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Published in: Journal of Medical Case Reports 1/2013

Open Access 01-12-2013 | Case report

Primary intestinal lymphangiectasia diagnosed by double-balloon enteroscopy and treated by medium-chain triglycerides: a case report

Authors: Yu Lai, Tao Yu, Xiao-yu Qiao, Li-na Zhao, Qi-kui Chen

Published in: Journal of Medical Case Reports | Issue 1/2013

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Abstract

Introduction

Primary intestinal lymphangiectasia is a disorder characterized by exudative enteropathy resulting from morphologic abnormalities of the intestinal lymphatics. Intestinal lymphangiectasia can be primary or secondary, so the diagnosis of primary intestinal lymphangiectasia must first exclude the possibility of secondary intestinal lymphangiectasia. A double-balloon enteroscopy and biopsy, as well as the pathology can be used to confirm the diagnosis of intestinal lymphangiectasia. A polymeric diet containing medium-chain triglycerides and total parenteral nutrition may be a useful therapy.

Case presentation

A 17-year-old girl of Mongoloid ethnicity was admitted to our hospital with a history of diarrhea and edema. She was diagnosed with protein-losing enteropathy caused by intestinal lymphangiectasia. This was confirmed by a double-balloon enteroscopy and multi-dot biopsy. After treatment with total parenteral nutrition in hospital, which was followed by a low-fat and medium-chain triglyceride diet at home, she was totally relieved of her symptoms.

Conclusion

Intestinal lymphangiectasia can be diagnosed with a double-balloon enteroscopy and multi-dot biopsy, as well as the pathology of small intestinal tissue showing edema of the submucosa and lymphangiectasia. Because intestinal lymphangiectasia can be primary or secondary, the diagnosis of primary intestinal lymphangiectasia must first exclude the possibility of secondary intestinal lymphangiectasia. A positive clinical response to the special diet therapy, namely a low-fat and medium-chain triglyceride diet, can further confirm the diagnosis of primary intestinal lymphangiectasia.
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Literature
2.
go back to reference Mistilis SP, Stephen DD, Skyring AP: Intestinal lymphangiectasia. Mechanism of enteric loss of plasma-protein and fat. Lancet. 1965, 1: 77-81.CrossRefPubMed Mistilis SP, Stephen DD, Skyring AP: Intestinal lymphangiectasia. Mechanism of enteric loss of plasma-protein and fat. Lancet. 1965, 1: 77-81.CrossRefPubMed
3.
go back to reference Jeffries GH, Chapman A, Sleisenger MH: Low-fat diet in intestinal lymphangiectasia. Its effects on albumin metabolism. N Engl J Med. 1965, 270: 761-766.CrossRef Jeffries GH, Chapman A, Sleisenger MH: Low-fat diet in intestinal lymphangiectasia. Its effects on albumin metabolism. N Engl J Med. 1965, 270: 761-766.CrossRef
4.
go back to reference Kuroiwa G, Takayama T, Sato Y, Takahashi Y, Fujita T, Nobuoka A, Kukitsu T, Kato J, Sakamaki S, Niitsu Y: Primary intestinal lymphangiectasia successfully treated with octreotide. J Gastroenterol. 2001, 36: 129-132. 10.1007/s005350170142.CrossRefPubMed Kuroiwa G, Takayama T, Sato Y, Takahashi Y, Fujita T, Nobuoka A, Kukitsu T, Kato J, Sakamaki S, Niitsu Y: Primary intestinal lymphangiectasia successfully treated with octreotide. J Gastroenterol. 2001, 36: 129-132. 10.1007/s005350170142.CrossRefPubMed
5.
go back to reference Toskes P: Gastrointestinal diseases: malabsorption. Cecil Textbook of Medicine. Edited by: Wyngaarden J, Smith L. 1988, Philadelphia: WB Saunders, 732-745. 18 Toskes P: Gastrointestinal diseases: malabsorption. Cecil Textbook of Medicine. Edited by: Wyngaarden J, Smith L. 1988, Philadelphia: WB Saunders, 732-745. 18
6.
go back to reference Aoyagi K, Iida M, Yao T, Matsui T, Okada M, Oh K, Fujishima M: Characteristic endoscopic features of intestinal lymphangiectasia: correlation with histological findings. Hepatogastroenterology. 1997, 44: 133-138.PubMed Aoyagi K, Iida M, Yao T, Matsui T, Okada M, Oh K, Fujishima M: Characteristic endoscopic features of intestinal lymphangiectasia: correlation with histological findings. Hepatogastroenterology. 1997, 44: 133-138.PubMed
7.
go back to reference Aoyagi K, Iida M, Yao T, Matsui T, Okada M, Fujishima M: Intestinal lymphangiectasia: value of double-contrast radiographic study. Clin Radiol. 1994, 49: 814-819. 10.1016/S0009-9260(05)81974-9.CrossRefPubMed Aoyagi K, Iida M, Yao T, Matsui T, Okada M, Fujishima M: Intestinal lymphangiectasia: value of double-contrast radiographic study. Clin Radiol. 1994, 49: 814-819. 10.1016/S0009-9260(05)81974-9.CrossRefPubMed
8.
go back to reference Aoyagi K, Iida M, Matsumoto T, Sakisaka S: Enteral nutrition as a primary therapy for intestinal lymphangiectasia: value of elemental diet and polymeric diet compared with total parenteral nutrition. Dig Dis Sci. 2005, 50: 1467-1470. 10.1007/s10620-005-2863-7.CrossRefPubMed Aoyagi K, Iida M, Matsumoto T, Sakisaka S: Enteral nutrition as a primary therapy for intestinal lymphangiectasia: value of elemental diet and polymeric diet compared with total parenteral nutrition. Dig Dis Sci. 2005, 50: 1467-1470. 10.1007/s10620-005-2863-7.CrossRefPubMed
9.
go back to reference Rust C, Pratschke E, Hartl W, Kessler M, Weibecke B, Sauerbruch T, Paumgartner G, Beuers U: Fibrotic entrapment of the small bowel in congenital intestinal lymphangiectasia. Am J Gastroenterol. 1998, 93: 1980-1983. 10.1111/j.1572-0241.1998.00477.x.CrossRefPubMed Rust C, Pratschke E, Hartl W, Kessler M, Weibecke B, Sauerbruch T, Paumgartner G, Beuers U: Fibrotic entrapment of the small bowel in congenital intestinal lymphangiectasia. Am J Gastroenterol. 1998, 93: 1980-1983. 10.1111/j.1572-0241.1998.00477.x.CrossRefPubMed
10.
go back to reference Mine K, Matsubayashi S, Nakai Y, Nakagawa T: Intestinal lymphangiectasia markedly improved with antiplasmin therapy. Gastroenterology. 1989, 96: 1596-1599.CrossRefPubMed Mine K, Matsubayashi S, Nakai Y, Nakagawa T: Intestinal lymphangiectasia markedly improved with antiplasmin therapy. Gastroenterology. 1989, 96: 1596-1599.CrossRefPubMed
11.
go back to reference Bac DJ, Van Hagen PM, Postema PT, ten Bokum AM, Zondervan PE, van Blankenstein M: Octreotide for protein-losing enteropathy with intestinal lymphangiectasia. Lancet. 1995, 24: 1639-CrossRef Bac DJ, Van Hagen PM, Postema PT, ten Bokum AM, Zondervan PE, van Blankenstein M: Octreotide for protein-losing enteropathy with intestinal lymphangiectasia. Lancet. 1995, 24: 1639-CrossRef
12.
go back to reference Ballinger AB, Farthing MJ: Octreotide in the treatment of intestinal lymphangiectasia. Eur J Gastroenterol Hepatol. 1998, 10: 699-702.PubMed Ballinger AB, Farthing MJ: Octreotide in the treatment of intestinal lymphangiectasia. Eur J Gastroenterol Hepatol. 1998, 10: 699-702.PubMed
13.
go back to reference Rubin DC: Small intestine: anatomy and structural anomalies: Lymphangiectasia. Textbook of Gastroenterology. Edited by: Yamada T. 1999, Philadelphia: Lippincott Williams &Wilkins, 1578-1579. 3 Rubin DC: Small intestine: anatomy and structural anomalies: Lymphangiectasia. Textbook of Gastroenterology. Edited by: Yamada T. 1999, Philadelphia: Lippincott Williams &Wilkins, 1578-1579. 3
14.
go back to reference Goldberg RI, Calleja GA: Protein-losing gastroenteropathies. Boccus Gastroenterology. Edited by: Haubrich WS, Schaffner F, Berk JE. 1995, Philadelphia: WB Saunders, 1072-1086. 5 Goldberg RI, Calleja GA: Protein-losing gastroenteropathies. Boccus Gastroenterology. Edited by: Haubrich WS, Schaffner F, Berk JE. 1995, Philadelphia: WB Saunders, 1072-1086. 5
15.
go back to reference Alfano V, Tritto G, Alfonsi L, Cella A, Pasanisi F, Contaldo F: Stable reversal of pathologic signs of primitive intestinal lymphangiectasia with a hypolipidic, MCT-enriched diet. Nutrition. 2000, 16: 303-304. 10.1016/S0899-9007(00)00223-9.CrossRefPubMed Alfano V, Tritto G, Alfonsi L, Cella A, Pasanisi F, Contaldo F: Stable reversal of pathologic signs of primitive intestinal lymphangiectasia with a hypolipidic, MCT-enriched diet. Nutrition. 2000, 16: 303-304. 10.1016/S0899-9007(00)00223-9.CrossRefPubMed
Metadata
Title
Primary intestinal lymphangiectasia diagnosed by double-balloon enteroscopy and treated by medium-chain triglycerides: a case report
Authors
Yu Lai
Tao Yu
Xiao-yu Qiao
Li-na Zhao
Qi-kui Chen
Publication date
01-12-2013
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2013
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/1752-1947-7-19

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