Lesser Sac Cystic Lymphangioma: Gastric Outlet Obstruction

lesser Sac cystic Lymphangioma Gastric Outlet ObstructionCystic Lymhangioma of the Lesser Sac in adult presenting with features of Gastric Outlet Obstruction A Case declareS Suresh KumarSri Aurobindo Prasad DasVikram KateRunning title Lesser poke cystic lymphangioma causing gastric outlet obstructionKey Words abdominal cystic Lymphangioma Omental Bursa Outlet ObstructionArticle type Case ReportSection (speciality) Surgery- Gastrointestinal SurgeryAbstract Non- Structured (Case Report)ABSTRACTBackgroundCystic lymphangioma is a paediatric benign tumour, overtakes commonly in head and cervix uteri region. Adult Intraabdominal lymphangiomas are uncommon and only few cases of lesser scoop cystic lymphangioma are describe in the literature.Case presentationWe present a case of lesser sac cystic lymphangioma in a 26 year old lady who presented with features of gastric outlet obstruction. Clinical examination of the tum revealed 10 x 10 cm firm, non tender, mobile intraabdominal lump in the epigastrium.An upper gastrointestinal endoscopy showed possible external compression at the boy of the stomach. sonography of tum demonstrated a multicystic lesion measuring 10.2 X 8 cm, with versed echoes in one locule. Contrast enhanced computed tomography of the abdomen showed a deep multiloculated cystic lesion with hyper-dense content in the lesser sac extending up to the left over(p) 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 set in the lesser sac very close to the lesser curvature of stomach. The descending branches of left gastric which were found coursing through the cyst were ligated and the cyst was excised in Toto. Histopathological examination of the excised lesion showed features of cystic lymphangioma showing dilated lymphatic vessels with fibro-fatty tissue ingrained in the endothelial lining.Conclusionthough intraabdominal cystic lymph angiomas are very rare, it should be considered in all atypical cases of gastric outlet obstruction. A reasonable knowledge about these benign lesions go out aid in establishing the diagnosis and surgical management.Key WordsAbdominal cystic Lymphangioma Omental Bursa Outlet ObstructionBackgroundLymphagiomas are benign lesions generally occur as a result of lymphatic system malformation and infrequently reported after trauma. 90% of the cases are seen within the first two days of life and symptomatic presentation in adults is very rare. 1 Cystic lymphagiomas commonly involves neck and axilla but can occur anywhere in the body. though Intraabdominal lymphangioma accounts for Case presentationA 26-year old lady presented to surgical outpatient department with complaints of early satiety, vomiting few hours after food intake and vague impressiveness of upper abdomen. She also had features of dyspepsia for the past one year. She had developed pain and had a feeling of a mobile lump i n the upper abdomen for the past one month. There was no pancreatitis episode before. General physical examination was unremarkable. Clinical examination of the abdomen revealed 10 x 10 cm lump in the epigastrium which was firm in consistency. The lump was non tender, freely mobile and intra-abdominal.InvestigationsThe haemogram and biochemical profiles were within normal limits. An upper gastrointestinal endoscopy was make which showed normal mucous membrane of stomach with possible external compression at the boy of the stomach. Ultrasound of abdomen was suggestive of a multicystic lesion measuring 10 X 8 cm, with internal echoes in one locule, present superior to pancreas adjacent to the left lobe of the liver. Rest of abdomen was unremarkable.Contrast enhanced computed tomography of the abdomen showed a large multiloculated cystic lesion measuring 10.2 X 8 cm with hyper dense content in some loculi in the lesser sac extending up to the left lobe of the liver and indenting the s tomach, lesser sac, and upper border of pancreas with a few branches of left gastric vessels coursing through it. (Figure 1) Correlating the clinical, USG and CECT abdomen findings a diagnosis of possible lymphangioma was made and the patient was mean for exploratory laparotomy as the symptoms were persisting.Surgical managementOn 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. (Figure 2) The descending branches of left gastric which were found coursing through the cyst were ligated and the cyst was excised in Toto. (Figure 3) A poop out was placed in lesser sac and abdomen was closed in layers.Outcome and follow- upPostoperative course was uneventful. The drain was removed on the 3nd operative day and the patient was discharged on seventh post operative day. Histopathological examination of the excised lesion showed features of cystic lymphangioma showing dilated lymphatic vessels wit h fibro-fatty tissue ingrained in the endothelial lining. (Figure 4)Patient was followed up at one month, six months, one year and two year after operation. On each visit, clinical examination and USG was done which showed no evidence of recurrence. The lady is presently on regular follow up for past 2 years with no evidence recurrence trough date.DiscussionIntra-abdominal cystic lymphangiomas are uncommon benign tumours that can involve the mesentery, omentum, colon, spleen, pelvis, groin, and retroperitoneum 5, 6. We had a case of cystic lymphangioma in the lesser sac which is very rare. Cystic lymphangiomas are considered to originate from malformed or malpositioned lymphatic tissue. Factors such as abdominal trauma, inflammatory process, lymphatic obstruction due to radiation therapy or surgery may lead to secondary formation of such tumors. Our patient did not have any of the mentioned inciting factors the cause was probably congenital which had manifested late in life. commo nly known to present in childhood, lymphagiomas sometimes remain asymptomatic until late adulthood, and are discovered during evaluation of vague and chronic nonspecific symptoms like anorexia, nausea, vomiting, fatigue, and weighting loss, which are secondary to wad effect 4. This was seen in our patient who had early satiety and vomiting after food intake probably due to mass effect and had a vague feeling of mass in the abdomen. Other ways of presentation such as smashing abdomen, mimicking acute appendicitis has also been reported 3.Contrast CT done in our patient revealed multicystic lesion measuring 10.2 X 8 cm, present superior to pancreas, in the lesser sac adjacent to the left lobe of the liver. Cystic lymphangioma usually presents as a large multilocular cystic mass with enhanced walls with multiple thin septa containing uncomplicated fluid on abdominal echography and contrast enhanced computerized tomography which is very often diagnostic 7.The differential diagnosis inc ludes cystic lesions of, enteric urogenital or mesothelial origin. Pancreatic pseudocysts from trauma, dermoid cysts or teratomas and cystic degeneration of solid tumours are also to be considered before making this rare diagnosis.Management of intra-abdominal cystic tumours depends on the clinical symptoms, size of the cyst, and the degree of clinical suspicion for malignancy. Imaging usually provides useful information for the planning of treatment. Complete removal of the tumour offers an excellent prognosis and laparoscopic removal of lymphangioma also shown feasible in few reports. We therefore decided to perform a laparotomy and complete excision of the cyst was done. On the other hand, recurrence has been reported in 10% of patients in whom primary resection was incomplete.9 If the feeding lymphatics are not completely ligated, chylous ascites may also occur.ConclusionThough intra-abdominal cystic lymphangiomas are very rare, it should be considered in cases of gastric or bil iary obstruction where the cause could not be recognized clearly. Reasonable knowledge about these benign lesions will aid in establishing the diagnosis during such instances. Complete surgical excision offers the best chance for cure and should be attempted in all the cases to avoid recurrence.Consent written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for inspection by the Editor of this journal.Competing interests disclosuresThe authors have no potential or real conflict of interestsThe authors have no financial support or ties to discloseThe authors have no financial or non financial competing interests to declareAuthors contributionsSuresh Kumar S, Das S A and Kate V diagnosed and managed the case. Suresh Kumar S, Das S A did the literature search. Suresh Kumar S and Das S A collected the images. Suresh Kumar S and Kate V drafted the manuscript. All authors read and a pproved the final manuscript.ReferencesHanagiri T, Baba M, Shimabukuro T, Hashimoto M, Takemoto H, Inoue A, Sugitani A, Shirakusa T. Lymphangioma in the small intestine Report of a case and review of the Japanese literature. Surgery Today 1992 22 363-367Roisman I, Manny J, Fields S, Shiloni E. Intra-abdominal lymphangioma. Br J Surg 1989 76 485-489Benjamin HL Tan, Teegan Lim. Cystic lymphangioma of the lesser sac presenting as acute appendicitis A case report. Cases J. 2008 1 147.Massoud Baghai-Wadji, Azadeh JalalKamali, ToorajReza Mirshekari. Cystic lymphangioma of the lesser sac. Can J Surg. 2006 49 292.Fernandez Hurtado I, Bregant J, Mulet Ferragut JF, et al. abdominal cystic lymphangioma. Cir Pediatr. 1998 11 171-3Chuang-Wei Chen, Sheng-Der Hsu, Chien-Hua Lin, Ming-Fang Cheng, Jyh-Cherng Yu Cystic lymphangioma of the jejunal mesentery in an adult A case report. World J Gastroenterol 2005 11 5084-5086Angela D. Levy, Vito Cantisani, Markku Miettinen. Abdominal Lymphangiomas Imagi ng Features with Pathologic Correlation. AJR 2004 182 1485-14918. Kenney B, Smith B, Bensoussan AL. Laparoscopic excision of a cystic lymphangioma. J Laparoendosc Surg. 1996 6 S99-101.9. Steyaert H, Guitard J, Moscovici J, Juricic M, Vaysse P, Juskiewenski S Abdominal cystic lymphangioma in children benign lesions that can have a proliferative course. J Pediatr Surg 1996, 31677-680.1