Celiac Research Program, Harvard Medical School, Boston, USA.
Celiac Center, Beth Israel Deaconess Medical Center, 330 Brookline Ave, Boston, MA, 02215, USA.
Dig Dis Sci. 2021 Jun;66(6):1989-1997. doi: 10.1007/s10620-020-06454-5. Epub 2020 Jul 11.
Non-responsive celiac disease (NRCD) has many aetiologies, including gluten exposure. Budesonide may be used for refractory celiac disease (RCD) and celiac crisis.
We reviewed the effectiveness of budesonide to induce clinical and histologic response in NRCD with villous atrophy (VA).
Case series of adult cases with NRCD and VA prescribed budesonide at two celiac centers. Clinical variables and mucosal recovery (i.e., normal villous architecture within 1 year of treatment) were evaluated.
Forty-two cases [77% female, median age 45.0 (IQR 28.3-60.0) years] were included. Most common symptoms were diarrhea (64%) and abdominal pain (62%). Budesonide was initiated at 9 mg (83%) for a median duration of 16.0 weeks (IQR 6.8-25.0 weeks). In total, 57% exhibited a clinical response, positively associated with diarrhea (adjusted OR 6.08 95% CI 1.04-35.47) and negatively with fatigue (adjusted OR 0.18 95% CI 0.03-0.98). Clinical response was higher among those with dietitian counseling prior to budesonide (n = 29, 70 vs. 23%, p < 0.01). Mucosal recovery was observed in 11/24 with follow-up duodenal biopsies. There was no association between clinical response and mucosal recovery, and 79% of clinical responders had a symptomatic relapse. RCD (48%) and chronic gluten exposure (24%) were the main suspected aetiologies of NRCD. Most individuals without a clinical response subsequently received an IBS-related diagnosis.
Budesonide may be effective to induce clinical response in NRCD presenting with diarrhea and VA, but clinical recurrence and lack of mucosal recovery are frequent after tapering. Other diagnoses, including coexisting IBS, may be considered in non-responders to budesonide therapy.
非反应性乳糜泻（NRCD）有许多病因，包括麸质暴露。布地奈德可用于难治性乳糜泻（RCD）和乳糜泻危象。
我们回顾了布地奈德诱导伴有绒毛萎缩（VA）的 NRCD 临床和组织学反应的有效性。
在两个乳糜泻中心，对接受布地奈德治疗的 NRCD 和 VA 成人病例进行了病例系列研究。评估了临床变量和黏膜恢复（即治疗 1 年内正常的绒毛结构）。
共纳入 42 例病例[77%为女性，中位年龄 45.0（IQR 28.3-60.0）岁]。最常见的症状是腹泻（64%）和腹痛（62%）。布地奈德起始剂量为 9mg（83%），中位疗程为 16.0 周（IQR 6.8-25.0 周）。总共有 57%的患者表现出临床反应，与腹泻呈正相关（调整后的 OR 6.08，95%CI 1.04-35.47），与疲劳呈负相关（调整后的 OR 0.18，95%CI 0.03-0.98）。在接受布地奈德治疗前接受营养师咨询的患者中，临床反应更高（n=29，70% vs. 23%，p<0.01）。在 24 例有随访十二指肠活检的患者中观察到黏膜恢复。临床反应与黏膜恢复之间无相关性，79%的临床反应者出现症状复发。RCD（48%）和慢性麸质暴露（24%）是 NRCD 的主要疑似病因。大多数无临床反应的患者随后被诊断为 IBS 相关疾病。
布地奈德可能有效诱导伴有腹泻和 VA 的 NRCD 临床反应，但在逐渐减少剂量后，临床复发和缺乏黏膜恢复较为常见。对于布地奈德治疗无反应者，可能需要考虑其他诊断，包括并存的 IBS。
