Primary Intestinal Lymphangiectasia Presenting as Recurrent Chylous Ascites: A Rare Case

Primary intestinal lymphangiectasia (PIL) is a rare protein-losing enteropathy, typically diagnosed in childhood. Adult-onset PIL is exceptionally rare and poses a significant diagnostic challenge, often diagnosed as other gastrointestinal diseases. A 28-year-old female, presented with recurrent chylous ascites, and hypoalbuminemia (2.3 g/dL); no hepatic dysfunction or immunoglobulin deficiency was evident; comprehensive evaluation excluded hepatic, malignant (lymphoma), and infectious etiologies (tuberculosis, filariasis). Past history is significant for a presumptive diagnosis of Crohn's disease, which was initially made based on clinical picture and laboratory finding of elevated fecal calprotectin, however, endoscopy and histology studies were inconclusive. On further investigations, imaging showed colonic wall thickening with mesenteric lymphadenopathy, and ascitic fluid analysis showed chylous ascites without evidence for malignant or infectious etiology. Upper endoscopy revealed multiple white duodenal plaques, and terminal ileal biopsies confirmed notable lymphatic dilation. Treatment was initiated with dietary modification (low-fat, high-protein, medium-chain triglyceride supplementation) and budesonide; the patient showed partial response. Later on, Octreotide therapy was initiated, and led to gradual resolution of ascites. Adult onset PIL is challenging to diagnose, particularly when initially misdiagnosed as an inflammatory bowel disease. For correct diagnosis, thorough evaluation, by histopathology and exclusion of secondary causes, is essential. Dietary therapy is the mainstay of management; additional benefits can be obtained by pharmacologic options like octreotide in refractory cases. This case is among the first reported cases of adult-onset PIL from Palestine, contributing to the limited literature and highlighting the need for heightened clinical awareness of such rare presentations.

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Journal
Journal of Investigative Medicine High Impact Case Reports
Published
2026-04-01
DOI
https://doi.org/10.1177/23247096261440327
Primary Topic
Lymphatic Disorders and Treatments
Type
article
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article

Primary Intestinal Lymphangiectasia Presenting as Recurrent Chylous Ascites: A Rare Case

Motaz Natsheh, F Jobran, Basheer Babaa, Hani Hour et al.
Journal of Investigative Medicine High Impact Case Reports
Lymphatic Disorders and Treatments
article

Primary Intestinal Lymphangiectasia Presenting as Recurrent Chylous Ascites: A Rare Case

Motaz Natsheh, F Jobran, Basheer Babaa, Hani Hour, Saed I Y Attawna, Abdallah Dwayat, Badawi Eltamimi, Malak Ramzy Hroub, Maha Ramzi, Yousef Habes, Omar H. Maree
article en

Abstract

Primary intestinal lymphangiectasia (PIL) is a rare protein-losing enteropathy, typically diagnosed in childhood. Adult-onset PIL is exceptionally rare and poses a significant diagnostic challenge, often diagnosed as other gastrointestinal diseases. A 28-year-old female, presented with recurrent chylous ascites, and hypoalbuminemia (2.3 g/dL); no hepatic dysfunction or immunoglobulin deficiency was evident; comprehensive evaluation excluded hepatic, malignant (lymphoma), and infectious etiologies (tuberculosis, filariasis). Past history is significant for a presumptive diagnosis of Crohn's disease, which was initially made based on clinical picture and laboratory finding of elevated fecal calprotectin, however, endoscopy and histology studies were inconclusive. On further investigations, imaging showed colonic wall thickening with mesenteric lymphadenopathy, and ascitic fluid analysis showed chylous ascites without evidence for malignant or infectious etiology. Upper endoscopy revealed multiple white duodenal plaques, and terminal ileal biopsies confirmed notable lymphatic dilation. Treatment was initiated with dietary modification (low-fat, high-protein, medium-chain triglyceride supplementation) and budesonide; the patient showed partial response. Later on, Octreotide therapy was initiated, and led to gradual resolution of ascites. Adult onset PIL is challenging to diagnose, particularly when initially misdiagnosed as an inflammatory bowel disease. For correct diagnosis, thorough evaluation, by histopathology and exclusion of secondary causes, is essential. Dietary therapy is the mainstay of management; additional benefits can be obtained by pharmacologic options like octreotide in refractory cases. This case is among the first reported cases of adult-onset PIL from Palestine, contributing to the limited literature and highlighting the need for heightened clinical awareness of such rare presentations.

Journal of Investigative Medicine High Impact Case ReportsVol. 14
Al-Quds University (PS), Rafik Hariri University Hospital (LB), Jahra Hospital (KW), Palestine Polytechnic University (PS), Al-Ahli Hospital (QA), Rochester General Hospital (US)
Openalex Percentile: Top 33%
Lymphatic Disorders and Treatments
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