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Duve R, Robillard K, Kanehira K. Therapeutic Banding for Bleeding Duodenal Lymphangiectasias: A Novel Approach. ACG Case Rep J 2023; 10:e01087. [PMID: 37342653 PMCID: PMC10278729 DOI: 10.14309/crj.0000000000001087] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/16/2023] [Accepted: 05/30/2023] [Indexed: 06/23/2023] Open
Abstract
With endoscopic advancements, the number of detected intestinal lymphangiectasias has been on the rise. They are generally considered benign and incidental; occasionally, these lesions carry complications, and best management options need to be established. Bleeding intestinal lymphangiectasias should be considered a rare cause in the differential diagnosis for gastrointestinal bleeding. References in the literature primarily indicate surgical treatment in these situations. In this study, we report an uncommon case of a man with esophageal adenocarcinoma who developed acute gastrointestinal bleeding from duodenal lymphangiectasias that were successfully banded.
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Affiliation(s)
- Robert Duve
- Department of Internal Medicine, Jacobs School of Medicine and Biomedical Sciences, University at Buffalo, Buffalo, NY
| | - Kevin Robillard
- Division of Gastroenterology, Roswell Park Cancer Institute, Buffalo, NY
- Division of Gastroenterology, Hepatology and Nutrition, Jacobs School of Medicine and Biomedical Sciences, University at Buffalo, Buffalo, NY
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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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Iwamuro M, Tanaka T, Kanzaki H, Kawano S, Kawahara Y, Iwasaki Y, Okada H. Deterioration of duodenal lymphangiectasia after radiotherapy for gastric MALT lymphoma. Ecancermedicalscience 2017; 11:752. [PMID: 28798810 PMCID: PMC5533599 DOI: 10.3332/ecancer.2017.752] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/12/2016] [Indexed: 12/19/2022] Open
Abstract
A 68-year-old Japanese woman underwent radiotherapy for gastric lymphoma. Although lymphangiectasia was sparsely observed in the second portion of the duodenum before radiotherapy, the number of pinpoint white spots obviously increased after the treatment. Although the duodenal lymphangiectasia gradually progressed, the patient had no features of protein-losing enteropathy. This case highlights the importance of endoscopic observation of the duodenum after irradiation to the abdomen as radiotherapy may secondarily cause intestinal lymphangiectasia.
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Affiliation(s)
- Masaya Iwamuro
- Department of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama 700–8558, Japan
- Department of General Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama 700–8558, Japan
| | - Takehiro Tanaka
- Department of Pathology, Okayama University Hospital, Okayama 700–8558, Japan
| | - Hiromitsu Kanzaki
- Department of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama 700–8558, Japan
| | - Seiji Kawano
- Department of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama 700–8558, Japan
| | - Yoshiro Kawahara
- Department of Endoscopy, Okayama University Hospital, Okayama 700–8558, Japan
| | - Yoshiaki Iwasaki
- Health Service Centre, Okayama University, Okayama 700–0082, Japan
| | - Hiroyuki Okada
- Department of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama 700–8558, Japan
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Abstract
Two patients with lymphangioma and one with hemangioma of the duodenum are described. The radiologic presentation of duodenal angiomas is that of multiple submucous, soft, polypoid, non-infiltrating tumors. During propagation of peristalsis, on compression, or gas distension of the duodenum, they change in shape and dimensions. Deep peristaltic waves could cause apparent vanishing of the angiomas. Possible mechanisms of such behaviour of angiomas are discussed. Listed characteristics enabled the radiologic distinction of angiomas from solid submucous duodenal tumors. Duodenoscopy allows differentiation of duodenal lymphangiomas from hemangiomas and duodenal varices. Final diagnoses were based on histologic analysis of surgical specimens.
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Park MS, Lee BJ, Gu DH, Pyo JH, Kim KJ, Lee YH, Joo MK, Park JJ, Kim JS, Bak YT. Ileal polypoid lymphangiectasia bleeding diagnosed and treated by double balloon enteroscopy. World J Gastroenterol 2013; 19:8440-8444. [PMID: 24363538 PMCID: PMC3857470 DOI: 10.3748/wjg.v19.i45.8440] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/22/2013] [Revised: 09/04/2013] [Accepted: 09/29/2013] [Indexed: 02/06/2023] Open
Abstract
Intestinal lymphangiectasia is a rare disease characterized by focal or diffuse dilated enteric lymphatics with impaired lymph drainage. It causes protein-losing enteropathy and may lead to gastrointestinal bleeding. Commonly, lymphangiectasia presents as whitish spots or specks. To our knowledge, small bowel bleeding resulting from polypoid intestinal lymphangiectasia has not been reported. Here, we report a rare case of active bleeding from the small bowel caused by polypoid lymphangiectasia with a review of the relevant literature. An 80-year-old woman was hospitalized for melena. Esophagogastroduodenoscopy could not identify the source of bleeding. Subsequent colonoscopy showed fresh bloody material gushing from the small bowel. An abdominal-pelvic contrast-enhanced computed tomography scan did not reveal any abnormal findings. Video capsule endoscopy showed evidence of active and recent bleeding in the ileum. To localize the bleeding site, we performed double balloon enteroscopy by the anal approach. A small, bleeding, polypoid lesion was found in the distal ileum and was successfully removed using endoscopic snare electrocautery.
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Freeman HJ, Nimmo M. Intestinal lymphangiectasia in adults. World J Gastrointest Oncol 2011; 3:19-23. [PMID: 21364842 PMCID: PMC3046182 DOI: 10.4251/wjgo.v3.i2.19] [Citation(s) in RCA: 60] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/26/2010] [Revised: 01/31/2011] [Accepted: 02/07/2011] [Indexed: 02/05/2023] Open
Abstract
Intestinal lymphangiectasia in the adult may be characterized as a disorder with dilated intestinal lacteals causing loss of lymph into the lumen of the small intestine and resultant hypoproteinemia, hypogammaglobulinemia, hypoalbuminemia and reduced number of circulating lymphocytes or lymphopenia. Most often, intestinal lymphangiectasia has been recorded in children, often in neonates, usually with other congenital abnormalities but initial definition in adults including the elderly has become increasingly more common. Shared clinical features with the pediatric population such as bilateral lower limb edema, sometimes with lymphedema, pleural effusion and chylous ascites may occur but these reflect the severe end of the clinical spectrum. In some, diarrhea occurs with steatorrhea along with increased fecal loss of protein, reflected in increased fecal alpha-1-antitrypsin levels, while others may present with iron deficiency anemia, sometimes associated with occult small intestinal bleeding. Most lymphangiectasia in adults detected in recent years, however, appears to have few or no clinical features of malabsorption. Diagnosis remains dependent on endoscopic changes confirmed by small bowel biopsy showing histological evidence of intestinal lymphangiectasia. In some, video capsule endoscopy and enteroscopy have revealed more extensive changes along the length of the small intestine. A critical diagnostic element in adults with lymphangiectasia is the exclusion of entities (e.g. malignancies including lymphoma) that might lead to obstruction of the lymphatic system and “secondary” changes in the small bowel biopsy. In addition, occult infectious (e.g. Whipple’s disease from Tropheryma whipplei) or inflammatory disorders (e.g. Crohn’s disease) may also present with profound changes in intestinal permeability and protein-losing enteropathy that also require exclusion. Conversely, rare B-cell type lymphomas have also been described even decades following initial diagnosis of intestinal lymphangiectasia. Treatment has been historically defined to include a low fat diet with medium-chain triglyceride supplementation that leads to portal venous rather than lacteal uptake. A number of other pharmacological measures have been reported or proposed but these are largely anecdotal. Finally, rare reports of localized surgical resection of involved areas of small intestine have been described but follow-up in these cases is often limited.
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Affiliation(s)
- Hugh James Freeman
- Hugh James Freeman, Department of Medicine (Gastroenterology), University of British Columbia, Vancouver, BC, V6T 1W5, Canada
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Agret F, Nahon S, Tuszinsky T, Caugant H, Bonyhay G, Delas N. Lymphangiome kystique duodénal hémorragique traité avec succès par plasma argon. ACTA ACUST UNITED AC 2004; 28:1181-2. [PMID: 15657548 DOI: 10.1016/s0399-8320(04)95204-9] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/22/2022]
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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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Hizawa K, Aoyagi K, Kurahara K, Suekane H, Kuwano Y, Nakamura S, Fujishima M. Gastrointestinal lymphangioma: endosonographic demonstration and endoscopic removal. Gastrointest Endosc 1996; 43:620-4. [PMID: 8781946 DOI: 10.1016/s0016-5107(96)70204-3] [Citation(s) in RCA: 28] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Affiliation(s)
- K Hizawa
- Second Department of Internal Medicine, Faculty of Medicine, Kyushu University, Fukuoka, Japan
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Ikura Y, Hashimoto T, Takamine Y, Tani T, Konishi Y, Uchida H, Shirane H, Kajiwara T. Lymphangioma of the duodenum: report of a case. Surg Today 1994; 24:160-163. [PMID: 8054798 DOI: 10.1007/bf02473401] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/28/2023]
Abstract
A case of lymphangioma of the duodenum with a successful preoperative diagnosis is reported herein. A 76-year-old man who complained of tarry stool was found to have a hemorrhagic polypoid tumor in the third portion of the duodenum. The tumor was diagnosed histologically as cavernous lymphangioma by endoscopic biopsy. Since such a tumor is essentially benign, a partial resection of the duodenum including the tumor was performed. Therefore, an endoscopic biopsy seems to be valuable in the diagnosis of duodenal lymphangioma.
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Affiliation(s)
- Y Ikura
- Department of Surgery, Kobe City General Hospital, Japan
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