Case Report Open Access
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Nov 26, 2023; 11(33): 8030-8037
Published online Nov 26, 2023. doi: 10.12998/wjcc.v11.i33.8030
Giant complex hepatic cyst causing pseudocystitis: A case report
Song Li, De-Sheng Ni, A-Dong Xia, Guo-Liang Chen, Department of Hepatobilary Pancreatic Gastrointestinal Surgery, Jinhua People's Hospital, Jinhua 321000, Zhejiang Province, China
Jie Tang, Department of General Surgery, The Second Affiliated Hospital of Bengbu Medical College, Bengbu 233000, Anhui Province, China
Jie Tang, Anhui Key Laboratory of Tissue Transplantation, Bengbu Medical College, Bengbu 233000, Anhui Province, China
ORCID number: Song Li (0009-0004-6327-8525); De-Sheng Ni (0009-0008-4168-7335); A-Dong Xia (0009-0006-1412-0064); Guo-Liang Chen (0009-0002-1750-3256).
Co-first authors: Song Li and Jie Tang.
Author contributions: Li S, Tang J, Ni DS, and Xia AD conceived, designed, and refined the study protocol; Li S, Tang J, Xia AD, and Chen GL were involved in data collection; Li S, Tang J, and Chen GL analyzed the data; Li S, Tang J, and Ni DS drafted the manuscript; all authors were involved in the critical review of the results and have contributed to, read, and approved the final manuscript. Li S and Tang J contributed equally to this work as co-first authors. The reasons for designating Li S and Tang J as co-first authors are threefold. First, the research was performed as a collaborative effort, and the designation of co-first authors accurately reflects the distribution of responsibilities and burdens associated with the time and effort required to complete the study and the resultant paper. This also ensures effective communication and management of post-submission matters, ultimately enhancing the paper's quality and reliability. Second, the overall research team encompassed authors with a variety of expertise and skills from different fields, and the designation of co-first authors best reflects this diversity. This also promotes the most comprehensive and in-depth examination of the research topic, ultimately enriching readers' understanding by offering various expert perspectives. Third, Li S and Tang J contributed efforts of equal substance throughout the research process. The choice of these researchers as co-first authors acknowledges and respects this equal contribution, while recognizing the spirit of teamwork and collaboration of this study. In summary, we believe that designating Li S and Tang J as co-first authors is fitting for our manuscript as it accurately reflects our team's collaborative spirit, equal contributions, and diversity.
Informed consent statement: Informed consent was obtained from the patient before the publication of this case report.
Conflict-of-interest statement: The authors declare no competing financial interest for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: De-Sheng Ni, MD, Chief Physician, Department of Hepatobilary Pancreatic Gastrointestinal Surgery, Jinhua People's Hospital, No. 267 Danxi East Road, Jindong District, Jinhua 321000, Zhejiang Province, China. 13868987870@163.com
Received: September 14, 2023
Peer-review started: September 14, 2023
First decision: September 28, 2023
Revised: October 9, 2023
Accepted: November 13, 2023
Article in press: November 13, 2023
Published online: November 26, 2023
Processing time: 70 Days and 23.3 Hours

Abstract
BACKGROUND

Hepatic cysts are common benign liver tumors that are typically asymptomatic. However, larger cysts, particularly giant liver cysts, can potentially induce symptoms. If the diameter of the cyst exceeds 10 cm, it can exert pressure on adjacent organs, leading to manifestations of corresponding symptoms. Here, we report the case of a complex giant hepatic cyst that caused pseudocystitis.

CASE SUMMARY

A 16-year-old girl was admitted to our hospital with frequent and urgent urination. Ultrasonography revealed no obvious uterine adnexal abnormalities but showed a hypoechoic, cystic mass (173 mm × 84 mm × 138 mm) with clear boundaries, and an unclear blood flow signal in the abdominal cavity (extending from the lower edge of the left lobe of liver to the upper edge of the bladder). Abdominal contrast-enhanced computed tomography revealed a giant cystic mass in the abdominal and pelvic cavities, possibly originating from the liver, and a small amount of free fluid in the pelvic cavity, which subsequent magnetic resonance imaging confirmed. The imaging characteristics were consistent with a benign lesion. The patient underwent laparoscopic resection of the giant liver cyst with partial liver resection. Post-surgery her symptoms urinary symptoms were relieved completely and she was discharged on the sixth postoperative day.

CONCLUSION

Our patient presented with symptoms suggestive of pseudocystitis, stressing the need for considering possibilities of other etiologies and differential diagnoses.

Key Words: Giant hepatic cyst; Pseudocystitis; Symptoms; Case report

Core Tip: Giant hepatic cysts can cause symptoms upon reaching a significant size. We present a case of a complex giant liver cyst causing pseudocystitis in a 16-year-old girl with frequent and urgent urination. Imaging revealed a large cystic lesion in the liver. Laparoscopic resection with partial liver resection was performed, resulting in the resolution of the urinary symptoms. Therefore, clinicians should consider the possibility of giant liver cysts in patients presenting with similar symptoms.



INTRODUCTION

Liver cysts are common asymptomatic benign tumors with an incidence of 4.5%–7.0%[1,2] and a female preponderance (1.5:1)[3,4]. Liver cysts are mostly diagnosed in adults aged around 40 years and are usually detected incidentally during imaging using computed tomography and/or ultrasonography[5-7]. Frequent and urgent micturition in women, especially married women, is mostly caused by urinary tract and gynecological pathologies. Liver cysts do not cause symptoms related to other abdominal organs unless they expand in size, and cause compression on adjacent structures and symptoms subsequent to that[8,9]. Pseudocystitis resulting from a liver cyst is rare. Here, we report a case of pseudocystitis caused by a giant liver cyst.

CASE PRESENTATION
Chief complaints

Frequent and urgent urination and discovery of an abdominal mass.

History of present illness

A 16-year-old girl was admitted to our hospital with frequent and urgent urination. She had no nausea, vomiting, abdominal pain, or feeling of bloating. Gynecological examination using ultrasonography revealed no obvious uterine adnexal abnormalities. A hypoechoic cystic mass measuring approximately 173 mm × 84 mm × 138 mm with clear boundaries and an unclear blood flow signal was observed in the abdominal cavity, extending from the lower edge of the left liver lobe to the upper edge of the bladder.

History of past illness

The patient was in good health condition with no history of acute or chronic infectious diseases, no history of drug or food allergies, no history of surgery or trauma, and no history of blood transfusion. She was fully vaccinated per societal schedules.

Personal and family history

The patient had a history of pancreatitis in the previous year, no history of hypertension or diabetes, and no family history of liver or renal cysts. Her family members had no similar medical history. Furthermore, she denied any history of familial hereditary diseases.

Physical examination

Physical examination revealed the following: Temperature, 36.8°C; heart rate, 108 beats/min; respiratory rate, 18 beats/min; and blood pressure, 114/82 mmHg. There was no yellowing of the skin or sclera, or swelling of superficial lymph nodes throughout the body. The neck was soft, and the chest was symmetrical, with no obvious abnormalities heard during cardiac and pulmonary auscultation. Her abdomen was flat. A mass measuring approximately 12 cm × 10 cm was palpated in the lower abdomen. The mass was soft in texture, with smooth surface, rounded edges, and clear boundaries, had a range of motion, and could be pushed forward without tenderness. It did not extend to the liver or spleen under the ribs, and Murphy’s sign was negative. There was no pain on percussion in the renal area and no bilateral lower limb edema. Her examination was negative for shifting dullness, and bowel sounds were normal.

Laboratory examinations

Laboratory examination revealed the following: White blood cell count, 3.93 × 109/L; neutrophil percentage, 36.00%; neutrophil count, 1.421 × 109/L; hemoglobin concentration, 109.00 g/L; serum C-reactive protein level, 0.23 mg/L; β-human chorionic gonadotropin level, 0.23 mIU/mL; serum albumin level, 38.6 g/L; total bilirubin level, 21.0 μmol/L; and serum alanine aminotransferase level, 12.4 U/L. After 2 d of bladder fluid cultivation, no bacterial or fungal growth was observed. The remaining findings are shown in Tables 1-5.

Table 1 Patient laboratory data at the time of admission.
Parameter
Patient’s value
Reference value
White blood cell count (109/L)3.933.50–9.50
C-reactive protein (mg/L) 0.230–6
Neutrophils (%)36.0040–75
Lymphocytes (%)55.7020–50
Monocytes (%)5.203–10
Hemoglobin (g/L)109.00115.0–150.0
Platelet count (109/L)172125–350
Activated thromboplastin time (s)13.509.4-12.5
Activated partial thromboplastin time (s)11.011–14
Albumin (g/L)38.640.0–55.0
Total protein (g/L)62.865.0–85.0
Total bilirubin (μmol/L)21.00.0–21.0
Aspartate aminotransferase (U/L)15.113.0–35.0
Alanine aminotransferase (U/L)12.47.0–40.0
Blood creatinine (μmol/L)54.541.0–81.0
Table 2 Tumor indicators at the time of admission.
Parameter
Patient’s value
Reference value
Carcinoembryonic antigen (ng/mL)1.360–5.0
Alpha-fetoprotein (ng/mL)1.71< 9.01
CA-199 (U/mL)3.400–25
β-human chorionic gonadotropin (mIU/mL)0.240–35
CA-125 (U/mL)15.170–5
CA-15-3 (U/mL)3.100–14
CA-50 (U/mL)3.91< 25
CA-72-4 (U/mL)4.020–10
CA-24-2 (U/mL)4.27< 25
Squamous cell carcinoma-associated antigen (ng/mL)0.780–1.5
Cytokeratin 19 fragment (ng/mL)1.59< 3.3
Neuron specific enolase (ng/mL)9.930–20
Table 3 Cystic fluid - routine testing findings.
Parameter
Patient’s value
Reference value
ColorYellow
SolidificationNo solidification
PelluciditySlightly turbid
Proportion1.010Leakage fluid < 1.015
Exudate > 1.018
Rivalta test+Leakage fluid: -
Exudate: +
White blood cell count (109/L)0.04Leakage fluid < 0.1
Exudate > 0.5
Red blood cell count (109/L)0.400
OtherCholesterol crystals detected
Table 4 Biochemical indicators of cystic fluid.
Parameter
Patient’s value
TG (mmol/L)0.12
GLU (mmol/L)0.37
CH (mmol/L)0.67
AMY (U/L)55
ADA (U/L)25.6
LDH (U/L)58.6
ACE (U/L)6.1
Total protein (g/L)66.4
Albumin (g/L)42.4
Imaging examinations

Abdominal contrast-enhanced computed tomography revealed a giant cystic mass in the abdominal and pelvic cavities, possibly originating from the liver. Furthermore, a small amount of free fluid was observed in the pelvic cavity (Figure 1). Magnetic resonance imaging revealed a large cystic mass in the abdominal and pelvic cavities, with features suggesting a benign lesion (Figure 2).

Figure 1
Figure 1 Abdominal contrast-enhanced computed tomography images. Images show a huge cystic mass in the abdominal and pelvic cavities, possibly arising from the liver. A small amount of free fluid is present in the pelvic cavity. A: Middle abdomen; B: Lower abdomen; C: Pelvic cavity; D: Coronal plane of the abdomen; E: Sagittal plane of the abdomen.
Figure 2
Figure 2 Magnetic resonance imaging. Images show a large cystic mass in the abdominal and pelvic cavities that was considered a benign lesion. A small amount of free fluid is present in the pelvic cavity. A: Pelvic cavity; B: Enhanced pelvic cavity; C: Sagittal plane of the abdomen; D: Enhanced sagittal plane of the abdomen.
Table 5 Cystic fluid alpha-fetoprotein and carcinoembryonic antigen.
Parameter
Patient’s value
Reference value
Carcinoembryonic antigen (ng/mL)0.430–5.0
Alpha-fetoprotein (ng/mL)0.72 < 9.01
FINAL DIAGNOSIS

Bile duct derived complex liver cyst.

TREATMENT

The patient underwent laparoscopic resection of the giant liver cyst along with partial liver resection. Her presenting symptoms of frequent or urgent urination were completely relieved post-surgery and she was discharged on the sixth postoperative day.

OUTCOME AND FOLLOW-UP

The patient recovered well, with no symptoms of frequent or urgent urination, and no specific discomfort was observed during follow-up at 0.5, 1, and 3 mo after discharge.

DISCUSSION

Liver cysts are a benign disease with genetic characteristics[10,11]. Simple liver cysts are typical cystic thin-walled masses that originate from bile duct cells that form abnormally during embryonic development[5]. In most cases, cysts occur only in the liver and patients generally have no obvious clinical symptoms. However, in some patients, the expansion of liver cysts can cause abdominal symptoms, mainly due to a series of corresponding clinical symptoms caused by compression of the surrounding tissues or organs caused by oversized liver cysts[12]. Asymptomatic simple liver cysts usually do not require treatment. The treatments for liver cysts with obvious clinical symptoms include percutaneous puncture, aspiration, sclerotherapy, and surgery[5].

Herein, we report a case of pseudocystitis caused by a giant complex liver cyst that was pathologically suggestive of a bile duct derived liver cyst and was different from a conventional liver cyst (Figure 3). There are no reports in the literature of complex liver cysts causing symptoms of pseudocystitis. Liver cysts are typically asymptomatic. An increase in cyst size, particularly in giant liver cysts, can initiate symptoms. If the diameter of the cyst exceeds 10 cm, it can expand and cause pressure effect on adjacent organs and corresponding symptoms may appear[8], including abdominal pain, nausea, vomiting, obstructive jaundice, superior vena cava thrombosis, acute pulmonary embolism, and acute pancreatitis[6,13-15]. Giant liver cysts should be differentiated from Caroli disease[16], giant mesenchymal hamartomas of the liver[17], teratomas[18], and other diseases[19-21]. Our patient had previously experienced symptoms of pancreatitis. Therefore, we assumed that the pain was caused by a liver cyst. At the time of presentation, the liver cyst caused symptoms in distant organs that disappeared postoperatively. Thus, it was confirmed that the giant liver cyst caused the pain. These cysts usually require surgical intervention. In our patient, the giant cyst was located at the edge of the liver; thus, the liver was stretched and deformed under the influence of gravity. During the surgical process, we obtained biopsy samples of adjacent tissues to determine the possibility of further deterioration of liver architecture, and to rule out possible recurrence, which the liver cysts are prone to.

Figure 3
Figure 3 Postoperative specimen and pathological diagnosis. A: Postoperative specimen; B: Bile duct derived cyst liver tissue showing bleeding and a compression injury (liver tissue); C: A few red blood cells and histiocytes (ascites cell mass).
CONCLUSION

Our case report highlights that the diagnosis and treatment of complex giant liver cysts that cause pseudocystitis should be comprehensive and multidimensional. The differential diagnosis of such abdominal masses should be considered before treatment. The patient was a young, unmarried girl, and a detailed plan was specified to minimize major trauma and achieve the best treatment outcomes. These patients require close follow-up because liver cysts are prone to recurrence.

Footnotes

Provenance and peer review: Unsolicited article; Externally peer reviewed.

Peer-review model: Single blind

Specialty type: Medicine, research and experimental

Country/Territory of origin: China

Peer-review report’s scientific quality classification

Grade A (Excellent): 0

Grade B (Very good): 0

Grade C (Good): C

Grade D (Fair): 0

Grade E (Poor): 0

P-Reviewer: Kodama T, Japan S-Editor: Lin C L-Editor: Wang TQ P-Editor: Lin C

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