Academic Unit of Gastroenterology, Royal Hallamshire Hospital, Sheffield S10 2JF, UK.
Department of Gastroenterology, Vrije Universiteit Medical Center, 1117 Amsterdam, The Netherlands.
Nutrients. 2022 Jul 5;14(13):2776. doi: 10.3390/nu14132776.
We characterised the aetiology of non-responsive coeliac disease (NRCD) and provided contemporary mortality data in refractory coeliac disease (RCD) from our centre. We also measured urine gluten immunogenic peptides (GIPs) in patients with established RCD1 to evaluate gluten exposure in these individuals.
This was a longitudinal cohort study conducted in Sheffield, UK. Between 1998 and 2019, we evaluated 285 adult (≥16 years) patients with NRCD or RCD. Patients with established RCD1 and persisting mucosal inflammation and/or ongoing symptoms provided three urine samples for GIP analysis.
The most common cause of NRCD across the cohort was gluten exposure (72/285; 25.3%). RCD accounted for 65/285 patients (22.8%), 54/65 patients (83.1%) had RCD1 and 11/65 patients (16.9%) had RCD2. The estimated 5-year survival was 90% for RCD1 and 58% for RCD2 ( = 0.016). A total of 36/54 (66.7%) patients with RCD1 underwent urinary GIP testing and 17/36 (47.2%) had at least one positive urinary GIP test.
The contemporary mortality data in RCD2 remains poor; patients with suspected RCD2 should be referred to a recognised national centre for consideration of novel therapies. The high frequency of urinary GIP positivity suggests that gluten exposure may be common in RCD1; further studies with matched controls are warranted to assess this further.
本研究旨在分析非应答性乳糜泻（NRCD）的病因，并提供本中心难治性乳糜泻（RCD）的当代死亡率数据。我们还测量了已确诊的 RCD1 患者的尿中谷蛋白免疫原性肽（GIP），以评估这些个体中的谷蛋白暴露情况。
这是一项在英国谢菲尔德进行的纵向队列研究。在 1998 年至 2019 年期间，我们评估了 285 名成年（≥16 岁）NRCD 或 RCD 患者。已确诊的 RCD1 且持续存在黏膜炎症和/或持续症状的患者提供了三份尿液样本进行 GIP 分析。
整个队列中 NRCD 最常见的原因是谷蛋白暴露（72/285；25.3%）。RCD 占 285 例患者中的 65 例（22.8%），其中 54 例（83.1%）为 RCD1，11 例（16.9%）为 RCD2。RCD1 的估计 5 年生存率为 90%，RCD2 为 58%（=0.016）。总共 54 例 RCD1 患者中的 36 例（66.7%）进行了尿 GIP 检测，其中 17 例（47.2%）至少有一次尿 GIP 检测阳性。
RCD2 的当代死亡率仍然很差；疑似 RCD2 的患者应转介至认可的国家中心，以考虑新的治疗方法。尿 GIP 阳性率高表明 RCD1 中可能普遍存在谷蛋白暴露；需要进一步进行匹配对照研究以进一步评估。
