Laparoscopy-assisted extended right hepatectomy for giant hemorrhagic hepatic cyst mimicking biliary cystadenocarcinoma: a case report
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Hemorrhagic hepatic cysts infrequently involve several iconographic changes requiring a differential diagnosis, primarily with a cystic malignancy. We herein report a case of laparoscopy-assisted extended right hepatectomy for a giant hemorrhagic hepatic cyst with an enhancing mural nodule that was clinically suspected of being biliary cystadenocarcinoma.
A 73-year-old woman was followed up for giant hepatic cyst occupying the right lobe of the liver. During the follow-up, an enhancing mural nodule was newly noted on computed tomography in 2016. Based on additional clinical examinations, biliary cystadenocarcinoma was undeniable, and laparoscopy-assisted extended right hepatectomy was performed for diagnostic and therapeutic purposes. She had no perioperative complications and was discharged on postoperative day 13. A histological examination of the mural nodule showed neovascularization within an organized hematoma.
We herein report a rare case of giant hemorrhagic hepatic cyst mimicking biliary cystadenocarcinoma that was successfully treated with laparoscopy-assisted extended right hepatectomy. Laparoscopic surgery in our case was an effective procedure performed with the utmost care.
KeywordsHemorrhagic hepatic cyst Organized hematoma Neovascularization Laparoscopy-assisted surgery
Apparent diffusion coefficient
Elastica van Gieson
Fluorodeoxyglucose-positron emission tomography
Hemorrhagic hepatic cyst
Laparoscopic liver resection
Murine double minute
Magnetic resonance imaging
Smooth muscle actin
Standardized uptake value
- T1 and T2WI
T1- and T2-weighted imaging
Intracystic hemorrhage of the liver (i.e., hemorrhagic hepatic cyst (HHC)) is an infrequent complication occurring in simple hepatic cysts with complicated iconographic characteristics (e.g., a thickened cyst wall, a solid septal structure or a mural nodule) [1, 2, 3, 4, 5]. In particular, when enhancing solid components which are newly detected in the cyst, it is sometimes difficult to deny the presence of cystic neoplasms, such as biliary cystadenoma or cystadenocarcinoma, and surgical resection is considered for diagnostic and therapeutic purposes. When attempting a minimally invasive approach, it is of paramount importance to ensure the surgical and oncological safety [6, 7].
We herein report a case of giant HHC with an enhancing mural nodule newly arising during follow-up for hepatic giant cyst and clinically suspicious for biliary cystadenocarcinoma that was successfully treated with laparoscopy-assisted extended right hepatectomy and was pathologically diagnosed as HHC with neovascularization within an organized hematoma.
A 73-year-old woman had been followed for giant hepatic cyst occupying the right lobe of the liver with a maximum diameter of 20 cm since 2005. Her medical history included a benign tumor in the transverse colon and an unruptured cerebral aneurysm. She had no remarkable family history.
We encountered a case of successful laparoscopic surgery performed for giant HHC with an enhancing mural nodule that was clinically suspected of being biliary cystadenocarcinoma.
Laparoscopic liver resection (LLR) has rapidly gained acceptance over the past decade as a viable treatment for several malignant and benign hepatic diseases, given its prominent advantages over an open approach with regard to post-surgical pain relief, better cosmetic results, and a shorter hospital stay due to the smaller surgical wound and less bleeding. The recent international consensus is that minor LLR is a safe and feasible procedure that is superior to an open approach for select patients; in contrast, major LLR remains a technically demanding procedure, and its safety, feasibility and preliminary evidence are still limited .
At our institute, we have expanded the indication for LLR as we accumulate experience, and laparoscopic-assisted extended right hepatectomy was completed with sufficient surgical and oncological safety in the present case. Remarkable surgical techniques applied in this case included the shrinkage of the cyst and the hanging maneuver technique. First, we carefully removed the cyst contents without any spillage because shrinkage of the cyst was necessary in order to create an intraabdominal working space. We were subsequently able to mobilize the right lobe of the liver laparoscopically. Second, the hanging maneuver technique was effective for ensuring the safe performance of parenchymal division through the small laparotomy wound (7 cm), as this technique makes it possible to pull up the liver so that the resection line comes just beneath the laparotomy wound .
Previous studies have discussed the imaging characteristics of HHCs; however, they still remain challenging to describe [1, 2, 3, 4, 5]. Very recently, Kohno et al. summarized the imaging characteristics of HHCs with enhancing mural nodules . In all the resected cases in the literature, the nodules were histologically diagnosed with neovascularization within organized hematoma, as was observed in our case. As they mentioned, the ventral enhancing mural nodule in our series showed persistent centripetal filling on delayed CT images and central high signal intensity on T2WI. In addition, the calcification of the cyst wall was found close to the dorsal mural nodule. However, unlike their report, the ventral mural nodule in the present case demonstrated a weak but abnormal uptake on FDG-PET. This abnormal uptake on PDG-PET may have been associated with the high Ki-67 value. Previous studies have shown that the SUVmax on FDG-PET was correlated with the Ki-67 index of various tumors [10, 11]. Considering that the ventral mural nodule rapidly grew within a year, it is conceivable that the nodule included highly proliferating endothelial cells. Moreover, we found a high signal intensity area on DWI with a low ADC value on MRI in the ventral mural nodule, and this was retrospectively considered to be consistent with organized hematoma. However, these MRI findings were also observed in biliary cystadenocarcinoma, which led to the preoperative misdiagnosis.
Because HHC is a rare and heterogeneous entity, its cause and pathogenesis have not been fully discussed. In the present case, we hypothesize that giant HHC was formed as a result of repeated rupture of venous malformation and that neovascularization developed reactively in association with the thrombus undergoing organization. Hepatic hemangioma with cyst formation has been also reported as a rare condition [12, 13], and our case bore some similarities to this entity. However, the dilated vessels in the ventral mural nodule were immunohistochemically negative for αSMA and EVG, indicating reactive angiogenesis mimicking cavernous hemangioma.
In conclusion, we reported a rare case of HHC mimicking biliary cystadenocarcinoma successfully treated with laparoscopy-assisted extended right hepatectomy. When a new enhancing solid nodule appears within HHC, it is difficult to rule out cystic neoplasms preoperatively in many cases. However, we should bear in mind the possible presence of HHC with neovascularization within the organized hematoma, and laparoscopic surgery can be a diagnostic and therapeutic option if technically possible and performed with the utmost surgical and oncological care.
The authors declare that this study was not funded externally.
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Data sharing not applicable to this article as no datasets were generated or analyzed during the current study. Please contact author for data requests.
YF and TA designed this study and drafted the manuscript. TA and HE were the patient’s attending physicians, and TA, HE, TN and KG performed the hepatectomy. KH and EM were the expert pathologists. YI, HA, SK, MM, and YD were the supervising surgeons. All authors read and approved the final manuscript.
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Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent document is available for review by the Editor-in-Chief of this journal.
The authors declare that they have no competing interests.
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