Lesser Sac Cystic Lymphangioma: Gastric Outlet Obstruction

Lesser Sac Cystic Lymphangioma: Gastric Outlet Obstruction Cystic Lymhangioma of the Lesser Sac in adult presenting with features of Gastric Outlet Obstruction A Case Report S Suresh Kumar Sri Aurobindo Prasad Das Vikram Kate Running title: Lesser sac cystic lymphangioma causing gastric outlet obstruction Key Words: Abdominal cystic Lymphangioma; Omental Bursa; Outlet Obstruction Article type: Case Report Section (speciality): Surgery- Gastrointestinal Surgery Abstract: Non- Structured (Case Report) ABSTRACT: Background: Cystic lymphangioma is a paediatric benign tumour, occurs commonly in head and neck region. Adult Intraabdominal lymphangiomas are uncommon and only few cases of lesser sac cystic lymphangioma are reported in the literature. Case presentation: We 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 abdomen 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. Ultrasound of abdomen demonstrated a multicystic lesion measuring 10.2 X 8 cm, with internal echoes in one locule. Contrast enhanced computed tomography of the abdomen showed a large multiloculated cystic lesion with hyper-dense content in the lesser sac extending up to the left 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 found 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. Conclusion: Though intraabdominal cystic lymphangiomas are very rare, it should be considered in all atypical cases of gastric outlet obstruction. A reasonable knowledge about these benign lesions will aid in establishing the diagnosis and surgical management. Key Words: Abdominal cystic Lymphangioma; Omental Bursa; Outlet Obstruction Background Lymphagiomas 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 years 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 presentation A 26-year old lady presented to surgical outpatient department with complaints of early satiety, vomiting few hours after food intake and vague fullness 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 in 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. Investigations The haemogram and biochemical profiles were within normal limits. An upper gastrointestinal endoscopy was done which showed normal mucosa 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 stomach, 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 planned for exploratory laparotomy as the symptoms were persisting. Surgical management On 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 drain was placed in lesser sac and abdomen was closed in layers. Outcome and follow- up Postoperative course was uneventful. The drain was removed on the 3nd postoperative 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 with 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 surgery. 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 till date. Discussion Intra-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. Commonly 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 weight loss, which are secondary to mass 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 acute 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 ultrasonography and contrast enhanced computerized tomography which is very often diagnostic [7]. The differential diagnosis includes 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. Conclusion: Though intra-abdominal cystic lymphangiomas are very rare, it should be considered in cases of gastric or biliary 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 review by the Editor of this journal. Competing interests disclosures The authors have no potential or real conflict of interests The authors have no financial support or ties to disclose The authors have no financial or non financial competing interests to declare Authors contributions Suresh 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 approved the final manuscript. References Hanagiri 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: 363-367 Roisman I, Manny J, Fields S, Shiloni E. Intra-abdominal lymphangioma. Br J Surg 1989; 76: 485-489 Benjamin 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: 171-3 Chuang-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: 5084-5086 Angela D. Levy, Vito Cantisani, Markku Miettinen. Abdominal Lymphangiomas: Imaging Features with Pathologic Correlation. AJR 2004; 182: 1485-1491 8. 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, 31:677-680. 1