Medicine, Dalhousie Medical School, Halifax, Nova Scotia, Canada
Medicine, Dalhousie Medical School, Halifax, Nova Scotia, Canada.
BMJ Case Rep. 2024 Jun 19;17(6):e256225. doi: 10.1136/bcr-2023-256225.
Non-hepatic causes of hyperammonaemia are uncommon relative to hepatic aetiologies. An adolescent female was admitted to the hospital with a diagnosis of very severe aplastic anaemia. During her treatment with immunosuppressive therapy, she developed neutropenic enterocolitis, pseudomonal bacteraemia and hyperammonaemia. A combination of intermittent haemodialysis and high-volume continuous veno-venous haemodiafiltration (CVVHDF) was required to manage the hyperammonaemia. Despite a thorough investigation, there were no hepatic, metabolic or genetic aetiologies identified that explained the hyperammonaemia. The hyperammonaemia resolved only after the surgical resection of her inflamed colon, following which she was successfully weaned off from the renal support. This is a novel case report of hyperammonaemia of non-hepatic origin secondary to widespread inflammation of the colon requiring surgical resection in an immunocompromised patient. This case also highlights the role of high-volume CVVHDF in augmenting haemodialysis in the management of severe refractory hyperammonaemia.
与肝性病因相比,非肝脏原因引起的高血氨血症不常见。一名青春期女性因极重度再生障碍性贫血被收入院。在接受免疫抑制治疗期间,她发生中性粒细胞减少性肠炎、假单胞菌菌血症和高血氨血症。需要间歇性血液透析和高容量连续性静脉-静脉血液透析滤过(CVVHDF)联合治疗来控制高血氨血症。尽管进行了全面的检查,但仍未发现能解释高血氨血症的肝、代谢或遗传病因。在她发炎的结肠接受手术切除后,高血氨血症才得以解决,此后她成功地脱离了肾脏支持。这是一例因广泛结肠炎症导致非肝脏来源的高血氨血症的新型病例报告,该炎症在免疫功能低下的患者中需要手术切除。该病例还强调了高容量 CVVHDF 在增强血液透析治疗严重难治性高血氨血症中的作用。