Volume 83, Issue 7 (October 2025)
Tehran Univ Med J 2025, 83(7): 552-557 |
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Khorsand Zak H, Seyed Sharifi S H, Abbasi Y, Nikdel R. Small intestinal obstructive lymphangiomatosis in an infant: a case report. Tehran Univ Med J 2025; 83 (7) :552-557
URL: http://tumj.tums.ac.ir/article-1-13747-en.html
URL: http://tumj.tums.ac.ir/article-1-13747-en.html
1- Department of Pediatrics, School of Medicine North Khorasan University of Medical Sciences, Bojnurd, Iran.
2- Department of General Surgery, School of Medicine, North Khorasan University of Medical Sciences, Bojnurd, Iran.
3- Department of Pediatrics, School of Medicine North Khorasan University of Medical Sciences, Bojnurd, Iran. ,rasul.nikdel@yahoo.com
2- Department of General Surgery, School of Medicine, North Khorasan University of Medical Sciences, Bojnurd, Iran.
3- Department of Pediatrics, School of Medicine North Khorasan University of Medical Sciences, Bojnurd, Iran. ,
Abstract: (14 Views)
Background: Intestinal lymphangiomatosis is a rare benign disorder of the gastrointestinal tract characterized by abnormal proliferation and dilatation of lymphatic channels within the intestinal wall and mesentery. It accounts for less than 1% of gastrointestinal tumors and predominantly affects the small intestine, particularly in pediatric patients. Clinical manifestations vary widely depending on the size, location, and extent of the lesions, ranging from asymptomatic presentations to acute complications. Due to its rarity and nonspecific features, early diagnosis is often difficult. Imaging modalities, especially ultrasonography, computed tomography, and magnetic resonance imaging, are crucial for initial evaluation and preoperative planning.
Case Presentation: We report an infant who presented with failure to thrive, progressive abdominal distension, and acute symptoms of gastrointestinal obstruction. The patient was admitted to Imam Reza Hospital, Bojnurd, in February 2024. A detailed clinical assessment and laboratory evaluation were performed, followed by abdominal ultrasonography, which revealed a multiloculated cystic mass originating from the small bowel, highly suggestive of a lymphatic malformation. Due to complete small bowel obstruction and worsening clinical status, urgent exploratory laparotomy was undertaken. Intraoperative findings demonstrated a cystic lesion involving a segment of the small intestine, causing total luminal obstruction. Segmental resection of the affected bowel with primary anastomosis was performed, and the resected specimen was submitted for histopathological analysis. The postoperative course was uneventful, with gradual resolution of abdominal distension and successful reintroduction of enteral feeding. Histopathological examination confirmed intestinal lymphangioma, showing dilated lymphatic channels lined by endothelial cells within the intestinal wall, with no evidence of malignancy. During follow-up, the patient demonstrated satisfactory weight gain and normalization of gastrointestinal function, without recurrence or postoperative complications.
Conclusion: Although rare, intestinal lymphangiomatosis should be considered in infants presenting with failure to thrive and signs of bowel obstruction. Timely imaging, particularly ultrasonography, is essential for early diagnosis. Surgical resection remains the definitive treatment in symptomatic cases and is associated with favorable outcomes. Increased clinical awareness can help prevent delayed diagnosis and minimize associated morbidity.
Case Presentation: We report an infant who presented with failure to thrive, progressive abdominal distension, and acute symptoms of gastrointestinal obstruction. The patient was admitted to Imam Reza Hospital, Bojnurd, in February 2024. A detailed clinical assessment and laboratory evaluation were performed, followed by abdominal ultrasonography, which revealed a multiloculated cystic mass originating from the small bowel, highly suggestive of a lymphatic malformation. Due to complete small bowel obstruction and worsening clinical status, urgent exploratory laparotomy was undertaken. Intraoperative findings demonstrated a cystic lesion involving a segment of the small intestine, causing total luminal obstruction. Segmental resection of the affected bowel with primary anastomosis was performed, and the resected specimen was submitted for histopathological analysis. The postoperative course was uneventful, with gradual resolution of abdominal distension and successful reintroduction of enteral feeding. Histopathological examination confirmed intestinal lymphangioma, showing dilated lymphatic channels lined by endothelial cells within the intestinal wall, with no evidence of malignancy. During follow-up, the patient demonstrated satisfactory weight gain and normalization of gastrointestinal function, without recurrence or postoperative complications.
Conclusion: Although rare, intestinal lymphangiomatosis should be considered in infants presenting with failure to thrive and signs of bowel obstruction. Timely imaging, particularly ultrasonography, is essential for early diagnosis. Surgical resection remains the definitive treatment in symptomatic cases and is associated with favorable outcomes. Increased clinical awareness can help prevent delayed diagnosis and minimize associated morbidity.
Type of Study: Case Report |
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