Mohammad Sultan Khuroo, MD, Echinococcu.s Management hepatic Echinococcus cysts (maximal diameter, ± 4.0) in 12 patients were aspi- 7.5 cm rated and (20%) irrigated saline with under the cysts, which hypertonic sonographic ance. All patients had symptoms of a hepatic by guid- signs mass had and caused a prominent of scoleces were observed Mean hospital stay was ± 3.4. Serial sonographic days nations revealed high-level the cyst cavity (heterogeneous pattern), uniformly which ultimately in all 4.0 exami- echoes in echo became echogenic (pseudotumor). After follow-up of 14.0 months ± 5.5, maximal cyst diameter decreased to 4.1 cm ± 3.1 (P < .001). One patient died of unrelated causes; the remaining 11 patients experienced relief of symptoms and a decrease in liver span. Index terms: Cyst, percutaneous drainage, 761.3121 #{149} Echinococcosis, 761.2083 #{149} Liver, cysts, 761.3121 #{149} Liver, echinococcosis, 761.2083 #{149} Liver, interventional procedure, 761.12986 #{149} Liver,USstudies,761.12986 #{149} Parasites, 76.2083 Radiology 1991; Ali Zargar, H YDATID man and larval 180:141-145 (2). to the of the cases 0 RSNA, 1991 Received in primary cysts, of disseminated or disease is a much (3). Medical therapy has especially advantages, October 15, 1990; re11. in patients with recurrent disease or in patients for whom surgery is otherwise madvisable (1,3-5). There are some questions about its efficacy and safety (57). Percutaneous puncture or aspiration of hydatid cysts has been contraindicated (8). However, there are reports of hydatid cysts being punc- tured and neither anaphylaxis nor peritoneal soilage resulting (2,9,10). In this study, we report the results of ultrasound (US)-guided percutaneous transhepatic patic hydatid drainage cysts MATERIALS of 21 he- in 12 patients. AND METHODS From June 1988 onward, all patients with hydatid disease of the liver being treated at our institution were considered for treatment with percutaneous drainage (a) November 15; revision 26, 1991; accepted March requests to M.S.K. re- successful but management recurrent hydatid greater problem symptoms caused by cyst with that appeared wall in Table fluid anechoic a or hypoechoic, primary echoes, form excluded solid of treat- if the pattern (b) was cyst with- infected, The cysts, clinical formed parameters the study of the intradermal 1gM antibodies (measured test of immunogbobulin to G Echinococcus by means of the en- zyme-linked immunosorbent assay [ELISA]) were performed and the results evaluated, as reported earlier (12). The patient fasted overnight. The procedure group. group 2. The Casoni measurement was performed the next morning, or had ruptured into the biliary tree or pleural or peritoneal cavity. Twelve patients, harboring 21 hepatic hydatid and (IgG) and granulosus component enhancement of back wall (c) gave informed consent to Patients were had a hyperechoic out back shown in Table 1. The types and descriptions of the hydatid cysts (11) are shown if the patient and signs of a hepatic a hydatid cyst; (b) had a prominent (a) (c) ment. India. MD the surgical cysts-is echoes; and use of PD as the requested February reprint Mahajan, In the Liver: Drainage’ bordering of uncomplicated with marked (Kashmir), areas Treatment-usually moval had revision ceived Address #{149} Rakesh Cysts (PD). PD was performed the Department of Gastroenterology, of Medical Sciences, Srinagar 190 011 DM Mediterranean and Baltic seas, South America, Australia, the Middle East, and New Zealand. Immigration has bed to an increased prevalence of the disease in Europe and North America mass From Institute MD, disease is the commonest one of the most severe hucestodiases (1). The disease is endemic fluid component that appeared anechoic or hypoechoic, with marked enhancement of back wall echoes. The amounts of cyst fluid aspirated and of hypertonic saline used were 190 mL ± 240 and 120 mL ± 90, respectively. Separation of the endocyst from the pericyst and nonviability cysts. #{149} Showkat granulosus with Percutaneous Twenty-one granulosus DM are Abbreviations: ELISA = enzyme-linked munosorbent assay, Ig = immunoglobulin, PD = percutaneous drainage. im- 141 Table 2 Table Distribution of 21 Cysts into Five According et al (11) Types Gharbi 3 of Cyst Fluid Aspirated Giaracterisfics to Classification of before and after Infusion of Saline into Cyst Cavity No. of Cysts with Characteristic No. of Description Type Before Cysts of Characteristic I Pure fluid collection; ifi Iv V After Infusion of Saline 14 Appearance rounded with welldefined borders Fluid collection with split wall (localized) Fluid collection with multiple septa; honeycomb appearance Hypoechoic with high internal echoes Cyst with reflecting H Infusion Saline and watery Clear 0 20 Opalescent 1 0 1 Turbid 21 0 Culture 3 Negative 19 Positive Cytologic 2 thick walls * t Escherichia 13 (12) 4 21 (1)* 7 3 1 coil (one patient) Staphylococcusepidermidis S Numbers 1 findings Scoleces Hookiets Membrane 1 20 2* and (skin in parentheses Salmonella typhimurium (one patient). contaminant). are number of cysts with scoleces. viable with close monitoring. Facilities were available to treat any potential complication. The location of the cyst was defined in three planes with sonography by using a real-time linear scanner (SSD 256; Aloka, Tokyo) with a 3.5-MHz probe. The relation of the cyst to the normal liver was delineated, and a site for puncture was marked such that the cyst could be approached through thick normal liver tissue. If possi- visit, patients underwent clinical examina- patients masses, tion and had a blood sample drawn for serum chemistry and serologic testing. Sonography was performed to PD, were examination. to evaluate the diameter and appearance of the cyst. All values were expressed as means ± one ble, the right intercostal route was preferred to minimize the chances of peritoneal soilage. Under aseptic conditions, USguided cyst puncture was performed through the biopsy port of the puncture probe (3.5 MHz). A transhepatic catheter needle (5-F, 40-cm-long radiopaque polyethylene catheter; Cook Europe, Bjaerver- standard skov, All of the 21 cysts in the 12 patients were successfully treated with PD. The transhepatic catheter needle was used to drain 13 cysts, while the cholangiographic needle was used to drain eight cysts. The amounts of cyst fluid aspirated and of saline infused into the cyst cavity were 190 mL ± 240 (range, 2.5-600 mL) and 120 mL ± 90 (range, 2-450 mL), respectively. The characteristics of the cyst fluid obtained before and after infusion of saline into the cyst cavity are shown in Table 3. In one patient, scoleces were viable in the cyst fluid aspirated after infusion of saline. PD was repeated, and the scoleces became nonviable. The mean hospital stay was 4.0 Denmark) was tion of unilocular cysts containing giography employed for aspira- large-volume daughter needle (22 cysts. cysts, gauge, In a cholan- 20 cm bong Cook Europe) was used to puncture each daughter cyst; individual daughter cysts were then aspirated and irrigated with hypertonic saline. In small-volume cysts, the cholangiography needle was the preferred choice. puncture, Immediately after cyst cyst fluid was aspirated. was sterile ration hypertonic (20%) saline. Cyst aspiand reffiling was monitored contin- uously ifiled by means with an equal The cavity volume of sonography. of The hy- pertonic saline was left in the cyst for 20 minutes, after which the fluid was aspirated. At this time, separation of the endocyst from reduce exudation cavity the pericyst was Cyst with filled with fluid observed. To from the cyst wall, the washed left partly was was normal saline and to cytologic and microbiologic examination. For cytologic examination, the fluid was centrifuged and the sediment examined for fragments hooklets, of the laminated and scoleces. The scoleces was motility at immediate microscopy staining with eosin tient assessed membrane, viability neutral was observed charged from by observing for 48 hours the hospital #{149} Radiology their and (13). The pa- and dis- if the procedure had been uneventful. Thereafter, were followed up every month. 142 of patients At each The in statistical paired not palpable Cystic prior at follow-up Student analysis of the Cyst Size and Appearance results. Repeated tions RESULTS PD days Outcome ± 3.4 (range, patients left the hours after PD. solution. subjected deviation. test was used with hepatomegaly. palpable in six patients 4-12 hospital days). within Nine 5.5 (range, and pearance diameter 4.1 cm size patients cysts) were (P < After from ± 3.2 .001). PD, the 3-18 months). One patient died of unrelated causes. Eleven patients remained alive. All were relieved of clinical symptoms. They noticed relief of pain in the abdomen, and the liver span decreased in all 10 decreased ± 3.1 (P ruptured. of three from for diam- ± 4.0 to echo cysts revealed floating high-level within the the hydatid During 7.2 .001). (with a to- separated Sonography cavity. 3 in < in all 21 cysts; echogenic cyst than up patients endocyst less cyst 7.9 cm the PD. diameter from cysts erogeneous ± for cyst in these pericyst daughter aminations, appeared followed up every of 14.0 months The 10 patients decreased 3.4 cm ap- of seven up PD. remaining the were a period followed after after a total tab of 14 cysts) were followed 9-18 months after PD, and eter in in cyst soon (with these two patients cm ± 4.0 to 4.8 cm The decrease alteration occurred Two months an follow-up (Figs 1-3). The initial cyst of 7.5 cm ± 4.0 decreased to 3.1 (P < .001). The decrease ± in cyst linear Follow-up All patients month over a progressive cyst size examina- during revealed also 48 sonographic performed structures in follow-up ex- internal cyst cavity pattern). echoes (het- These be- came more abundant and denser until the cyst cavity was uniformly echogenic (representing a pseudotu- mor). On cysts lost their further and were sonography. not examination, rounded visible five appearance at repeated July 1991 b. Figure 1. dimensions separated C. Serial US scans of a patient with type 1 hydatid cyst. of cyst (C) are 6.5 x 5.7 cm. Note separated endocyst. endocyst and high-level internal echoes. a. (a) Before (c) Five PD, dimensions months after of cyst are PD, dimensions 14.5 x 11.2 cm. (b) One month of cyst are 4.5 x 3.5 cm. Cyst after PD, is filled by b. Figure daughter echoes. 2. Serial US scans of a patient with cysts. (b) Three months after PD, (c) Six months after PD, dimensions Serologic type III hydatid cyst. (a) Before PD, dimensions of cyst are 8.2 x 6.2 cm. dimensions of cyst (C) are 5.5 x 5.0 cm. Note linear echogenic structures of cyst are 3.5 x 3.5 cm. Cyst is replaced by echogenic mass (pseudotumor). Testing Of the eight patients with a positive ELISA for IgG antibodies (titer> 1:160), seven had a fourfold drop in absorbance and a negative serologic titer at follow-up. One patient with persistently positive titer was fob- ter PD. To evaluate this cyst contents, US-guided aspect of the aspiration performed six cysts were cysts. Another at autopsy. was The a lowed up for 3 months after PD. All three patients with a positive ELISA for 1gM antibodies (titer > 1:160) had a fourfold drop in absorbance and a negative serologic titer. None of the patients had a rise in the hydatid antibody titer, either of the IgG or the 1gM type. cyst in eight examined contents were studied ± 1.5 (range, 1-12 weeks) 10.3 after weeks PD. The fluid was turbid in all 14 cysts, and bile stained in three cysts. Cultures for both aerobic and anaerobic microorganisms and microscopy were bris, and ples. hydatid membranes, hooklets, dead scobeces in all the fluid revealed negative, cellular desam- Complications Cyst Reaspiration We were term Volume viability 180 During concerned about of the cyst Number 1 #{149} long- contents af- PD and the low-up period, none developed anaphylaxis, immediate fol- of the patients asthma, or Note and laryngeal edema. type I) developed after the procedure. lasted for 48 hours, multiple high-level septa and internal One patient (cyst urticaria 6 hours The urticaria and the patient responded to antihistaminic therapy. Two patients (cyst types I and V) developed fever within 48 hours after PD. Cyst fluid from both patients showed growth of microorganisms (E coli in one and S typhimurium in the other). Specific antibiotic therapy was instituted, resulting in clinical recovery. One patient (cyst type II) developed biliary rupture of a hydatid cyst 4 weeks after PD. At endoscopic retrograde cholangiopancreatography, a wide endoscopic sphincterotomy was performed and laminated membranes were basketed out of the bile duct. At Radiology 143 #{149} serial sonographic cyst cavity examinations, was replaced by the an echogenic mass that eventually lost its rounded shape and was not visible at repeated sonography. One patient with six liver cysts died of massive hemoptysis 3 months after PD. Autopsy revealed a mediastinal tubercular gland eroding into the pubmonary vein and bronchial tree. and cyst bly risks puncture has been due to the potential of anaphylactic shock, peritoneal seeding and dissemination, and growth of secondary peritoneab cysts. Although anaphylactic shock from rupture of an echinococcal cyst has been documented, its exact frequency and mechanism have not been well studied (9). Lewall and McCorkell studied 20 patients with rupture of echinococcal cysts in the liver; none developed anaphylaxis (14). Schiller studied complications of echinococcal cyst rupture in 30 patients. tients had tachypnea, and hypotension and hours of cyst rupture; patients had generalized autopsy (15). These Two pa- tachycardia, died within 48 both of these peritonitis at authors con- cluded that an anaphylactic reaction occurs infrequently following cyst rupture and spillage. Percutaneous catheter drainage is the treatment of choice for the majority of intraabdominal regardless no evidence ity contents ous occurs aspiration abscesses fluid of etiology that any not during of fluid (16,18). The There is of cay- percutane- collections use of fine and nee- dles and catheters; advances in radiographic, sonographic, and CT techniques; an approach through thick liver tissue; a preference for the right intercostal approach; and sudden complete decompression immediately after puncture make the chance of spillage extremely low and perhaps less than what might occur with sur- gical manipulation. PD was performed with close monitoring and with facilities available for treating any potential complication, especially anaphylaxis. Hydatid cyst aspiration and/or drainage has been performed and reported in the literature (2,9,10,19). Of the 15 patients whom we are aware of in whom hydatid cysts were aspirated, none deveboped anaphylaxis. In the present study, 21 cysts were aspirated in 12 patients, none of whom developed asthma, laxis. 144 laryngeal Two patients #{149} Radiology related edema, developed or anaphy- fever, to PD. This findings contained infu- patient had eventually are caused by rupture (14). can lead rupture due to high sure. Two earlier reports therapeutic drainage cysts in the reported pigtail cavity of a patient with six hepatic hydatid cysts. Scan was obtained 15 days after four cysts were drained in a single session. Note separated endocyst in four cysts and intact endocyst in two cysts. Arrows indicate sites of cysts. to biliary intracystic pres- have described of echinococcal (2,9). Mueller patient who et ab (9) underwent PD of a recurrent hydatid liver. In their case, a 8.3-F catheter was for 3 months. tinuous ity liver one successful cyst of the catheter left in place This allowed drainage for a prolonged (2) treated three cysts of the liver in the con- of the cay- Bret et al period. stage. We datid cysts tinuous endocyst all of the in a single With catheter drainage, membrane would quently blocked the and helped introduce residual cavity. catheter lumen infection into Also, have been impractical ters in small cyst cavities. However, the cyst ducts and/or endoscopic cyst from contents the common common bile duct may function: agent it and the endocyst also from pattern; months ± 5.5 the follow-up (range, 3-18 of 14.0 months) in insufficient to clinical out- To conclusively we are follow-up answer this performing of the long- patients in this study. However, the earliest response to cyst regrowth or dissemination is tive in all three patients at the last follow-up. None of the 12 patients had a rise in 1gM antibody titer at serial follow-up. We believe that the period of follow-up was sufficient for 1gM antibody levels to rise if peritoneal dissemination and cyst growth had occurred. Serial sonographic examination of of bile at endoscopic retrograde cholangiopancreatography in one patient. Such patients to the appearance of an 1gM-type antibody response to E granulosus (12). All three of our patients with a positive 1gM ELISA titer showed a fall in the antibody titers, and the test was nega- duct has been performed (20). In the present study, we were successful in removing laminated membrane from the growth term contents gablbladremoval separate question, meticulous evacuation of cyst contents and surgery. Cysts that communicate with the biliary tree require to evacuate (20%) the pericyst. This agent is effective and safe, has no systemic toxic effects, and does not cause secondary sclerosing chobangitis when bile duct communication is present (21). E granulosus cysts have a slow tion? successful in cysts with a hyperechoic solid pattern without back wall shadows. Infected hydatid cysts require from the bile der. However, and come of PD. Can cyst growth occur locally or in the peritoneal cavity due to cyst fluid soilage and dissemina- cathe- patients with suspected bibiary communication might benefit from prolonged cyst drainage, as did the patient reported by Mueller et al. Such cavities continue to drain for a long time; in many there is frank biliary rupture of cyst material, as occurred in one of our patients. We performed PD in hydatid cysts with a prominent fluid component. The procedure would not have been surgery a dual the present study was evaluate the long-term it would to place saline as a scolexcidal helped con- hypertonic performed acted the thick have fre- of PD the cysts. The saline, which left in the cyst cavity for 20 mm- utes, 21 hy- stage. We used was patients with hydatid with PD. After aspi- drained benefit from a combination endoscopic basketing. irrigate ration, scolexcidab irrigation, and reaspiration of the cyst cavity, the catheter was immediately removed and the cyst was thus treated in a single the collections, (16,17). spillage before a cyst with a split wall at US before the procedure was performed. Such This cyst aspirated showed growth of miThis led us to believe that the cysts were already harboring bacteria, which were reactivated by aspiration. Similarly, biliary rupture of a type II cyst in one patient was possi- sonographic cysts with DISCUSSION Hydatid contraindicated fluid sion of saline croorganisms. all of the treated changes. high-level The cavities were echoes consisting bar debris, dead and membranes. slowly solidified further fluid docyst, and cysts revealed scoleces, These hooklets, contents owing secretion the cyst similar filled with of cellu- to the by the gave the lack of enappearJuly 1991 ance of a pseudotumor. Similar sonographic appearances have been reported by other researchers after PD (2,8) long-term Until treatment cysts and in patients receiving albendazole therapy (4). now, surgery has been the of choice for E granulosus (21). Surgery of uncomplicated sterile, unilocular cysts is effective, with a morbidity of approximately 8% and a mean hospital stay of 11.8 days. The surgical morbidity increases-as does the hospital stay-in cases in which cyst drainage is performed; in patients with diseases; cardiac and cessible and in cases sites, multiple pulmonary of cysts may and have fatal at mac- disseminated those recurring after surgical procedures. Peritoneal age is known to occur at surgery cysts, consequences; prior spilland 50% of surviving patients continue to harbor the disease (21). PD-as assessed in 12 patients in the present study-was effective and safe and resulted in a much shorter hospital stay (4.0 days ± 3.4). PD has a particular advantage over surgery, as it can be performed in patients whose cysts are inaccessible to surgery or who are high surgical risks due to systemic illnesses. PD is particularly safe in patients with recurrence of cysts after surgery, as the aspirating needle can be directed through the scar tissue to reduce the chances of cyst leakage and peritoneal soilage. Albendazole, a benzimidazole, has been shown to be effective in human hydatid disease (1,3-5). The drug produces high concentrations of albendazobe sulfoxide in the blood, cyst fluid, and cyst wall (1). It causes death of scoleces, and cysts show objective evidence of reduction in size and may even disappear (5). However, pro- Volume 180 #{149} Number 1 longed therapy is needed, the drug is teratogenic and hepatotoxic, and some patients fail to respond (4-7). PD can be supplemented with albendazole therapy in a number of ways to achieve better results: abbendazole can be used before PD to make cysts albendazobe sulfoxide can ud-Din, The PA, for secretarial authors thank 9. 10. be introduced into the cyst cavity as a scobexcidab agent, and PD can be foblowed by albendazole therapy to kill any live scoleces in the cyst or in the peritoneum. We believe that, in the future, management of most hydatid cysts of the liver will be possible with a combination of drug therapy and PD. Surgery will be needed for those cysts that defy this combination treatment. U Acknowledgment 8. 11. 12. 13. 14. Mehraj- assistance. 15. References 1. 2. 3. 4. 5. 6. Saimot AG, Meulemans A, Cremieux AC, et al. Albendazole as a potential treatment for human hydatidosis. Lancet 1983; 2:652656. Bret PM, Fond A, Bretagnolle M, et al. Percutaneous aspiration and drainage of hydatid cysts in the liver. Radiology 1988; 168:617-620. Bezzi M, Teggi A, Rosa FD, et al. Abdominal hydatid disease: US findings during medical treatment. Radiology 1987; 162:9195. Morris DL, Dykes PW, Dickson B, Marriner SE, BoganjA, Burrows FGO. Albendazole in hydatid disease. Br Med J 1983; 286:103104. Morris DL, Dykes PW, Marriner 5, et al. Albendazole: objective evidence of response in human hydatid disease. JAMA 1985; 253:2053-2057. Gil-Grande LA, Boixeda D, Garcia-Hoz F, et al. 7. Treatment of liver hydatid disease with mebendazole: a prospective study of 13 cases. Am J Gastroenterol 1983; 78:584588. Schantz PM, Van den Bossche H, EckertJ. Chemotherapy for larval echinococcosis in animals and humans: report of a workshop. Z Parasitenkd 1982; 67:5-26. 16. 17. 18. 19. 20. Lewis JW, Koss N, Kerstein MD. A review of echinococcal disease. Ann Surg 1975; 181:390-396. Mueller PR, Dawson SL, Ferrucci JTJr, Nardi GL. Hepatic echinococcal cyst: successful percutaneous drainage. Radiology 1985; 155:627-628. McCorkell SJ. Unintended percutaneous aspiration of pulmonary echinococcal cyst. AJR 1984; 143:123-126. Gharbi HA, Hassine W, Brauner MW, Dupuch K. Ultrasound examination of the hydatid liver. Radiology 1981; 139:459-463. Wattal C, Malla N, Khan 5, Agarwal C. Comparative evaluation of enzyme-linked immunosorbent assay for the diagnosis of pulmonary echinococcosis. J Clin Microbiol 1986; 24:41-46. Smyth JD, Barrett NS. Procedures for testing the viability of human hydatid cysts following surgical removal, especially after chemotherapy. Trans R Soc Trop Med Hyg 1980; 74:649-652. Lewall DB, McCorkell SJ. Rupture of echinococcal cysts: diagnosis, classification, and clinical implications. AJR 1986; 146: 391-394. Schiller CF. Complications of Echinococcus cyst rupture: a study of 30 cases. JAMA 1966; 195:220-222. vanSonnenberg E, Mueller PR, FerrucciJT Jr. Percutaneous drainage of 250 abdominal abscesses and fluid collections. I. Results, features, and complications. Radiology 1984; 151:337-341. Mueller PR, vanSonnenberg E, Ferrucci JT Jr. Percutaneous drainage of 250 abdominal abscesses in fluid collections. II. Current procedural concepts. Radiology 1984; 151: 343-347. Welch CE, Malt RA. Abdominal surgery. N EngI J Med 1983; 308:753-760. Livraghi T, Bosoni A, Giordano F, Lai N, Vettori C. Diagnosis of hydatid cyst by percutaneous aspiration: value of electrolyte determinations. JCU 1985; 13:333-337. Karawi MA, Hanid MA. Endoscopic moval of daughter Echinococcus cysts the common bile duct. Hepatogastroenterology 1985; 32:296-298. 21. LangerJC, Rose B, Keystone JS, Taylor Langer B. Diagnosis and management hydatid disease of the liver. Ann Surg 199:412-417. Radiology refrom BR, of 1984; #{149} 145
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