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Okuda M, Yoshioka T, Tsunemitsu R, Inoeu H, Inada R. A Case of Colonic Intussusception Caused by Cecal Lymphangioma and Requiring Emergency Surgery. Cureus 2024; 16:e63029. [PMID: 39050313 PMCID: PMC11268082 DOI: 10.7759/cureus.63029] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 06/22/2024] [Indexed: 07/27/2024] Open
Abstract
Lymphangiomas are benign tumors of dilated lymphatic vessels often found in the head, neck, and axilla of children. Lymphangiomas rarely occur in the abdomen, much less in the colon of adults. Colonic lymphangioma can cause symptoms and signs such as abdominal pain and abdominal distension and complications of gastrointestinal bleeding and intussusception. Intussusceptions are rarer in adults than in children. Most cases of intussusception in adults have a distinct cause, and a substantial number are related to malignant tumors. Herein, we report a rare case of ileocecal intussusception caused by cecal lymphangioma that required emergency surgery. A 40-year-old woman presented with severe abdominal pain of a two-day duration. Her abdomen was tender and showed muscle rigidity in the right lower quadrant. Contrast-enhanced CT of the abdomen revealed ileocecal intussusception and a cystic mass, which was the leading point, with no evidence of bowel strangulation. The patient underwent emergency laparoscopic surgery because she had severe abdominal pain and showed peritoneal irritation signs. During surgery, the cecum was found to have invaginated into the ascending colon, almost to the hepatic flexure. Laparoscopic ileocecal resection with central vascular ligation was performed without intraoperative reduction of the intussusception because the cystic lesion could have a malignant component. Upon inspection, the lesion was a 60 mm × 50 mm submucosal mass located in the cecum. It was filled with clear serous fluid and thin walls. Pathology revealed the cystic mass to be a cecal lymphangioma with no evidence of malignancy. The patient was discharged seven days after emergency surgery with no complications. Our case adds to evidence that cecal lymphangiomas can cause colonic intussusception in adults. Although rare, the risk of colonic intussusception must be considered in the management of colonic lymphangiomas.
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Affiliation(s)
- Mitsutoshi Okuda
- Department of Gastroenterological Surgery, Kochi Health Sciences Center, Kochi, JPN
| | - Takahiro Yoshioka
- Department of Gastroenterological Surgery, Kochi Health Sciences Center, Kochi, JPN
| | - Ryosuke Tsunemitsu
- Department of Gastroenterological Surgery, Kochi Health Sciences Center, Kochi, JPN
| | - Hiroaki Inoeu
- Department of Gastroenterological Surgery, Kochi Health Sciences Center, Kochi, JPN
| | - Ryo Inada
- Department of Gastroenterological Surgery, Kochi Health Sciences Center, Kochi, JPN
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Park JH, Park J, Ku GY, Moon DK, Ahn JS, Kim HJ, Kim MJ, Park JW, Ryoo SB, Jeong SY, Park KJ. Cystic lymphangioma causing intussusception of the right colon: a case report and review of current literature. J Surg Case Rep 2023; 2023:rjad628. [PMID: 38026739 PMCID: PMC10663064 DOI: 10.1093/jscr/rjad628] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/15/2023] [Accepted: 11/01/2023] [Indexed: 12/01/2023] Open
Abstract
Among intraabdominal lymphangiomas, colonic lymphangiomas are rare. These cystic tumors are generally asymptomatic and incidentally found but may present with bleeding or obstructive symptoms. Intussusception by such tumors is scarcely reported, with only nine previously reported cases listed in Pubmed. We report a case of a 41-year-old female Asian patient who presented with acute abdomen and was diagnosed with colonic intussusception caused by lymphangioma. She received emergent right hemicolectomy, recovered well without complications, and was discharged on the 5th postoperative day.
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Affiliation(s)
- Jae Hyun Park
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Jesung Park
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Ga Yoon Ku
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Do Kyoon Moon
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Jong Sung Ahn
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Hyo Jun Kim
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Min Jung Kim
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Ji Won Park
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Seung-Bum Ryoo
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Seung-Yong Jeong
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
| | - Kyu Joo Park
- Department of Surgery, Seoul National University College of Medicine, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Korea
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Dessai GS, Soni NK, Pujari S, Prabhu R, Kantharia CV. Cystic Lymphangioma of Rectum-A Case Report and Review of Literature. THE KOREAN JOURNAL OF GASTROENTEROLOGY = TAEHAN SOHWAGI HAKHOE CHI 2023; 82:91-95. [PMID: 37621244 DOI: 10.4166/kjg.2023.057] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/16/2023] [Revised: 07/18/2023] [Accepted: 07/21/2023] [Indexed: 08/26/2023]
Abstract
Colorectal cystic lymphangiomas are rare benign lesions. They are characterized by the presence of either single or multi-cystic spaces lined by endothelium. Though there are multiple case reports of right and transverse colonic lymphangioma; only around 10 cases of lymphangioma of the rectum have been reported. We present a case report of rectal lymphangioma with the relevant literature review.
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Affiliation(s)
- Govindraj S Dessai
- Department of Surgical Gastroenterology, Seth G S Medical College and KEM Hospital, Mumbai, India
| | - Nikhil Kumar Soni
- Department of Surgical Gastroenterology, Seth G S Medical College and KEM Hospital, Mumbai, India
| | - Sharvari Pujari
- Department of Surgical Gastroenterology, Seth G S Medical College and KEM Hospital, Mumbai, India
| | - Ramkrishna Prabhu
- Department of Surgical Gastroenterology, Seth G S Medical College and KEM Hospital, Mumbai, India
| | - Chetan V Kantharia
- Department of Surgical Gastroenterology, Seth G S Medical College and KEM Hospital, Mumbai, India
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Ly MMG, De Robles MS, Mckenzie C, Young CJ. Colonic lymphangioma presenting with intermittent pain and intussusception. J Surg Case Rep 2019; 2019:rjy336. [PMID: 30651966 PMCID: PMC6329364 DOI: 10.1093/jscr/rjy336] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/17/2018] [Accepted: 11/29/2018] [Indexed: 11/13/2022] Open
Abstract
BACKGROUND Cystic lymphangiomas are rare benign tumours and their actual incidence in the colon is unclear. Within the large bowel, these lesions are often submucosal and are incidental findings on colonoscopy. CASE REPORT A 43-year-old man with colonic lymphangioma presented with a 7-month history of abdominal pain and altered bowel habits. Computed tomography showed a cystic mass lesion at the distal descending colon. Colonoscopy revealed a smooth, polypoid mass, initially thought as an inverting diverticulum. He underwent laparoscopic high anterior resection. The pathological analysis revealed the descending colon mass to be a lymphangioma with no evidence of diverticulosis. CONCLUSION There is increasing incidence in lymphangioma due to the increasing accessibility to colonoscopy, particularly for asymptomatic or subacute disease. Although colonoscopic excision, sclerotherapy and use of steroids or fibrin glue have been advocated as possible modes of treatment, surgical excision is still considered to be the treatment of choice.
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Affiliation(s)
- Mark M G Ly
- Department of Colorectal Surgery, Royal Prince Alfred Hospital, Camperdown, New South Wales, Australia
| | - Marie Shella De Robles
- Department of Colorectal Surgery, Royal Prince Alfred Hospital, Camperdown, New South Wales, Australia
| | - Catriona Mckenzie
- Department of Tissue Pathology and Diagnostic Oncology, Royal Prince Alfred Hospital, Camperdown, New South Wales, Australia
| | - Christopher J Young
- Department of Colorectal Surgery, Royal Prince Alfred Hospital, Camperdown, New South Wales, Australia
- Discipline of Surgery, Sydney Medical School, The University of Sydney, New South Wales, Australia
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Kohga A, Kawabe A, Hasegawa Y, Yajima K, Okumura T, Yamashita K, Isogaki J, Suzuki K, Komiyama A. Ileo-ileal intussusception caused by lymphangioma of the small bowel treated by single-incision laparoscopic-assisted ileal resection. World J Gastroenterol 2017; 23:167-172. [PMID: 28104992 PMCID: PMC5221280 DOI: 10.3748/wjg.v23.i1.167] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/27/2016] [Revised: 10/07/2016] [Accepted: 10/31/2016] [Indexed: 02/06/2023] Open
Abstract
Intraabdominal lymphangiomas are uncommon; additionally, those affecting the gastrointestinal tract are rare and account for less than 1% of cases. Intussusception caused by a cystic lymphangioma of the small bowel is extremely rare. The patient was a 20-year-old woman who visited our emergency room with a complaint of abdominal pain. A computed tomography image revealed ileo-ileal intussusception with a leading hypovascular mass measuring 1 cm in a diameter. Single-incision laparoscopic-assisted ileal resection was performed. The surgical specimen consisted of a soft polycystic mass. Macroscopically, a pedunculated polyp with a convolutional pattern was found. Microscopically, the inner surfaces of the cysts were covered with a single layer of endothelial cells. On immunohistochemical examination, the endothelial cells were partially positive for D2-40 and CD34. Smooth muscle cells were also found around the cysts. The lesion was diagnosed as a cystic lymphangioma. Dozens of cases of small bowel lymphangiomas have previously been reported. Of these, cases with intussusception were very rare. This is the first case of small bowel intussusception due to lymphangioma treated by single-incision laparoscopic-assisted surgery.
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Zhu H, Wu ZY, Lin XZ, Shi B, Upadhyaya M, Chen K. Gastrointestinal tract lymphangiomas: findings at CT and endoscopic imaging with histopathologic correlation. ACTA ACUST UNITED AC 2009; 33:662-8. [PMID: 18180982 DOI: 10.1007/s00261-007-9354-6] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
Abstract
OBJECTIVE To evaluate and describe CT and endoscopic imaging findings in patients with pathologically confirmed gastrointestinal tract lymphangiomas. METHODS Findings from imaging examinations in 6 patients with pathologically confirmed lymphangioma were retrospectively reviewed (computed tomographic images obtained in 6 patients and endoscopic images obtained in 4 patients were available for review). Two radiologists evaluated lesion location, size, shape, edge, number, attenuation, the thickness of capsule wall and the degree of enhancement through PACS or workstation. RESULTS Lymphangiomas in gastrointestinal tract showed similar features in CT imaging: Contrast-enhanced CT scan showed the oval submucosal masses with homogeneous low attenuation. The lymphangioma appeared as a well-defined, smoothly marginated and non-enhancing cystic mass with intact overlying intestinal mucosa. In one case, the lymphangiomas were multiple with volvulus. Remarkably, CT imaging showed 2 patients with intussusception due to the mass. Endoscopic photographs manifested multiple colplanate mucosal protrusions in the gastric wall or intestinal wall. One case showed submucosal pedunculated proliferative lesion with adenomatous surface. CONCLUSIONS Lymphangiomas exhibited typical appearances that reflected their cystic pathologic features. They showed similar features in CT imaging: well-demarcated, non-enhancing, homogeneous low attenuation cystic masses with intact overlying mucosa. Endoscopic photographs showed submucosal masses with distension of overlying mucosal vessels.
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Affiliation(s)
- Hui Zhu
- Department of Radiology, Rui Jin Hospital, Shanghai Jiao Tong University School of Medicine, No. 197, The Second Rui Jin Rd, Shanghai, 200025, PR China
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