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Chohan S, Zaheer S. Mucinous Cystic Neoplasm of Gall Bladder Lacking the Usual Ovarian-Like Mesenchymal Stroma. Indian J Surg Oncol 2025; 16:717-721. [PMID: 40337016 PMCID: PMC12052720 DOI: 10.1007/s13193-024-02128-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/04/2024] [Accepted: 10/24/2024] [Indexed: 05/09/2025] Open
Affiliation(s)
- Shikhar Chohan
- Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
| | - Sufian Zaheer
- Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
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Ramavath K, Kaman L, Gupta A, Singh A, Das A. Biliary cystadenoma in an endemic zone of hydatid cyst: A rare surgical surprise. Ann Hepatobiliary Pancreat Surg 2020; 24:85-89. [PMID: 32181435 PMCID: PMC7061038 DOI: 10.14701/ahbps.2020.24.1.85] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/05/2019] [Revised: 12/02/2019] [Accepted: 01/19/2020] [Indexed: 11/23/2022] Open
Abstract
The advancement of radiological investigations has led to the early and incidental detection of hepatic cystic lesions. These are most commonly the simple cysts but can be malignant as well. Despite the recent advances, these lesions still pose a diagnostic as well as therapeutic challenge. The biliary cystadenomas and carcinomas form around 5% of all the malignant cystic lesions of liver. These lesions are hardly diagnosed preoperatively and are usually a histopathological surprise. They warrant a surgical excision. Herewith, the authors are describing a case of cystic hepatic neoplasm initially misdiagnosed as hydatid cyst of liver and discovered to be a vascular cystic lesion intraoperatively. This patient underwent resection of the lesion and was discovered to harbour biliary cystadenoma on histopathological specimen.
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Affiliation(s)
- Krishna Ramavath
- Department of General Surgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Lileswar Kaman
- Department of General Surgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Ashish Gupta
- Department of General Surgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Abhinav Singh
- Department of General Surgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Asim Das
- Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
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3
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Pitchaimuthu M, Duxbury M. Cystic lesions of the liver-A review. Curr Probl Surg 2017; 54:514-542. [PMID: 29173653 DOI: 10.1067/j.cpsurg.2017.09.001] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/30/2017] [Accepted: 09/08/2017] [Indexed: 01/10/2023]
Affiliation(s)
- Maheswaran Pitchaimuthu
- Department of General Surgery, Glasgow Royal Infirmary, Glasgow, United Kingdom; Department of HPB and Transplant Surgery, Cleveland Clinic Foundation, Cleveland, Ohio, USA.
| | - Mark Duxbury
- Department of General Surgery, Glasgow Royal Infirmary, Glasgow, United Kingdom
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Outcome following Resection of Biliary Cystadenoma: A Single Centre Experience and Literature Review. Int J Hepatol 2015; 2015:382315. [PMID: 26839708 PMCID: PMC4709616 DOI: 10.1155/2015/382315] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/12/2015] [Revised: 12/14/2015] [Accepted: 12/15/2015] [Indexed: 02/06/2023] Open
Abstract
Background. Biliary cystadenomas (BCAs) are rare, benign, potentially malignant cystic lesions of the liver, accounting for less than 5% of cystic liver tumours. We report the outcome following resection of biliary cystadenoma from a single tertiary centre. Methods. Data of patients who had resection of BCA between January 1993 and July 2014 were obtained from liver surgical database. Patient demographics, clinicopathological characteristics, operative data, and postoperative outcome were analysed. Results. 29 patients had surgery for BCA. Male : female ratio was 1 : 28. Clinical presentation was abdominal pain (74%), jaundice (20%), abdominal mass (14%), and deranged liver function tests (3%). Cyst characteristics included septations (48%), wall thickening (31%), wall irregularity (38%), papillary projections (10%), and mural nodule (3%). Surgical procedures included atypical liver resection (52%), left hemihepatectomy (34%), right hemihepatectomy (10%), and left lateral segmentectomy (3%). Median length of stay was 7 (IQ 6.5-8.5) days. Two patients developed postoperative bile leak. No patients had malignancy on final histology. Median follow-up was 13 (IQ 6.5-15.7) years. One patient developed delayed biliary stricture and one died of cholangiocarcinoma 11 years later. Conclusion. Biliary cystadenomas can be resected safely with significantly low morbidity. Malignant transformation and recurrence are rare. Complete surgical resection provides a cure.
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Hemorrhagic hepatic cyst: report of a case and review of the literature with emphasis on clinical approach and management. J Gastrointest Surg 2012; 16:1782-9. [PMID: 22688416 DOI: 10.1007/s11605-012-1922-6] [Citation(s) in RCA: 32] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/28/2012] [Accepted: 05/30/2012] [Indexed: 01/31/2023]
Abstract
OBJECTIVE Hemorrhage within a hepatic cyst (hemorrhagic hepatic cyst, HHC) is a complication of liver cysts that is difficult to differentiate from other neoplastic entities on imaging. Even when accurately diagnosed, there has been a lack of consensus on the optimal treatment strategy. After presenting our experience with a patient treated via laparoscopy, we aimed to conduct a review of the literature on HHCs. METHODS A computerized search in Medline, PubMed, Google Scholar, and The Cochrane Collaboration was carried out for journal articles or abstracts published from 1950 to 2011. RESULTS A total of 24 patients with HHCs were identified from 1983 to 2011. The cohort had an even gender distribution with a mean age of 62.7 years. Most patients presented with abdominal pain (80 %), while three (14 %) patients were asymptomatic at the time of presentation. CT imaging and ultrasound were unable to accurately diagnose HHC, whereas hyperintensity on MRI was a reliable diagnostic tool. Three (13 %) patients were managed conservatively with observation. Seven (30 %) patients had percutaneous transhepatic drainage. Among these, two patients experienced recurrence that required repeat treatment. Two (9 %) patients underwent open unroofing of their HHC and one (4 %) laparoscopically, without recurrences. Seven (30 %) patients underwent hepatic resection, whereas six (26 %) patients had a cyst enucleation for their HHCs. All patients had uneventful recoveries, with a mean follow-up of 25 months. CONCLUSIONS MRI is a reliable diagnostic tool in the setting of an HHC. Laparoscopic unroofing of HHCs may represent a less morbid and safe treatment modality with low recurrence rates. Given the low level of clinical evidence available so far, these results should be interpreted with caution.
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Biliary cystadenomas: a case for complete resection. HPB SURGERY : A WORLD JOURNAL OF HEPATIC, PANCREATIC AND BILIARY SURGERY 2012; 2012:501705. [PMID: 22778493 PMCID: PMC3388282 DOI: 10.1155/2012/501705] [Citation(s) in RCA: 31] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 03/14/2012] [Revised: 04/30/2012] [Accepted: 05/08/2012] [Indexed: 12/13/2022]
Abstract
Introduction and Objective. Biliary cystadenoma is a rare benign neoplasm of the liver with less than 200 cases being reported allover the world. We report a series of 13 cases highlighting the radiological findings and problems related to its management. Materials and Methods. Records of thirteen patients who underwent surgery for biliary cystadenomas, between March 2006 and October 2011, were reviewed retrospectively. Results. Majority of the patients were females (11 out of 13), with a median age of 46 (23–65) years. The most frequent symptom was abdominal pain (92%). Seven patients had presented with history of previous surgery for liver lesions. Five patients had presented with recurrence after partial resection for a suspected hydatid cyst and two after surgery for presumed simple liver cyst. Ten of the 13 patients had complete resection of the cyst with enucleation in 3 patients, 2 of whom in addition required T-tube drainage of the bile duct. There has been no recurrence during the follow-up period ranging from 3 months to 5 years. Conclusion. Biliary cystadenoma must be differentiated from other benign cysts. Hepatic resection or cyst enucleation is the recommended treatment option.
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Sang X, Sun Y, Mao Y, Yang Z, Lu X, Yang H, Xu H, Zhong S, Huang J. Hepatobiliary cystadenomas and cystadenocarcinomas: a report of 33 cases. Liver Int 2011; 31:1337-44. [PMID: 21745301 DOI: 10.1111/j.1478-3231.2011.02560.x] [Citation(s) in RCA: 43] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
BACKGROUND Hepatobiliary cystadenomas and cystadenocarcinomas are rare and often misdiagnosed. AIMS We report our experience with 33 cases over 20 years to discuss an algorithm for these diseases. METHODS Patients presenting with a diagnosis of hepatobiliary cystadenomas and cystadenocarcinomas were retrospectively reviewed from January 1991 to October 2010. Clinical data were collected by examining hospital records and by follow-up questionnaire interviews. RESULTS Thirty-three patients had pathologically diagnosed hepatobiliary cystadenomas (19/33, 17 females and two males) or cystadenocarcinomas (14/33, five females and nine males). Symptoms of cystadenomas at hospitalization were abdominal bloating or pain (9/19). Nine patients had an elevated level of carbohydrate antigen (CA) 19-9. The surgical procedures, i.e. cyst enucleation, segmentectomy, sectionectomy and hemihepatectomy, were performed with satisfactory outcomes. Symptoms of cystadenocarcinomas included abdominal bloating or pain (8/14) and fever (3/14). Seven patients had elevated CA19-9. The imaging characteristics of cystadenocarcinomas were similar to those of cystadenomas. The clinical outcomes for cystadenocarcinomas were mostly poor after either surgical or conservative treatment. CONCLUSIONS Clinical symptoms are unreliable for these diagnoses and their differential diagnosis. Imaging evaluations and CA19-9 are of value for the recognition of cystadenoma and cystadenocarcinoma, but not for their differential diagnosis. Any recurrence of liver cyst after surgery or other treatments should lead one to suspect one of these diseases. Invasive examination and percutaneous fine-needle aspiration cytology are not recommended. Complete excision or careful enucleation should be the first treatment choice for a better prognosis.
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Affiliation(s)
- Xinting Sang
- Department of Liver Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & PUMC, Beijing, China
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8
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Ghole SA, Bakhtary S, Staudenmayer K, Sze DY, Pai RK, Visser BC, Norton JA, Poultsides GA. Ruptured biliary cystadenoma managed by angiographic embolization and interval partial hepatectomy. Dig Dis Sci 2011; 56:1949-53. [PMID: 21445579 DOI: 10.1007/s10620-011-1677-z] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/07/2011] [Accepted: 03/09/2011] [Indexed: 12/22/2022]
Affiliation(s)
- Saif A Ghole
- Department of Surgery, Stanford University Medical Center, Stanford, CA, USA
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Affiliation(s)
- W. Y. LAU
- Surgical Unit C, Queen Elizabeth Hospital, Kowloon, Hong Kong
| | - C. H. CHOW
- Surgical Unit C, Queen Elizabeth Hospital, Kowloon, Hong Kong
| | - M. L. LEUNG
- Surgical Unit C, Queen Elizabeth Hospital, Kowloon, Hong Kong
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Manouras A, Markogiannakis H, Lagoudianakis E, Katergiannakis V. Biliary cystadenoma with mesenchymal stroma: Report of a case and review of the literature. World J Gastroenterol 2006; 12:6062-9. [PMID: 17009411 PMCID: PMC4124420 DOI: 10.3748/wjg.v12.i37.6062] [Citation(s) in RCA: 50] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023] Open
Abstract
Biliary cystadenomas are rare, cystic neoplasms of the biliary ductal system that usually occur in middle-aged women. They cannot be safely differentiated from cystadenocarcinomas before operation and should always be considered for resection. Cystadenomas have a strong tendency to recur, particularly following incomplete excision, and a potential of malignant transformation. Therefore, complete resection is the therapy of choice and thorough histopathologic evaluation is imperative. A case of benign biliary cystadenoma with mesenchymal stroma is presented along with a review of the relative literature addressing the clinical presentation, histology, histogenesis, differential diagnosis, imaging features, treatment and prognosis of this interesting and rare entity.
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Affiliation(s)
- Andreas Manouras
- 1st Department of Propaedeutic Surgery, Hippokration Hospital, Athens, Greece
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Kazama S, Hiramatsu T, Kuriyama S, Kuriki K, Kobayashi R, Takabayashi N, Furukawa K, Kosukegawa M, Nakajima H, Hara K. Giant intrahepatic biliary cystadenoma in a male: a case report, immunohistopathological analysis, and review of the literature. Dig Dis Sci 2005; 50:1384-9. [PMID: 16047491 DOI: 10.1007/s10620-005-2791-6] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/19/2022]
Affiliation(s)
- Shinsuke Kazama
- Department of Surgery, Yaizu Municipal Hospital, 1000 Dobara, Yaizu-city, Shizuoka, 425-8505, Japan.
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Yoshida H, Tajiri T, Mamada Y, Taniai N, Akimaru K, Kawano Y, Mizuguchi Y, Shimizu T, Takahashi T, Naito Z. Rapidly enlarging hepatobiliary cystadenoma. J Med Ultrason (2001) 2003; 30:257-62. [DOI: 10.1007/bf02481290] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/11/2003] [Accepted: 05/12/2003] [Indexed: 10/24/2022]
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Florman SS, Slakey DP. Giant Biliary Cystadenoma: Case Report and Literature Review. Am Surg 2001. [DOI: 10.1177/000313480106700802] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/02/2022]
Abstract
Biliary cystadenoma is a very rare cystic neoplasm of the liver. This tumor is insidiously progressive and usually presents in white females in their fifth decade. It has a characteristic appearance on ultrasound, computed tomography, and angiography. The exact etiology of these tumors is unknown, but several theories have been proposed. Historically these cystic tumors have been treated by a variety of techniques including aspiration, fenestration, internal drainage, and resection. Previously reported series have confirmed a >90 percent recurrence rate with anything less than complete excision. In addition biliary cystadenoma is a premalignant lesion and only surgical excision can differentiate it from its malignant counterpart, biliary cystadeno-carcinoma.
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Abstract
A 53-yr-old woman with a history of hepatic cystadenoma 25 yr before presented with a simple hepatic cyst, which evolved over 9 yr into a complex cystadenoma with septations and internal bleeding. She was treated with a left hepatectomy. Review of the literature shows that hepatic cystadenomas, although rare, frequently can recur years later and have potential for malignant transformation. Histologic similarities of one variant with ovarian stroma raises interesting possibilities regarding the origin of these lesions. The best treatment results are obtained with radical excision.
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Affiliation(s)
- G E Catinis
- Department of Gastroenterology, Louisiana State University Medical Center, New Orleans, USA
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Hanazaki K, Wakabayashi M, Mori H, Sodeyama H, Yoshizawa K, Yokoyama S, Sode Y, Kawamura N, Miyazaki T. Hemorrhage into a simple liver cyst: diagnostic implications of a recent case. J Gastroenterol 1997; 32:848-51. [PMID: 9430029 DOI: 10.1007/bf02936967] [Citation(s) in RCA: 36] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
Abstract
Hemorrhage complicating simple liver cyst is rare. On imaging studies, the differential diagnosis between intracystic hemorrhage and cystadenocarcinoma of the liver is unreliable, and hepatectomy has been performed for benign liver cyst in this situation. We describe the characteristics of hemorrhage into a liver cyst in a patient who underwent dome resection of the cyst. In our patient, important diagnostic findings included benign cytologic features in a cyst fluid specimen aspirated with ultrasonographic guidance and a fall in hemoglobin and hematocrit, suggestive of hemorrhage. Other informative features were absence of communication between the intrahepatic bile ducts and the cystic liver lesion upon endoscopic retrograde cholangiopancreatography, as well as benign intraoperative frozen-section histology.
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Affiliation(s)
- K Hanazaki
- Department of Surgery, Nagano Red Cross Hospital, Japan
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Abstract
In a detailed review of cystic hepatobiliary neoplasms, we identified a subset of 50 cases in which tumors were characterized by the presence of a mesenchymal cell layer interposed between an inner epithelial lining and an outer connective tissue layer. We have recently seen three such patients, making a total of 53 patients reported in the English literature. All of the patients were female, 44 of whom, with an average age of 41 years, had benign tumors. The average age of the remaining nine patients was 57 years and these patients had malignant tumors. In seven patients, the malignancy arose from the epithelial layer, but in two patients sarcomatous changes were identified in the mesenchymal tissues. The older age of the patients with malignant tumors with adequate serial biopsies in two cases supported the thesis that malignant transformation may occur in the benign tumors. Moreover the location of the tumor in one of our patients in whom the resected tumor was associated with anomalous right hepatic ducts and portal veins supported the theory that these tumors develop embryologically from nests of primitive hepatobiliary endodermal and mesodermal cells. Although surgical treatment was performed in all patients, 25% of the patients with benign hepatobiliary cystadenoma with mesenchymal stroma (CMS), and 33% of the patients with malignant CMS had tumor recurrence after primary resection. Ninety per cent of these patients had an incomplete resection at the time of their initial operations. Forty-four per cent of the patients with malignant CMS died after a mean follow-up of 17 months. We conclude that CMS (Edmonson's tumor) occurs uniquely in young female patients, develops from nests of primitive embryonal cells, has the potential for malignant transformation, and should be completely resected at primary operation to avoid recurrence.
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Marcial MA, Hauser SC, Cibas ES, Braver J. Intrahepatic biliary cystadenoma. Clinical, radiological, and pathological findings. Dig Dis Sci 1986; 31:884-8. [PMID: 3731980 DOI: 10.1007/bf01296059] [Citation(s) in RCA: 24] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/07/2023]
Abstract
A 24-year-old white woman presented with abdominal discomfort associated with a firm, nonpulsatile hepatic mass. Radiological examination, which included radionuclide liver scan, abdominal ultrasonography, computed tomography, and hepatic angiography, demonstrated a large, globular, multilocular cyst arising from the left lobe of the liver. A left hepatic lobectomy was performed, and gross examination of the specimen confirmed the cystic nature of the lesion. Histologically, the multiple locules were lined by a single layer of tall cuboidal to columnar cells, which were comparable to those of native biliary tract epithelium. There was little evidence of cellular pleomorphism, and invasion into the underlying mesenchymal stroma was not present. A diagnosis of intrahepatic biliary cystadenoma was made. The clinical, radiological, and pathological findings of this neoplasm are presented and its differential diagnosis discussed.
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Tomioka T, Tsuchiya R, Harada N, Tsunoda T, Matsuo T. Cystadenoma and cystadenocarcinoma of the liver: localization of carcinoembryonic antigen. THE JAPANESE JOURNAL OF SURGERY 1986; 16:62-7. [PMID: 3515006 DOI: 10.1007/bf02471072] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/06/2023]
Abstract
Cystadenomas and cystadenocarcinomas of the liver are rare tumors. The distribution of carcinoembryonic antigen (CEA) in two cystadenomas and a cystadenocarcinoma was examined immunohistochemically by the peroxidase-labeled antibody method, at the light microscopic level. In the cystadenomas and areas consisting of benign-appearing cells in the cystadenocarcinoma, CEA was localized to the luminal surfaces of the glandular cells appearing as thin linear stains. In malignant epithelial cells forming nests and daughter cysts of the cystadenocarcinoma, CEA was demonstrated throughout the cytoplasm, in a diffuse pattern. Thus, the immunohistochemical localization of CEA may be helpful in the diagnosis of borderline lesions and in determining the distribution of benign and malignant epithelium in cystadenocarcinomas. The etiology, clinical features, pathology and treatment of these rare tumors are discussed.
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Wheeler DA, Edmondson HA. Cystadenoma with mesenchymal stroma (CMS) in the liver and bile ducts. A clinicopathologic study of 17 cases, 4 with malignant change. Cancer 1985; 56:1434-45. [PMID: 4027877 DOI: 10.1002/1097-0142(19850915)56:6<1434::aid-cncr2820560635>3.0.co;2-f] [Citation(s) in RCA: 170] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/08/2023]
Abstract
This study of 17 cases describes a homogeneous clinicopathologic group of tumors previously subsumed under the general term, hepatobiliary cystadenoma. This unique group of neoplasms that the authors have termed "cystadenoma with mesenchymal stroma" (CMS), occurred exclusively in women and showed two essential tumor components: a cyst lining of columnar to cuboidal, mucin secreting epithelium; and a moderately to densely cellular stroma composed of spindle (rarely oval) cells. The stromal element appeared similar to primitive mesenchyme, both on light and electron microscopic study, showing variable differentiation toward fibroblasts, smooth muscle, adipose tissue and capillaries. Cases of hepatobiliary "cystadenoma" in the literature with histology similar to CMS had similar sex and age incidences, while histologically dissimilar cases, lacking the mesenchymal stroma, had significantly different parameters. Four cases of CMS in this series showed malignant change that was manifested as papillary adenocarcinoma, suggesting the need for complete surgical removal of these premalignant neoplasms.
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