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Belhasan D, Ghalim F. Diffuse abdominal lymphangiomatosis without tumoral masses: a case report. Clin J Gastroenterol 2024; 17:430-433. [PMID: 38460084 DOI: 10.1007/s12328-024-01941-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/08/2023] [Accepted: 02/17/2024] [Indexed: 03/11/2024]
Abstract
Diffuse abdominal lymphangiomatosis is a rare and complex disease. It typically presents with non-specific gastrointestinal symptoms and characteristic cystic lesions or tumoral masses on imaging based on the literature to date. This report presents the rare case of a young man with an atypical form of diffuse abdominal lymphangiomatosis in the complete absence of cystic lesions or lymphangioma tumoral masses, thus presenting a unique diagnostic challenge. It was successively treated by surgery, gastric electrical stimulator, sirolimus, and imatinib.
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Affiliation(s)
- Dina Belhasan
- University of Minnesota School of Medicine, 420 Delaware Street SE, Minneapolis, MN, 55455, USA.
| | - Fahd Ghalim
- Mekka Clinic Digestive Endoscopy Unit, Boulevard de la Mecque, 2000, Casablanca, Morocco
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Liu KR, Zhang S, Chen WR, Huang YX, Li XG. Intermittent melena and refractory anemia due to jejunal cavernous lymphangioma: A case report. World J Gastrointest Surg 2024; 16:1208-1214. [PMID: 38690049 PMCID: PMC11056656 DOI: 10.4240/wjgs.v16.i4.1208] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/16/2024] [Revised: 02/14/2024] [Accepted: 03/21/2024] [Indexed: 04/22/2024] Open
Abstract
BACKGROUND Lymphangiomas in the gastrointestinal tract are extremely rare in adults. As a benign lesion, small intestine lymphangiomas often remain asymptomatic and pose challenges for definitive diagnosis. However, lymphangiomas can give rise to complications such as abdominal pain, bleeding, volvulus, and intussusception. Here, we report a case of jejunal cavernous lymphangioma that presented with intermittent melena and refractory anemia in a male adult. CASE SUMMARY A 66-year-old man presented with intermittent melena, fatigue and refractory anemia nine months prior. Esophagogastroduodenoscopy and colonoscopy were performed many times and revealed no apparent bleeding. Conservative management, including transfusion, hemostasis, gastric acid secretion inhibition and symptomatic treatment, was performed, but the lesions tended to recur shortly after surgery. Ultimately, the patient underwent capsule endoscopy, which revealed a more than 10 cm lesion accompanied by active bleeding. After single-balloon enteroscopy and biopsy, a diagnosis of jejunal cavernous lymphangioma was confirmed, and the patient underwent surgical resection. No complications or recurrences were observed postoperatively. CONCLUSION Jejunal cavernous lymphangioma should be considered a cause of obscure gastrointestinal bleeding. Capsule endoscopy and single-balloon enteroscopy can facilitate diagnosis. Surgical resection is an effective management method.
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Affiliation(s)
- Kai-Rui Liu
- Department of Abdominal Surgery, The Second Affiliated Hospital of Guangzhou University of Chinese Medicine, Guangzhou 510000, Guangdong Province, China
| | - Sheng Zhang
- Department of Abdominal Surgery, The Second Affiliated Hospital of Guangzhou University of Chinese Medicine, Guangzhou 510000, Guangdong Province, China
| | - Wei-Run Chen
- Department of Abdominal Surgery, The Second Affiliated Hospital of Guangzhou University of Chinese Medicine, Guangzhou 510000, Guangdong Province, China
| | - You-Xing Huang
- Department of Abdominal Surgery, The Second Affiliated Hospital of Guangzhou University of Chinese Medicine, Guangzhou 510000, Guangdong Province, China
| | - Xu-Guang Li
- Department of General Surgery, The Affiliated Brain Hospital of Guangzhou Medical University, Guangzhou 510000, Guangdong Province, China
- Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and The Ministry of Education of China, Guangzhou Medical University, Guangzhou 510000, Guangdong Province, China
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Ding XL, Yin XY, Yu YN, Chen YQ, Fu WW, Liu H. Lymphangiomatosis associated with protein losing enteropathy: A case report. World J Clin Cases 2021; 9:3758-3764. [PMID: 34046480 PMCID: PMC8130063 DOI: 10.12998/wjcc.v9.i15.3758] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/09/2021] [Revised: 02/13/2021] [Accepted: 03/08/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Lymphangiomatosis is a multisystem disorder that is rarely localized to the gastrointestinal tract. Lymphangiomatosis usually has no specific clinical presentation and is easily misdiagnosed. A case report and review of the literature on lymphangiomatosis associated with protein-losing enteropathy will help to improve the overall understanding of this disease.
CASE SUMMARY We report a case of lymphangiomatosis of the bowel and other solid organs. A 78-year-old man presented with recurrent bowel bleeding and protein-losing enteropathy, as well as cystic lesions in the spleen, liver, and kidney. Imaging examinations revealed many cystic lesions on the spleen, liver, kidney, and thickened wall of the ascending colon, as well as pleural effusion and ascites. Colonoscopy revealed a strawberry mucosa, variable spontaneous bleeding, and surface erosion located in the terminal ileum. Several cystic masses with a translucent and smooth surface as well as diffuse white spots were located in the colon. A laterally spreading tumor (LST) was located in the ascending colon. Pathology indicated highly differentiated adenocarcinoma (LST) and lymphangiomatoid dilation, and D2-40 was positive. The final diagnosis was lymphangiomatosis. The patient underwent surgery for LST and then was administered thalidomide 75-150 mg/d. His condition, however, did not improve. He eventually died 6 mo after the initial diagnosis.
CONCLUSION Lymphangiomatosis usually occurs diffusely and can involve many organs, such as the spleen, kidney, liver, lung, mesentery, and bowel. Recurrent bowel bleeding or protein-losing enteropathy is an important indicator that should alert clinicians about the possibility of this disease when it afflicts the bowel. Doctors should improve the medical understanding of lymphangiomatosis.
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Affiliation(s)
- Xue-Li Ding
- Department of Gastroenterology, The Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
| | - Xiao-Yan Yin
- Department of Gastroenterology, The Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
| | - Ya-Nan Yu
- Department of Gastroenterology, The Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
| | - Yun-Qing Chen
- Department of Pathology, The Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
| | - Wei-Wei Fu
- Department of Pathology, The Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
| | - Hua Liu
- Department of Gastroenterology, The Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
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Van Meerhaeghe T, Vandenbroucke F, Velkeniers B. Systemic generalised lymphangiomatosis: unknown aetiology and a challenge to treat. BMJ Case Rep 2021; 14:14/1/e237331. [PMID: 33462010 PMCID: PMC7813405 DOI: 10.1136/bcr-2020-237331] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022] Open
Abstract
We describe a case of a woman diagnosed at the age of 35 years with a generalised mediastinal and abdominal lymphangiomatosis associated with a protein losing enteropathy, who successfully improved when treatment with sirolimus was initiated.
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Liu L, Ke G, Shen WM, Jia CK. Giant cystic lymphangioma in the mesoileum: A case report and literature review. TURKISH JOURNAL OF GASTROENTEROLOGY 2020; 31:603-606. [PMID: 32915149 DOI: 10.5152/tjg.2020.19256] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/22/2022]
Affiliation(s)
- Ling Liu
- Department of General Surgery, Affiliated Hangzhou First People's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang Province, China
| | - Ge Ke
- Department of General Surgery, Affiliated Hangzhou First People's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang Province, China
| | - Wei-Min Shen
- Department of General Surgery, Affiliated Hangzhou First People's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang Province, China
| | - Chang-Ku Jia
- Department of General Surgery, Affiliated Hangzhou First People's Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang Province, China
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Giuliani A, Romano L, Coletti G, Walid A Fatayer M, Calvisi G, Maffione F, Muolo C, Vicentini V, Schietroma M, Carlei F. Lymphangiomatosis of the ileum with perforation: A case report and review of the literature. Ann Med Surg (Lond) 2019; 41:6-10. [PMID: 30992989 PMCID: PMC6449703 DOI: 10.1016/j.amsu.2019.03.010] [Citation(s) in RCA: 21] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/14/2019] [Accepted: 03/24/2019] [Indexed: 01/10/2023] Open
Abstract
Lymphangiomatosis is a benign proliferation of lymph vessels. Lymphatic diseases can vary from small lymphangioma to generalized lymphangiomatosis, which is a rare condition and can have several clinical manifestations. The gastrointestinal tract may be affected, but the incidence in the intestinal wall is very low. We propose in our study a case of ileal lymphangiomatosis presenting with perforation, in which the diagnosis was made after the pathological analysis of the resected intestinal tract. Although rare and not described in the literature, intestinal lymphangiomatosis could manifest itself with acute abdomen and could be a surgical urgency. This disease should be considered when intestinal perforation is observed.
Lymphangiomatosis is a multisystemic disorder. The GI tract may be affected, but the incidence of lymphangioma of the intestinal wall is very low. Lymphangiomatosis of the small bowel is a rare disease, so it is easy for a clinician to make a misdiagnosis. Intestinal lymphangiomatosis could manifest itself with an acute abdomen and surgical urgency. The pathologist should keep it in mind in the differential diagnosis, when he analyses a case of perforation.
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Affiliation(s)
| | - Lucia Romano
- Department of Surgery, University of L'Aquila, Italy
- Corresponding author. University of L'Aquila, Dipartimento di Scienze Chirurgiche, Ospedale San Salvatore, 67100, Coppito, AQ, Italy.
| | - Gino Coletti
- UOC Anatomia Patologica, ASL1 Abruzzo, Ospedale San Salvatore, L'aquila, Italy
| | | | - Giuseppe Calvisi
- UOC Anatomia Patologica, ASL1 Abruzzo, Ospedale San Salvatore, L'aquila, Italy
| | | | - Chiara Muolo
- Department of Surgery, University of L'Aquila, Italy
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