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[胶原性口炎性腹泻]

[Collagenous sprue].

作者信息

Hafkemeyer P, Herbst E, Köhler G, Fuhrmann K, Kreisel W

机构信息

Abteilung Innere Medizin II, Universität, Freiburg.

出版信息

Dtsch Med Wochenschr. 1995 Oct 20;120(42):1430-4. doi: 10.1055/s-2008-1055495.

DOI:10.1055/s-2008-1055495
PMID:7555672
Abstract

HISTORY AND FINDINGS

A 45-year-old man with type I diabetes mellitus was admitted to hospital because of colicky abdominal pain and 5-6 watery stools daily. Upper gastrointestinal endoscopy showed nearly total atrophy of the villi in the duodenum and jejunum suggesting coeliac disease. However, gluten-free diet for 2 weeks brought no improvement. Another examination of the biopsy 6 weeks after the first endoscopy revealed extensive collagen deposition in the lamina propria of the small intestine, giving the diagnosis of collagenous sprue.

TREATMENT AND COURSE

Parenteral nutrition, lactulose, cisapride, cholestyramine, doxycycline, paromomycin, vancomycin and octreotide failed to affect the loss of fluid from the gut which 12 weeks after admission had increased to 221 daily. However, it was stopped after prednisolone was administered (100mg daily). 7 months after starting the steroid treatment the collagen layer had disappeared and the villous atrophy had partially regressed. Over the next 6 months the prednisolone dosage was decreased to 10 mg daily. Shortly thereafter a perimembranous glomerulonephritis occurred, with proteinuria (up to 60 g/d) and oedema. It regressed to 6 g/d when the steroid dose was increased and cyclosporin, 0.5 g/d, had been added. On maintenance dosage of cyclosporin the histological and clinical remission of the collagenous sprue has now lasted for over 2 years.

CONCLUSIONS

This case suggests that steroid administration is an effective treatment of collagenous sprue. The presence of diabetes and other immune-related diseases in this case also suggests that an immunological mechanism may play a causative role in collagenous sprue.

摘要

病史与检查结果

一名45岁的I型糖尿病男性因腹部绞痛和每日5 - 6次水样便入院。上消化道内镜检查显示十二指肠和空肠绒毛几乎完全萎缩,提示乳糜泻。然而,2周的无麸质饮食并未带来改善。首次内镜检查6周后对活检组织的再次检查显示小肠固有层有广泛的胶原沉积,从而诊断为胶原性口炎性腹泻。

治疗过程

肠外营养、乳果糖、西沙必利、考来烯胺、强力霉素、巴龙霉素、万古霉素和奥曲肽均未能改善肠道液体丢失,入院12周后肠道液体丢失增加至每日221毫升。然而,在给予泼尼松龙(每日100毫克)后,液体丢失停止。类固醇治疗开始7个月后,胶原层消失,绒毛萎缩部分消退。在接下来的6个月里,泼尼松龙剂量减至每日10毫克。此后不久,发生了膜性肾小球肾炎,伴有蛋白尿(高达60克/天)和水肿。当增加类固醇剂量并加用环孢素(每日0.5克)后,蛋白尿降至6克/天。目前,在环孢素维持剂量下,胶原性口炎性腹泻的组织学和临床缓解已持续超过2年。

结论

该病例表明类固醇给药是治疗胶原性口炎性腹泻的有效方法。该病例中存在糖尿病和其他免疫相关疾病也提示免疫机制可能在胶原性口炎性腹泻中起致病作用。

相似文献

1
[Collagenous sprue].[胶原性口炎性腹泻]
Dtsch Med Wochenschr. 1995 Oct 20;120(42):1430-4. doi: 10.1055/s-2008-1055495.
2
[Long-term collagenous sprue--remission with a gluten-free diet].[长期胶原性口炎性腹泻——无麸质饮食缓解]
Z Gastroenterol. 1999 Dec;37(12):1163-8.
3
[Collagenous sprue--a rare form of celiac disease in adulthood].胶原性口炎性腹泻——成人乳糜泻的一种罕见形式
Z Gastroenterol. 1993 Feb;31(2):144-6.
4
Non-gluten sensitivity-related small bowel villous flattening with increased intraepithelial lymphocytes: not all that flattens is celiac sprue.非麸质敏感性相关的小肠绒毛变平伴上皮内淋巴细胞增多:并非所有变平的都是乳糜泻。
Am J Clin Pathol. 2004 Apr;121(4):546-50. doi: 10.1309/10FC-NCTC-56NM-N0YE.
5
Collagenous enterocolitis: a case of collagenous colitis with involvement of the small intestine.胶原性小肠结肠炎:1例累及小肠的胶原性结肠炎病例。
Am J Gastroenterol. 1988 Jul;83(7):767-71.
6
Collagenous gastritis: histopathologic features and association with other gastrointestinal diseases.胶原性胃炎:组织病理学特征及其与其他胃肠道疾病的关联。
Am J Surg Pathol. 2009 May;33(5):788-98. doi: 10.1097/PAS.0b013e318196a67f.
7
[Collagenous sprue. Clinical case].
Rev Med Chil. 1998 Sep;126(9):1100-2.
8
TNF-alpha antibody treatment in refractory collagenous sprue: report of a case and review of the literature.难治性胶原性口炎的肿瘤坏死因子-α抗体治疗:一例报告及文献复习
Z Gastroenterol. 2009 Jun;47(6):575-8. doi: 10.1055/s-0028-1109057. Epub 2009 Jun 16.
9
A highly accurate method for monitoring histological recovery in patients with celiac disease on a gluten-free diet using an endoscopic approach that avoids the need for biopsy: a double-center study.一种使用内镜方法监测乳糜泻患者在无麸质饮食时组织学恢复情况的高度准确的方法,该方法无需活检:一项双中心研究。
Endoscopy. 2007 Jan;39(1):46-51. doi: 10.1055/s-2006-945044.
10
[Celiac disease with subepithelial deposition of collagen].伴有上皮下胶原沉积的乳糜泻
Ugeskr Laeger. 2000 Jun 19;162(25):3615-6.

引用本文的文献

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Collagenous sprue: a case report and literature review.胶原性口炎性腹泻:一例病例报告及文献综述。
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2
[Recurrent tetany as the first symptom of late manifesting celiac disease].
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