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anagement of Nonparasitic Hepatic Cystsscar M Mazza, MD, Diego L Fernandez, MD, Juan Pekolj, MD, PhD, FACS, Guillermo Pfaffen, MD,odrigo Sanchez Clariá, MD, Ernesto P Molmenti, MD, PhD, MBA, FACS,duardo de Santibañes, MD, PhD, FACS

BACKGROUND: The optimal management of nonparasitic hepatic cysts (NPHC) is a topic of debate. Thepurpose of this study was to evaluate our 17-year experience with NPHC.

STUDY DESIGN: From January 1990 to August 2007, 131 consecutive patients with NPHC were evaluated andtreated at our institution. Seventy-eight patients (60%) had simple hepatic cysts (SHC). Theremaining 53 (40%) had polycystic liver disease (PLD). Morbidity, mortality, and recurrencerates for each of the two groups were evaluated.

RESULTS: Thirty-seven patients underwent open deroofing (SHC, 24; PLD,13), 66 had laparoscopicderoofing (SHC, 46; PLD, 20), 19 had percutaneous drainage (SHC, 4; PLD, 15), 3 had majorhepatic resections (PLD, 3), 4 had cystojejunostomy (SHC, 4), and 2 had combined hepato-renal transplantation (PLD, 2). Corresponding morbidity, mortality, and recurrence rates were,respectively: conventional deroofing: SHC, 29%, 0%, 8%; PLD, 8%, 0%, 0%; laparoscopicderoofing: SHC, 2%, 0%, 2%; PLD, 25%, 0%, 5%; percutaneous drainage: SHC, 0%, 0%,75%; PLD, 0%, 0%, 20%; cystojejunostomy: SHC, 75%, 0%, 25%; major hepatic resections:PLD, 66%, 0%, 0%; and hepatorenal transplantation: PLD, 50%, 50%, 0%.

CONCLUSIONS: Laparoscopic deroofing provided complete relief of symptoms for both SHC and PLD. Percu-taneous drainage was our procedure of choice for infected liver cysts and potentially for patientswho cannot tolerate general anesthesia. Liver and liver-kidney transplantations were reserved forpatients with end-stage PLD alone and in association with end-stage renal disease, respectively.
( J Am Coll Surg 2009;209:733–739. © 2009 by the American College of Surgeons)
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onparasitic hepatic cysts (NPHC) present with aroad range of anatomic and clinical manifestations.lthough laparoscopic and percutaneous drainage con-

titute minimally invasive therapeutic options, resec-ion, fenestration, and liver transplantation allow for thereatment of massive liver enlargement. Differentiationetween simple hepatic cysts (SHC) and adult polycys-ic liver disease (PLD) is essential to determine the op-imal therapeutic approach.1 The purpose of thistudy was to evaluate our 17-year experience treatingPHCs.

isclosure Information: Nothing to disclose.

eceived June 29, 2009; Revised September 1, 2009; Accepted September 1,009.rom the Hepato-Pancreatico-Biliary and Liver Transplantation Units, De-artment of General Surgery, Hospital Italiano, Buenos Aires, ArgentinaMazza, Fernandez, Pekolj, Pfaffen, Sanchez Clariá, Santibañes) and theepartment of Surgery, North Shore University Hospital, Manhasset, NY

Molmenti).orrespondence address: Eduardo de Santibañes, MD, Hospital Italiano deuenos Aires, General Surgery and Liver Transplant Unit, Gascon 450, Bue-

pos Aires 1181, Argentina.

7332009 by the American College of Surgeons

ublished by Elsevier Inc.

ETHODSetween January 1990 and August 2007, 131 consecu-

ive patients with NPHCs were evaluated and treated atur institution. There were 41 men and 90 women, withmean age of 62.5 years (range 19 to 87 years). All

atients underwent an initial ultrasonography diagnos-ic screening. The surgical approach (deroofing or resec-ion) was based on the anatomic information providedy CT scans. Polycystic liver lesions complicated byleeding or infection were evaluated by additional ultra-onography or MRI examinations. Malignancy was sus-ected in cases of cystic wall thickening or papillaryrowth within the cyst. Contrast-enhanced CT scansombined with MRI and �-fetoprotein, CEA, and CA9-9 serum levels were used for preoperative diagnoses.ncologic resections were based on intraoperative find-

ngs. Malignant cystic tumors were not included in thistudy.

Patients were divided into two groups according to cystype: simple hepatic cysts and polycystic liver disease. Table

depicts their demographic information. Symptoms atresentation, therapeutic procedures, postoperative com-
lications according to Dindo’s classification,2 and short-
ISSN 1072-7515/09/$36.00doi:10.1016/j.jamcollsurg.2009.09.006

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734 Mazza et al Management of Nonparasitic Hepatic Cysts J Am Coll Surg

nd longterm results were considered when performing ournalysis of prospectively collected data.

imple hepatic cystsimple hepatic cysts, with a mean diameter of 9.2 cmrange 3 to 20 cm), were encountered in 78 patients. Forty-hree cases involved the right lobe and 29 the left lobe. Sixere bilateral. Presenting symptoms are summarized in Ta-le 1. In 20 instances (25.6%), initial findings were asso-iated with cyst complications: 11 bleeds, 8 infections, andspontaneous rupture. Therapeutic procedures are shown

n Table 2. Elective surgical deroofing, as described in anpen fashion by Lin and colleagues3 and laparoscopicallyy Vauthey and associates,4 was the procedure of choice.nly the cyst wall (rather than compressed liver paren-

hyma) was resected to avoid postoperative bile leaks. De-ermination of an open or laparoscopic approach was basedolely on the experience of the surgeon. In open cases, aight subcostal incision was made, intraoperative ultra-onography was not routinely performed, and resection ofhe cystic wall was carried out with electrocautery.

Intraoperative cholangiograms were done only when theystic contents suggested a biliary communication. Ab-ominal drains were always used after deroofing. Resectedystic wall specimens were routinely sent for pathologicvaluation to exclude malignancies. In laparoscopic cases, a0- to 12-mm port was inserted at the umbilicus, anddditional ports were positioned according to the locationf the cyst. In most instances, a second port was placed inhe epigastric area, and a third one in the right or left upperuadrants. During our early laparoscopic experience, theyst wall was resected with a monopolar hook and scissors.ost recently, we have opted for an ultrasonic scalpel. Four

atients who presented with infected cysts underwent per-utaneous drainage with multipurpose 8- to 12-F drains.oux-en-Y cystojejunostomies were performed in fourases of giant cysts located in the posterior aspect of theight lobe of the liver. Three of these four patients hadecurrent cysts after unsuccessful open deroofing attemptstwo of them at outside hospitals).

olycystic liver diseasehere were 53 patients with PLD who were divided into

hree groups according to Gigot’s classification.5 Group I

Abbreviations and Acronyms

NPHC � nonparasitic hepatic cystsPLD � polycystic liver diseaseSHC � simple hepatic cysts

n � 33) included patients with a few large cysts. Group II H

n � 14) was composed of patients with diffuse cysticnvolvement that retained areas of preserved hepatic paren-hyma. Group III (n � 6) encompassed patients with mas-ive cystic involvement and little uninvolved parenchyma.ysts had a mean diameter of 11.5 cm (range 3 to 25 cm)

nd were predominant in the right lobe in 9 patients, in theeft lobe in 3, and bilateral in 41. Presenting symptoms areetailed in Table 1. In nine patients (20.7%), initial find-

ngs were associated with cyst complications: four bleedsnd five infections. Treatment modalities (Table 2) wereased on evaluation of both clinical symptoms and Gigot’slassification. Open and laparoscopic surgical deroofingas performed according to the techniques described in thereceding text. Patients selected for this procedure wereostly in groups I and II. Only dominant and symptom-

tic lesions were deroofed. Percutaneous drainage was re-erved for infected cysts (as identified by ultrasonographyr MRI or both) and for patients who could not tolerateeneral anesthesia. Multipurpose drains were used in allases. Patients with massive hepatomegaly associated withespiratory and digestive impairment underwent liver re-ections through a bilateral subcostal incision with a mid-ine extension. Liver transection was with an ultrasonic

able 1. Demographic and Clinical Characteristics

linical features Simple cystsPolycystic

liver disease

78 53ge, y (range) 61.3 (29–87) 64 (22–85)ender, male:female 19:59 14:39ain, n (%) 45 (57.7) 32 (60.3)ausea/vomiting/earlysatiety, n (%) 12 (15.4) 3 (5.6)

atigue/dyspnea, n (%) 9 (11.5) 7 (13.2)bdominal mass, n (%) 13 (16.6) 6 (11.3)ever, n (%) 5 (6.4) 5 (7.54)iameter of dominant cyst,cm (range) 9.2 (3–20) 11.5 (3–25)

able 2. Therapeutic Procedures in Patients with Simpleepatic Cysts and with Polycystic Liver Disease

herapeutic procedures

Patients withsimple hepatic

cysts(n � 78)

Patients withpolystic liver

disease(n � 53)

n % n %

pen deroofing 24 30.76 13 24.5aparoscopic deroofing 46 58.9 20 37.7ercutaneous drainage 4 5.12 15 28.3ystojejunostomy 4 5.12epatectomy 3 5.7
epatorenal transplant 2 3.8

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735Vol. 209, No. 6, December 2009 Mazza et al Management of Nonparasitic Hepatic Cysts

issector. We performed a left hepatectomy, a right trisec-ionectomy, and one nonanatomic resection. Two patientsith type III disease who presented with massive hepa-

omegaly and end-stage renal failure underwent com-ined liver-kidney transplantation. One of them hadreviously undergone bilateral nephrectomies.

ESULTSimple hepatic cystsonventional deroofingean hospital stay was 7.4 days (range 2 to 19 days). Thereere seven short-term postoperative complications: one in-

raabdominal abscess successfully treated by percutaneousrainage, one bile leak that resolved spontaneously afterercutaneous drainage, four cases of small bowel obstruc-ion treated nonsurgically with nasogastric decompression,nd one pneumothorax (5 grade II according to Dindo’slassification, 2 grade IIIa). Cyst recurrences developed inwo patients (5.2%) after 21 and 35 months. A third pa-ient presented with an incisional hernia.

aparoscopic deroofingean hospital stay was 1.47 days (range 1 to 3 days). One

atient presented with a bile leak (grade II) that resolvedpontaneously after percutaneous drainage. Another4.3%) presented with a recurrent cyst 15 months afterhe initial operation and was treated with an openystojejunostomy.

ercutaneous drainagell four patients who underwent percutaneous drainageresented with sepsis originating from infected cysts, andequired antibiotics tailored to the organisms identified byulture (Escherichia coli in three patients and Klebsiella inhe remaining one). All patients responded favorably toreatment. The percutaneous drains were removed onceoth clinical and radiologic resolution was achieved. Re-urrent lesions developed in three patients (75%). Onesymptomatic patient required no treatment. Another un-erwent laparoscopic deroofing. The third was treated withcystojejunostomy. Except for the recurrences, there waso associated morbidity or mortality. Mean hospital stayas 7 days (range 2 to 15 days).

ystojejunostomyour patients with giant lesions located in the right poste-ior lobe of the liver underwent conventional cystojejunos-omies. Fevers developed in three suspected to originaterom bacterial contamination of the remaining cavity andere successfully treated with empiric oral antibiotics. Inne case, a persistent symptomatic lesion required addi-
ional percutaneous drainage and alcoholization. There T
ere no recurrences. Mean hospital stay was 9.2 daysrange 3 to 29 days).

Mean followup for the simple hepatic cysts group was 43onths (range 11 to 98 months). There was no mortality,

nd the incidence of morbidity was 8.9%. Mean hospitaltay for laparoscopic deroofing was significantly lowerhan that for conventional deroofing (p � 0.05). Thereere no differences in morbidity or mortality between

aparoscopic and open approaches.

olycystic liver diseasepen deroofingpen deroofing was performed in 13 patients with Gigot’s

ype I and II disease. A patient with type III PLD whonderwent open deroofing of multiple cysts during ourarly experience, experienced ascites and renal and respira-ory failure unresponsive to medical treatment. An emer-ency combined liver-kidney transplantation was under-aken, but the patient died postoperatively from a cardiacrrest. Mean hospital stay was 8.6 days (range 7 to 17 days).

aparoscopic deroofingaparoscopic deroofing was performed in 20 patients.nly large dominant and symptomatic cysts were treated,

s with the open approach. One patient (5%) with type IIigot’s disease experienced a recurrence and was treatedith a second laparoscopic fenestration. She eventually un-erwent bilateral nephrectomies and a combined liver-idney transplantation. Mean hospital stay was 3 daysrange 1 to 6 days). The incidence of morbidity for deroof-ng among PLD patients was 9.4%. There was 1 case eachn � 5) of postoperative bleed, small bowel obstruction,cute renal failure, post drainage bacteremia, and soft tissuenfection. All were grade II. The mean hospital stay foraparoscopic deroofing was significantly lower than thator conventional deroofing (p � 0.05). There were noifferences in morbidity between the open and laparo-copic approaches.

ercutaneous drainageifteen patients presented with infected cysts and systemicepsis. All underwent successful treatment with percutane-us drainage and received intravenous antibiotics. Cultureesults showed gram-negative bacilli (E coli, Klebsiella, En-erobacter, and Serratia) in all instances. Drains were re-

oved upon resolution of clinical symptoms and radio-ogic findings. There were three (20%) symptomaticecurrences. One was treated with laparoscopic deroofing.he remaining two were drained percutaneously for a sec-nd time, in one occasion with additional alcohol sclerosis.
here was no morbidity or mortality.

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iver resectioniver resection was the treatment of choice in three selectedatients with massive hepatomegaly and type III PLD whoresented with gross abdominal distension and weight loss.rocedures performed included a left hepatectomy, a rightrisectionectomy, and a nonanatomic resection. One of thehree is currently listed for a combined liver-kidney trans-lantation because of continued hepatic enlargement andeterioration of renal function. There were two complica-ions. One patient required reexploration for bleeding andemodialysis for acute renal failure (grade IVa). Persistentscites developed in another requiring prolonged hospital-zation (grade II). Mean hospital stay was 17.3 days (rangeto 28 days).

ombined liver-kidney transplantationombined liver-kidney transplantation was undertaken in

wo patients with PLD. One was transplanted emergentlyfter experiencing severe ascites after deroofing of a domi-ant cyst. He died posttransplantation of a cardiac arrest (grade V). The second

ad previously undergone two laparoscopic deroofings andilateral nephrectomies. She is currently 3 years posttrans-lantation, alive and well with normal liver and kidneyunction.

Mean followup time for the entire PLD group was 59.5onths (range 17 to 96 months).

ISCUSSIONhe differential diagnosis between parasitic cysts, pseudo-

ysts, true cysts, and malignant cysts may be difficult.1

chinococcal (hydatid) cysts represent the major pathologyo exclude in many regions of the world, such as in Argen-ina. Although epidemiologic data, serologic results, andmaging studies are frequently useful, when in doubt weerform (like many other authors) a laparoscopic explora-ion. If a diagnosis of hydatidosis is made,6 we convert to anpen approach for resection. Pseudocysts (intrahepaticilomas and abscesses) are infrequent lesions, devoid ofpithelial lining, that often develop as complications ofepatic surgery and abdominal trauma.7 Simple hepaticysts originate in embryologic blind ducts with no biliaryree communication. They have an epithelial cover witherous contents, and can be divided into two differentypes: simple cysts and PLD.4 Simple cysts can be single orultiple. PLD has a familial component, and may involve

ther organs in addition to the liver. Neither carries malig-ant potential. Symptoms develop as a result of mass effectr secondary to complications such as bleeding and infec-ions.8 Treatment is reserved for symptomatic or compli-ated cysts, or when malignancy cannot be excluded.9

Multiple reports confirm the effectiveness of surgical s

eroofing for hepatic cysts. This technique, originally de-cribed by Lin and coworkers3 in 1968, entails the resectionf the largest possible amount of emergent cystic wall.aparoscopic deroofing, with lower morbidity rates, simi-

ar recurrence rates, and shorter hospital stays than its openounterpart,10-12 has become the treatment of choice foroth symptomatic large simple and PLD lesions.13,14 Pa-ients treated with the conventional open technique in oureries, as in many other institutions, represent the laparo-copic learning curve. When deroofing, special care shoulde taken not to resect hepatic parenchyma, given thatransected bile ducts may lead to postoperative bile leaks (Fig.). In their impressive series, Gamblin and colleagues15 usedascular staplers or tissue sealing devices and considered aand-assisted technique for large lesions requiring exten-

igure 1. Laparoscopic views of a simple hepatic cyst as seen (A)n initial laparoscopic inspection and (B) after successful deroofing.

ive liver mobilization. In our work, we opted for monopo-

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ar scissors or ultrasonic scalpel to minimize our costs with-ut compromising patient safety. Some authors recom-end filling the cyst cavity with omentum16 to achieve a

etter drainage of the epithelial secretions into the perito-eal cavity. We do not routinely perform this maneuver.e believe that our series provides additional support to

he previous observations by Gamblin and coworkers15 thataparoscopic resections have become the standard of careor the treatment of benign symptomatic cysts.

Percutaneous drainage of symptomatic hepatic cysts haseen popularized recently. Because recurrence after simpleuncture and aspiration of the cyst is almost guaranteed,17

he use of sclerosing substances after evacuation of theyst has been advocated as a way to improve recurrenceates.18-20 Such substances, however, are formally contra-ndicated in cases of suspected or proven cyst-biliary treeommunications21 or in infected cysts. We use this ap-roach selectively, mostly for infected lesions,22 in patientsith systemic manifestations. In all cases, we obtainedrompt relief of septic conditions.Large cysts in the right posterior lobe have a high recur-

ence rate.23 In these lesions, the intimate contact betweeniver and diaphragm prevents an adequate continuousrainage of the deroofed cyst into the peritoneal cavity,

eading to reaccumulation of its contents. Three of the fourosterior right giant cysts that we treated with Roux-en-Y

imbs had had previous unsuccessful deroofing attempts.hree resolved promptly, as documented by postoperativeT scans. The fourth patient presented with a sympto-atic persistent leslon 1 month later and was successfully

reated with additional percutaneous drainage and alcohol-zation. Three of these patients presented fever during theostoperative period, and they were all successfully treatedith empiric oral antibiotics. We believe that this unusual

reatment may prove helpful to prevent recurrence of largeosterior cyst. Our experience also seems to suggest thatntibiotics should be routinely administered postopera-ively after this procedure.

PLD exhibits major differences. It is characterized byrequent renal involvement and an autosomal dominantamilial history with high genetic penetrance.24,25 PLDysts are multiple and tend to increase in number andize throughout life, as opposed to simple cysts. Becauseeterioration of hepatic function is very rare, prognosis

s usually determined by the concomitant renal dis-ase.4,26,27 Cyst infection, a life-threatening condition inatients with PLD, usually presents with severe systemiceterioration superimposed on chronic renal failure. Weuccessfully treated all these patients with prompt per-utaneous drainage of the infectious focus, minimizing
ny surgical aggression.22,25 Correct identification of the T
nfected cysts may be difficult. CT scanning usually pro-ides a view of the anatomy, but MRI or ultrasonogra-hy examination at the time of drainage may be neces-ary to identify their heterogeneous content (Fig. 2).

igure 2. Complex cyst in a patient with polycystic liver disease asiewed by (A) CT scan and (B) ultrasound imaging. Heterogeneousontents (arrows) in one of the cysts can be clearly identified byltrasound.

he advantage of MRI in these cases is based on the fact

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hat the high protein content of complicated cysts con-ers on them a characteristic hyperintensity in T1-eighted sequences.7

Symptoms in PLD type I are usually from the mass effectf a few well-defined lesions. Laparoscopic deroofing haseen effective in our experience and in that of other au-hors,5,14,25,28 and should be considered as the first choicepproach for this group of patients.29 Laparoscopy is asso-iated with fewer adhesions, facilitating reoperationshould more interventions be necessary.30

Finally, there is a third group of patients, Gigot’s PLDypes II and III, with symptomatic massive hepatomegaly.eroofing does not offer great relief to the respiratory fail-

re, digestive symptoms, and cachexia of this group.5,25

ur first patient experienced severe respiratory and renalnsufficiency soon after the procedure, prompting an emer-ency hepatorenal transplantation. Combined hepatic re-ection and cyst fenestration in the remnant liver seems toe a more appropriate option in these patients.27,31,32

ymptomatic relief with low morbidity was achieved inost cases.31,33,34 We undertook this approach in three pa-

ients, with good results.In patients with type III disease showing diffuse liver

nlargement and small cysts not suitable to deroofing oresection, direct evaluation for liver transplantation shoulde considered.26,35 Nevertheless, liver transplantation con-inues to have a limited role in the management of thisisease. In spite of many series reporting good results, PLDoes not usually evolve into end-stage liver disease, makinghis decision difficult. Transplantation must be balancedgainst the risks of operation, immunosuppression, quality ofife, and possible benefits of a combined renal implant.36-38 Weerformed two combined transplantations. One patientho received a transplant emergently died as a result ofcardiac arrest. The other remains alive and well with noomplications.

Various liver malignancies may present as cystic lesions.uring the time period of our study, we diagnosed seven

atients with cystic neoplasms. All of them underwent ma-or hepatic resections. Final pathologic diagnoses were: twoystoadenocarcinomas, two cystic sarcomas, one cysticepatocarcinoma, one hepatic adenoma with areas of se-ere dysplasia in the setting of Caroli’s disease, and oneystic adenoma. Tumor cavities generally developed as aesult of necrosis and subsequent liquefaction of the malig-ant mass, induced by rapid cellular proliferation or localnd systemic chemotherapy. The presence of thickened orrregular cyst walls should raise the suspicion of a neo-lasm. When in doubt, intraoperative ultrasonographynd frozen examination of surgical specimens is recom-
ended. If a malignant diagnosis is confirmed, com-
lete resection of the lesion with oncologic margins isequired whenever possible. Outcomes depend on theype of tumor present.

Laparoscopic deroofing provided complete relief ofymptoms for both SHC and PLD. Percutaneous drainageas our procedure of choice for infected liver cysts andotentially for patients who could not tolerate general an-sthesia. Liver and liver-kidney transplantation was re-erved for cases of end-stage PLD alone and in associationith end-stage renal disease, respectively.

uthor Contributions

tudy conception and design: Mazza, Fernandez, Pekolj, deSantibañes

cquisition of data: Fernandez, Pffafen, Sanchez Clariánalysis and interpretation of data: Mazza, Fernandez, Pekolj,

de Santibañesrafting of manuscript: Mazza, Fernandez, Molmentiritical revision: Molmenti, de Santibañes

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Management of Nonparasitic Hepatic Cysts

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