/SSB concentrations and dry eye symptom score (r = 0.56, p = 0.02) and also rose bengal staining scores of the ocular surface (r = 0.44, p = 0.02). This study shows that autoantibodies against Ro/SSA and La/SSB antigens are present in the tear fluid of some patients with SS and their presence in serum or tear fluid is associated with the severity of keratoconjunctivitis sicca. Additional measurement of tear fluid levels of anti-Ro/SSA and anti-La/SSB may serve as a valuable diagnostic indicator of SS.
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                Author and article information

                Journal
                rcp
                Revista chilena de pediatría
                Rev. chil. pediatr.
                Sociedad Chilena de Pediatría (Santiago, , Chile )
                0370-4106
                August 2009
                : 80
                : 4
                : 361-366
                Affiliations
                [01] orgnameUniversidad de Zulia orgdiv1Facultad de Odontología orgdiv2Instituto de Investigaciones Venezuela
                [02] Maracaibo orgnameHospital Universitario de Maracaibo Venezuela
                Article
                S0370-41062009000400008 S0370-4106(09)08000408
                10.4067/S0370-41062009000400008
                d44eb5f1-96d5-4683-8c9f-80ac672b66c0

                This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

                History
                : 04 July 2008
                : 11 May 2009
                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 26, Pages: 6
                Product

                SciELO Chile

                Categories
                CASOS CLINICOS

                Sjógren,Recurrent parotitis,parotiditis recurrente,auto immunity,autoinmune

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