Pediatric Nephrology, Oregon Health & Science University and OHSU Doernbecher Children's Hospital, Portland, OR, USA.
Pediatric Nephrology, Harvard Medical School and Boston Children's Hospital, Boston, MA, USA.
Pediatr Nephrol. 2024 Oct;39(10):2845-2860. doi: 10.1007/s00467-023-06211-6. Epub 2024 Feb 23.
Gastrointestinal (GI) sequelae, such as vomiting, hyperacidity, dysphagia, dysmotility, and diarrhea, are nearly universal among patients with nephropathic cystinosis. These complications result from disease processes (e.g., kidney disease, cystine crystal accumulation in the GI tract) and side effects of treatments (e.g., cysteamine, immunosuppressive therapy). GI involvement can negatively impact patient well-being and jeopardize disease outcomes by compromising drug absorption and patient adherence to the strict treatment regimen required to manage cystinosis. Given improved life expectancy due to advances in kidney transplantation and the transformative impact of cystine-depleting therapy, nephrologists are increasingly focused on addressing extra-renal complications and quality of life in patients with cystinosis. However, there is a lack of clinical data and guidance to inform GI-related monitoring, interventions, and referrals by nephrologists. Various publications have examined the prevalence and pathophysiology of selected GI complications in cystinosis, but none have summarized the full picture or provided guidance based on the literature and expert experience. We aim to comprehensively review GI sequelae associated with cystinosis and its treatments and to discuss approaches for monitoring and managing these complications, including the involvement of gastroenterology and other disciplines.
胃肠道(GI)后遗症,如呕吐、胃酸过多、吞咽困难、运动障碍和腹泻,在患有肾源性胱氨酸病的患者中几乎普遍存在。这些并发症是由疾病过程(例如,肾病、胃肠道中胱氨酸晶体的积累)和治疗的副作用(例如,半胱氨酸、免疫抑制治疗)引起的。胃肠道受累会通过影响药物吸收和患者对管理胱氨酸病所需的严格治疗方案的依从性,对患者的健康状况产生负面影响,并危及疾病结局。由于肾移植的进步和胱氨酸耗竭疗法的变革性影响,患者的预期寿命得到了改善,因此肾病学家越来越关注解决胱氨酸病患者的肾脏以外的并发症和生活质量问题。然而,目前缺乏临床数据和指导,无法为肾病学家提供与胃肠道相关的监测、干预和转诊建议。各种出版物已经研究了胱氨酸病中某些胃肠道并发症的患病率和病理生理学,但没有一份出版物能够全面总结全貌,或根据文献和专家经验提供指导。我们旨在全面审查与胱氨酸病及其治疗相关的胃肠道后遗症,并讨论监测和管理这些并发症的方法,包括胃肠病学和其他学科的参与。