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Spontaneous rupture of a giant non parasitic hepatic cyst presenting as an acute surgical abdomen

Annals of Hepatology 2007; 6(3): July-September: 190-193 Case Report
Spontaneous rupture of a giant non parasitic
hepatic cyst presenting as an acute surgical abdomen
Nikolaos S. Salemis;1,2 Epameinondas Georgoulis;1 Stavros Gourgiotis;2 Efstathios Tsohataridis1 Abstract
10:1 ratio, with an age range 50-60 years.2-4 The vast ma-jority are asymptomatic being detected incidentally during Spontaneous rupture of a non parasitic hepatic cyst is
abdominal imaging3 and are not associated with hepatic an extremely rare occurrence. A 50 -year- old male,
function abnormalities.5 Clinical symptoms occur in about was admitted with typical clinical manifestations of
5% of the patients due to a progressive increase of cyst’s acute surgical abdomen. At exploratory laparotomy, a
size, or the development of a complication.6 Complications giant ruptured non parasitic cyst occupying the entire
of NPHC are unusual and may include intracystic hemor- left liver lobe was found, along with a large amount of
rhage, torsion, infection, malignant transformation or com- free intraperitoneal fluid. The cyst was widely un-
pression of adjacent structures resulting most commonly roofed very close to the liver parenchyma. The patient
in obstructive jaundice.7,8 More rarely, spontaneous rup- had an uneventful postoperative course and was dis-
ture,9,10 inferior vena cava thrombosis11 and acute pulmo- charged six days later. The clinical presentation, diag-
nary embolism,12 have all been reported. We herein de- nostic evaluation and surgical management of this ex-
scribe the case of a spontaneous rupture of a giant non- tremely rare clinical entity are discussed, along with a
parasitic hepatic cyst in a 50 year old male, who presented review of the literature. This case, which according to
with acute surgical abdomen and was successfully treat- our best knowledge is the fourth reported in the litera-
ed with open wide unroofing. To our knowledge, this case ture, highlights the considerable risk of serious com-
appears to be the fourth reported in the literature.
plications associated with the presence of a large symp-
tomatic nonparasitic hepatic cyst. Prophylactic treat-

Case presentation
ment should be considered in all these cases.
A 50- year- old male was admitted with a 4 hour history Key words: Non parasitic hepatic cyst, spontaneous
of a sudden onset of severe abdominal pain, associated rupture, acute abdomen.
with nausea and vomiting. Although the pain was initiallyepigastric, it progressively became generalized. He also Introduction
complained of a dull epigastric pain and right upperguardant discomfort, which began to experience two Non parasitic hepatic cysts (NPHC) are detected in months prior to the current admission. His past medical 4.65% of the population, as a result of the increasing use history included chronic obstructive pulmonary disease, and refinement of abdominal imaging and ultrasonogra- phy.1 They are encountered more often in women by a 4- On clinical examination he was distressed, his abdo- men was distended with marked diffuse tenderness, 1 Army Veterans General Hospital NIMTS.
guarding and rebound tenderness. Bowel sounds were ab- sent. His blood pressure was 160/100 mmHg, pulse rate 90beats/minute and body temperature 37.6ºC. The haemo- 2nd Department of Surgery. Athens, Greece.
gram revealed a white cell count of 15,100 x103/ML with 91.3% neutrophils, a haemoglobin level of 14.2 g/dL and a platelet count of 231 x 103/mL. Liver function tests were normal. Abdominal ultrasonography demonstrated echo- patterns consistent with a large cystic lesion occupying almost the entire left liver lobe as well as fluid in the peri- toneal cavity. Abdominal computerized tomographic (CT) scans confirmed the presence of a huge homogeneous cyst with irregular borders in the left live lobe measuring 17 x 12 cm (Figure 1) and free intraperitoneal fluid espe- Manuscript received and accepted: 25 June and 12 July 2007 cially in the lower abdomen (Figures 2 and 3).
NS Salemis et al. Spontaneous rupture of a giant non parasitic hepatic cyst presenting as an acute surgical abdomen At emergency exploratory laparotomy, a giant rup- the possibility of spontaneous regression.16 The most tured cyst that occupied the left liver lobe was found and common presenting features in symptomatic patients are approximately 1,000 mL of intraperitoneal fluid were as- epigastric pain, nausea, vomiting, a feeling of fullness or pirated. A wide excision of the cyst was undertaken very close to the liver parenchyma. A running locking suture To our knowledge, only 14 cases of spontaneous rup- was used along the edge of the resected cyst wall, in order ture of NPHC have been reported in the English litera- to prevent bleeding and bile leakage. Intraoperative fro- ture.9,15 Sudden abdominal pain was the most frequent zen section of multiple specimens from the cystic wall symptom in 13 cases, while one patient was asymptomat- showed no evidence of malignancy, while cytology and ic. We were able to find only three cases of patients who cultures of the cystic fluid were negative. Serological developed acute abdomen following spontaneous rupture tests for echinococcus as well as tumor markers includ- of a NPHC.18-20 Our case appears to be the fourth reported.
ing CEA and CA 19/9 were negative. Histological exam- Several therapeutic approaches have been described ination of the entire resected specimen measuring 27 x for the management of NPHC, including guided aspiration 12 cm confirmed the diagnosis of a benign non hydatid, with or without injection of a sclerosant, open unroofing, non neoplastic simple liver cyst. The patient had an un-eventful postoperative course and was discharged sixdays later. Follow-up examinations performed six andtwelve months after surgery showed no findings of recur-rence.
Benign nonparasitic hepatic cysts are rare congenital lesions that arise from the development of aberrant intra-hepatic bile ducts in utero and are lined with cuboidalepithelium.13 They can be classified as congenital or ac-quired. Congenital cysts can be further divided into soli-tary benign nonparasitic cysts, polycystic liver disease,or Caroli’s disease. The acquired type includes neoplas-tic, traumatic and infectious cysts.14 The majority of he-patic cysts remain asymptomatic and managed conserva-tively,2,15 due to the risk of complications in therapy and Figures 2 and 3. Computerized tomographic scan demonstrates
Figure 1. Computerized tomographic scan demonstrates a huge
the presence of free intraperitoneal fluid, especially in the lower cyst with irregular borders, occupying the left liver lobe.
Annals of Hepatology 6(3) 2007: 190-193 laparoscopic unroofing, radical excision, cyst enterosto- In cases where the cystic fluid is bile stained and a my, hepatic resection and liver transplantation.4,13,21 Prior communication with the biliary tree is suspected, a cys- to any kind of intervention, a thorough diagnostic evalu- toenteric anastomosis using a Roux-en-Y jejunal loop is ation is mandatory. Ecchinococcal serology should be indicated.2,14,17 Intraperitoneal drainage in the above men- obtained in all patients with cystic liver lesions and rare tioned cases is unadvisable and usually results in the for- cases of cystic tumors need to be excluded.6,14 mation of a postoperative biloma.14,21 However the con- Ultrasonography or CT-guided cyst aspiration can be struction of a Roux-en-Y cystoenterostomy carries a con- easily performed in cysts larger than 5 cm in diameter,17 siderable risk of infectious complications and additional but is associated with recurrence in 100% of the cases21-23 demanding surgery. Tocchi et al,21 reported that all three and additionally carries a considerable risk of complica- patients who underwent a cystojejunostomy, developed tions. The only indications of the procedure are either pa- relapsing cholangitis requiring repeated antibiotic treat- tients with high operative risk or the need to perform a ment, while in one patient hepatic resection due to septic cytologic diagnosis.17 Additionally, it is a good thera- complications was performed one year later. More radical peutic test to ascertain whether abdominal symptoms are procedures like hepatic resection and cystectomy have related to the liver cyst14 in order to avoid an unnecessary also been advocated in the management of NPHC. Al- intervention.3 In order to destroy the secretory epithelial though these procedures obtain the best results in terms layer of the cyst, aspiration can be combined with the in- of recurrence rate,4,17,21 they are however associated with jection of a sclerosing agent such as ethanol, minocy- increased morbidity, which may not be acceptable for a cline chloride, or tetracycline chloride. This procedure benign disease. Martin et al,4 reported a morbidity rate of results to a much lower recurrence rate which may still be 50% among 16 patients who underwent hepatic resection as high as 20%13 but should be considered only after a for NPHC. Hepatic resections may be better reserved for malignant or infectious etiology and a biliary communi- patients with multiple recurrences or when a possibility of cation have all been ruled out.2,21 However, series of this a cystic tumor can not be ruled out.2,25 Radical cystectomy technique involve small number of patients and insuffi- is indicated in symptomatic cysts that can not be ade- cient follow-up do not allow adequate evaluation of its quately unroofed due to a small superficial component,14 while liver transplantation may be indicated in polycystic Laparoscopic unroofing has been increasingly used in liver patients with symptoms refractory to other therapeu- the management of NPHC. Various studies have shown tic approaches or with combined renal failure.2 that it is associated with reduced morbidity, shorter hospi- In conclusion, we describe an extremely rare case of a tal stay and more rapid return to normal activity, when spontaneous rupture of a huge nonparasitic hepatic cyst compared with open unroofing.24 Laparoscopic treatment in a male patient who presented with clinical manifesta- has been also been reported in the emergency setting, tions of acute surgical abdomen. Prophylactic treatment with good results.15 The two methods appear to have al- should be considered in symptomatic large non parasitic most similar recurrence rates.2,4,17,21,22 These findings are hepatic, cysts as they carry a considerable risk of serious however influenced by case selection and should be treat- complications. Adequate patient selection is of great im- ed with some caution.2,4 Additionally larger periods of fol- portance for achieving acceptable long term results.
low-up are also necessary.4 Gigot et al,3 in a series of 26patients found that laparoscopic fenestration was techni- References
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may affect recurrence have been identified, including in- Echographic epidemiology of non-parasitic hepatic cysts. J Clin adequate extent of deroofing, deep cysts and cysts in Ultrasound 1993; 21: 115-118.
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M, Kinoshita H, Shirouzou K. Nonparasitic solitary huge liver NS Salemis et al. Spontaneous rupture of a giant non parasitic hepatic cyst presenting as an acute surgical abdomen cysts causing intracystic hemorrhage or obstructive jaundice. J 17. Koperna T, Vogl S, Satzinger U, Schulz F. Nonparasitic cysts of Hepatobiliary Pancreat Surg 2002; 9: 764-768.
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10. Yamaguchi M, Kuzume M, Matsumoto T, Matsumiya A, Nakano Acute abdomen in a haemodialyzed patient with polycystic kid- H, Kumada K. Spontaneous rupture of a nonparasitic liver cyst ney disease-rupture of a massive liver cyst. Nephrol Dial Trans- complicated by intracystic hemorrhage. J Gastroenterol 1999; 20. Morano C, Fornari L, Persico SL. Acute abdomen caused by the 11. Torzilli G, Santambrogio R, Vellini S, Palmisano A, Donadon M, rupture of a solitary non-parasitic cyst of the liver. Minerva Chir Cornalba G. Montorsi. Inferior vena cava thrombosis: an un- usual complication of a large simple non-parasitic liver cyst re- 21. Tocchi A, Mazzoni G, Costa G, Cassini D, Bettelli E, Agostini N, quiring an intergrated approach. Hepatogastroenterology 2003; Miccini M. Symptomatic nonparasitic hepatic cysts. Arch Surg 12. Buyse S, Asselah T, Vilgrain V, Paradis V, Sauvanet A, Consigny 22. Regev A, Reddy KR, Berho M, Sleeman D, Levi JU, Livingstone Y, Dufour V, Fantin B, Valla D, Marcellin P. Acute pulmonary AS, Levi D, Ali U, Molina E, Schiff ER Large cystic lesions of the embolism: a rare complication of a large non-parasitic hepatic liver in adults: a 15-year experience in a tertiary center. J Am Col cyst. Eur J Gastroenterol Hepatol 2004; 16: 1241-1244.
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