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小儿干燥综合征合并远端肾小管酸中毒和自身免疫性甲状腺功能减退:一种罕见的关联。

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

作者信息

Agarwal Amit, Kumar Pradeep, Gupta Nomeeta

机构信息

Department of Pediatrics, Batra Hospital and Medical Research Centre, Delhi, India.

出版信息

CEN Case Rep. 2015 Nov;4(2):200-205. doi: 10.1007/s13730-015-0169-y. Epub 2015 Feb 19.

Abstract

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.

摘要

一名14岁女性因突发肌无力前来就诊;在检查过程中,发现她患有高氯性代谢性酸中毒,阴离子间隙正常,肾功能正常,提示可能患有肾小管酸中毒(RTA)。在对RTA进行进一步评估时,她的抗核抗体、抗Ro和抗La抗体呈阳性。唾液腺核素扫描显示,她的左侧腮腺无功能。腮腺活检显示小叶间导管扩张,被淋巴细胞包绕。她还患有自身免疫性甲状腺功能减退,促甲状腺激素升高及抗甲状腺过氧化物酶抗体阳性提示了这一点。入院时,她的血清钾水平较低,接受了静脉注射氯化钾治疗。在从急性低钾性麻痹恢复后,她开始服用枸橼酸钾口服液,同时补充磷酸盐、羟氯喹、口服泼尼松龙和甲状腺素补充剂。在接下来的6个月里,她的钾、碳酸氢盐和磷酸盐用量显著减少,体重增加了3千克,身高增长了3.5厘米。由于原发性干燥综合征在儿科人群中本身就很罕见,其与肾小管酸中毒的关联更为罕见,我们建议在RTA检查过程中将干燥综合征作为鉴别诊断值得一试。

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