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A Child With Primary Sjögren Syndrome and a Review of the Literature
Mahmut Civilibal, MD*,
Nur Canpolat,
Ayse Yurt, MD,
Sebuh Kurugoglu, MD,
Sibel Erdamar, MD,
Onur Bagci, MD,
Lale Sever, MD,
Ozgur Kasapcopur, MD,
Salim Caliskan, MD,
Nil Arisoy, MD
Istanbul University Cerrahpasa Medical Faculty
* To whom correspondence should be addressed. E-mail: drcivilibal{at}hotmail.com.
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Abstract |
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Primary Sjögren syndrome (pSS) is an uncommon disease in childhood. Childhood pSS might have different clinical manifestations than adult pSS. We describe a 13-year-old girl with multiple episodes of bilateral parotid swelling lasting 2 years. Her history included severe arthralgia, local edema, and purpura episodes since 9 years of age. During her 3-week hospitalization, 2 episodes of parotid swelling occurred, which both resolved in 48 hours. Ultrasonography and magnetic resonance images of parotid glands showed parenchymal inhomogeneity related to adipose degeneration and nodular pattern. Investigations showed elevated erythrocyte sedimentation rate, the presence of hypergammaglobulinemia, positive antinuclear antibody, and elevated rheumatoid factor, anti-Sjögren syndrome antigen A, and anti-Sjögren syndrome antigen B. Histopathologic examination of labial minor salivary glands revealed focal periductal lymphocytic infiltrate and sialoduct ectasia. She was diagnosed as having pSS. Recurrent parotid swelling is a more characteristic feature of disease in children, and this finding should alert the clinician to the possible diagnosis of pSS.
First published on May 16, 2007, doi:10.1177/0009922807301945
Clinical Pediatrics 2007;46:738.
A more recent version of this article appeared on October 1, 2007
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