Pediatric Sjogren syndrome with distal renal tubular acidosis and autoimmune hypothyroidism: an uncommon association

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• 作者：Amit Agarwal ; Pradeep Kumar ; Nomeeta Gupta
• 关键词：Renal tubular acidosis ; Pediatric Sjogren syndrome ; Autoimmune hypothyroidism
• 刊名：CEN Case Reports
• 出版年：2015
• 出版时间：November 2015
• 年：2015
• 卷：4
• 期：2
• 页码：200-205
• 全文大小：327 KB
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• 作者单位：Amit Agarwal (1)
  Pradeep Kumar (1)
  Nomeeta Gupta (1)
  
  1. Department of Pediatrics, Batra Hospital and Medical Research Centre, Delhi, India
  
• 刊物主题：Nephrology; Urology/Andrology;
• 出版者：Springer Japan
• ISSN：2192-4449

文摘

A 14-year-old female came with the history of sudden onset weakness; during work up, she was found to have hyperchloremic metabolic acidosis with normal anion gap and normal renal function suggesting the possibility of renal tubular acidosis (RTA). On further evaluation of RTA, she had positive antinuclear antibody, anti-Ro, and anti-La antibodies. On nuclear scan of salivary glands, her left parotid gland was nonfunctional. Her parotid biopsy revealed dilated interlobular ducts engulfed by lymphoid cells. She also had autoimmune hypothyroidism as suggested by raised TSH and positive anti-TPO antibodies. At admission, her serum potassium levels were low and she was treated with intravenous potassium chloride. After she recovered from acute hypokalemic paralysis, she was started on oral potassium citrate along with phosphate supplements, hydroxychloroquine, oral prednisolone and thyroxine supplements. Over the next 6 months, she has significant reduction in the dosage of potassium, bicarbonate and phosphate and gained 3 kg of weight and 3.5 cm of height. As primary Sjogren syndrome itself is rare in pediatric population and its association with renal tubular acidosis is even rarer, we suggest considering Sjogren syndrome as a differential diagnosis during the RTA work-up is worth trying. Keywords Renal tubular acidosis Pediatric Sjogren syndrome Autoimmune hypothyroidism