Amaro, T

Castro-Poças, F

Lobo, L

Saraiva, MM

Soares, J

English

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LETTER TO THE EDITORColon hemangiolymphangioma—a rare caseof subepithelial polypF. Castro-Poças & Luís Lobo & Teresina Amaro & JoséSoares & Miguel Mascarenhas SaraivaAccepted: 20 November 2014 /Published online: 4 December 2014# Springer-Verlag Berlin Heidelberg 2014Dear Editor:The majority of colon polyps have an epithelial nature.There is a small subset of them that are submucosalproliferations of mesenchymal cells, such as lipomas,leiomyomas, schwannomas, gastrointestinal stromal tu-mours and vascular lesions [1]. These last ones are ex-tremely rare when responsible for polypoid lesions of thecolon [2].Clinical caseAn asymptomatic 64-year-old man without a relevantclinical history was referred for endoscopic colorectalcancer screening.In the colonoscopy, a polypoid lesion was identified, whichwas pediculated and measuring about 25 mm at the proximaltransverse colon. The lesion was covered by normal-appearing mucosa.We performed a miniprobe endoscopic ultrasonogra-phy, 12 MHz, which showed a polypoid morphologylesion, which heterogeneous and cystic, with variousanechoic cavities separated by different thickness septa,being a part of them incomplete. The lesion was welldelimited with regular boarders and located in the submu-cosa, with interface preservation with the adjacent layers.The size of the lesion was 27.1 by 20 mm.The patient required a definitive diagnosis. The endo-scopic resection was performed with a hot snare. Therewere no complications.The patient is well with no additional colorectal lesions2 years after the polyp’s resection.The macroscopic examination of the excised speci-men showed a circumscribed round polyp, which is25 mm in diameter, of soft consistency and coveredby normal-appearing mucosa. The histological examina-tion showed a lesion with capillary and lymphatic-typeblood vessels in mucosa and submucosa; positivity forCD31 and D2-40 was expressed in the endothelial cellsof lymphatic vessels. A definitive diagnosis of colonhemangiolymphangioma was made.DiscussionIt is well known that the vast majority of polyp lesionshave epithelial nature, rarely having a subepithelial mes-enchymal cells one [1].Excluding certain vascular lesions, such as angioectasias, thereal vascular tumours, such as haemangiomas, are rare in thedigest ive t r ac t [3] . Among vascu la r tumours,hemangiolymphangiomas are a rare entity. Recently Andra-Luca and Montgmory published the first description ofhemangiolymphangiomas in the digestive tract [2]. During aperiod of 22 years, they identified 27 patients with vasculartumours, being 12 cases of hemangiolymphangiomas, eightlocated in the small bowel and four in the colon.F. Castro-Poças :M. M. SaraivaDepartment of Gastroenterology, ManoPh-CUF Institute, Porto,PortugalL. Lobo : J. SoaresDepartment of Gastroenterology, Pedro Hispano Hospital,Matosinhos, PortugalT. AmaroDepartment of Pathology, Pedro Hispano Hospital, Matosinhos,PortugalF. Castro-Poças (*)R. Prof. Egas Moniz, n.° 1868, 4510-119 Gondomar, Portugale-mail: castro.pocas@sapo.ptInt J Colorectal Dis (2015) 30:989–990DOI 10.1007/s00384-014-2077-6Hemangiolymphangiomas consist of a proliferation [3]or a network of vascular spaces of varied nature (veins,venules, arteries, arterioles, capillaries or lymphatic) lim-ing by a benign endothelial lining with intervening con-nective tissue stroma and forming a tumour. Therefore thehemangiolymphangiomas show haemangioma andlymphangioma characteristics with an anastomosis networkcomposed by blood and lymphatic vessels [4].In our case, the identification of this lesion was anincidental finding which is in agreement with the fewreported cases in the literature, where in the majority ofcases, the lesions are associated with non-specific signs orsymptoms and rarely anaemia or gastrointestinal bleeding[2].In the series published by Andra-Luca, of the fiveresected cases, one was performed by surgery and fourby endoscopy. The authors call our attention to the factthat we showed a considered eventual follow-up of thelesions, due to the risk of bleeding or recurrent subsequentlesions.The colonoscopy performed in our patient 2 years afterthe lesion’s resection was normal.According to our knowledge, this was the first caseof hemangiolymphangioma characterised by endoscopicultrasonography. As we know, this technique has ac-quired a main role in the characterisation and clinicalorientation in the subepithelial lesions of the digestivetract.Naturally we need more cases of hemangiolymphangiomato try to define its ultrasonographic semiology, which can helpin the clinical orientation, namely in the asymptomatic cases.On the one hand, when clarifying what the digestive walllayers involved by the lesion, it can help to decide betweenendoscopic or surgical resection. On the other hand, whencontributing in a decisive way to different diagnosispossibilities, it can identify lesions that do not requireany therapeutic approach or follow-up, such as lipoma.In our case, the ultrasonographic information that thelesion was located in the submucosa was fundamentalfor choosing endoscopic resection and not surgical.In spite of the colon polyps with subepithelial naturebeing rare, particularly vascular tumours, we can spec-ulate that in the future, we will find more cases, sincecolonoscopy was recognised as the gold standardscreening procedure for colorectal cancer.In summary, we have reported a rare case of submu-cosal hemangiolymphangioma of the colon. Maybe itwas also the first description, in the English literature,of a hemangiolymphangioma by endoscopic ultrasonog-raphy. These lesions may be treated successfully bystandard polypectomy techniques.References1. Flejou JF (2013) Non-epithelial tumors of the large intestine. In:Shepherd NA, Warren BF, Williams GT, Greenson JH, Lauwers GY,Novelli MR (eds) Morson and Dawson’s gastrointestinal pathology,5th edn. Wiley-Blackwell, Oxford, pp 739–442. Handra-Luca A, Montgomery E (2011) Vascular malformationsand hemangiolymphangiomas of the gastrointestinal tract: mor-phological features and clinical impact. Int J Clin Exp Pathol4(5):430–433. Fenoglio-Preiser CM, Noffsinger AE, Lantz PE, Isaacson PG (2008)Gastrointestinal pathology. Lippincott Williams and Wilkins,Philadelphia4. Fenoglio-Preiser C, Noffsinger A, Stemmermann G, Lantz P, IsaacsonP (2008) Gastrointestinal pathology: an atlas and text. LippincottWilliams & Wilkins, Philadelphia990 Int J Colorectal Dis (2015) 30:989–990

Colon hemangiolymphangioma-a rare case of subepithelial polyp.

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Colon hemangiolymphangioma-a rare case of subepithelial polyp.

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