Renal Medicine, Freeman Hospital, Newcastle-upon-Tyne, UK
Hepatology, Freeman Hospital, Newcastle-upon-Tyne, UK.
BMJ Case Rep. 2022 Aug 16;15(8):e248461. doi: 10.1136/bcr-2021-248461.
A woman in her 50s was referred to nephrology clinic due to progressive chronic kidney disease. She exhibited features of proximal renal tubulopathy, namely Fanconi syndrome, including normoglycaemic glycosuria, normal anion gap metabolic acidosis, and intermittent hypouricaemia and hypophosphataemia. Kidney biopsy showed tubulointerstitial inflammation and focal chronic damage. In addition, antimitochondrial antibodies were present and she had abnormal liver blood tests. A unifying diagnosis of primary biliary cholangitis with an associated renal tubulopathy and interstitial nephritis was made. She was commenced on sodium bicarbonate, ursodeoxycholic acid and oral prednisolone, leading to an improvement in liver biochemistry. Kidney function was stabilised, but a sustained improvement was not seen. This case acts as a reminder of the rare association of tubulointerstitial nephritis and Fanconi syndrome with primary biliary cholangitis, which may be an under-recognised phenotype.
一位 50 多岁的女性因进行性慢性肾脏病被转至肾病科就诊。她表现出近端肾小管病变的特征,即范可尼综合征,包括血糖正常的糖尿、正常阴离子间隙代谢性酸中毒、间歇性低尿酸血症和低磷血症。肾脏活检显示肾小管间质性炎症和局灶性慢性损伤。此外,存在抗线粒体抗体,且肝功能检查异常。明确诊断为原发性胆汁性胆管炎伴肾小管病和间质性肾炎,开始给予碳酸氢钠、熊去氧胆酸和口服泼尼松龙治疗,导致肝功能生化指标改善。肾功能稳定,但未持续改善。该病例提醒人们注意原发性胆汁性胆管炎罕见合并肾小管间质性肾炎和范可尼综合征,这种表型可能认识不足。