Open Access

Rerupture of nonparasitic liver cyst treated with cyst fenestration: a case report

  • Kentaro Inoue1, 2Email author,
  • Tomohiro Iguchi2,
  • Shuhei Ito2,
  • Takefumi Ohga2,
  • Tadahiro Nozoe2,
  • Ken Shirabe1,
  • Takahiro Ezaki2 and
  • Yoshihiko Maehara1
Surgical Case Reports20151:71

DOI: 10.1186/s40792-015-0075-8

Received: 20 April 2015

Accepted: 21 August 2015

Published: 2 September 2015

Abstract

We herein describe a case involving spontaneous rerupture of a nonparasitic liver cyst successfully treated with cyst fenestration and an omental flap. A 59-year-old Japanese woman was transferred to our hospital for evaluation of acute abdominal pain. She had a history of conservative treatment with antibiotics for spontaneous rupture of a liver cyst 1 month previously. On arrival, she exhibited abdominal tenderness and muscular defense. Enhanced computed tomography showed ascites and a large ruptured hepatic cyst (diameter of 10 cm). We diagnosed rerupture of a liver cyst and performed laparotomy for cyst fenestration and intraperitoneal drainage. During the operation, we found the perforation site on the ventral side of the cyst and brown, muddled ascitic fluid. Cholangiography showed no bile leakage on the inner wall. Pathological investigation revealed no evidence of malignancy. The patient recovered without any adverse events and was discharged on postoperative day 8. No recurrences or complications occurred for 2 years.

Keywords

Nonparasitic liver cyst rupture Cyst fenestration Acute abdomen

Background

A nonparasitic liver cyst (NLC) is a common benign liver disease. It is potentially asymptomatic and is often incidentally diagnosed with abdominal imaging such as ultrasonography or computed tomography (CT). With the advancements and spread of these abdominal imaging techniques, NLCs are becoming more frequently detected and have been found in approximately 5 % of the population [1]. In many cases, an NLC is asymptomatic and is conservatively followed up without treatment. However, NLCs are sometimes associated with various complications such as rupture, infection, hemorrhage, obstructive jaundice, portal hypertension, and pulmonary embolism. These complications occur in less than 5 % of all patients with NLC [2].

We herein describe a rare case of spontaneous rerupture of an NLC that had become exacerbated after conservative treatment and was successfully treated with surgical fenestration.

Case presentation

A 59-year-old Japanese woman was transferred to the emergency unit of our hospital for evaluation of acute abdominal pain. She had a history of conservative treatment for a spontaneous NLC rupture 1 month previously in another hospital (Fig. 1a).
Fig. 1

CT images of progression of hepatic cyst rupture. a CT image 1 month before presentation to our hospital. The largest cyst showed an irregularly shaped wall on the ventral side (above, yellow arrows). At that time, the caudal part of the cyst kept circular (below). Some ascitic fluid was found around the spleen. b CT image on arrival to our hospital. Volume of the irregularly shaped cyst had obviously decreased (red arrows) and was present within a relatively high dense lesion (red circle)

On examination, she had a pulse rate of 115 beats/min, blood pressure of 112/68 mmHg, and no fever. Her abdomen was flat but hard and painful. She also exhibited obvious tenderness and muscular defense upon arrival. Blood tests revealed acute inflammation and anemia (Table 1). The levels of the tumor markers carcinoembryonic antigen and carbohydrate antigen 19-9 were within normal limits. Enhanced CT showed hepatic cysts and ascites. The largest cyst was found on the lateral segment; it exhibited an irregularly shaped surface and was present within a partially high dense lesion (Fig. 1b). The cyst volume had obviously decreased during the 1-month period before presentation to our hospital (Fig. 1, below). However, no neoplastic features such as thickened walls, papillary projections, or calcifications were found. The ascitic fluid collected by abdominal puncture was brown and muddled. The bilirubin level of the ascitic fluid was normal; however, the neutrophil and hemoglobin levels were high. Bacterial culture of ascetic fluid was negative (Table 2).
Table 1

Blood examination on arrival

White blood cells

17400

/μl

Neutrophil

89.8

%

Hemoglobin

10.7

g/dl

Platelets

247,000

/μl

Albumin

4.0

g/dl

Total bilirubin

0.53

mg/dl

Lactate dehydrogenase

255

IU/l

Aspartate aminotransferase

26

IU/l

Alanine transaminase

24

IU/l

Alkaline phosphatase

298

IU/l

Creatinine

0.5

mg/dl

C-reactive protein

0.26

mg/dl

Carcinoembryonic antigen

2.5

ng/ml

Carbohydrate antigen 19-9

<2.0

U/ml

α-fetoprotein

4.9

ng/ml

Table 2

Examination of ascitic fluid on arrival

Property

Brown and slightly muddled

Cell counts

43980

/μl

Neutrophils

88

%

Total bilirubin

<0.01

mg/dl

Hemoglobin

1.0

g/dl

Bacterial culture

Negative

Based on the patient’s clinical course and investigation findings, we diagnosed panperitonitis associated with rerupture of the liver cyst and accompanied by hemorrhage. Laparotomy was performed for cyst fenestration and intraperitoneal drainage.

During the operation, we found the perforation site on the ventral side of the cyst (Fig. 2). The perforation was approximately 3 cm, and the cyst wall was fibrous. Although no obvious hematoma was detected, approximately 600 ml of ascitic fluid was found. The ascitic fluid was brown and slightly muddled. No nodules or other specific findings, indicating signs of malignancy, were found. We resected the ventral wall of the cyst followed by cholecystectomy and cholangiography. Cholangiography showed no bile leakage on the inner wall. We performed cyst argon beam coagulator ablation of the inner wall and covered the site with an omental transposition flap. The patient tolerated these procedures well and was transferred to the intensive care unit in a hemodynamically stable condition. Pathological examination showed only fibrous connective tissue covered with simple cuboidal epithelium; there was no evidence of malignancy (Fig. 3). The patient received antibiotics (PIPC/TAZ) until postoperative day 5. She recovered without any adverse events and was discharged on postoperative day 8. She was in good condition without recurrent symptoms 2 years postoperatively.
Fig. 2

Perforation lesion of hepatic cyst. The perforation lesion was on the ventral side of the cyst. The lesion was approximately 3 cm, and the cyst wall was fibrous

Fig. 3

Pathological examination of cyst wall. Only fibrous connective tissue covered with simple cuboidal epithelium was observed; no evidence of malignancy was present

Conclusions

Rupture of parasitic liver cysts, which are mainly caused by the Echinococcus species, is a well-known complication of such cysts and is often reported as hydatid cyst rupture [3, 4]. In contrast, rupture of NLCs is highly rare. The frequency is unknown, but Morgenstern [5] stated that only four cases of rupture are present among approximately 250 reports of solitary NLC published before 1958. In our computerized search of English-language reports of NLC rupture published from 1959 to 2013, we identified only 17 publications describing NLC rupture (Table 3) [319]. The causes of NLC rupture are variable and include infection, trauma, iatrogenic injury, and spontaneity [11, 16, 20]. In the current report, we presented a case of the second rupture without a specific cause such as infection or trauma after previous conservative treatment. The patient had acute abdomen and signs of preshock on arrival; clinical investigations showed mild anemia, acute systemic inflammation, and muddy ascitic fluid. The preoperative CT showed an irregularly shaped NLC with a high dense lesion. Therefore, we diagnosed the spontaneous rerupture of the NLC with hemorrhage and performed acute surgery. As intraoperative findings, no obvious hematoma was detected. However, comparing with the previous reports in Table 3, brown muddled ascites indicated the presence of hemorrhage. Therefore, in our case, the slight bleeding in the ruptured NLC could exist, and it might be the reason why the patient exhibited the acute abdomen.
Table 3

Review of nonparasitic liver cyst rupture

Year

Reference

Age

Sex

Symptoms

Peritoneal irritation

Cyst (cm)

Location (segments)

Ascites

property of ascites

Hemorrhage

Emergency procedures

Treatment

Outcome

2014

Our case

59

F

Acute abdominal pain

Yes

10

Left

Yes

Brown and slightly muddled

No active bleeding

Yes

Laparotomy and cyst fenestration

Uneventful

Tenderness and muscular defense

Placing omentum over the ruptured cyst

2013

Marion

37

F

Pain in the right hypochondrium

No

18

Right lobe S4

Yes

Hemoperitoneum blood clots

Yes

Yes

Cystectomy

Uneventful

Tenderness in the right subcostal region

Pallor

Dyspnea

2010

Ueda

64

F

Right upper quadrant pain

No

10

Right lobe

Yes

Serous brown

No

No

Percutaneous aspiration

Uneventful

Injection of minocycline hydrochloride

2010

Miliadis

70

M

Sudden diffuse abdominal pain

Yes

13

Right lobe

Yes

Opaque-yellowish peritoneal fluid

Unknown

Yes

Deroofing of the cyst

Uneventful

Diffuse guarding

Omentoplasty

Rebound tenderness

Cholecystectomy

2007

Salemis

50

M

Sudden severe abdominal pain

Yes

17

Left lobe

Yes

Unknown

Unknown

Yes

Wide excision of the cyst

Uneventful

Nausea

Running locking suture along the edge of the resected cyst wall

Vomiting

Diffuse tenderness

Rebound tenderness

2005

Cheung

73

F

Sever abdominal pain

Yes

17

Right lobe

Yes

Blood stained

Yes

Yes

Laparoscopic deroofing of ruptured cyst

Good condition

2003

Shutsha

67

F

Sudden sharp abdominal pain in the right upper abdomen after coughing fit

No

Unknown

Multiple

Yes

Unknown

No

-

None because abdominal pain spontaneously disappeared within 2 days

Good condition

2003

Kanazawa

78

M

Sudden onset of sever right hypochondralgia

No

Unknown

Right lobe

Yes

Dark, bloody-colored pus

Yes intracystic

No

Antibiotics

Good condition

Drainage and alcohol injection

Tenderness in the right hypochondral region without muscle defense

2002

Ishikawa

42

F

Discomfort in upper abdomen

No

10

S4 and S5

Yes

Muddy, dark brown

Yes

No

Transcatheter arterial embolization (TAE)

Uneventful

13 after TAE

Drainage

Cystectomy

2002

Carel

76

M

Progressive abdominal pain

Yes

9

Right lobe

Yes

hemoperitoneum

Yes

Yes

Laparotomy

Death 4 weeks after admission due to complications (hemodynamic instability, arrhythmias, bacterial pneumonia)

Severe tenderness

Placing omentum over the ruptured cyst

Diffuse rebound pain

1999

Yamaguchi

61

M

Spontaneous pain in the right upper quadrant of the abdomen

Yes

13

Left and S5

Yes

With blood clot

Yes

no

Hepatectomy due to involving anterior branch of right portal vein

Uneventful

No preoperative investigation

Tenderness

Muscular defense

1999

Payatakes

62

unknown

Acute right upper quadrant abdominal pain

-

9.5

Right

-

-

-

-

Partial excision

Symptom free

External drainage

1989

Akriviadis

48

F

Sever epigastric pain

-

Unknown

Left

-

-

-

No

Conservatively

Uneventful

1988

Ayyash

36

M

Sudden epigastric pain

-

4

Right

-

-

-

No

Conservatively

Uneventful

Vomiting

1974

Brunes

54

F

Diffuse abdominal pain

-

25

Left

-

-

-

-

Partial removal of the ruptured cyst

Symptom free

1972

Russell

68

M

Sudden severe abdominal pain

-

12

Left

-

-

-

-

Left lobectomy

Uneventful

1960

Johnston

82

F

Right-sided abdominal pain

-

15

Right

-

-

-

-

Catheter drainage

Died on third postoperative day

Vomiting

1959

Morgenstern

56

F

Sudden severe abdominal pain

Yes

35

Left

Yes

Dark greenish brown

Unknown

Yes

Lobectomy

Uneventful

No vomiting

Decompressing cholecystostomy

In general, treatment options for symptomatic NLCs include surgical procedures and conservative management such as percutaneous needle aspiration and drainage [21]. Percutaneous needle aspiration is a less invasive intervention than a surgical operation and can also be used to examine the properties of the cyst contents. However, it is associated with high relapse rates of >80 %. This high recurrence rate can be decreased by about 20 % when percutaneous needle aspiration is combined with alcohol minocycline chloride or tetracycline chloride injection [22, 23]. In our case, the patient underwent the only conservative management after the initial rupture of NLC without any adjunctive procedures. This could be one reason why the rerupture occurred. With respect to surgical management, open or laparoscopic cyst fenestration, also termed deroofing, is a definitive and widespread treatment [24]. Argon beam coagulation and electrocoagulation to destroy the remaining epithelium and placement of an omental transposition flap after fenestration can also contribute to reduced relapse rates [25]. Complete cyst excision and partial hepatectomy have been performed in some cases because of concern regarding malignancy. However, these operations are highly invasive and almost unacceptable for benign diseases despite the fact that the reported recurrence rate is 0 % [11, 25]. Therefore, in the present case, we performed emergent laparotomy, cyst fenestration, argon beam coagulation of the remaining cyst wall, and placement of an omental transposition flap.

The optimal treatment strategy and surgical indications for NLC rupture are not clearly defined. Conservative management including percutaneous drainage might be useful for cases without critical features such as signs of peritoneal irritation and shock [7]. However, as shown in the current case, rerupture of an NLC after conservative treatment should be considered. In terms of curability, the risk of relapse, and the possibility of other complications such as hemorrhage, cyst fenestration might be more favorable in most cases.

In conclusion, rupture of an NLC is a highly rare complication but can be a cause of the acute abdomen. Clinical observation and conservative treatment including percutaneous needle aspiration and drainage might be beneficial; however, careful consideration of the optimal therapy and performance of close follow-up are necessary owing to the possibility of relapse.

Consent

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Abbreviations

CT: 

computed tomography

NLC: 

nonparasitic liver cyst

Declarations

Acknowledgements

We thank Dr. Sueishi, a pathology faculty member, for the initial pathologic diagnosis of the patient of this case report.

Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.

Authors’ Affiliations

(1)
Department of Surgery and Science, Kyushu University
(2)
Department of Surgery, Fukuoka Higashi Medical Center

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Copyright

© Inoue et al. 2015