Transactions of the Royal Society of Tropical Medicine and Hygiene Advance Access published January 30, 2014
Abdominal echinococcosis: outcomes of conservative surgery Anshuman Pandeya,*, Abhijit Chandrab and Shakeel Masooda a
Department of Surgical Gastroenterology, Dr Ram Manohar Lohia Institute of Medical Sciences, Vibhuti Khand, Gomti Nagar, Lucknow 226010, India; bDepartment of Surgical Gastroenterology, King George’s Medical University, Lucknow 226003, India
ORIGINAL ARTICLE
Trans R Soc Trop Med Hyg doi:10.1093/trstmh/tru003
*Corresponding author: Tel: +91 522 4918504; fax: +91 522 4918506; E-mail: [email protected]
Background: Hydatid disease, infection with the larval stage of the cestode Echinococcus spp., represents a substantial disease burden worldwide. We report here the outcomes of conservative surgery in patients with abdominal echinococcosis. Methods: We carried out a retrospective review of patients who underwent conservative surgery for abdominal hydatid disease during the period January 2008 to December 2011. Perioperative outcomes were analysed after a mean follow-up of 24 months (range 6–36 months). Results: Thirty patients (mean age 40.4 years; male:female¼7:3) underwent surgery. Most (29 patients) had a hepatic hydatid cyst and underwent partial cystectomy with omentoplasty; surgery was open in 22 cases (73%), laparoscopic in six cases (20%) and laparoscopic converted to open in one case (3%); one patient with a splenic cyst underwent open splenectomy. Cystobiliary communication was present in 10 cases (3%). Postoperative complications included transient biliary leak in two cases (7%), grade 1 surgical site infection in five cases (17%) and respiratory tract infection in three cases (10%), with no mortality. Mean hospital stay was 17+9.2 days. None of the patients had recurrence of disease on follow-up imaging. Conclusion: Conservative surgery offers an effective approach for abdominal echinococcosis, with minimal morbidity or recurrence, and is an alternative to radical procedures. Keywords: Abdominal echinococcosis, Conservative surgery, Hydatid disease, Morbidity, Outcomes, Recurrence
Introduction Hydatid disease is endemic mainly in the Mediterranean countries, the Middle East, the Baltic areas, South America, India and Northern China.1,2 In Southern India the incidence is 1–200 per 100 000 population. The disease occurs where livestock, mainly sheep and cattle, are raised with dogs, the dog being the definitive hosts for the adult phase of the echinococcal tapeworm. However, with increasing worldwide travel and tourism, hydatid disease can occur anywhere, even in developed countries. Cystic echinococcosis is among the most neglected parasitic diseases. Little effort is being made to develop newer drugs and treatment modalities to improve outcomes, and in managing the disease little attention is paid to monitoring efficacy, relapse, adverse reactions or cost-effectiveness. The disease is a zoonotic infection caused by adult or larval stages of the cestode Echinococcus granulosus. The liver is the organ commonly affected.3,4 Once a cyst has developed in the human liver, it grows by as much as 1 cm during the first 6 months and 2–3 cm every year thereafter, depending on the host’s resistance.5,6 Liver cysts may remain asymptomatic for years or may
spontaneously regress.3 More commonly the disease is slowly progressive and if not treated causes symptoms and complications. The cysts may form fistulas into adjacent organs or rupture into the peritoneal cavity. Older cysts tend to form exogenous daughter cysts, a significant factor for recurrence of the disease after surgery.7,8 When the results of medical therapy for hydatid disease are not satisfactory, surgery remains the mainstay of treatment. Several surgical techniques have been proposed for liver disease, including classic open surgical techniques like cyst ablation, deroofing of the cyst with omentoplasty, cyst drainage, marsupialisation, pericystectomy and, for large cysts, liver resection.8–10 Resection of small, superficial cysts is a simple and fast procedure; however, in the case of large cysts, cystectomy involves a major liver resection with its associated operative risk.9–11 Conservative procedures, such as cyst drainage or omentoplasty on the residual cavity, are easier to perform, but they carry a considerable morbidity and recurrence rate.2,13 More recently, minimally invasive procedures, such as percutaneous aspiration of cysts and laparoscopic surgery have been developed.14,15 The choice of surgical therapy depends on the patient’s general condition, the number and localisation of cysts, and the available expertise.
# The Author 2014. Published by Oxford University Press on behalf of Royal Society of Tropical Medicine and Hygiene. All rights reserved. For permissions, please e-mail: [email protected].
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Received 29 October 2013; revised 3 January 2014; accepted 3 January 2014
A. Pandey et al.
We describe here a retrospective review and the long-term results of conservative surgical procedures performed in 30 patients treated for hydatid disease in our centre over a 3-year period.
Methods
Results Between 2008 and 2011, 30 patients were operated upon for liver hydatid cysts at our institution. The study group consisted of nine women (9/30; 30%) and 21 men (21/30; 70%). The age range was 5–73 years (mean age 40.4 years), and patients came to our referral hospital from both rural and urban areas. The most common complaints, present in 21/30 (70%) and 6/30 (20%) of cases respectively, were dull pain at the right upper quadrant and epigastrium and a palpable mass. Less common symptoms were dyspepsia associated with other vague complaints and weight loss (Table 1). Cholangitis was the initial presentation in four patients
Discussion Table 1. Presenting complaints in 30 patients who underwent conservative surgery for abdominal hydatid disease in Lucknow, India, between January 2008 and December 2011 Complaint
No. of patients (%)
Abdominal pain Abdominal lump Cholangitis Dyspepsia Weight loss
21 (70) 5 (16.7) 4 (13.4) 2 (6.7) 2 (6.7)
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Hydatid disease is a continuing public health problem in endemic countries. The liver is the most common site for hydatid disease (75% of cases), followed by the lungs (15%), the spleen (5%) and other organs (5%).2,16,17 Although the disease is usually asymptomatic for many years because of the slow growth of the cyst, it is progressive and can cause severe complications. It tends to recur.8,10 The hydatid cyst slowly enlarges in the liver parenchyma and can cause symptoms such as dull pain in the right upper quadrant, hepatomegaly and formation of a palpable mass.16 The germinal layer gives rise to daughter cysts, which may arise exogenously. This is the plausible explanation for multiple liver cysts in certain patients.8 Pressure may increase within the cyst, causing it to rupture into the adjacent bile ducts with the
Downloaded from http://trstmh.oxfordjournals.org/ at Belgorod State University on February 4, 2014
All patients undergoing surgical treatment for abdominal hydatid disease at King George’s Medical University and Dr Ram Manohar Lohia Institute of Medical Sciences, Lucknow, India, during January 2008 to December 2011 were included in the study. A written informed consent was taken from all the patients. A complete detailed history was taken from each patient, and patients received a complete general physical examination and abdominal examination. Preoperative evaluation included blood tests (complete blood count, liver function tests and anti-echinococcus antibody testing) and radiological imaging; abdominal ultrasonography and a CT scan were done routinely. Endoscopic retrograde cholangiopancreatography (ERCP) and stenting were carried out routinely in patients presenting with preoperative cholangitis. All patients received albendazaole for 6 weeks before surgery and for 2 weeks postoperatively. All the patients underwent partial cystectomy, with evacuation of cyst content by a laparoscopic or open technique; adequate precautions were taken to prevent spillage and peritoneal contamination. A cholecystectomy was performed routinely in those who had undergone preoperative stenting. After surgery, patients were followed up with an abdominal ultrasound after a 6 month interval to document any residual cyst or recurrence.
(4/30; 13%). The diagnosis was established by combining the physical findings and results of the laboratory tests. The haematological tests had a very low sensitivity, with eosinophilia present in only nine patients (9/30; 30%). Liver function test results were abnormal in 13 patients (13/30; 43%), elevation of serum alkaline phosphatase being the most common finding. Among the serological tests the ELISA for hydatid was positive in 21 patients (21/30; 70%). Ultrasonography and CT scans were used to support the diagnosis. Ultrasonography was diagnostic in all but two patients in whom it was used (sensitivity, 93%). Computed tomography (Figure 1) was diagnostic in all patients (sensitivity, 100%). The imaging revealed that liver cysts were solitary in 23 cases (23/30; 77%) and multiple in six cases (6/30; 20%). Cysts were found in the right lobe in 21 cases (21/30; 70%), the left lobe in five cases (5/30; 18%), and in both lobes in three cases (3/30; 10%). One patient (1/30; 3%) had an isolated hydatid cyst in the spleen. Cyst diameter ranged from 1.3 to 15 cm, with a mean size of 7.6 cm. Preoperative ERCP was done in four (4/30; 13%) patients with preoperative cholangitis. Treatment was surgical in all cases. Partial cystectomy with omentoplasty (Figure 2) was performed in all patients; 22 (22/ 30; 73%) underwent open surgery and six (6/30; 20%) a laparoscopic procedure. One patient had the procedure converted from laparoscopic to open because of technical difficulty and one (1/30; 3%) underwent open splenectomy. Abdominal packing was done with pads soaked in betadine and hypertonic saline. Visible biliary communication was seen in 10 patients (10/30; 33%), and these were managed by suturing with fine absorbable sutures. Of the 21 cysts in the right lobe, most were in segments V and VIII (90%). There was one cyst in segment VI and one in segment VII. In four patients who had undergone preoperative biliary stenting a concomitant cholecystectomy was performed. The postoperative course in most patients was satisfactory. Mean hospital stay was 17+9.2 days. The main postoperative complications were septic wound infection in five patients and respiratory infection in three patients. All these complications were managed routinely. Two patients had biliary leak in the postoperative period, with an output of less than 100 ml/day that had stopped spontaneously by seventh postoperative day. No patient required postoperative ERCP and stenting. The mean follow up was 24 months (range 6–36 months) and during this period there was no recurrence in any of the patients.
Transactions of the Royal Society of Tropical Medicine and Hygiene
Figure 2. Intraoperative images of hydatid cysts showing (A) multiple liver cysts; (B) a single liver cyst; (C) a splenic cyst (splenectomy specimen).
release of daughter cysts, resulting in biliary colic and jaundice.18 This occurred in four of our patients (13%). Infection is another complication, which occurs when both the pericyst (outer layer of the cyst wall) and endocyst (middle and inner layers) perforate, allowing bacteria to pass easily into the cyst and give rise to a hepatic abscess.19 Diagnosis is based on ultrasonography and CT scans. Imaging shows large cystic lobulated structures containing multiple daughter vesicles or membranes, septa, and hydatid sand (sediment of scolices and vesicles). The cyst wall may be partially or heavily calcified. Heavily calcified cysts are considered inactive, but partial calcification of the cyst does not always indicate death of the parasite. In our study ultrasonography and CT scans had a diagnostic sensitivity of more than 90%. Hydatid disease is also confirmed by a high antibody titre to hydatid antigen using the counter immunoelectrophoresis test. Enzyme-linked immunosorbent assay, with a sensitivity rate of 56.7–70%, is considered highly specific for the diagnosis of human echinococcosis, especially when used for locations other than the liver or the lung, or for calcified cysts.20 Other laboratory tests, such as haematological tests and biochemical liver tests, are considered less sensitive. Some authors consider the absolute and relative eosinophil count to be important,20 but eosinophilia was present in only 11 (11/30; 35%) of our patients, and abnormal liver function in 13 (13/30; 43%).
Medical treatment of hydatid liver cysts is based on the use of benzoimidazole carbamates, such as mebendazole and albendazole. The aim of treatment is to reduce the size of cysts and prevent distal metastasis, and the success of drug therapy relies on the drug’s ability to penetrate the cyst wall and on persistence of adequate levels of the active metabolites. Albendazole is more effective than mebendazole, because of superior cyst penetration and absorption. Both agents have been used in several studies as a conservative treatment, achieving stabilisation of cyst size or some reduction, especially in patients with small cysts.21–23 However, their clinical efficacy remains doubtful.8,22,24 They are used mainly to manage disseminated systemic disease and in patients with inoperable disease, and are used in combination with surgical treatment, as we did, to prevent postoperative recurrence.3,8,22,25 Chemotherapy is contraindicated in patients with large cysts that have a risk of rupture, especially if the cysts are superficial or infected, because of the possibility of hydatid abscess. Follow-up of medical treatment with ultrasound examinations is considered necessary, as progression of hydatid disease does not usually produce specific symptoms.20 Medical therapy may be inadequate in disease management, so surgery remains the primary treatment for liver hydatidosis. However, the surgical approach may vary in different institutions and choice of the appropriate surgical procedure is controversial. The principle of surgical treatment is to evacuate the cyst while
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Figure 1. CT scans showing (A) single and (B) multiple hepatic hydatid cysts, in the right lobe.
A. Pandey et al.
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drainage or radical surgery combined with albendazole therapy.36,37 Although morbidity in the current study was less than has been reported in other published studies, and our patients experienced no disease recurrence, a limitation of our study is that the follow-up period remains short at only up to 36 months. The study also does not clearly indicate criteria for the selection of patients for a laparoscopic procedure rather than open surgery. However, our results emphasise the superior outcomes achieved with conservative techniques, with reduced morbidity and no mortality.
Conclusion Conservative techniques offer an effective means of controlling hepatic hydatidosis and can replace unnecessary radical procedures when appropriate. When an appropriate operative strategy is chosen after consideration of multiple factors relating to the patient’s particular medical situation, such as the size and location of the cyst, existing complications, and other diseases, the ideal result can be achieved with a minimal risk of postoperative morbidity or recurrence.
Authors’ contributions: AP and AC conceived the study; AP, AC and SM undertook the surgical procedures and documented the details; AP and AC carried out endoscopic procedures as required. All authors took part in the analysis and interpretation of the data. AP drafted the manuscript and dealt with queries. All authors read and approved the final version. AP is guarantor of the paper. Acknowledgements: We thank the surgical and nursing team for their contributions to the study and the patients for their faith in us and giving us an opportunity to serve them. Funding: None. Competing interests: None declared. Ethical approval: Not required.
References 1 Doty JE, Tompkins RK. Management of cystic disease of the liver. Surg Clin North Am 1989;69:285–95. 2 Safioleas M, Misiakos EP, Kakisis J et al. Surgical treatment of human echinococossis. Int Surg 2000;85:358–65. 3 Chautems R, Buhler L, Gold B et al. Long term results after complete or incomplete surgical resection of liver hydatid disease. Swiss Med Wkly 2003;133:258–62. 4 Amr SS, Amr ZS, Jitawi S et al. Hydatidosis in Jordan: an epidemiological study of 306 cases. Ann Trop Med Parasitol 1994;88:623–7. 5 Pedrosa I, Saiz A, Arrazola J et al. Hydatid disease: radiologic and pathologic features and complications. Radiographics 2000;20: 795–817. 6 Lewall DB. Hydatid disease: biology, pathology, imaging and classification. Clin Radiol 1998;52:863–74. 7 Guntz M. Anatomo-physio-pathological bases for surgical treatment of hydatid cyst of the liver [in French]. Minerva Chir 1973;28:708–16.
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avoiding spillage of cyst contents, neutralise the cyst and obliterate the residual cavity.11,12,26 During evacuation it is highly important to prevent spillage of daughter cysts. Different scolicidal agents have been used to irrigate the cyst cavity, together with abdominal packing, and patients can develop severe hypernatremia after acute absorption of sodium chloride in high concentrations.2 Several precautions can be taken to prevent intraoperative contamination of the peritoneal cavity during cyst evacuation; these include partially or gradually decompressing the cyst before opening the entire cyst wall. Use of wide tubing or multiple tubes is recommended for the evacuation of cyst contents, as these often block the suction devices during evacuation.8,27 Adequate drainage and obliteration of the remaining cavity is necessary to minimise the possibility of serum or blood accumulation or liver abscess formation.25 These sequelae, along with biliary leakage, constitute the main postoperative complications and are the main reasons for morbidity and mortality among these patients.3,8,25 Conservative surgical techniques are external drainage or marsupialisation and internal drainage, evacuation and capitonnage (closure of a cyst using sutures to approximate the opposing surfaces of the cavity) or omentoplasty (use of a flap of omentum to fill the cyst cavity).12 In previous studies, a remarkably lower morbidity and mortality rate has been observed among patients treated with cyst excision and omentoplasty.1,12,26,28–30 Furthermore, in the current study, partial cyst excision and omentoplasty had a significantly lower complication rate. Recent studies indicate that evacuation of the cyst content can be carried out successfully using laparoscopic methods. It is suggested that this method has a better yield for posterior cysts and it reduces the risk of spillage, thus decreasing the rate of recurrence.31,32 In addition, it provides better aesthetic results, and reduces the length of hospital stay. This procedure was performed successfully in six of our patients who had superficial cysts close to the liver capsule. Radical techniques, such as total pericystectomy, when feasible, provide better results, because they minimise the risk of spillage and recurrence3,33,34 However all radical procedures carry the risk of intraoperative or postoperative bleeding, owing to the absence of a true surgical cleavage with the liver parenchyma.2,8 This risk is much larger for cysts adherent to the hepatic veins, inferior vena cava or hepatic hilum.35 In our study, there were minimal postoperative complications in patients undergoing nonradical surgery for liver hydatid. The morbidity rate after surgery for hepatic hydatidosis has been reported to range from 6% to 47%.13,25,28,33 In our study the overall morbidity rate was 22% (7/30 patients), with the main complications being wound infection and respiratory tract infection. There was no mortality in the study. Recurrence of hydatid disease in the liver constitutes a main concern in these patients. It usually results from spillage of hydatid fluid containing daughter cysts during the operation or incomplete evacuation of the cyst following conservative procedures, leaving residual vesicles in place.3,8 Reported recurrence rates range from 6% to 25% when omentoplasty is used.2,13,36 In our study, with a follow up of 6–36 months, there has been no recurrence to date. Recommended practice is to follow up patients with ultrasonography every 6 months or an annual CT scan for at least 3 years, as most recurrences of hydatid disease are observed in this time period. Recurrent cysts should be managed by percutaneous
Transactions of the Royal Society of Tropical Medicine and Hygiene
8 Magistrelli P, Masetti R, Coppola R et al. Surgical treatment of hydatid disease of the liver: a 20-year experience. Arch Surg 1991;126:518–22.
22 Aktan AO, Yalin R. Preoperative albendazole treatment for liver hydatid disease decreases the viability of the cyst. Eur J Gastroenterol Hepatol 1996;8:877–9.
9 Filippou DK, Kolimpiris C, Anemodouras N et al. Modified capitonage in partial cystectomy performed for liver hydatid disease: report of 2 cases. BMC Surg 2004;4:8.
23 Blanton RE, Wachira TM, Zeyhle EE et al. Oxfendazole treatment for cystic hydatid disease in naturally infected animals. Antimicrob Agents Chemother 1998;42:601–5.
10 Langer JC, Rose DB, Keystone JS et al. Diagnosis and management of hydatid disease of the liver: a 15-year North American experience. Ann Surg 1984;199:412–7.
24 Teggi A, Lastilla MG, De Rosa F. Therapy of human hydatid disease with mebendazole and albendazole. Antimicrob Agents Chemother 1993;37:1679–84.
11 Balik AA, Basoglu M, Celebi F et al. Surgical treatment of hydatid disease of the liver: review of 304 cases. Arch Surg 1999;134:166–9.
25 Agaoglu N, Turkyilmaz S, Arslan MK. Surgical treatment of hydatid cysts of the liver. Br J Surg 2003;90:1536–41.
13 Vagianos CE, Karavias DD, Kakkos SK et al. Conservative surgery in the treatment of hepatic hydatidosis. Eur J Sur 1995;161:415–20. 14 Men S, Hekimoglu B, Yucesoy C et al. Percutaneous treatment of hepatic hydatid cysts: an alternative to surgery. Am J Roentgenol 1999;172:83–9. 15 Bickel A, Daud G, Urbach D et al. Laparoscopic approach to hydatid liver cysts: Is it logical? Physical, experimental, and practical aspects. Surg Endosc 1998; 12:1073–7. 16 Safioleas M, Stamoulis I, Theocharis S et al. Primary hydatid disease of the gallbladder: a rare clinical entity. J Hepatobiliary Pancreat Surg 2004;11:352–6. 17 Safioleas MC, Moulakakis KG, Manti C et al. Clinical considerations of primary hydatid disease of the pancreas. Pancreatology 2005;5:457–61. 18 Ovnat A, Peiser J, Avinoah E et al. Acute cholangitis caused by ruptured hydatic cyst. Surgery 1984;95:497–500. 19 Marti-Bonmati L, Menor Serrano F. Complications of hepatic hydatid cysts: ultrasound, computed tomography, and magnetic resonance diagnosis. Gastrointest Radiol 1990;15:119–25. 20 Niscigorska J, Sluzar T, Marczevska M et al. Parasitic cysts of the liver: practical approach to diagnosis and differentiation. Med Sci Monit 2001;7:737–41. 21 Haddad MC, Al-Awar G, Huwaijah SH et al. Echinococcal cysts of the liver: a retrospective analysis of clinico-radiological findings and different therapeutic modalities. Clin Imaging 2001;25:403–8.
26 Safioleas M, Misiakos E, Manti C et al. Diagnostic evaluation and surgical management of hydatid disease of the liver. World J Surg 1994;18:859–65. 27 Grosdidier J, Boissel P, Bresler L et al. Retrospective study of a series of 62 cases of hydatid liver cysts [in French]. J Chir (Paris) 1985;122:163–9. 28 Sayek I, Yalin R, Sanac Y. Surgical treatment of hydatid disease of the liver. Arch Surg 1980;115:847–50. 29 Mentes A, Yuzer Y, Ozbal O et al. Omentoplasty versus introflection for hydatid liver cysts. J R Coll Surg Edin 1993;38:82–5. 30 Uravic M, Stimac D, Lenac T et al. Diagnosis and treatment of liver hydatid disease. Hepatogastroenterology. 1998;45:2265–9. 31 Kayaalp C. Evacuation of hydatid cysts using laparoscopic trocar. World J Surg 2002;26:1324–7. 32 Bensaadi H, Champault G. Laparoscopic hand-assisted surgery for hydatid cysts of the liver. Surg Laparosc Endosc Percutan Tech 2004;14:91–2. 33 Alfieri S, Doglietto GB, Pacelli F et al. Radical surgery for liver hydatid disease: a study of 89 patients. Hepatogastroenterology 1997;44:496–500. 34 Nardo B, Patrici A, Piazzese E et al. Radical surgical treatment of recurrent hepatic hydatidosis. Hepatogastroenterology 2003;50: 1478–81. 35 Belli L, Aseni P, Rondinara GF et al. Improved results with pericystectomy in normothermic ischemia for hepatic hydatidosis. Surg Gynecol Obstet 1986;163:127–32. 36 Sielaff TD, Taylor B, Langer B. Recurrence of hydatid disease. World J Surg 2001;25:83–6. 37 Dziri C. Hydatid disease: continuing serious public health problem. World J Surg 2001;25:1–3.
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12 Dziri C, Paquet JC, Hay JM et al. Omentoplasty in the prevention of deep abdominal complications after surgery for hydatid disease of the liver: a multicenter, prospective, randomized trial. J Am Coll Surg 1999;188:281–9.
Abdominal echinococcosis: outcomes of conservative surgery Anshuman Pandeya,*, Abhijit Chandrab and Shakeel Masooda a
Department of Surgical Gastroenterology, Dr Ram Manohar Lohia Institute of Medical Sciences, Vibhuti Khand, Gomti Nagar, Lucknow 226010, India; bDepartment of Surgical Gastroenterology, King George’s Medical University, Lucknow 226003, India
ORIGINAL ARTICLE
Trans R Soc Trop Med Hyg doi:10.1093/trstmh/tru003
*Corresponding author: Tel: +91 522 4918504; fax: +91 522 4918506; E-mail: [email protected]
Background: Hydatid disease, infection with the larval stage of the cestode Echinococcus spp., represents a substantial disease burden worldwide. We report here the outcomes of conservative surgery in patients with abdominal echinococcosis. Methods: We carried out a retrospective review of patients who underwent conservative surgery for abdominal hydatid disease during the period January 2008 to December 2011. Perioperative outcomes were analysed after a mean follow-up of 24 months (range 6–36 months). Results: Thirty patients (mean age 40.4 years; male:female¼7:3) underwent surgery. Most (29 patients) had a hepatic hydatid cyst and underwent partial cystectomy with omentoplasty; surgery was open in 22 cases (73%), laparoscopic in six cases (20%) and laparoscopic converted to open in one case (3%); one patient with a splenic cyst underwent open splenectomy. Cystobiliary communication was present in 10 cases (3%). Postoperative complications included transient biliary leak in two cases (7%), grade 1 surgical site infection in five cases (17%) and respiratory tract infection in three cases (10%), with no mortality. Mean hospital stay was 17+9.2 days. None of the patients had recurrence of disease on follow-up imaging. Conclusion: Conservative surgery offers an effective approach for abdominal echinococcosis, with minimal morbidity or recurrence, and is an alternative to radical procedures. Keywords: Abdominal echinococcosis, Conservative surgery, Hydatid disease, Morbidity, Outcomes, Recurrence
Introduction Hydatid disease is endemic mainly in the Mediterranean countries, the Middle East, the Baltic areas, South America, India and Northern China.1,2 In Southern India the incidence is 1–200 per 100 000 population. The disease occurs where livestock, mainly sheep and cattle, are raised with dogs, the dog being the definitive hosts for the adult phase of the echinococcal tapeworm. However, with increasing worldwide travel and tourism, hydatid disease can occur anywhere, even in developed countries. Cystic echinococcosis is among the most neglected parasitic diseases. Little effort is being made to develop newer drugs and treatment modalities to improve outcomes, and in managing the disease little attention is paid to monitoring efficacy, relapse, adverse reactions or cost-effectiveness. The disease is a zoonotic infection caused by adult or larval stages of the cestode Echinococcus granulosus. The liver is the organ commonly affected.3,4 Once a cyst has developed in the human liver, it grows by as much as 1 cm during the first 6 months and 2–3 cm every year thereafter, depending on the host’s resistance.5,6 Liver cysts may remain asymptomatic for years or may
spontaneously regress.3 More commonly the disease is slowly progressive and if not treated causes symptoms and complications. The cysts may form fistulas into adjacent organs or rupture into the peritoneal cavity. Older cysts tend to form exogenous daughter cysts, a significant factor for recurrence of the disease after surgery.7,8 When the results of medical therapy for hydatid disease are not satisfactory, surgery remains the mainstay of treatment. Several surgical techniques have been proposed for liver disease, including classic open surgical techniques like cyst ablation, deroofing of the cyst with omentoplasty, cyst drainage, marsupialisation, pericystectomy and, for large cysts, liver resection.8–10 Resection of small, superficial cysts is a simple and fast procedure; however, in the case of large cysts, cystectomy involves a major liver resection with its associated operative risk.9–11 Conservative procedures, such as cyst drainage or omentoplasty on the residual cavity, are easier to perform, but they carry a considerable morbidity and recurrence rate.2,13 More recently, minimally invasive procedures, such as percutaneous aspiration of cysts and laparoscopic surgery have been developed.14,15 The choice of surgical therapy depends on the patient’s general condition, the number and localisation of cysts, and the available expertise.
# The Author 2014. Published by Oxford University Press on behalf of Royal Society of Tropical Medicine and Hygiene. All rights reserved. For permissions, please e-mail: [email protected].
1 of 5
Downloaded from http://trstmh.oxfordjournals.org/ at Belgorod State University on February 4, 2014
Received 29 October 2013; revised 3 January 2014; accepted 3 January 2014
A. Pandey et al.
We describe here a retrospective review and the long-term results of conservative surgical procedures performed in 30 patients treated for hydatid disease in our centre over a 3-year period.
Methods
Results Between 2008 and 2011, 30 patients were operated upon for liver hydatid cysts at our institution. The study group consisted of nine women (9/30; 30%) and 21 men (21/30; 70%). The age range was 5–73 years (mean age 40.4 years), and patients came to our referral hospital from both rural and urban areas. The most common complaints, present in 21/30 (70%) and 6/30 (20%) of cases respectively, were dull pain at the right upper quadrant and epigastrium and a palpable mass. Less common symptoms were dyspepsia associated with other vague complaints and weight loss (Table 1). Cholangitis was the initial presentation in four patients
Discussion Table 1. Presenting complaints in 30 patients who underwent conservative surgery for abdominal hydatid disease in Lucknow, India, between January 2008 and December 2011 Complaint
No. of patients (%)
Abdominal pain Abdominal lump Cholangitis Dyspepsia Weight loss
21 (70) 5 (16.7) 4 (13.4) 2 (6.7) 2 (6.7)
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Hydatid disease is a continuing public health problem in endemic countries. The liver is the most common site for hydatid disease (75% of cases), followed by the lungs (15%), the spleen (5%) and other organs (5%).2,16,17 Although the disease is usually asymptomatic for many years because of the slow growth of the cyst, it is progressive and can cause severe complications. It tends to recur.8,10 The hydatid cyst slowly enlarges in the liver parenchyma and can cause symptoms such as dull pain in the right upper quadrant, hepatomegaly and formation of a palpable mass.16 The germinal layer gives rise to daughter cysts, which may arise exogenously. This is the plausible explanation for multiple liver cysts in certain patients.8 Pressure may increase within the cyst, causing it to rupture into the adjacent bile ducts with the
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All patients undergoing surgical treatment for abdominal hydatid disease at King George’s Medical University and Dr Ram Manohar Lohia Institute of Medical Sciences, Lucknow, India, during January 2008 to December 2011 were included in the study. A written informed consent was taken from all the patients. A complete detailed history was taken from each patient, and patients received a complete general physical examination and abdominal examination. Preoperative evaluation included blood tests (complete blood count, liver function tests and anti-echinococcus antibody testing) and radiological imaging; abdominal ultrasonography and a CT scan were done routinely. Endoscopic retrograde cholangiopancreatography (ERCP) and stenting were carried out routinely in patients presenting with preoperative cholangitis. All patients received albendazaole for 6 weeks before surgery and for 2 weeks postoperatively. All the patients underwent partial cystectomy, with evacuation of cyst content by a laparoscopic or open technique; adequate precautions were taken to prevent spillage and peritoneal contamination. A cholecystectomy was performed routinely in those who had undergone preoperative stenting. After surgery, patients were followed up with an abdominal ultrasound after a 6 month interval to document any residual cyst or recurrence.
(4/30; 13%). The diagnosis was established by combining the physical findings and results of the laboratory tests. The haematological tests had a very low sensitivity, with eosinophilia present in only nine patients (9/30; 30%). Liver function test results were abnormal in 13 patients (13/30; 43%), elevation of serum alkaline phosphatase being the most common finding. Among the serological tests the ELISA for hydatid was positive in 21 patients (21/30; 70%). Ultrasonography and CT scans were used to support the diagnosis. Ultrasonography was diagnostic in all but two patients in whom it was used (sensitivity, 93%). Computed tomography (Figure 1) was diagnostic in all patients (sensitivity, 100%). The imaging revealed that liver cysts were solitary in 23 cases (23/30; 77%) and multiple in six cases (6/30; 20%). Cysts were found in the right lobe in 21 cases (21/30; 70%), the left lobe in five cases (5/30; 18%), and in both lobes in three cases (3/30; 10%). One patient (1/30; 3%) had an isolated hydatid cyst in the spleen. Cyst diameter ranged from 1.3 to 15 cm, with a mean size of 7.6 cm. Preoperative ERCP was done in four (4/30; 13%) patients with preoperative cholangitis. Treatment was surgical in all cases. Partial cystectomy with omentoplasty (Figure 2) was performed in all patients; 22 (22/ 30; 73%) underwent open surgery and six (6/30; 20%) a laparoscopic procedure. One patient had the procedure converted from laparoscopic to open because of technical difficulty and one (1/30; 3%) underwent open splenectomy. Abdominal packing was done with pads soaked in betadine and hypertonic saline. Visible biliary communication was seen in 10 patients (10/30; 33%), and these were managed by suturing with fine absorbable sutures. Of the 21 cysts in the right lobe, most were in segments V and VIII (90%). There was one cyst in segment VI and one in segment VII. In four patients who had undergone preoperative biliary stenting a concomitant cholecystectomy was performed. The postoperative course in most patients was satisfactory. Mean hospital stay was 17+9.2 days. The main postoperative complications were septic wound infection in five patients and respiratory infection in three patients. All these complications were managed routinely. Two patients had biliary leak in the postoperative period, with an output of less than 100 ml/day that had stopped spontaneously by seventh postoperative day. No patient required postoperative ERCP and stenting. The mean follow up was 24 months (range 6–36 months) and during this period there was no recurrence in any of the patients.
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Figure 2. Intraoperative images of hydatid cysts showing (A) multiple liver cysts; (B) a single liver cyst; (C) a splenic cyst (splenectomy specimen).
release of daughter cysts, resulting in biliary colic and jaundice.18 This occurred in four of our patients (13%). Infection is another complication, which occurs when both the pericyst (outer layer of the cyst wall) and endocyst (middle and inner layers) perforate, allowing bacteria to pass easily into the cyst and give rise to a hepatic abscess.19 Diagnosis is based on ultrasonography and CT scans. Imaging shows large cystic lobulated structures containing multiple daughter vesicles or membranes, septa, and hydatid sand (sediment of scolices and vesicles). The cyst wall may be partially or heavily calcified. Heavily calcified cysts are considered inactive, but partial calcification of the cyst does not always indicate death of the parasite. In our study ultrasonography and CT scans had a diagnostic sensitivity of more than 90%. Hydatid disease is also confirmed by a high antibody titre to hydatid antigen using the counter immunoelectrophoresis test. Enzyme-linked immunosorbent assay, with a sensitivity rate of 56.7–70%, is considered highly specific for the diagnosis of human echinococcosis, especially when used for locations other than the liver or the lung, or for calcified cysts.20 Other laboratory tests, such as haematological tests and biochemical liver tests, are considered less sensitive. Some authors consider the absolute and relative eosinophil count to be important,20 but eosinophilia was present in only 11 (11/30; 35%) of our patients, and abnormal liver function in 13 (13/30; 43%).
Medical treatment of hydatid liver cysts is based on the use of benzoimidazole carbamates, such as mebendazole and albendazole. The aim of treatment is to reduce the size of cysts and prevent distal metastasis, and the success of drug therapy relies on the drug’s ability to penetrate the cyst wall and on persistence of adequate levels of the active metabolites. Albendazole is more effective than mebendazole, because of superior cyst penetration and absorption. Both agents have been used in several studies as a conservative treatment, achieving stabilisation of cyst size or some reduction, especially in patients with small cysts.21–23 However, their clinical efficacy remains doubtful.8,22,24 They are used mainly to manage disseminated systemic disease and in patients with inoperable disease, and are used in combination with surgical treatment, as we did, to prevent postoperative recurrence.3,8,22,25 Chemotherapy is contraindicated in patients with large cysts that have a risk of rupture, especially if the cysts are superficial or infected, because of the possibility of hydatid abscess. Follow-up of medical treatment with ultrasound examinations is considered necessary, as progression of hydatid disease does not usually produce specific symptoms.20 Medical therapy may be inadequate in disease management, so surgery remains the primary treatment for liver hydatidosis. However, the surgical approach may vary in different institutions and choice of the appropriate surgical procedure is controversial. The principle of surgical treatment is to evacuate the cyst while
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Figure 1. CT scans showing (A) single and (B) multiple hepatic hydatid cysts, in the right lobe.
A. Pandey et al.
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drainage or radical surgery combined with albendazole therapy.36,37 Although morbidity in the current study was less than has been reported in other published studies, and our patients experienced no disease recurrence, a limitation of our study is that the follow-up period remains short at only up to 36 months. The study also does not clearly indicate criteria for the selection of patients for a laparoscopic procedure rather than open surgery. However, our results emphasise the superior outcomes achieved with conservative techniques, with reduced morbidity and no mortality.
Conclusion Conservative techniques offer an effective means of controlling hepatic hydatidosis and can replace unnecessary radical procedures when appropriate. When an appropriate operative strategy is chosen after consideration of multiple factors relating to the patient’s particular medical situation, such as the size and location of the cyst, existing complications, and other diseases, the ideal result can be achieved with a minimal risk of postoperative morbidity or recurrence.
Authors’ contributions: AP and AC conceived the study; AP, AC and SM undertook the surgical procedures and documented the details; AP and AC carried out endoscopic procedures as required. All authors took part in the analysis and interpretation of the data. AP drafted the manuscript and dealt with queries. All authors read and approved the final version. AP is guarantor of the paper. Acknowledgements: We thank the surgical and nursing team for their contributions to the study and the patients for their faith in us and giving us an opportunity to serve them. Funding: None. Competing interests: None declared. Ethical approval: Not required.
References 1 Doty JE, Tompkins RK. Management of cystic disease of the liver. Surg Clin North Am 1989;69:285–95. 2 Safioleas M, Misiakos EP, Kakisis J et al. Surgical treatment of human echinococossis. Int Surg 2000;85:358–65. 3 Chautems R, Buhler L, Gold B et al. Long term results after complete or incomplete surgical resection of liver hydatid disease. Swiss Med Wkly 2003;133:258–62. 4 Amr SS, Amr ZS, Jitawi S et al. Hydatidosis in Jordan: an epidemiological study of 306 cases. Ann Trop Med Parasitol 1994;88:623–7. 5 Pedrosa I, Saiz A, Arrazola J et al. Hydatid disease: radiologic and pathologic features and complications. Radiographics 2000;20: 795–817. 6 Lewall DB. Hydatid disease: biology, pathology, imaging and classification. Clin Radiol 1998;52:863–74. 7 Guntz M. Anatomo-physio-pathological bases for surgical treatment of hydatid cyst of the liver [in French]. Minerva Chir 1973;28:708–16.
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avoiding spillage of cyst contents, neutralise the cyst and obliterate the residual cavity.11,12,26 During evacuation it is highly important to prevent spillage of daughter cysts. Different scolicidal agents have been used to irrigate the cyst cavity, together with abdominal packing, and patients can develop severe hypernatremia after acute absorption of sodium chloride in high concentrations.2 Several precautions can be taken to prevent intraoperative contamination of the peritoneal cavity during cyst evacuation; these include partially or gradually decompressing the cyst before opening the entire cyst wall. Use of wide tubing or multiple tubes is recommended for the evacuation of cyst contents, as these often block the suction devices during evacuation.8,27 Adequate drainage and obliteration of the remaining cavity is necessary to minimise the possibility of serum or blood accumulation or liver abscess formation.25 These sequelae, along with biliary leakage, constitute the main postoperative complications and are the main reasons for morbidity and mortality among these patients.3,8,25 Conservative surgical techniques are external drainage or marsupialisation and internal drainage, evacuation and capitonnage (closure of a cyst using sutures to approximate the opposing surfaces of the cavity) or omentoplasty (use of a flap of omentum to fill the cyst cavity).12 In previous studies, a remarkably lower morbidity and mortality rate has been observed among patients treated with cyst excision and omentoplasty.1,12,26,28–30 Furthermore, in the current study, partial cyst excision and omentoplasty had a significantly lower complication rate. Recent studies indicate that evacuation of the cyst content can be carried out successfully using laparoscopic methods. It is suggested that this method has a better yield for posterior cysts and it reduces the risk of spillage, thus decreasing the rate of recurrence.31,32 In addition, it provides better aesthetic results, and reduces the length of hospital stay. This procedure was performed successfully in six of our patients who had superficial cysts close to the liver capsule. Radical techniques, such as total pericystectomy, when feasible, provide better results, because they minimise the risk of spillage and recurrence3,33,34 However all radical procedures carry the risk of intraoperative or postoperative bleeding, owing to the absence of a true surgical cleavage with the liver parenchyma.2,8 This risk is much larger for cysts adherent to the hepatic veins, inferior vena cava or hepatic hilum.35 In our study, there were minimal postoperative complications in patients undergoing nonradical surgery for liver hydatid. The morbidity rate after surgery for hepatic hydatidosis has been reported to range from 6% to 47%.13,25,28,33 In our study the overall morbidity rate was 22% (7/30 patients), with the main complications being wound infection and respiratory tract infection. There was no mortality in the study. Recurrence of hydatid disease in the liver constitutes a main concern in these patients. It usually results from spillage of hydatid fluid containing daughter cysts during the operation or incomplete evacuation of the cyst following conservative procedures, leaving residual vesicles in place.3,8 Reported recurrence rates range from 6% to 25% when omentoplasty is used.2,13,36 In our study, with a follow up of 6–36 months, there has been no recurrence to date. Recommended practice is to follow up patients with ultrasonography every 6 months or an annual CT scan for at least 3 years, as most recurrences of hydatid disease are observed in this time period. Recurrent cysts should be managed by percutaneous
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8 Magistrelli P, Masetti R, Coppola R et al. Surgical treatment of hydatid disease of the liver: a 20-year experience. Arch Surg 1991;126:518–22.
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9 Filippou DK, Kolimpiris C, Anemodouras N et al. Modified capitonage in partial cystectomy performed for liver hydatid disease: report of 2 cases. BMC Surg 2004;4:8.
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10 Langer JC, Rose DB, Keystone JS et al. Diagnosis and management of hydatid disease of the liver: a 15-year North American experience. Ann Surg 1984;199:412–7.
24 Teggi A, Lastilla MG, De Rosa F. Therapy of human hydatid disease with mebendazole and albendazole. Antimicrob Agents Chemother 1993;37:1679–84.
11 Balik AA, Basoglu M, Celebi F et al. Surgical treatment of hydatid disease of the liver: review of 304 cases. Arch Surg 1999;134:166–9.
25 Agaoglu N, Turkyilmaz S, Arslan MK. Surgical treatment of hydatid cysts of the liver. Br J Surg 2003;90:1536–41.
13 Vagianos CE, Karavias DD, Kakkos SK et al. Conservative surgery in the treatment of hepatic hydatidosis. Eur J Sur 1995;161:415–20. 14 Men S, Hekimoglu B, Yucesoy C et al. Percutaneous treatment of hepatic hydatid cysts: an alternative to surgery. Am J Roentgenol 1999;172:83–9. 15 Bickel A, Daud G, Urbach D et al. Laparoscopic approach to hydatid liver cysts: Is it logical? Physical, experimental, and practical aspects. Surg Endosc 1998; 12:1073–7. 16 Safioleas M, Stamoulis I, Theocharis S et al. Primary hydatid disease of the gallbladder: a rare clinical entity. J Hepatobiliary Pancreat Surg 2004;11:352–6. 17 Safioleas MC, Moulakakis KG, Manti C et al. Clinical considerations of primary hydatid disease of the pancreas. Pancreatology 2005;5:457–61. 18 Ovnat A, Peiser J, Avinoah E et al. Acute cholangitis caused by ruptured hydatic cyst. Surgery 1984;95:497–500. 19 Marti-Bonmati L, Menor Serrano F. Complications of hepatic hydatid cysts: ultrasound, computed tomography, and magnetic resonance diagnosis. Gastrointest Radiol 1990;15:119–25. 20 Niscigorska J, Sluzar T, Marczevska M et al. Parasitic cysts of the liver: practical approach to diagnosis and differentiation. Med Sci Monit 2001;7:737–41. 21 Haddad MC, Al-Awar G, Huwaijah SH et al. Echinococcal cysts of the liver: a retrospective analysis of clinico-radiological findings and different therapeutic modalities. Clin Imaging 2001;25:403–8.
26 Safioleas M, Misiakos E, Manti C et al. Diagnostic evaluation and surgical management of hydatid disease of the liver. World J Surg 1994;18:859–65. 27 Grosdidier J, Boissel P, Bresler L et al. Retrospective study of a series of 62 cases of hydatid liver cysts [in French]. J Chir (Paris) 1985;122:163–9. 28 Sayek I, Yalin R, Sanac Y. Surgical treatment of hydatid disease of the liver. Arch Surg 1980;115:847–50. 29 Mentes A, Yuzer Y, Ozbal O et al. Omentoplasty versus introflection for hydatid liver cysts. J R Coll Surg Edin 1993;38:82–5. 30 Uravic M, Stimac D, Lenac T et al. Diagnosis and treatment of liver hydatid disease. Hepatogastroenterology. 1998;45:2265–9. 31 Kayaalp C. Evacuation of hydatid cysts using laparoscopic trocar. World J Surg 2002;26:1324–7. 32 Bensaadi H, Champault G. Laparoscopic hand-assisted surgery for hydatid cysts of the liver. Surg Laparosc Endosc Percutan Tech 2004;14:91–2. 33 Alfieri S, Doglietto GB, Pacelli F et al. Radical surgery for liver hydatid disease: a study of 89 patients. Hepatogastroenterology 1997;44:496–500. 34 Nardo B, Patrici A, Piazzese E et al. Radical surgical treatment of recurrent hepatic hydatidosis. Hepatogastroenterology 2003;50: 1478–81. 35 Belli L, Aseni P, Rondinara GF et al. Improved results with pericystectomy in normothermic ischemia for hepatic hydatidosis. Surg Gynecol Obstet 1986;163:127–32. 36 Sielaff TD, Taylor B, Langer B. Recurrence of hydatid disease. World J Surg 2001;25:83–6. 37 Dziri C. Hydatid disease: continuing serious public health problem. World J Surg 2001;25:1–3.
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12 Dziri C, Paquet JC, Hay JM et al. Omentoplasty in the prevention of deep abdominal complications after surgery for hydatid disease of the liver: a multicenter, prospective, randomized trial. J Am Coll Surg 1999;188:281–9.
