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Found 6 results

  1. Celiac.com 07/11/2016 - Collagenous sprue is a rare form of small bowel enteropathy characterized by a thickened basement membrane and is considered to be directly related to celiac disease. Doctors have numerous treatment strategies for celiac sprue, but there is currently no effective standardized therapy. One medical team recently described four cases of celiac sprue and proposes thioguanine (6-TG) treatment, based on their results. The research team included Tom van Gils, Tine van de Donk, Gerd Bouma, Foke van Delft, E Andra Neefjes-Borst, and Chris JJ Mulder. They are variously affiliated with the Department of Gastroenterology and Hepatology, VU University Medical Center, Amsterdam, The Netherlands, Department of Pathology, VU University Medical Center, Amsterdam, The Netherlands. The team reviewed 4 cases of celiac sprue. They got their data from the prospective database of patients referred to their celiac centre. The team had an expert pathologist evaluate the small bowel biopsies. None of the patients had ever shown celiac-specific antibodies, and all were negative for HLA-DQ2 and HLA-DQ8 phenotype. Three patients were treated with a combination of 6-TG and budesonide, and 1 patient received 6-TG only. All patients improved remarkably. They found normalized thickening of the basement membrane in 2 patients, and complete histological improvement, including full recovery of villi, in 1 patient. In the third patient, the thickened basement membrane was only very focally recognized. The thickened membrane remained in the last patient, likely due to the short follow-up time. Celiac sprue should be separated from celiac disease. Based on the lack of typical HLA phenotyping and the absence of celiac-specific antibodies, there seems to be no relation with celiac disease in these four patients. A promising treatment option might be 6-TG with or without budesonide. Larger study groups are needed to develop an effective standardized treatment for celiac sprue. This is exciting for folks with celiac sprue, as they previously had no good treatment options at all. Source: BMJ Open Gastro 2016; 3:e000099. doi:10.1136/bmjgast-2016-000099
  2. Collagenous Sprue is a distinctive lesion of the intestinal mucosa associated with progressive malabsorption. The intestinal pathology is initially identified with the characteristic flat lesion of untreated Celiac Sprue. Thereafter, bands of eosinophilic hyaline material within the lamina propria become increasingly apparent. As the disease progresses, the mucosa becomes progressively thinner. Therapy, including the gluten-free diet, does not help. Some cases currently designated refractory or unclassified Sprue many prove to be Collagenous Sprue.
  3. Celiac.com 12/07/2009 - Collagenous sprue is associated with high morbidity, but the etiology of this condition is poorly understood. There is little data concerning the pathological and clinical manifestations of patients with collagenous sprue. The research team set out shed some light on the etiology, disease manifestations and outcomes of collagenous sprue. A team of researchers recently undertook a clinico-pathological study of 19 patients with collagenous sprue and found that the condition does not always end badly for the patient. The research team was made up of Efsevia Vakiani, Carolina Arguelles-Grande, Mahesh M Mansukhani, Suzanne K Lewis, Heidrun Rotterdam, Peter H Green and Govind Bhagat. They are associated with either the Department of Pathology at New York's Memorial Sloan-Kettering Cancer Center, or with Columbia University's Department of Medicine or of Pathology. The team searched their departmental database covering the periods from 1999–2008 to identify cases of collagenous sprue and to gather clinical and lab data. The team evaluated small bowel histology, including thickness of sub-epithelial collagen, intra-epithelial lymphocyte phenotype and results of T-cell clonality assays. The found nineteen patients (15 women, 4 men, age 22–80 years, mean 57 years). Seventeen (89%) suffered from celiac disease and two from unclassified sprue. 9 of 17 (53%) celiac disease patients had refractory disease; 5 of 15 (33%) presented atypically without diarrhea, including 2 of 6 (33%) with active (untreated) celiac disease, and 3 of 9 (33%) with refractory celiac disease. They found autoimmune disorders in 12 of 19 (63%) patients and microscopic colitis (n¼7), lymphocytic gastritis (n¼2) or collagenous gastritis (n¼2) in nine patients. Thickness of subepithelial collagen increase varied from mild (n¼6), moderate (n¼10), or marked (n¼3), and villous atrophy from total (n¼13) to subtotal (n¼6). In no case did they find phenotypically aberrant intraepithelial lymphocytes. The only patient with refractory celiac disease type II showed a dominant T-cell clone with polymerase chain reaction analysis. 7 of 11 (64%) patients showed histological improvement. Overall, 8 of 19 (42%) responded favorably to a gluten-free diet, including 2 of 9 (22%) with refractory celiac disease. 10 of the 19 patients responded to immuno-modulatory therapy, including 6 of 9 (67%) with refractory celiac disease. Only one patient died from the effects of refractory celiac disease. No patient developed lymphoma. The vast majority of patients with collagenous sprue did have celiac disease. Even though numerous patients required immuno-modulatory therapy to control symptoms, many responded to gluten-free diet alone. The researchers conclude that most collagenous sprue patients have relatively good clinical outcomes. Source: Modern Pathology 23 October 2009; doi:10.1038/modpathol.2009.151
  4. Celiac.com 05/04/2010 - A team of clinicians recently set out to assess the effectiveness of treating collagenous sprue with a combination of gluten-free diet and steroids. The team was made up of Alberto Rubio-Tapia, Nicholas J. Talley, Suryakanth R. Gurudu, Tsung-Teh Wu, and Joseph A. Murray. They are affiliated variously with the Division of Gastroenterology and Hepatology of the Mayo Clinics in Scottsdale, Arizona, Jacksonville, Florida, and Rochester, Minnesota, and the Division of Anatomic Pathology in Rochester Mayo Clinic. Deposits of subepithelial collagen that form a distinctive band in the small bowel are one of the clinical hallmarks of collagenous sprue. For the study, the team evaluated clinical characteristics, treatments, and outcomes of patients with collagenous sprue. The team looked at medical records for thirty patients with collagenous sprue from the Mayo Clinics from Scottsdale, Jacksonville, and Rochester, for the periods covering 1993 and 2009. 21 of the patients were female (70%), ranging in age from 53–91 years. The majority of patients suffered from severe diarrhea and weight loss. However, collagenous spore is commonly associated with collagen deposits or chronic inflammation in other parts of the gastrointestinal tract, as well as other immune-mediated disorders. 16 patients (53%) were hospitalized to treat dehydration, while 21 patients (70%) suffered from associated immune-mediated diseases, the most common of which was celiac disease. Other common associated diseases included microscopic colitis, hypothyroidism, and autoimmune enteropathy. Subjects showed subepithelial layers of collagen deposits in the small bowel ranging from 20 –56.5μm, and averaging 29 μm thickness. Eight patients showed subepithelial collagen deposits in the colon or stomach. 24 patients (80%) showed a positive clinical response to treatment with a combination of a gluten-free diet and immunosuppressive drugs. Nine patients showed confirmed histologic improvement, while five patients experienced complete remission. Of two patients who died, one succumbed to complications from collagenous sprue, while one died of another illness. Most patients with collagenous sprue show a positive clinical response to a combination of gluten-free diet and steroids. Source: Clinical Gastroenterology and Hepatology 2010;8:344–349. doi:10.1016/j.cgh.2009.12.023
  5. Celiac.com 02/05/2010 - Collagenous sprue is classically understood as a disorder of the small intestinal mucosa marked by persistent diarrhea, severe malabsorption with multiple nutrient deficiencies, and progressive weight loss. H. J. Freeman of the Department of Medicine, University of British Columbia Hospital, Vancouver, BC, Canada offers an update on collagenous sprue. Patients with collagenous sprue typically show a severe to variably severe "flattened" mucosal biopsy lesion with distinctive sub-epithelial deposits in the lamina propria region. These deposits contain collagens, as demonstrated by both histochemical stains and ultrastructural studies. Moreover, permanent disappearance of these deposits after resection of a localized colon cancer suggests that this disorder might actually involve a para-neoplastic morphologic marker of an occult malignancy. In collagenous sprue cases, physicians often first consider a diagnosis of simple celiac disease, until the patient fails to respond to a gluten-free diet. Recent studies portray a close association between collagenous sprue and celiac disease, sometimes with concomitant T-cell enteropathy. A number of studies demonstrate gastric and/or colonic associations with the unusual inflammatory mucosal process in collagenous sprue, which suggests that the condition may be more complex and have more varied contributing causes than presently understood. Source: World Journal of Gastroenterology. 2010 Jan 21; 16(3):296-8
  6. Celiac that do not remain on a gluten-free diet can develop Refractory Sprue. Refractory Sprue and Collagenous Sprue patients who initially respond to a gluten-free diet many subsequently relapse despite maintaining their diet. Such patients are then refractory to further dietary therapy. In contrast, others are refractory to dietary therapy from its inception and, assuming they are truly on a gluten-free diet, may not have celiac disease; these patients are said to have unclassified Sprue. Some refractory patients with celiac disease, typical or atypical, respond to treatment with corticosteroids or other immunosuppressive drugs. In others, there is no response and malabsorption may be progressive. Collagenous Sprue is characterized by the development of a thick band of collagen-like material directly under the intestinal epithelial cells and has been regarded by some as a separate entity from celiac disease. However, subepithelial collagen deposition has been noted in up to 36% of patients with classic Celiac Disease and in Tropical Sprue. Although individuals with large amounts of subepithelial collagen may be refractory to therapy, the presence of collagen does not , a riori, preclude a successful response to a gluten-free diet. Collagenous colitis accompanying celiac disease also has been observed and would be considered in the diagnosis of diarrhea occurring in celiac disease patients on a gluten-free diet.
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