Saudi Journal of Gastroenterology

CASE REPORT
Year
: 2001  |  Volume : 7  |  Issue : 2  |  Page : 74--76

Meso-sigmoidcolon hamartoma


Mohammed H Al Akeely, Atilio B Giangreco 
 Department of Surgery, King Khalid University Hospital, Riyadh, Saudi Arabia

Correspondence Address:
Mohammed H Al Akeely
King Khalid University Hospital, P.O. Box 7805, Riyadh 11472
Saudi Arabia




How to cite this article:
Al Akeely MH, Giangreco AB. Meso-sigmoidcolon hamartoma.Saudi J Gastroenterol 2001;7:74-76


How to cite this URL:
Al Akeely MH, Giangreco AB. Meso-sigmoidcolon hamartoma. Saudi J Gastroenterol [serial online] 2001 [cited 2021 Jun 20 ];7:74-76
Available from: https://www.saudijgastro.com/text.asp?2001/7/2/74/33407


Full Text

Intra-abdominal lymphangiohamartomas are rare. About 60% of them occurred in patients under a five-year of age and a significant number does not manifest until adult life [1] . Lymphangiohamartomas are generally regarded as a developmental malformation of the lymphatic and vascular system. They are non-neoplastic swellings due to inborn error in tissue development. Their capacity for growth normally parallels that of the surrounding normal tissue. They usually behave in benign fashion [2] . We report a case of lymphangiohamartoma involving the sigmoid mesocolon in a 33-year old man.

 Case report



A 33-year old Egyptian male was admitted to Riyadh Central Hospital (RCH) complaining of swelling in the left side of the abdomen for the last three months. It was associated with dragging abdominal pain. There was neither change in bowel habits nor passage of mucus or blood per rectum. He was treated for schistosomiasis during his childhood in Egypt. Physical examination revealed a well nourished young man with a firm, non tender, nodular and mobile mass on the left iliac fossa measuring approximately 14.0 x 12.0 cm. It was dull on percussion and has no bruit on auscultation. Lymph nodes were not enlarged and no other abnormalities detected on examination of the respiratory, cardiac and central nervous system. Serologic tests revealed positive HbsAg, anti Hbc and anti Hbe. However, chest and abdominal X­ray's were unremarkable. Ultrasonography revealed evidence of liver cirrhosis. A CT scan of the lower abdomen and pelvis showed a large well defined fibro fatty mass surrounding the distal descending and sigmoid colon. The bowel wall was thickened and the loop appeared stretched [Figure 1], supporting the clinical diagnosis of lymphoma of the sigmoid mesocolon. Colonoscopy showed only congested mucosa of the sigmoid colon. Punch biopsies from that area showed non specific chronic colitis. Barium enema showed no significant abnormality. Laparoscopy revealed thick nodular appearance of the serosal surface on the left side of the colon. Punch biopsy of this nodule showed only fatty tissue with non specific chronic inflammation. Tuberculosis was suspected and empirical treatment was given for ten weeks with no significant improvement. The patient was readmitted ten weeks later because of vague abdominal pain, weight loss, constipation and occasional passage of mucus and blood per rectum. Physical examination showed no change in the size of the mass. CT scan of the mass also showed the same configuration except increase of the soft tissue component. Cystoscopic examination was normal. On laparotomy, a left sided nodular congested mass covered by greater omentum was found. The mass measured 14.0 x 12.0 cm, attached to the sigmoid colon and its mesentery. Resection of the mass with 29.0 cm of the colon was performed [Figure 2]. Hi stopatho logical examination showed empty capillaries and cavernous spaces different in size, partially lined by endothelial cells. In some areas capillaries with erythrocytes in the lumen were easily seen. The interstitial tissue is scanty and also showed fatty tissue with edema and degenerative changes as well as focal collection of lymphocytes. The hamartomatous lesion was involving mainly the mesocolon and extending focally to the superficial external area of the muscular coat of the colon. Some of the vessels revealed thickened wall while others had only a single layer of endothelial cells. Congestion, telangiectasia and lymphangiectasia were also present in the colonics submucosa but not in the mucosa [Figure 3]. The surrounding lymphnodes also showed lymphangiectasia. Resection margins were free of lesion. Postoperatively, the recovery was uneventful. Over four years follow-up, the patient was symptoms free and without evidence of tumor recurrence.

 Discussion



Lymphangiohamartomas are generally regarded as a developmental malformation of the lymphatic and vascular system. Most lymphangiomas occur in the neck (75%), axillary region (20%) and rarely in the retroperitoneum, mesentery, omentum, colon, pelvis groin, spleen, bone or skin [3] . Abdominal location of these angiomatous tumors is mainly observed in childhood. The age ranges from 3-63 years. Seventeen cases of abdominal lymphangiomas have been located as follows: five at the mesentery, four at retroperitoneum, three at the colon, three at omentum, one at the gallbladder and one at the mesocolon.

In the present case, the hamartomatous malformation was located at the mesocolon with minimal extension to the muscular coat of the colon. On CT no definitive large cystic configuration was noted, therefore supporting the presumptive clinical diagnosis of lymphoma. Histological examination revealed lymphangiectasia and telangiectasia of the submucosa and congestion of the mucosa. Usually CT scan demonstrates the nature of these masses when large cystic changes are present, however, a definitive diagnosis is established by histological examination of the surgical specimen. When the lymphangiomatous process involve the colonic mucosa, colonoscopic examination usually reveals some smooth soft polypoid mass [4] . In some of these cases endoscopic resection has been performed with excellent results [5],[6] . All seven cases of lymphangiomas involving the mesocolon, reported in the medical literature are located at the transverse mesocolon [1],[3],[7],[8],[9],[10],[11] . In three of mesocolon lymphangiomas, cystic configuration of the tumor is detected by CT scan, while in the other four similar to our report, the diagnosis has been established by the evaluation of the resected specimen.

In conclusion, lymphangiohamartomas involving the mesocolon are very rare, characterized by absence of specific clinical symptoms and signs, therefore the correct diagnosis, is usually established after surgical resection. Awareness of physicians will contribute to make a correct preoperative diagnosis more feasible.

References

1Yuen S.T, Ng I. O. L, Na L. Mesocolonic lymphangioma: A case report with immunoperoxidase and electron microscopic studies. Pathology 1992: 221-3.
2Willis RA. The borderline of embryology and pathology: London, Butterworth, 1958: 348-50.
3Chung J H, Suh Y. L, Park LA, Hang JJ, Chi J.G, Kim Y. I, Kim WH. A pathologic study of abdominal lymphangiomas. J. Korean Med. Sci. 1999; 14: 257-62.
4Young T.H, Ho A.S, Tnag H S, Hsu C.T, Lee H.S, ChaoY.C. Cystic lymphangioma of the transverse colon: report of case and review of literature. Abdomen. Imagin 1996; 21:515-7.
5Yoshikane H, Hamajima E, Yokoi T, Suzuki T, Leda H, Miyachi N, Fukui A. Lymphangioma of the colon treated by endoscopic resection following find needle puncture aspiration. Endoscopy 1996; 28: 407-8.
6Iwakiri Y. et al. Endoscopic removal of a lymphangioma of the colon: case report of the diagnostic value of endoscopic ultrasound probe. Endocopy, 1997, 29 : 32-3.
7Zago A, Tosi D, Portuese D, Rosa G. Cystic Lymphangioma of the transverse mesocolon simulating neoplasm of the pancreatic tail. Ann Ital. Chir. 1997; 68: 95-8.
8Mayer M, Fartab M, Villinger, Yurtsever H. Cystic lymphangioma of the transverse mesocolon. Chiruf. 1994; 65: 561-3.
9Dorf G, Renoux M, Marcelli D, Duchier J, Olivier C, Bour H. Cystic lymphangioma of the transverse mesocolon. A further case. Arch. Fr. Mal. App. Dig 1970; 59: 5889-95.
10Hauser H, Mischinger HJ, Beham A, Berger A, Cerwenka H, Rasmara J, Fruhwirth H, Werkgarner G. Cystic retroperitoneal lymphangiomas in adults. Eur. J. Surg. Oncol. 1997;23:322-6.
11Nordshus T, Lotveit T. Multiple mesenteric cystis diagnosed by ultrasound. A case report. Ann. Chit. Gynaecolo. 1976; 65: 234-8.