Multilocular cystadenoma and cystadenocarcinoma of the prostate

BACKGROUND: Multicystic prostatic tumors are rare, with only a few reported cases of prostatic cystadenoma and cystadenocarcinoma in the scientific literature. METHODS: A retrospective review of our tumor registry over the last 25 years identified 2 rare cystic tumors of the prostate: 1 multilocular cystadenoma and 1 multilocular cystadenocarcinoma. RESULTS: The first case illustrates the clinical and pathologic features of prostatic multilocular cystadenoma. A 42-year-old man presented with a 16-cm suprapubic mass causing displacement of adjacent visceral organs. Pathologic examination after prostatectomy confirmed it to be a multilocular cystadenoma of the prostate. The patient's postoperative course was uneventful, and his serum prostate-specific antigen level remained at < or =0.04 ng/ml throughout the course of his disease. In the second case, we present an 80-year-old male presenting with a 12-cm cystic mass of the prostate. His serum prostate-specific antigen level remained at > or =9.0 ng/ml throughout the course of his disease. The tumor had an aggressive local growth pattern, with invasion into perirectal adipose tissue. This patient underwent a pelvic exenteration, followed by adjuvant systemic chemotherapy and complete androgen blockade. Despite aggressive treatment, he had 3 recurrences over 4 months but remains alive with disease at 23-month follow-up. CONCLUSIONS: Cystadenocarcinoma of the prostate is locally aggressive and should be included in the differential diagnosis of cystic lesions of the prostate.     

肝胆管囊腺瘤及囊腺癌的CT、MRI诊断

目的总结肝胆管囊腺瘤和囊腺癌的CT、MRI表现,探讨CT、MRI诊断该肿瘤的价值。方法回顾性分析经手术病理证实为肝胆管囊腺瘤(3例)和囊腺癌(5例)的CT和(或)MRI表现、术前诊断,并与病理所见进行对照分析。结果3例囊腺瘤均为单房或多房的囊性肿物,无壁结节,术前均误诊为肝包虫或肝囊肿;5例恶性者中囊腺癌3例,囊腺瘤伴局部癌变2例。5例均为单房或多房的囊实性肿物,有壁结节。CT扫描囊性部分呈均匀液性密度,囊壁、分隔厚薄不均,壁结节大小不一,增强扫描囊壁、分隔及壁结节强化。MR示囊性部分呈均匀液性信号,增强扫描囊壁、分隔及壁结节强化。5例恶性者术前4例诊断为囊腺瘤或囊腺癌,1例诊为胆管癌。CT、MRI表现与病理所见对应良好。结论胆管囊腺癌在CT、MRI上具有特征性表现,可以做到术前正确诊断;但胆管囊腺瘤如果无壁结节则缺乏特征性表现,需要和肝复杂性囊肿或肝包虫病等鉴别。     

原发性肝内胆管囊性肿瘤

原发性肝内胆管囊性肿瘤（ＰＣＴＩＨＢＤ）比较罕见可分为囊腺瘤和囊腺癌。它们分泌大量的粘液形成单房和多房性囊肿，囊内壁发出乳头状赘生物突入囊腔，囊腺癌通常被认为由囊腺瘤恶变而来。文章报告ＰＣＴＩＨＢＤ５例，其中囊腺癌４例，囊腺瘤１例。５例经Ｂ超和ＣＴ检查后均诊断为肝癌，只是在剖腹探查和病理学检查后才获得正确的诊断，手术切除为唯一有效的治疗。４例囊腺癌中１例术后第６天死于大出血，１例随访７个月后死于肝内复发；另２例分别随访４年及５年均存活。１例囊腺瘤手术７年仍健在。     

Hepatobiliary cystadenoma combined with multiple liver cysts: report of a case

Hepatobiliary cystadenomas are rare benign tumors with malignant potential. They are almost always solitary lesions accompanied by multilocular cysts in the liver, and are difficult to differentiate from cystadenocarcinoma, despite the diagnostic modalities available. This report describes a case of hepatobiliary cystadenoma with multiple cysts in the left hepatic lobe, diagnosed by magnetic resonance imaging in a 48-year-old woman. Abdominal computed tomography revealed only multiple cystic lesions in the left lobe, but cholangiography via a nasogastric biliary drainage tube combined with percutaneous transhepatic cholangiography showed a stenotic region with fine irregularity in the left lateral posterior segmental bile duct and left lateral anterior segmental bile duct. Hepatobiliary cystadenocarcinoma with multiple liver cysts was suspected. We performed left hepatectomy, and microscopic examination confirmed a diagnosis of hepatic cystadenoma with multiple liver cysts. There was no nuclear atypia or mitosis in the epithelium of the locus, which was constructed of simple columnar-to-cuboidal epithelium with basal nuclei. The patient is well without recurrence more than 4 years after surgery. Received: July 5, 2000 / Accepted: January 9, 2001     

Primary smooth muscle tumor of the liver encasing hepatobiliary cystadenoma without mesenchymal stroma.

We describe a 59-year-old Japanese woman with a large mass of her liver encasing cystic components. Radiologic imaging showed the mass to be hypervascular, and surgical resection disclosed a white tumor. The solid portion was immunohistochemically characterized as a smooth muscle tumor. The cystic components were multilocular and lined with columnar epithelium, consistent with a hepatobiliary cystadenoma. The epithelium strongly stained for CA19-9. The subepithelial space was occupied by collagenous connective tissue interspersed with a small number of spindle-shaped cells. The cystic lesions lacked the mesenchymal stroma between the epithelium and connective tissue layer. There have been no previous reports of a hepatic smooth muscle tumor encasing a hepatobiliary cystadenoma. Because of the pathogenesis of the cystadenoma, it is possible to assume that the smooth muscle tumor also arose from the cells composing the biliary duct in association with the development of the cystadenoma.     

肝内胆管囊腺瘤的诊治分析

目的：提高对肝内胆管囊腺瘤的认识。 方法：回顾性分析8年来收治的18例肝内胆管囊腺瘤患者的临床表现、影像特点及外科治疗的方式。 结果：4例为体检发现,其他14例均有不同症状,主要表现为右上腹不适或疼痛11例,腹部肿块3例,伴有黄疸发热等胆管炎症状2例。B超和CT提示肝内囊性占位性病变,可呈多囊或单囊,内壁可见乳头状物。均获得手术切除,18例肝内胆管囊腺瘤中6例发生癌变。 癌变者术后平均生存时间为35个月(27-58个月)。结论：术前确诊肝内胆管囊腺瘤困难,肝内胆管囊腺瘤易恶变,发现后应早期手术切除,可有效防止复发或恶变。     

Lymphoepithelial Cyst of the Pancreas: Report of a Case

An extremely rare case of a lymphoepithelial cyst (LEC) of the pancreas is described herein. A pancreatic cystic tumor was initially detected in a 50-year-old man at a medical checkup. On admission, his serum carbohydrate antigen (CA) 19-9 level was 8 100 U/ml and a computed tomography scan revealed a well-circumscribed multilocular cystic tumor in the pancreatic head and body. Magnetic resonance cholangiopancreatography showed no communication between the pancreatic ducts and the tumor. A distal pancreatectomy with lymph node dissection was performed because the lesion was suspected to be a mucinous cystadenoma or cystadenocarcinoma of the pancreas. However, histological examination revealed that the cyst was lined by stratified squamous epithelium and surrounded by lymphoid tissue, thereby confirming the diagnosis of LEC of the pancreas. The superficial layer of squamous epithelium and the cystic contents were found to be immunohistologically positive for CA19-9. Establishing a preoperative diagnosis of LEC is quite difficult because it resembles other cystic neoplasms of the pancreas in radiographic features and is frequently associated with an elevation of serum tumor markers such as CA19-9. Received: June 21, 1999 / Accepted: March 24, 2000     

Mucinous cystadenocarcinoma of the pancreas diagnosed in postpartum

Background Cystic tumors of the pancreas are uncommon. They account for 10–15% of all pancreatic cystic masses and only 1% of pancreatic malignancies. Mucinous cystadenocarcinoma is the most frequent pancreatic cystadenocarcinoma and it is mainly seen in women, suggesting a sex hormone influence. Its presentation during pregnancy is infrequent and entails difficult diagnostic and therapeutic decisions. We report the case of a 31-year-old woman who presented a pancreatic cystadenocarcinoma 2 months after delivery. Materials and methods A 31-year-old woman was referred to our service because of abdominal pain and mass. She had given birth to her first child 2 months previous. Abdominal ultrasound demonstrated a poorly circumscribed cystic mass in the left upper abdominal quadrant, and the computed tomography scan showed a multilocular cystic lesion located in the body of pancreas. There was no seric alteration of specific pancreatic enzymes or tumor markers. Results Laparoscopic examination showed a large cystic tumor (12 × 11 × 5.5 cm) in the pancreas involving the body and the tail. It extended to the spleen and was highly vascularized, precluding a minimal invasive resection. An open body–tail pancreatectomy and splenectomy was performed. The resection margins were free of tumor, and the histological study showed a mucinous pancreatic cystadenocarcinoma with mucin-producing columnar epithelium and associated papillae patterns, reminiscent of ovarian stroma. Immunohistochemical studies were negative for hormonal receptors. The patient had no post-surgical complications and was discharged home in 4 days. Conclusions Cystic tumors of the pancreas are infrequent, and cancer of the pancreas during pregnancy is extremely rare. Insidious symptoms and bodily changes due to pregnancy may mask diagnosis. Aggressive surgery is currently the only chance of cure.     

胰腺囊性肿瘤的诊治98例报告

目的探讨胰腺囊性肿瘤的常见类型、诊断及合理手术方式的选择。方法对2000年1月至2008年12月期间收治98例胰腺囊性肿瘤患者的临床资料进行回顾性分析。结果本病无特征性的临床表现,B超、CT和Mill等影像学检查也能发现胰腺囊肿病灶但不能确定具体类型。本组胰腺囊腺瘤57例（浆液性囊腺瘤32例,黏液性囊腺瘤25例）,黏液性囊腺癌13例,实性假乳头状瘤19例,导管内乳头状黏液性肿瘤9例。所有患者均手术治疗,胰十二指肠切除26例,保留十二指肠胰头切除28例,胰腺节段切除术7例,胰体尾切除加脾脏切除25例,肿瘤摘除术12例。术后均进行随访,3例胰腺囊腺癌患者于术后6个月到4年期间死于癌转移或其他疾病,1例囊腺癌患者术后9个月肿瘤复发,再次手术后现仍生存,其他均存活。结论B超,CT和MRI等影像学技术是胰腺囊性肿瘤的主要诊断方法。手术切除为治疗该肿瘤的最有效手段。正确的诊断和合理手术方式的选择是提高其临床治愈率的关键。     

A case of retroperitoneal mucinous cystadenoma treated successfully by laparoscopic excision

A case of retroperitoneal mucinous cystadenoma treated laparoscopically is reported. A 26-year-old woman, who complained of abdominal fullness, was referred to us after a large intra-abdominal mass was discovered at her local hospital. Ultrasound and computed tomography showed a fluid-filled mass, 9 cm in diameter, in the left retroperitoneal space. Laparoscopic surgery was performed. We found a smooth and thin-walled cystic tumor that displaced the descending colon to the right and arose from the retroperitoneum, loosely adhering to the psoas muscle. We successfully extirpated the tumor laparoscopically. The surgical specimen consisted of a multilocular, red to yellowish-gray, cystic mass with a smooth, glistening external surface, and inner lining. The mass was filled with thin, yellowish fluid. The histologic diagnosis was benign mucinous cystadenoma. The postoperative course was uneventful, and the patient remains free of recurrence 15 months after surgery. Retroperitoneal mucinous cystadenoma is a rare entity, and the treatment of choice is surgical excision. Laparoscopic extirpation should be considered for the treatment of selected retroperitoneal cystic lesions.     

Giant multilocular cystadenoma of the prostate: a rare cause of huge cystic pelvic mass

Giant multilocular prostatic cystadenoma is a rare benign tumor that evolves from the prostate gland. Obstructive voiding symptoms occur in all reported cases. These lesions do not invade adjacent structures. Preoperative radiologic evaluation can define the benign nature of the lesion. Here we report a case of large cystic lesions identified by magnetic resonance imaging and sonographic findings that caused an extensive mass effect in the pelvis. When retrovesical, huge cystic lesions fill the pelvis completely in young men, with high levels of serum prostate-specific antigen, giant multilocular prostatic cystadenoma should be considered as a differential diagnosis. To our knowledge, this is the youngest case of prostatic cystadenoma reported in the literature.     

Solitary hepatic lymphangioma: Report of a case

A 52-year-old woman presented with upper abdominal pain. Abdominal ultrasonography showed a 4-cm well-defined mass containing solid and cystic components in segment IV of the liver, and contrast-enhanced T1-weighted magnetic resonance imaging revealed heterogeneous enhancement within the tumor, indicating a solid or fibrous component. There were no cystic lesions in any other organs. A partial hepatectomy was performed, based on a preoperative diagnosis of sclerosing hemangioma and biliary cystadenoma or cystadenocarcinoma. Pathologically, the tumor appeared to be a multilocular and cystic lesion lined by attenuated endothelial- like cells with no atypia. Immunohistochemistry demonstrated the endothelial-like cells to be positive for the lymphatic-specific markers D2-40, LYVE-1, and Prox-1, which proved helpful for confirming the diagnosis as solitary hepatic lymphangioma. This case is presented with details of the pathologic and radiologic findings, because solitary hepatic lymphangioma is an extremely rare tumor and no previous reports have provided details of the immunohistochemical characteristics.     

A case of papillary cystadenoma of the epididymis mimicking a testicular tumor

A 68-year-old male presented with painless left scrotal enlargement of one year duration. Ultrasound, computed tomography (CT) and magnetic resonance imaging showed a multilocular cystic mass, 10×7.5× 8.5 cm in size, in the left scrotum. The intracystic fluid was partially hemorrhagic. A solid part of the tumor, seen at the base of the scrotum, was partially calcified and was enhanced by contrast medium. The left testis could not be identified by imaging studies. Although CT imaging showed a simple cyst in the right kidney, no other lesions in the kidneys, adrenal glands, pancreas or the central nervous system were detected. Serum tumor marker values for testicular cancer were within the normal range. Under the pre-operative diagnosis of a left testicular tumor, left high orchiectomy was performed. Grossly the specimen consisted of a multilocular cystic tumor, 12.5×8.5×8.5 cm in size, with a 2.7 cm tan-colored solid component within the wall of the cyst. The left testis was atrophic, 1.3 cm in size, and demonstated no continuity with the solid part of the tumor. Histologically, the solid component of the tumor showed tubular and papillary growth of cuboidal and columnar tumor cells with clear cytoplasm. Histopathological diagnosis of papillary cystadenoma of the epididymis (PCE) was made. Von Hippel-Lidau disease was ruled out by subsequent genetic analysis. After follow up for 18 months, there was no sign of recurrence. To our knowledge, this is the 33rd and the largest case of PCE reported in Japan.     

Hepatic endometrioma: a case report and review of the literature: report of a case

Hepatic endometriosis has an extremely rare occurrence characterized by the presence of ectopic endometrium in the liver. A diagnosis of hepatic endometriosis is established after surgery. A 51-year-old multiparous female was referred to our unit for investigation of a liver tumor. The patient reported a 6-month history of epigastric pain and vomiting. She had undergone conservative hysterectomy for uterine leiomyomas several years earlier. The results of liver function tests and the levels of tumor markers (CA 19.9, CEA, CA125, αFP) were normal. Radiological imaging (USS, CT and MRI) suggested the presence of liver cystadenoma, liver cystadenocarcinoma or cystic metastasis of the liver in the left liver lobe extending to the diaphragm with left hepatic vein compression. Laparotomy was performed. The intraoperative frozen sections suggested a diagnosis of endometriosis. Anatomical resection was performed, including left lobectomy with diaphragm resection. The final histology confirmed the presence of hepatic endometrioma without malignant transformation. Fourteen cases of hepatic endometrioma have been described in the medical literature. We herein report the 15th case. Making a preoperative diagnosis of hepatic endometriosis is very difficult, despite conducting a complete investigation, in the absence of clinical and radiological characteristics. The diagnosis is made according to a histological examination of the whole surgical sample.     

胰腺囊性肿瘤40例诊疗分析

目的探讨胰腺囊性肿瘤的诊治方法。方法对笔者所在医院科室2001年10月至2013年10月期间收治的40例胰腺囊性肿瘤患者的临床资料进行回顾性分析。结果胰腺囊性肿瘤无特殊临床表现,B超和CT检查对胰腺囊性肿瘤的诊断正确率分别为57．5％（23／40）和72．5％（29／40）,但不能准确区分其组织学类型。40例患者均行手术治疗,其中2例患者误诊为假性囊肿而行内引流术,另外38例行胰体尾切除术。术后病理学检查证实浆液性囊腺瘤23例,黏液性囊腺瘤9例,导管内乳头状黏液性腺瘤3例,黏液性囊腺癌5例。5例失访,35例患者获随访,随访时间为（74．2±12．8）个月（2个月～8年）;3例囊腺癌患者中1例肿瘤切除者至今存活（已随访8年）,2例肿瘤未切除者分别于术后4个月和7个月因肿瘤转移死亡;其余32例获访的囊腺瘤患者均存活至今。结论外科切除是治疗胰腺囊性肿瘤最有效的手段,即使是对于无任何症状的患者也应行积极的手术治疗。     

Giant mesenchymal hamartoma of the liver in an adult

We report a case of hepatic mesenchymal hamartoma in an adult; this condition is extremely rare, with only 15 cases having been reported in the English-language literature worldwide. The patient was a 36-year-old woman who was seen at her local hospital for upper abdominal distension. A giant multilocular cystic tumor, which had almost entirely replaced the normal parenchyma of the right lobe of the liver, was diagnosed. She was referred to our hospital, where, with a diagnosis of biliary cystadenoma, the tumor was successfully removed by right hemihepatectomy. After an uneventful postoperative course, the patient was discharged from our hospital. On histological examination, the tumor consisted of numerous cystic lesions without epithelial lining cells; hepatocytes, bile duct, and vascular components, without either lobular structure or atypia, were observed in the pseudocyst wall, leading to a diagnosis of hepatic mesenchymal hamartoma. There have been a few previously reported cases of multifocal hepatic mesenchymal hamartoma reappearing in the remaining liver after hepatectomy, although these cases are considered to be extremely rare. Therefore, periodic follow-up will be necessary for the patient.     

Giant biliary cystadenoma: case report and literature review

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 cystadenocarcinoma.     

A case of papillary cystadenocarcinoma of the prostate

A 63-year-old man presented with dysuria. Ultrasonography revealed a cystic intravesical mass. During needle aspiration we aspirated bloody fluid. The result cytology was class II. We gave medication on an outpatient basis, but symptoms became worse. We performed transurethral resection (TUR) of prostate. Histopathological examination of the TUR specimen revealed a papillary cystadenocarcinoma of the prostate. We diagnosed the tumor as a stage B prostate cancer, and performed total prostatectomy. Histopathological examination of prostate revealed no residual cancer. Eleven cases of papillary cystadenocarcinoma of the prostate in the Japanese literature are reviewed briefly.     

A case of prostatic cystadenoma

We report a case of prostatic cystadenoma. A 81-year-old man visited our hospital with the chief complaint of urinary difficulty. RUG and DIP showed bladder and prostatic urethra deviated to the left side. TRUS, CT and MRI revealed multilocular cyst in the pelvic cavity. Resection of the cystic wall was performed under the diagnosis of pelvic cyst. The cysts were originated from the right side of the prostate and adhered with the bladder severely. The histopathological diagnosis was prostatic cystadenoma. Fortyfive cases of prostatic cyst in the Japanese literature are reviewed.