Bilateral submandibular gland nodular oncocytic hyperplasia with papillary cystadenoma-like areas.

A 57-year-old man presented in 2016 with a 4-month history of a right submandibular mass, having undergone left submandibular gland (SMG) excision in 2003. Imaging suggested a benign tumour and subsequent core biopsy findings suggested a nodular oncocytic hyperplasia, similar to the tumour removed from the contralateral side. This was confirmed on histological analysis following right submandibular gland excision which showed characteristic features of nodular oncocytic hyperplasia along with an unusual diffuse papillary cystadenoma-like ductal proliferation, similar to that seen in the 2003 specimen. A diagnosis of multinodular adenomatous oncocytic hyperplasia (MAOH) was rendered in order to communicate the unique histological features that have otherwise not been described in the literature. We believe that this is the first reported case of non-synchronous multinodular oncocytic hyperplasia and the first case affecting the submandibular glands.

Adolescent Hydrocele Carrying a Surprise: A Case of Papillary Cystadenoma of the Epididymis.

Papillary cystadenoma of the epididymis (PCE) is a rare benign epithelial tumor remarkable for its association with von Hippel-Lindau disease. A 12-year-old boy consulted for a progressive enlargement of the left testicle. At time of surgery, the whole epididymis was enlarged. Pathologic diagnosis was PCE with a focus on borderline malignancy. Scrotal left epididymectomy was performed. von Hippel-Lindau disease screening was negative. No relapse has been detected 2 years later. In case of atypical clinical examination of a hydrocele, unusual presentations such as PCE should be considered. The main differential diagnoses were adenomatoid tumor, nonpapillary cystadenoma, and metastatic clear cell renal carcinoma.

Oncocytic papillary cystadenoma with prominent mucinous differentiation of parotid gland: A case report.

We describe the case of an oncocytic papillary cystadenoma with mucinous differentiation of the parotid gland in a 64-year-old male. Histologically, the tumor exhibited distinctive areas of intracystic papillary growth pattern with microcystic and macrocystic spaces containing mucinous secretions and small crystals. The cyst wall and papillary fronds were lined by oncocytic admixed with numerous mucocytes. Lymphoid tissue and invasive features were not identified. The tumor showed strong expression of CK7 and mammaglobin in oncocytes, and BRST-2 and MUC4 in mucocytes. p63, ER, PR, SOX10, DOG-1, and S100 stains were negative. No rearrangement of the MAML2 gene region or ETV6-NTRK3 fusion transcript was detected. The diagnosis of oncocytic papillary tumor with prominent mucinous differentiation is particularly problematic owing to the large number of potential mimics and should prompt consideration of appropriate molecular studies.

Laparoscopic Anatomical Left Hepatectomy for Intrahepatic Bile Duct Papillary Mucinous Cystadenoma With Intraoperative Vascular Repair: A Case Report.

Laparoscopic hepatectomy has been widely performed for patients with benign liver tumors such as hepatic hemangioma, focal nodular hyperplasia, and hepatic adenoma.We here present a case of a 78-year-old female patient who was initially admitted to our department due to fever and jaundice for 2 days. Abdominal enhanced computed tomography scan showed intrahepatic and extrahepatic bile duct dilatation with liver atrophy of left lobe. Unenhanced nodules were seen within the left intrahepatic bile duct. Ultrasonography revealed intrahepatic and extrahepatic bile duct dilatation with viscous fluid, tubular adenoma? Tumor markers including alpha fetoprotein, carcinoembryonic antigen, and CA19-9 were normal. Preoperative total bilirubin was 64.4 mmol/L.Laparoscopic anatomical left hepatectomy and common bile duct exploration were performed. In this procedure, a lot of mucus was seen within the common bile duct and left intrahepatic bile duct. No bile duct stones were found after the exploration. During parenchymal transection, intraoperative hemorrhage from middle hepatic vein was met, and we repaired middle hepatic vein by laparoscopic suture (5-0 Prolene). No air embolism and hypotension were met. This operation took 232 minutes and estimated blood loss was 300 mL. Postoperative ultrasonography indicated a normal outflow of middle hepatic vein and there was no stricture. The patient's postoperative course was uneventful and was discharged on the 6th day after surgery. Postoperative pathological diagnosis was intrahepatic bile duct papillary mucinous cystadenoma.

Serous papillary cystadenofibroma of the fallopian tube: A case report and short review of literature.

Serous papillary cystadenofibromas (SPCAFs) of the fallopian tube are very rare benign tumors of the female genital tract. They are usually asymptomatic and are found incidentally. Until now, only 18 cases of this tumor have been reported in the world literature. We report a case of SPCAF of the left fallopian tube in a 30-year-old female who presented with a large abdominal mass and pain. On computed tomography, a diagnosis of ovarian neoplasm was given. However, during surgery the tumor was found to arise from the fallopian tube and was treated with tubal cystectomy with sparing of the ovary. We present this unique case on account of its rarity, unusual presentation, and huge size along with a short review of literature.

Papillary cystadenoma of anterior one-third of the tongue.

Papillary cystadenoma is an unusual benign cystic neoplasm with cystic spaces of diverse sizes and intraluminal papillary projections histologically, which is commonly seen in the lips, cheek and palate. We report a case of papillary cystadenoma in a 40-year-old man on the anterior one-third of the tongue with classical histological features. The paper highlights the rarity of the site of occurrence with emphasis on differential diagnosis and the need for considering papillary cystadenoma when benign cystic lesions are encountered in the anterior one-third of the tongue.

Syringocysadenoma papilliferum of the vulva: a rarity in gynaecology.

A case of syringocystadenoma papilliferum of the vulva in a 36-year-old woman is reported .The patient presented with a single cystic lesion on the left labia minora of 8 years duration with a recent increase in size and redness around the lesion. Examination revealed a polypoid cystic lesion with no regional lymphadenopathy. An excision biopsy was performed under general anaesthesia and the specimen was subjected to histological examination. Histopathology revealed closely excised syringocystadenoma papilliferum. On follow-up, the patient was asymptomatic and the wound had healed well.

Intraparenchymal serous papillary cystadenoma of the testis: a case report.

A case is presented of a 58-year old man with a double multilocular cystic intratesticular tumour exhibiting the morphological features described by the WHO for diagnosis of a serous papillary cystadenoma of the ovary. We classified this tumour as the male analogue of a respective ovarian growth.

Primary mucinous cystadenoma of the spermatic cord within the inguinal canal.

We report a hitherto not documented case of primary mucinous cystadenoma arising in the spermatic cord within the right inguinal canal of a78-year-old man. The tumor was painless, hard and mobile. A computed tomography scan on the pelvis revealed an oval shaped, low attenuation mass, measuring 5.0x2.5x2.1 cm, that was present adjacent to the vas deferens. Grossly, the excised mass was multicystic mucinous tumor, filled with thick mucoid materials. Microscopically, the cystic wall was irregularly thickened. The cystic epithelium commonly showed short papillae lined by a single layer of columnar to cuboidal mucinous epithelial cells without significant stratification or cytologic atypia. Goblet cells were also frequently present. Immunohistochemically, the neoplastic cells showed positive reaction to carcinoembryonic antigen, cytokeratin 20, CDX2, epithelial membrane antigen, and CD15. However, they were negative for PAX8 and Wilms' tumor 1 protein. Pathological diagnosis was a papillary mucinous cystadenoma of the spermatic cord. Although mucinous cystadenoma in this area is extremely rare, it is important that these lesions be recognized clinically and pathologically in order to avoid unnecessary radical surgery. VIRTUAL SLIDES: The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1720965948762004.

Solid pseudopapillary tumor of the pancreas: a single-institution 20-year series of pediatric patients.

PURPOSE: Solid pseudopapillary tumor (SPT) of the pancreas is a rare neoplasm. The objective of this study was to review our institution's experience and provide an update on current management in the pediatric population. METHODS: Our pathology database identified all patients with SPT for a 20-year period (1991-2011). Demographics, clinical characteristics, operative details, pathology, and outcomes data were retrospectively reviewed. RESULTS: Eleven patients with SPT were identified. Most were female and Hispanic. Median age at resection was 14 years (9-17 years). Most patients presented with abdominal pain. Diagnostic imaging was most commonly an ultrasound or computed tomography. All tumors were resected en bloc. Median greatest tumor diameter was 5 cm (3.5-12 cm). Median length of stay was 8 days (5-19 days). Complications included pancreatic leak, chyle leak, delayed gastric emptying, fat malabsorption, and incisional keloid. Recurrence developed after 2.5 years in 1 patient with positive surgical margins. There were no metastases or deaths. Median follow-up was 1.4 years (0.6-5.9 years). CONCLUSION: This pediatric series of SPT from a single institution corroborates previous reports in the literature. In our experience, SPT behaves like a low-grade malignancy and has an excellent prognosis. Surgical resection is dictated by tumor location and remains the treatment of choice.

Proper management and follow-up strategy of branch duct intraductal papillary mucinous neoplasms of the pancreas.

BACKGROUND AND AIM: It has been reported that main duct intraductal papillary mucinous neoplasms are more invasive and have a worse prognosis than branch duct intraductal papillary mucinous neoplasms. Therefore, an aggressive surgical approach has mainly been recommended for all MD-IPMNs. However, the surgical management of BD-IPMNs has been controversial and the consensus guidelines are not specific for an indicator of malignancy in BD-IPMNs. The objective of this study was to determine the proper management and follow-up strategy of BD-IPMNs. METHODS: We monitored and analysed patients with presumed BD-IPMNs between March 1995 and March 2010. RESULT: The mean value of the initial cyst size in all patients with BD-IPMNs was 2.19 cm. Amongst 194 patients with BD-IPMNs, 34 underwent immediate surgical resection, 152 were followed conservatively. Amongst the 152 conservatively managed patients, 18 (11.8%) underwent surgical resection after a median follow-up of 12.7 months (range, 3-48 months). In 132 patients who were managed conservatively without surgery, the mean incremental rate of cyst size growth was 0.0038 cm/month during a median of 30.7 months of follow-up and there were no IPMN-related deaths. CONCLUSION: Amongst patients with BD-IPMNs, about 10% have surgery within approximately 1 year from the time of diagnosis because of the occurrence of new malignant stigmata. Therefore, a conservative approach without surgery and careful follow-up every 3 months or 6 months during the first year after diagnosis can be safely advocated in patients with BD-IPMNs larger than 10mm in size who have no risk factors for malignant IPMNs.

Role of laparoscopic distal pancreatectomy for solid pseudopapillary tumor.

BACKGROUND: Since the first case report regarding laparoscopic distal pancreatectomy (DP) for solid pseudopapillary tumor (SPT), few additional articles have been published. The objective of this study was to evaluate the feasibility, safety, and long-term outcome of the laparoscopic DP based on a series of adult SPT patients. METHODS: In a single-center study, we screened all adult patients undergoing a laparoscopic DP for SPT. Preoperative, operative, and postoperative data were retrospectively analysed and compared to the results of open DP for SPT published in the medical literature. RESULTS: From April 2000 to June 2010, 5 adult female patients (median age 34 y) underwent a laparoscopic DP for an SPT. No conversion to open surgery was required. The median size of the tumor was 45 mm. The postoperative mortality rate was 0%, and serious complications (Dindo IV) occurred in 2 patients. The postoperative quality of life was not significantly altered by the laparoscopic procedure. At a median follow-up of 60 mo, all patients were alive and without evidence of local recurrence, distant metastasis, diabetes, or exocrine insufficiency. CONCLUSION: Laparoscopy may offer an alternative to open surgery in the treatment of SPT of the distal pancreas in adult female patients. The laparoscopic procedure impacts neither the oncologic outcome nor the quality of life. However, due to the risk of postoperative complications, this procedure should be reserved for specialized centers.

Papillary cystadenofibroma of epididymis: a case report.

We present the first reported case of papillary cystadenofibroma of the epididymis. The tumor occurred in a 46-year-old man. The mass was 3.7 cm and included a hemorrhagic fluid-filled cyst. Microscopically, stromal-filled papillae were lined by low cuboidal to columnar epithelium. Epithelial cells were reactive for cytokeratin 7, cytokeratins AE1/3, and focally in the apical cytoplasm for CD10. Focal CD10 reactivity was also noted in the stroma. The lesion was negative for alpha-fetoprotein. These findings ruled out other lesions, including metastatic renal cell carcinoma.

Pancreatic true cysts--diagnosis and treatment difficulties.

Pancreatic true cysts represent a rare, heterogeneous group of pancreatic tumors; therapeutic strategy is based on patient's general status, cyst topography, and especially the estimated risk of malignancy. This paper aim is to present 7 cases of pancreatic true cysts, operated on a six years period (January 2004-January 2010) in our surgical clinic: 2 men and 5 women, aged between 24-61 years old; cyst diameter varies between 3.5-15 cm, tumor location being pancreatic head in two cases and the distal pancreas in 5 cases. Surgical treatment consisted in cyst enucleation (two cases), splenopancreatectomy (three cases), duodenopancreatectomy (one case), and subtotal splenopancreatectomy (one case). Histology was represented by serous cystadenoma (one case), mucinous cystadenoma (2 cases), intraductal papillary mucinous cystadenoma (one case), and papillary cystadenocarcinoma (3 cases).Postoperative results were good in all cases, with 3 postoperative pancreatic external fistulas, resolved conservatory; no case of post-pancreatectomy diabetes mellitus was registered. In conclusion, surgical removal of the pancreatic cystic tumors is necessary, especially due to the risk of malignancy, at least in the absence of rigorous histological proofs of benignancy. Postoperative results are favorable in terms of postoperative morbidity and mortality.