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Cab-Serrano BP, Ayuso-Diaz VM, Torres-Valdes EA, Chacon-Pacho RE, Moreno-Enriquez A. Giant Mesenteric Cavernous Lymphangioma in an Adult as a Cause of Chronic Intestinal Subocclusion: A Case Report. Cureus 2025; 17:e78934. [PMID: 40091914 PMCID: PMC11910691 DOI: 10.7759/cureus.78934] [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: 02/12/2025] [Indexed: 03/19/2025] Open
Abstract
Giant mesenteric cavernous lymphangioma is a rare lymphatic malformation in adults and an uncommon cause of chronic bowel obstruction. Although these lesions are typical of the paediatric population, their occurrence in adults poses significant diagnostic challenges due to their clinical and radiological similarity to malignant neoplasms such as liposarcoma, lymphoma and gastrointestinal stromal tumours. This report describes the case of a 58-year-old man with chronic bowel obstruction in whom the initial diagnosis suggested malignancy. After surgical resection, histopathological examination confirmed the benign nature of the lesion and identified it as a mesenteric cavernous lymphangioma. The present case contributes to the understanding of this entity in adults and emphasises its importance in the differential diagnosis of chronic intestinal obstruction. It also highlights the need to integrate clinical, radiological and anatomopathological evaluation to establish an accurate diagnosis and guide appropriate therapeutic decisions, with direct implications for the management of complex abdominal pathologies.
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Affiliation(s)
- Barbara P Cab-Serrano
- Surgery, Hospital Regional Elvia Carrillo Puerto, Institute for Social Security and Services for State Workers (ISSSTE), Yucatán, MEX
| | - Victor M Ayuso-Diaz
- Clinical Recruitment, Medical Care and Research, Yucatán, MEX
- Surgery, Elvia Carrillo Puerto Regional Hospital, Yucatán, MEX
- Genomic-Metabolic Unit, Marista University of Mérida, Yucatán, MEX
| | - Eduardo A Torres-Valdes
- Surgery, Hospital Regional Elvia Carrillo Puerto, Institute for Social Security and Services for State Workers (ISSSTE), Yucatán, MEX
| | - Ricardo E Chacon-Pacho
- Surgery, Hospital Regional Elvia Carrillo Puerto, Institute for Social Security and Services for State Workers (ISSSTE), Yucatán, MEX
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2
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Ono H, Honda S, Miyagi H, Minato M, Ara M, Kondo T, Okumura K, Okada T, Taketomi A. Management of severe symptomatic abdominal lymphatic malformation complicated by abscess formation, protein-losing gastroenteropathy, and bleeding. ANNALS OF PEDIATRIC SURGERY 2022. [DOI: 10.1186/s43159-022-00230-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/23/2022] Open
Abstract
Abstract
Background
The optimal strategy for the management of patients with severe symptomatic abdominal lymphatic malformation (ALM) complicated by abscess formation, protein-losing gastroenteropathy, and bleeding has not yet been established. The present study aimed to determine an appropriate management for patients with severe symptomatic ALM, particularly for those with abdominal complications.
Materials and methods
Eight infants and young adults who underwent emergency surgery for ALM in our department, between the years 1997 and 2020, were selected for the study. We also evaluated and compared the operative procedures, operative timing, and postoperative surgical outcomes.
Results
Emergency resection was performed in all patients with bleeding. Some patients presented with ALMs that invaded the adjacent organs. Resections that included the involved organs were necessary to achieve full resolution in three patients. After evaluating the diagnostic modalities for symptomatic ALM, we also performed elective surgery for patients without bleeding.
Conclusions
Clinicians should be aware of severe symptomatic ALM with or without bleeding, as well as its associated complications, in order to select the best surgical management plan.
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3
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Zhao N, Fu Y, Wang Z, An Q, Jia W. Case report: Submucosal cavernous lymphangioma causing jejuno-jejunal intussusception in an adult. Front Surg 2022; 9:953840. [PMID: 36338654 PMCID: PMC9632977 DOI: 10.3389/fsurg.2022.953840] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/26/2022] [Accepted: 09/05/2022] [Indexed: 04/22/2024] Open
Abstract
Cavernous lymphangioma often occurs in the head, neck, trunk, and extremities of infants and children, and it is rare to cause a small intestine intussusception in adults. In this case, a 32-year-old woman presented with abdominal pain, vomiting, and a 5 cm × 5 cm abdominal mass on the left side of the abdomen. Laboratory tests showed anemia and CT showed small intestinal intussusception. After conservative treatments, her symptoms disappeared. However, 18F-FDG PET/CT suggested malignancy and her symptoms reappeared after eating something. Segmental jejunal resection was performed and pathology showed submucosal cavernous lymphangioma. At the 1-year follow-up, the patient was asymptomatic. Then this paper reviewed the literature on small intestinal cavernous lymphangioma in adults and found that this is the first English case report of intussusception caused by a jejunal submucosal cavernous lymphangioma in an adult. Current problem is that adult intussusception and intestinal lymphangioma are difficult to diagnose preoperatively. Imaging techniques such as tomography and PET/CT aid in the diagnosis of these benign lesions. Surgical resection was considered to be the required treatment and seems to have had no recurrence in adults according to the literature.
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Affiliation(s)
- Ning Zhao
- Department of General Surgery, Department of Gastrointestinal Surgery, Beijing Hospital, National Center of Gerontology, Institute of Geriatric Medicine, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing, China
| | - Yuhang Fu
- Department of General Surgery, Peking University Fifth School of Clinical Medicine, Beijing Hospital, Beijing, China
| | - Zhongzheng Wang
- Department of General Surgery, Peking University Fifth School of Clinical Medicine, Beijing Hospital, Beijing, China
| | - Qi An
- Department of General Surgery, Department of Gastrointestinal Surgery, Beijing Hospital, National Center of Gerontology, Institute of Geriatric Medicine, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing, China
| | - Wenzhuo Jia
- Department of General Surgery, Department of Gastrointestinal Surgery, Beijing Hospital, National Center of Gerontology, Institute of Geriatric Medicine, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing, China
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4
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Laparoscopic Resection for Lymphangioma Originating From Small Intestine: A Case Report. Int Surg 2022. [DOI: 10.9738/intsurg-d-20-00018.1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022] Open
Abstract
Background
Lymphangioma originating from the small intestinal wall is a very rare finding. There have been only 15 case reports on the disease. Here, we report a case of lymphangioma in the small intestine that was resected laparoscopically. We also provide a literature review.
Case Presentation
A 37-year-old man came to our hospital with a chief complaint of abdominal pain. Physical examination showed mild tenderness in the lower abdomen, but rebound tenderness or guarding was absent. A contrast-enhanced computed tomography scan showed local poor enhancement in distal ileum, suggesting the possibility of mucous necrosis. Conservative therapy was started. On the eighth hospital day, although his abdominal symptoms improved, a follow-up computed tomography scan showed no improvement in the distal ileum lesion. Laparoscopic tumor resection was performed. The patient's postoperative course was unremarkable, and he was discharged on postoperative day 7. The pathologic result of the surgical specimen was lymphangioma of the ileum. The patient is still alive with no sign of recurrence 19 months after surgery.
Conclusion
Lymphangioma originating in the small intestinal wall is a very rare disease. Laparoscopic resection seems to be a preferable method for surgery.
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5
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Alhasan AS, Daqqaq TS. Extensive abdominal lymphangiomatosis involving the small bowel mesentery: A case report. World J Clin Cases 2021; 9:9990-9996. [PMID: 34877341 PMCID: PMC8610896 DOI: 10.12998/wjcc.v9.i32.9990] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/26/2021] [Revised: 07/16/2021] [Accepted: 07/29/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Abdominal lymphangiomatosis is a rare benign condition accounting for less than 1% of all the cases of lymphangiomatosis. Management usually involves radical surgical excision; however, depending upon the extent of involvement, patient condition, and absence of complications, conservative management can be also considered.
CASE SUMMARY We present the case of a 32-year-old male who presented with short onset abdominal pain and melena. Physical examination findings were within normal limits, except for left lower abdominal tenderness. Upper gastrointestinal endoscopy was within normal limits. Abdominal and pelvic ultrasound and computed tomography (CT) scan revealed numerous, variably-sized cystic lesions within the abdominal cavity, exclusively and extensively affecting the small bowel mesentery with sparing of the retroperitoneum. The diagnosis was confirmed by CT and cytological examination. Radical surgical excision was technically impossible in this patient because of the extensive involvement of the mesentery; therefore, the patient was managed conservatively.
CONCLUSION Extensive and exclusive small bowel mesentery involvement in abdominal lymphangiomatosis is rare. Imaging modalities play an important role in establishing the diagnosis and conservative management can be considered when surgery is technically impossible.
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Affiliation(s)
- Ayman S Alhasan
- Department of Radiology and Medical Imaging, College of Medicine Taibah University, Madinah 42353, Saudi Arabia
| | - Tareef S Daqqaq
- Department of Radiology and Medical Imaging, College of Medicine Taibah University, Madinah 42353, Saudi Arabia
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6
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Mohammed AA, Musa DH. Lymphangioma of the ileum causing acute abdominal pain in an adult, a very rare finding during surgery; Case report with literature review. Int J Surg Case Rep 2020; 66:319-321. [PMID: 31901559 PMCID: PMC6940693 DOI: 10.1016/j.ijscr.2019.12.031] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/21/2019] [Revised: 12/13/2019] [Accepted: 12/18/2019] [Indexed: 11/21/2022] Open
Abstract
Lymphangioma of the gastrointestinal tract is very rare, 75 % of the lesions affect the neck and 20 % affect the axillary region. The majority of cases are asymptomatic but some cases presented with emergency presentations. Complete surgical resection is the main form of therapy whenever possible. Introduction Lymphangioma of the gastrointestinal tract is very rare, 75 % of the lesions affect the neck and 20 % affect the axillary region, the rest are seen in the mediastinum, the visceral organs, the retroperitoneum, the mesentery, or in the bones. Lymphangioma of the gastrointestinal tract comprises less than 1 %. The majority of cases are asymptomatic but some cases presented with emergency presentations. Case presentation A 31-year-old male presented to emergency department complaining of central abdominal pain for 2 days, with nausea, dyspnea but no vomiting. The abdominal examination revealed a mildly distended abdomen, with guarding and tenderness mainly in the right lower abdominal quadrant. There were no any palpable masses or organ enlargement. The white blood cells count was elevated (14,000 c/mm). Abdominal ultrasound showed an evidence of 10*7 cm multiloculated thick walled lesion in the right lower abdomen, suggesting the possibility of an appendicular abscess. The patient was admitted for 2 days with little clinical improvement. Therefore, a laparotomy was performed at which there were 2 yellowish cystic lesions 60 cm from the ileocecal valve that were arising from each side of the small bowel and related to its mesentery causing luminal narrowing. Resection was done with end-end anastomosis. The histopathology results revealed a diagnosis of ill lymphangioma of the ileum. Conclusion Complete surgical resection is the main form of therapy whenever possible, sometimes is impossible when there is local invasion. When complete resection is performed long term follow up is not recommended as the recurrence is very rare.
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Affiliation(s)
- Ayad Ahmad Mohammed
- Department of Surgery, College of Medicine, University of Duhok, Kurdistan Region, Iraq.
| | - Dildar Haji Musa
- Department of Surgery, College of Medicine, University of Duhok, Kurdistan Region, Iraq
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7
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Tan B, Zhang SY, Wang YN, Li Y, Shi XH, Qian JM. Jejunal cavernous lymphangioma manifested as gastrointestinal bleeding with hypogammaglobulinemia in adult: A case report and literature review. World J Clin Cases 2020; 8:140-148. [PMID: 31970180 PMCID: PMC6962067 DOI: 10.12998/wjcc.v8.i1.140] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/22/2019] [Revised: 11/03/2019] [Accepted: 11/15/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Lymphangioma is a benign lesion that rarely involves the gastrointestinal tract, especially in adults. Small bowel lymphangioma is a rare cause of gastrointestinal bleeding. Here, we report a case of an adult diagnosed with jejunal lymphangioma presenting with melena, anemia and hypogammaglobulinemia. We also summarize and analyze all 23 reported cases from 1961 to 2019, and propose an algorithm for identification and management of small bowel lymphangioma.
CASE SUMMARY A case of a 29-year-old woman presented with persistent melena and iron-deficiency anemia, accompanied by hypogammaglobulinemia. No lesions were found in the initial workup with esophagogastroduodenoscopy, colonoscopy and computed tomography (CT) enterography. Ultimately, capsule endoscopy and double-balloon enteroscopy revealed a 3 cm × 2 cm primary lesion with intensive white lymphatic dilatatory changes and visible fresh blood stains, accompanied by a small satellite lesion. The patient underwent complete surgical resection of these lesions, and histopathological examination confirmed a diagnosis of cavernous lymphangioma of the jejunum. The patient showed no evidence of disease at the time of this report.
CONCLUSION We recommend CT, capsule endoscopy and enteroscopy to identify the lesions of lymphangioma. Laparoscopic surgery with histological diagnosis is an ideal curative method.
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Affiliation(s)
- Bei Tan
- Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Sheng-Yu Zhang
- Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
| | - Yi-Nan Wang
- Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing 100730, China
| | - Yuan Li
- Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing 100730, China
| | - Xiao-Hua Shi
- Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing 100730, China
| | - Jia-Ming Qian
- Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
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8
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Lymphangioma of the Small Intestine Case Report and Review of the Literature. SERBIAN JOURNAL OF EXPERIMENTAL AND CLINICAL RESEARCH 2019. [DOI: 10.2478/sjecr-2018-0001] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022] Open
Abstract
Abstract
Lymphangioma usually occurs in children and usually involves the skin. Mesenteric lymphangiomas are extremely rare in adults (1,2,3). Lymphangioma of the small-bowel mesentery is rare, representing less than 1% of all lymphangiomas (4).
We report a case of a 62-year-old female who presented with abdominal pain, discomfort, nausea and vomiting. Pre-operative tests including abdominal ultrasonography and magnetic resonance imaging were performed, but they could not accurately determine the nature of the tumour. Laparotomy was performed; the tumour was excised completely, and a large cystic tumour of the small bowel mesentery was found. Histopathological examination diagnosed the tumour as a cystic lymphangioma. Lymphangiomas are extremely rare, especially in the abdomen of adults, and are asymptomatic for the most part; they often present as acute abdominal conditions, causing life-threatening complications such as secondary infection, rupture with haemorrhage, and volvulus or intestinal obstruction when the tumour increases in size, requiring emergent surgery. Lymphangioma is often difficult to diagnose, and surgical resection is selected in many cases for both diagnosis and treatment.
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9
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A rare case of retroperitoneal and mesenteric lymphangiomatosis. Radiol Case Rep 2019; 15:11-14. [PMID: 31762861 PMCID: PMC6849486 DOI: 10.1016/j.radcr.2019.09.039] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/03/2019] [Revised: 09/27/2019] [Accepted: 09/30/2019] [Indexed: 12/15/2022] Open
Abstract
Lymphagiomatosis are rare benign malformations of the lymphatic system. They are more commonly seen during childhood and are frequently asymptomatic and incidentally found in the adult patient. We report a case of a 31-year-old male who presented initially with melena. Computer tomography scan revealed multiple confluent, fluid-density lesions encasing the retroperitoneum and mesentery. A laparotomy and incisional biopsy of the mesenteric lesion was performed. Histologic examination demonstrated fibrofatty tissue with prominent, thick-walled endothelial-lined vessels. The histologic and computer tomography findings were consistent with a diagnosis of retroperitoneal and mesenteric lymphangiomatosis. The patient was subsequently discharged home well.
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10
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Tan DTM, Chok AY, Farah BL, Yan YY, Toh EL. Spontaneous partial regression of a microcystic jejunal mesenteric lymphangioma and a proposed management algorithm. BMJ Case Rep 2019; 12:12/11/e231037. [PMID: 31767604 DOI: 10.1136/bcr-2019-231037] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
Mesenteric lymphangiomas are relatively rare, with clinical symptoms ranging from an asymptomatic presentation to an acute abdomen. The natural history and biological behaviour of this entity can range from slow indolent lesions to aggressive tumours with a risk of malignant transformation. Spontaneous regression of a mesenteric lymphangioma is rare. We herein report a case of a jejunal mesenteric lymphangioma that was initially detected incidentally in an asymptomatic patient with a subsequent sudden increase in size with resulting surrounding mass effect after 9 months and a spontaneous partial regression at surgical resection. Our case is the first reported case which outlines a period in the natural history and evolution of microcystic mesenteric lymphangioma, illustrating the sudden enlargement likely attributable to spontaneous and self-limiting haemorrhage and subsequent partial regression at surgical resection. We thenceforth propose a possible management algorithm for adult patients with mesenteric lymphangiomas.
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Affiliation(s)
- Dominic Ti Ming Tan
- College of Medical and Dental Sciences, University of Birmingham, Birmingham, UK .,Department of Surgery, Sandwell and West Birmingham Hospitals NHS Trust, Birmingham, UK
| | - Aik Yong Chok
- Department of Colorectal Surgery, Singapore General Hospital, Bukit Merah, Singapore
| | - Benjamin Livingston Farah
- Division of Pathology, Department of Anatomical Pathology, Singapore General Hospital, Bukit Merah, Singapore
| | - Yet Yen Yan
- Department of Radiology, Changi General Hospital, 2 Simei Street 3, Singapore
| | - Ee-Lin Toh
- Department of Colorectal Surgery, Singapore General Hospital, Bukit Merah, Singapore
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11
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Spolianski G, Kopelman D, Kimmel B, Hatoum OA. Laparoscopic exploration and treatment for a mesenteric cyst lymphangioma in adults. ANZ J Surg 2018; 89:1334-1336. [DOI: 10.1111/ans.14711] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/25/2018] [Accepted: 05/01/2018] [Indexed: 11/28/2022]
Affiliation(s)
- Gregory Spolianski
- Department of Surgery B, Faculty of Medicine, Emek Medical CenterTechnion‐Israel Institute of Technology Afula Israel
| | - Doron Kopelman
- Department of Surgery B, Faculty of Medicine, Emek Medical CenterTechnion‐Israel Institute of Technology Afula Israel
| | - Boaz Kimmel
- Department of Surgery B, Faculty of Medicine, Emek Medical CenterTechnion‐Israel Institute of Technology Afula Israel
| | - Ossama A. Hatoum
- Department of Surgery B, Faculty of Medicine, Emek Medical CenterTechnion‐Israel Institute of Technology Afula Israel
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12
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Kumar SS, Das SAP, Kate V. Cystic Lymhangioma of the Lesser Sac in Adult Presenting with Features of Gastric Outlet Obstruction - A Case Report. J Clin Diagn Res 2015; 9:PD15-6. [PMID: 26672967 DOI: 10.7860/jcdr/2015/14789.6830] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/06/2015] [Accepted: 09/18/2015] [Indexed: 11/24/2022]
Abstract
Cystic lymphangioma is a paediatric benign tumour, occurs commonly in head and neck region. Adult intraabdominal lymphangiomas are uncommon and only few cases of lesser sac cystic lymphangioma are reported in the literature. We present a case of lesser sac cystic lymphangioma in a 26-year-old lady who presented with features of gastric outlet obstruction. Clinical examination revealed a 10x10 cm firm, non tender, mobile intraabdominal lump in the epigastrium. An upper gastrointestinal endoscopy showed possible external compression at the body of the stomach. Ultrasound demonstrated a multicystic lesion, with internal echoes in one locule. CECT showed a large multiloculated cystic lesion with hyper-dense content in the lesser sac extending up to the left lobe of the liver and indenting the stomach, lesser sac, and upper border of pancreas. On surgical exploration, a 10X8 cm multi cystic lesion with clear fluid was found in the lesser sac very close to the lesser curvature of stomach. Histopathological examination of the excised lesion showed features of cystic lymphangioma. Though very rare, intraabdominal cystic lymphangiomas should be considered in all atypical cases of gastric outlet obstruction. A reasonable knowledge about these benign lesions will aid in establishing the diagnosis and surgical management.
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Affiliation(s)
- Sathasivam Suresh Kumar
- Assistant Professor, Department of Surgery, Jawaharlal Institute of Postgraduate Medical Education and Research (JIPMER) , Puducherry, India
| | - Sri Aurobindo Prasad Das
- Senior Resident, Department of Surgery, Jawaharlal Institute of Postgraduate Medical Education and Research (JIPMER) , Puducherry, India
| | - Vikram Kate
- Professor, Department of Surgery, Jawaharlal Institute of Postgraduate Medical Education and Research (JIPMER) , Puducherry, India
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13
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Hong IT, Cha JM, Lee JI, Joo KR, Baek IH, Shin HP, Jeon JW, Lim JU. [A Case of Cavernous Lymphangioma of the Small Bowel Mesentery]. THE KOREAN JOURNAL OF GASTROENTEROLOGY 2015; 66:172-5. [PMID: 26387702 DOI: 10.4166/kjg.2015.66.3.172] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/31/2023]
Abstract
Lymphangioma is a benign vascular lesion with characteristics of subepithelial tumor which can proliferate in the lymphatic system. Lymphangioma of the small-bowel mesentery is rare, having been reported in less than 2% of all lymphangiomas. Lymphangioma does not require any specific treatment because it is absolutely a benign tumor. However, surgical exploration is rarely required for cases with disease-related symptoms or complications, or for those misdiagnosed as a malignant lesion. We recently experienced a case of mesenteric cavernous lymphangomas in a 53-year-old female who was misdiagnosed as having a liposarcoma. The final diagnosis was confirmed by a pathologic examination of the specimen that was obtained via laparoscopic exploration. Herein, we report a very rare case of mesenteric cavernous lymphangioma along with a brief review of relevant literature.
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Affiliation(s)
- In Taik Hong
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Jae Myung Cha
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Joung Il Lee
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Kwang Ro Joo
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Il Hyun Baek
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Hyun Phil Shin
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Jung Won Jeon
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
| | - Jun Uk Lim
- Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea
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14
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Hureibi K, Sunidar OA. Mesenteric cystic lymphangioma mimicking malignancy. BMJ Case Rep 2014; 2014:bcr-2014-203560. [PMID: 25178885 DOI: 10.1136/bcr-2014-203560] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
Mesenteric cystic lymphangiomas are benign tumours arising from the mesentery, and have no known aetiology. Patients might be discovered incidentally to have asymptomatic mesenteric cysts, or they can present with symptoms such as pain, nausea and vomiting. A 27-year-old man presented with vague abdominal pain, loss of appetite, postprandial fullness and significant weight loss. There was no lymphadenopathy, and abdominal examination was unremarkable. CT showed a mesenteric mass and a diagnosis of abdominal lymphoma was suggested. There was no evidence of pulmonary tuberculosis on chest X-ray and the purified protein derivative test was negative. On laparotomy, a 5×9×7 cm sessile cyst containing thick white fluid and arising from the ileal mesentery was found and completely removed. Histopathology proved a diagnosis of mesenteric cystic lymphangioma. The patient made uneventful recovery, and was asymptomatic on clinical follow-up after 6 weeks.
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Affiliation(s)
- Khalid Hureibi
- Department of General Surgery, Royal Sussex County Hospital, Brighton, East Sussex, UK
| | - Osama A Sunidar
- Department of General Surgery, Al-Thawra Modern General and Teaching Hospital, Sanaa, Sanaa, Yemen
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15
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16
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Rai P, Rao RN, Chakraborthy SBD. Caecal lymphangioma: a rare cause of gastrointestinal blood loss. BMJ Case Rep 2013; 2013:bcr2013008866. [PMID: 23606386 PMCID: PMC3645253 DOI: 10.1136/bcr-2013-008866] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
Lymphangioma is the malformation of the lymphatic system. Lymphangioma is a benign tumour and most colonic lymphangiomas do not cause symptoms and do not require treatment. Resection is required in patients with bleeding or intussusceptions. We report a case of intestinal lymphangiomatosis in a 31-year-old man who presented with recurrent melena and anaemia, which were diagnosed endoscopically and treated with surgical resection.
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Affiliation(s)
- P Rai
- Department of Gastroenterology, SGPGIMS, Lucknow, Uttar Pradesh, India.
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17
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Morris-Stiff G, Falk GA, El-Hayek K, Vargo J, Bronner M, Vogt DP. Jejunal cavernous lymphangioma. BMJ Case Rep 2011; 2011:bcr.03.2011.4022. [PMID: 22696733 DOI: 10.1136/bcr.03.2011.4022] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022] Open
Abstract
Cavernous lymphangiomas are usually identified in infants and children with the majority of lesions found around the head and neck, trunk or extremities. Tumours affecting the intra-abdominal organs are rare. The authors report a case of small bowel cavernous lymphangioma arising within the jejunum of a 34-year-old woman presenting with dyspnoea and anaemia, and review the existing literature relating to this uncommon tumour.
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Affiliation(s)
- Gareth Morris-Stiff
- Department of Hepato-Pancreato-Biliary Surgery, Cleveland Clinic Foundation, Cleveland, Ohio, USA.
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18
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Kurokawa T, Kanai M, Kaneko Y, Takahashi H, Motohara T. A Case of Lymphangioma of the Jejunum Presenting with Intussusception. ACTA ACUST UNITED AC 2011. [DOI: 10.5833/jjgs.44.1597] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/14/2022]
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19
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Siddique K, Bhandari S, Basu S. Giant mesenteric lymphangioma: a rare cause of a life-threatening complication in an adult. BMJ Case Rep 2010; 2010:2010/sep06_1/bcr0420102896. [PMID: 22778193 DOI: 10.1136/bcr.04.2010.2896] [Citation(s) in RCA: 17] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022] Open
Abstract
Mesenteric cyst lymphangiomas (MCLs) are rare benign tumours of unknown aetiology seen mostly in children. Clinical presentation can be diverse ranging from incidental abdominal cysts to an acute abdomen. A 24-year-old previously fit and healthy young man presented with a 2-week history of cramping, central abdominal pain and vomiting requiring acute hospital admission. He was a diagnostic conundrum despite extensive preoperative investigations. Recurring symptoms required urgent surgical exploration, resection and histological confirmation with a successful outcome. We review the literature and present here this rare case in an adult of MCL that has the potential to grow, invade and develop major life-threatening complications.
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Affiliation(s)
- Khurram Siddique
- Department of General Surgery, William Harvey Hospital, Ashford, UK.
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20
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Limdi JK, Mehdi S, Sapundzieski M, Manu M, Abbasi AM. Cystic lymphangioma of the mesocolon. J Gastrointest Surg 2010; 14:1459-61. [PMID: 20221854 DOI: 10.1007/s11605-010-1176-0] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/16/2009] [Accepted: 02/09/2010] [Indexed: 01/31/2023]
Abstract
A 46-year-old gentleman presented to our hospital with a short story of abdominal pain and distension. On examination, gross ascites was noted and confirmed on subsequent imaging with no other notable features. CT scan after ascitic drainage showed a cystic mass extending from the lower pole of the spleen to the left iliac fossa in keeping with an intraperitoneal cyst. At laparatomy, a cystic lymphangioma was resected. Lymphangiomas are rare benign tumours and are reported to occur preferentially in the neck of axilla in children. Abdominal lymphangiomas are extremely rare particularly in adults but important to recognise due to a potential for serious consequences.
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Affiliation(s)
- Jimmy K Limdi
- The Pennine Acute Hospitals NHS Trust, Manchester, BL9 7TD, UK.
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21
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Alkhalil M, Brough WA, Hadjiloucas I. A rare cause of acute abdominal distension presenting as a surgical emergency. Br J Hosp Med (Lond) 2010; 71:172-3. [PMID: 20220727 DOI: 10.12968/hmed.2010.71.3.46985] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Affiliation(s)
- Mohammad Alkhalil
- Department of Surgery, Macclesfield District General Hospital, Macclesfield
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22
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Wani I. Mesenteric lymphangioma in adult: a case series with a review of the literature. Dig Dis Sci 2009; 54:2758-2762. [PMID: 19142726 DOI: 10.1007/s10620-008-0674-3] [Citation(s) in RCA: 28] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/24/2008] [Accepted: 12/08/2008] [Indexed: 01/18/2023]
Abstract
Mesenteric lymphangioma is a rare benign tumor with nonspecific clinical features. A case series of three adult patients who had mesenteric lymphangioma is reported along with a review of literature. Two patients presented with painless abdominal swelling who were being managed as a case of abdominal tuberculosis and a third one had features of intestinal obstruction. In our cases, preoperative diagnosis of mesenteric lymphangioma was not suspected and all the patients had laparotomy. Peroperative findings revealed mesenteric masses, which were simulating cavitatory form of mesenteric node tuberculosis. It is stressed that mimicking of mesenteric lymphangioma with a cavitatory form of tuberculosis of mesenteric nodes in areas of high prevalence may lead to its misdiagnosis. Histopathology confirmed diagnosis of mesenteric lymphangioma in each case. Although rare and difficult to diagnose preoperatively, surgical resection is to be considered gold standard treatment for mesenteric lymphangioma.
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Affiliation(s)
- Imtiaz Wani
- S.M.H.S Hospital, Shodi Gali, Amira Kada, Srinagar, Kashmir 190009, India.
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23
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Oshita M, Okazaki T, Yamataka A, Kusafuka J, Lane GJ, Matsumoto T, Miyano T. Jejuno-jejunal intussusception secondary to submucosal lymphangioma in a child. Pediatr Surg Int 2005; 21:1001-3. [PMID: 16235059 DOI: 10.1007/s00383-005-1529-z] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
Small bowel submucosal lymphangioma in children is extremely rare. We present the case of a 5-year-old boy with intussusception secondary to a submucosal lymphangioma in the jejunum. To the best of our knowledge, this is the first report of intussusception secondary to a small intestinal submucosal lymphangioma in a child.
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Affiliation(s)
- Masaaki Oshita
- Department of Pediatric General and Urogenital Surgery, Juntendo University School of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo 113-8421, Japan
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24
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Abstract
We herein describe the case of a 27-year-old female, who presented with a large mass of the upper left abdominal cavity discovered incidentally, through an annual health examination. Preoperative studies including abdominal ultrasonography and magnetic resonance imaging were performed, but they could not accurately determine the nature of the tumor. At laparotomy, a large cystic tumor of the small bowel mesentery was found. Histopathologic examination diagnosed the tumor as a cystic lymphangioma. Although lymphangiomas are rare, especially in the abdomen of adults, they may sometimes present as acute abdomen, causing complications that require emergent surgery.
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Affiliation(s)
- Chuang-Wei Chen
- Division of General Surgery, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan, China
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25
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Ugwonali O, Coady M, Saxena R, Robert M, Horowitz N, Topazian M. Intraoperative enteroscopy for diagnosis of a bleeding jejunal lymphangioma. J Clin Gastroenterol 2000; 31:333-5. [PMID: 11129277 DOI: 10.1097/00004836-200012000-00013] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/06/2023]
Abstract
A patient with recurrent gastrointestinal bleeding from a jejunal lymphangioma is described. Multiple preoperative diagnostic studies, including sonde enteroscopy, failed to reveal a bleeding source. A bleeding lymphangioma was subsequently detected by intraoperative enteroscopy of the small bowel. Intraoperative enteroscopy was a useful diagnostic technique in this patient with otherwise unexplained gastrointestinal bleeding.
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Affiliation(s)
- O Ugwonali
- Department of General Surgery, Yale University School of Medicine, New Haven, Connecticut 06520-8019, USA
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26
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Rieker RJ, Quentmeier A, Weiss C, Kretzschmar U, Amann K, Mechtersheimer G, Bläker H, Herwart OF. Cystic lymphangioma of the small-bowel mesentery: case report and a review of the literature. Pathol Oncol Res 2000; 6:146-8. [PMID: 10936792 DOI: 10.1007/bf03032366] [Citation(s) in RCA: 51] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Cystic lymphangioma of the small-bowel mesentery is a rare manifestation of an intraabdominal tumor in elderly patients. We present a case of a small-bowel mesentery lymphangioma, causing fever and chills and present clinical and pathologic features. Furthermore, etiology and differential diagnosis of this tumor are discussed.
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Affiliation(s)
- R J Rieker
- University of Heidelberg, Department of Pathology Pathologisches Institut, Im Neuenheimer Feld 220/221, Heidelberg, D-69120, Germany, ralf_rieker@med
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27
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Kok KY, Mathew VV, Yapp SK. Lymphangioma of the small-bowel mesentery: unusual cause of intestinal obstruction. J Clin Gastroenterol 1997; 24:186-7. [PMID: 9179742 DOI: 10.1097/00004836-199704000-00015] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Affiliation(s)
- K Y Kok
- Surgical Unit, RIPAS Hospital, Bandar Seri Begawan, Brunei
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