Apocrine cystadenoma: A long-standing apocrine hidrocystoma with an adenomatous proliferation.

BACKGROUND: Apocrine cystadenoma is a rare, benign adenomatous cystic neoplasm, the pathogenesis of which is not fully understood. We sought to characterize the clinical, dermatoscopic, and histopathologic features of apocrine cystadenoma and its relationship to hidrocystoma. METHODS: We retrospectively analyzed cases of apocrine cystadenoma and hidrocystoma retrieved from the dermatopathology laboratory information system. RESULTS: Of the 350 cases apocrine cystic lesions, 13 cases of apocrine cystadenomas met the inclusion criteria. The age ranged from 20 to 84 years with an average of 64 years. They were long-standing (duration 3-15 years), slow-growing, large tumors usually found on the scalp. Dermatoscopy accentuated translucent light to dark blue color and prominent vessels that were present more at the periphery. All lesions were multilocular with columnar to cuboidal lining and decapitation secretion. A large portion of the lesion consisted of a simple nonproliferative epithelial lining, identical to that observed in apocrine hidrocystomas, while the proliferative adenomatous component made up a smaller portion with two patterns: (1) tubular proliferation, which either protruded into the cystic cavity or expanded outward peripherally, or (2) papillary projections, which were multiple layers thick with fibrovascular core, sometimes accompanied by tubular proliferation. Immunohistochemical stains showed strong staining for p40 and a sparse number of cells stained for Ki-67 and p53. CONCLUSIONS: The long duration of the lesion and the large areas of simple apocrine epithelial lining suggest that apocrine cystadenomas arise from long-standing apocrine hidrocystomas. However, the retrospective nature of the study from a single institution is a limitation.

Caroli's disease misdiagnosed as bile duct cystadenoma: A case report.

Caroli's disease is also known as Congenital intrahepatic bile duct dilatation, and previously known as a congenital intrahepatic bile duct cyst; it is characterized by single or multiple intrahepatic cystic dilatations. In this article, we report a case of Caroli's disease (CT size 21.2 × 16.9 × 19.8 cm). Preoperative abdominal ultrasound and enhanced CT were misdiagnosed as biliary cystadenoma or hepatic echinococcosis, and finally diagnosed as Caroli's disease by postoperative histopathological examinations. Most of the disease is single or multiple cystic dilatation of small bile duct. Giant Caroli disease, cystic dilations with diameter >20 cm is very rarely seen in the clinic. The lack of experience of diagnosing giant cystic dilatation makes it difficult to make accurate diagnosis. Therefore, we analyze the causes of imaging misdiagnosis through this case report, and summarize the imaging diagnostic skills of the disease combined with relevant imaging diagnosis experience. The purpose of this study is to deepen the understanding of giant Caroli disease among imaging doctors so as to reduce the misdiagnosis of the disease in the future.

A CT-based radiomics nomogram for differentiating ovarian cystadenomas and endometriotic cysts.

AIM: To construct and validate a computed tomography (CT)-based radiomics nomogram integrating radiomics signature and clinical factors to distinguish ovarian cystadenomas and endometriotic cysts. MATERIALS AND METHODS: A total of 287 patients with ovarian cystadenomas (n=196) or endometriotic cysts (n=91) were divided randomly into a training cohort (n=200) and a validation cohort (n=87). Radiomics features based on the portal venous phase of CT images were extracted by PyRadiomics. The least absolute shrinkage and selection operation regression was applied to select the significant features and develop the radiomics signature. A radiomics score (rad-score) was calculated. The clinical model was built by the significant clinical factors. Multivariate logistic regression analysis was employed to construct the radiomics nomogram based on significant clinical factors and rad-score. The diagnostic performances of the radiomics nomogram, radiomics signature, and clinical model were evaluated and compared in the training and validation cohorts. Diagnostic confusion matrices of these models were calculated for the validation cohort and compared with those of the radiologists. RESULTS: Seventeen radiomics features from CT images were used to build the radiomics signature. The radiomics nomogram incorporating cancer antigen 125 (CA-125) level and rad-score showed the best performance in both the training and validation cohorts with AUCs of 0.925 (95% confidence interval [CI]: 0.885-0.965), and 0.942 (95% CI: 0.891-0.993), respectively. The accuracy of radiomics nomogram in the confusion matrix outperformed the radiologists. CONCLUSIONS: The radiomics nomogram performed well for differentiating ovarian cystadenomas and endometriotic cysts, and may help in clinical decision-making process.

Robot-Assisted Extraperitoneal Radical Prostatectomy for Giant Multilocular Prostatic Cystadenoma: A Case Report and Literature Review.

A 48-year-old man with a medical history of hypertension and no family history of prostate cancer presented with abdominal distension, lower abdominal pain, and lower urinary symptoms. Physical examination revealed a palpable mass in the lower abdomen, and a digital rectal examination detected a firm mass on the anterior side of the rectum. Laboratory tests showed an elevated PSA level (7.9 ng/mL). Imaging studies indicated a solid mass connected to the prostate's posterior and rectum's anterior walls, along with bladder compression. Transperitoneal biopsy and histological analysis led to a diagnosis of a stromal tumor with uncertain potential malignancy. Considering the absence of apparent malignancy signs and the smooth outer wall of the tumor, the patient underwent, for the first time in the literature, a robot-assisted radical extraperitoneal prostatectomy for complete macroscopic resection. The surgery involved excision of the bulky pelvic mass, preservation of the urethra, and anatomical reconstruction. The postoperative course was uneventful, and we discharged the patient with no complications. The pathological examination documented the diagnosis of multilocular prostatic cystadenoma. Post-surgery follow-up examinations, including PSA levels and imaging scans, showed no signs of tumor recurrence. At the 3-, 6-, and 9-month follow-ups, the patient was asymptomatic and had fully recovered, with no urinary or sexual dysfunction reported.

Apocrine Cystadenoma of the Eyelid, a Rare Neoplasm: Expanded Immunohistologic Profile.

An 83-year-old woman experienced the slow enlargement of a right lower eyelid mass. Histopathologic examination of the excised tissue showed a mucin-filled cystic tumor emanating from an apocrine bilayer that displayed bleb-like apocrine decapitation secretion. The outer flattened myoepithelial layer of the bilayer reacted with immunohistochemical stains for smooth muscle actin and calponin. In foci, the tumor exhibited a cribriform architecture with small pockets of mucin. Tumor cells were reactive for cytokeratin 7, Gross Cystic Disease Fluid Protein 15 (BRST-2), estrogen and progesterone receptors, androgen receptors, mammaglobin, epithelial membrane antigen, and GATA3. Ki67 showed a very low proliferation fraction. The lesion exemplifies the fourth instance of an eyelid apocrine cystadenoma in the literature.

METABOLOMIC MARKERS OF ENDOMETRIOSIS: PROSPECTS.

Endometriosis is a widespread pathology among women of reproductive age. The pathophysiological mechanisms of the disease aren't enough understood yet. In addition, the "gold" standard of diagnosis is still laparoscopy. Worldwide, patients may experience a delay in the diagnosis of endometriosis by approximately 6 to 12 years. Aim of the study - to conduct a systematic analysis of the data presented in modern literature on the metabolomic diagnosis of endometriosis in general and endometrioid cystadenomas in particular. The review includes data from world studies over the past 7 years regarding metabolomic screening for endometriosis. Metabolomic changes characteristic of endometriosis, noted in the metabolism of amino acids, organic acids, lipids, purines, are presented. The described disorders reflect the processes of oxidative stress, mitochondrial dysfunction, endothelial dysfunction, and active angiogenesis. The identified metabolomic changes may improve and speed up the process of diagnosing endometriosis in general and endometrioid cystadenomas in particular in a non-invasive way. Certain detailed violations of metabolic processes can become a promising point of application for the correction of symptoms and the treatment of this pathology.

Sex cord stromal tumors and tumors of the paratestis: new and old entities in a landscape of rare tumors.

PURPOSE OF REVIEW: The 5th edition of WHO classification incorporates the most relevant new data available in the literature regarding tumors of the male genitourinary tract. In this review, the authors summarize and critically discuss the most relevant new information regarding tumors occurring in the stromal testis and in the paratestis that will be reported in the new edition of WHO classification of tumors of the male genitourinary tract. RECENT FINDINGS: Signet-ring stromal tumors (SRST) and myoid gonadal stromal tumors (MGST) are two new entities brought in the 5th WHO classification of testicular tumors. All cases of SRST and MGST reported so far have behaved in a benign fashion after resection and whenever possible a conservative surgery is recommended. A future perspective is to aim at creating large multiinstitutional case series to link different morphologic patterns and molecular bases to the biologic behavior of these neoplasms. Another innovation in WHO consists in the inclusion in the group of Sertoli cell tumors of the sertoliform cystadenoma. The sertoliform cystadenoma is localized in the rete testis and it is of unknown origin. It was included in the group of gonadal stromal tumors because of a high morphological and immunohistochemical similarity to the Sertoli cell tumor. SUMMARY: Although further studies with long-term follow-up are needed to estimate the main oncologic outcomes in patients with rare gonadal stromal tumors, we highlight the importance of an accurate characterization by molecular and immunohistochemical assays of these entities.

Giant Multilocular Prostatic Cystadenoma in a 14-Year-Old Male: A Case Report of a Pediatric Pelvic Mass.

Giant multilocular prostatic cystadenoma (GMC) is an extremely rare, benign tumor seen in both adult and pediatric males. The neoplasm originates from prostatic tissue and is typically found within the rectovesical pouch, varying in both size and morphology. Microscopically, GMC contains both glandular and cystic prostatic tissue lined by cuboidal and columnar epithelium. Symptoms often arise once the pelvic mass begins to obstruct the surrounding structures and organs, although invasion into surrounding tissue is unlikely. Common symptoms include abdominal pain, urinary retention, and dysuria. The standard treatment for GMC is surgical removal of the mass with good outcomes and only 1 known case of recurrence. Here we present the case of a 14-year-old male with GMC-the youngest patient reported to date-who presented with abdominal pain, difficulty voiding, and hydroureteronephrosis.

Two-Port Laparoscopic Removal of Large Ovarian Cystadenoma.

An ovarian mucinous cystadenoma is a common benign tumour of the ovary that tends to reach very large sizes. Although traditional morcellation in the abdominal cavity is largely avoided in gynaecologic surgery, several authors have proposed other systems and techniques for the removal of large masses without resorting to laparotomy. We proposed an extremely minimally invasive technique to remove a large mass with a very low suspicion of malignancy, and created a video demonstration of the procedure. In this short film we illustrate our novel technique using only 2 laparoscopic ports, which maximizes both cosmesis and speed of recovery. The technique is not a completely closed system, therefore the potential for spreading an undiagnosed malignancy still exists. Accordingly, the authors do not suggest this technique for masses with a high potential for malignancy. However, in properly consented patients where there is very little suspicion for malignancy, our technique may be a reasonable option to promote fast recovery and provide excellent cosmesis.

Ovarian Cystadenomas: Growth Rate and Reliability of Imaging Measurements.

OBJECTIVES: To evaluate the growth rate of benign ovarian cystadenomas and the degree of variability in ultrasound measurements. METHODS: Two independent retrospective cohorts of women found to have benign cystadenomas at surgery were identified. To assess growth rate, ultrasounds on women in a community-based health system were reviewed and the growth rate was determined based on the maximum reported size dimension using a mixed effect model. To assess measurement variability, two radiologists independently measured presurgical adnexal imaging findings for women in a tertiary care referral setting. Interobserver, intra-observer, and intermodality (cine clip versus still images) variability in measurements was determined using correlation coefficients (CC) and Bland-Altman analysis, with the proportion of measurements varying by more than 1 cm calculated. RESULTS: For growth rate assessment, 405 women with 1412 ultrasound examinations were identified. The median growth rate was 0.65 cm/year with mucinous cystadenomas growing faster at 0.83 cm/year compared to 0.51 cm/year for serous cystadenomas (median test P < .0001). To evaluate measurement variability, 75 women were identified with 176 ultrasound studies. The within-subject standard deviations for ultrasound measurements were 0.74 cm for cine clip images and 0.41 cm for static images, with 11% of measurements overall differing by more than 1 cm. CONCLUSIONS: Cystadenomas grow on average 0.65 cm/year, which is similar in magnitude to the inherent error observed in measurement on ultrasound, suggesting that repeat ultrasound at intervals of longer than a year will often be needed to accurately assess growth if a cyst represents a benign cystadenoma.

Primary spindle cell sarcoma of the seminal vesicle: A case report and literature review.

We report a case of primary seminal vesicle spindle cell sarcoma of a 57-year-old man who underwent multiple surgical treatment. The first diagnosis of a local hospital was a right seminal vesicle cyst, so only laparoscopic decompression was performed. Postoperatively, the patient gradually developed lower abdominal discomfort, frequent and urgent urination, dysuria and constipation. Digital rectal examination palpated a heterogeneous mass. Magnetic resonance imaging showed a multilocular cystic mass of about 4.5 cm in diameter in the right seminal vesicle, which was diagnosed as a recurrent cyst. The patient underwent a second operation in our hospital, but the tumour could not be completely removed because of severe peripheral adhesions. The postoperative pathological diagnosis was seminal vesicle cystadenoma with spindle cell sarcoma. One month later, a computed tomography scan performed at another hospital showed that the mass had invaded the bladder and sigmoid colon. The pathological diagnosis of re-examination was spindle cell liposarcoma. After neoadjuvant chemotherapy, extended resection of the tumour was performed, and adjuvant chemotherapy was continued after surgery. The total duration of follow-up was 19 months and 3 months after the third surgery. The patient survived with no recurrence or metastasis.

Mucinous cystadenoma of the liver with pathological-radiological correlation.

The paper presents a pathological-radiological correlation of the manifestation of mucosal cystadenoma with ovarian stroma of the liver with examination and correlation with the new stroma nomenclature and differential diagnostic dilemmas of radiologists and pathologists.

Laryngeal oncocytic cystadenoma and sudden death.

An 86-year-old woman with Alzheimer disease collapsed in her nursing home and was not able to be resuscitated. At autopsy, the major findings were in the larynx where a pedunculated oncocytic cystadenoma had occluded the glottis. Oncocytic cysts or cystadenomas of the larynx are rare histologically benign lesions that account for only 0.1-1% of laryngeal lesions. While the usual presentation is of a sensation of a mass in the throat, hoarseness, or stridor, very occasionally, there may be acute airway compromise and sudden death. Oncocytic cystadenoma should, therefore, be included in the differential diagnosis of potentially lethal obstructive laryngeal lesions.

[A Case of Giant Multilocular Prostatic Cystadenoma Resected by Laparoscopic Pelvic Cystectomy : A Case Report].

A 60-year-old man visited our hospital to treat a large cystic mass in the pelvis which had been found by abdominal ultrasonography in December 201X. Computed tomography (CT) and magnetic resonance imaging (MRI) showed a multilocular cyst with a maximum diameter of about 10 cm. CT-guided drainage and sclerotherapy with minocycline reduced the size of tumor by 40%, but symptoms such as difficulty of defecation and urinary frequency appeared a year and a half later due to re-enlargement of the cysts. Laparoscopic resection of the multilocular cysts was performed, and all cysts were removed almost completely using transrectal ultrasonography. The multilocular cyst was positive for NKX3.1 by immunohistochemical staining, and was diagnosed as a giant multilocular prostatic cystadenoma. After surgery, the symptoms such as difficulty of defecation and urinary frequency were relieved promptly. One year after the surgery, the patient was free from recurrence of the disease.

Ovarian seromucinous cystadenomas and adenofibromas: first report of a case series.

AIMS: To report a series of benign ovarian seromucinous neoplasms, an uncommon and hitherto poorly described category of tumours included in the current 2014 World Health Organisation classification of tumours of the female reproductive organs. METHODS AND RESULTS: We report the clinicopathological features of a series of 22 benign ovarian seromucinous neoplasms (cystadenomas and adenofibromas or admixtures). The neoplasms occurred in patients aged 32-83 years (mean = 62, median = 65.5) and involved the left ovary (n = 14), the right ovary (n = 6) or both ovaries (n = 2). There was a common association with endometrioid elements (endometrioid differentiation within the cystadenoma/adenofibroma and/or endometriosis) and other endometriosis-associated neoplasms. CONCLUSIONS: We speculate that some of these represent benign ovarian endometrioid neoplasms with foci of mucinous and/or serous differentiation, while others represent true mixed neoplasms.

Factors Associated with Torsion in Pediatric Patients with Ovarian Masses.

BACKGROUND: Management of ovarian torsion has evolved toward ovarian preservation regardless of ovarian appearance during surgery. However, patients with torsion and an ovarian neoplasm undergo a disproportionately high rate of oophorectomy. Our objectives were to identify factors associated with ovarian torsion among females with an ovarian mass and to determine if torsion is associated with malignancy. METHODS: A retrospective review of females aged 2-21 y who underwent an operation for an ovarian cyst or neoplasm between 2010 and 2016 at 10 children's hospitals was performed. Multivariate logistic regression was used to assess factors associated with torsion. Imaging data were assessed for sensitivity, specificity, and predictive value in identifying ovarian torsion. RESULTS: Of 814 girls with an ovarian neoplasm, 180 (22%) had torsion. In risk-adjusted analyses, patients with a younger age, mass size >5 cm, abdominal pain, and vomiting had an increased likelihood of torsion (P < 0.01 for all). Patients with a mass >5 cm had two times the odds of torsion (odds ratio: 2.1; confidence interval: 1.2, 3.6). Imaging was not reliable at identifying torsion (sensitivity 34%, positive predictive value 49%) or excluding torsion (specificity 72%, negative predictive value 87%). The rates of malignancy were lower in those with an ovarian mass and torsion than those without torsion (10% versus 17%, P = 0.01). Among the 180 girls with torsion and a mass, 48% underwent oophorectomy of which 14% (n = 12) had a malignancy. CONCLUSIONS: In females with an ovarian neoplasm, torsion is not associated with an increased risk of malignancy and ovarian preservation should be considered.

Multifocal giant apocrine cystadenomas in a miniature poodle.

Apocrine cystadenomas have rarely been reported in dogs. Herein, a dog with multiple, large (1.0-7.0 cm) fluctuant and cystic skin masses on the right pelvic limb and chest is described. Histopathological evaluation revealed cystic cavities lined by single to multiple layers of apocrine epithelium with papillary projections, consistent with apocrine cystadenoma.

Les cystadénomes apocrines ont rarement été décrits chez le chien. Ici, un chien avec de multiples masses cutanées larges (1.0-7.0 cm), kystiques et fluctuantes sur le membre pelvien et le thorax, est décrit. L'évaluation histopathologique a montré des cavités kystiques entourées de couches simples à multiples d'épithélium apocrine avec projections papillaires, compatible avec un cystadénome apocrine.

Raramente se describen cistoadenomas apocrinos en perros. En este artículo se describe el caso de un perro con masas cutáneas múltiples, grandes (1,0 a 7,0 cm) fluctuantes y quísticas en la extremidad pélvica derecha y el pecho. La evaluación histopatológica reveló cavidades quísticas revestidas por capas individuales o múltiples de epitelio apocrino con proyecciones papilares, compatibles con cistoadenoma apocrino.

Os cistadenomas apócrinos já foram raramente descritos em cães. Descreve-se aqui o caso de um cão com múltiplas massas cutâneas grandes (1,0-7,0 cm), flutuantes e císticas no membro pélvico direito e no tórax. A avaliação histopatológica revelou cavidades císticas revestidas por uma a várias camadas de epitélio apócrino com projeções papilares, consistentes com cistadenoma apócrino.

Giant dumbbell-shaped prostatic cystadenoma presenting as pelvic and scrotal mass.

This study reports the case of an elderly man with a large tumour of the pelvic cavity and scrotum which was once diagnosed as a prostate cyst. Imaging studies considered the source of the tumour to be prostate, and the tumour was ultimately diagnosed by confirmed tissue expression of prostate specific antigen (PSA) and prostate acid phosphatase (PSAP) after surgery. This is the first report about dumbbell-shaped prostatic cystadenoma with invasive growth and even urethral damage, but there was no evidence of clear malignancy. Early diagnosis and treatment are crucial in such kinds of diseases.

Rapid Recurrence of Giant Multilocular Prostatic Cystadenoma after Laparoscopic Excision for Primary Case: A Case Report.

Giant multilocular prostatic cystadenoma is a rare benign tumor of the prostate gland that presents as a large retroperitoneal pelvic mass. The mass is usually located between the urinary bladder and rectum, and results in obstructive voiding symptoms and a change in bowel habits. Complete surgical excision is the treatment of choice. We present a case of rapid recurrent giant multilocular prostatic cystadenoma after laparoscopic excision for primary case. A previously healthy 54-year-old man presented with acute urinary retention. Prostate MRI showed a large cystic mass approximately 13 cm in size, multiple septa and lobulation in the prostate, and no visible solid lesions. Laparoscopic marsupialization of giant multilocular prostatic cystadenoma cysts was performed. One year later, the patient presented with local recurrence. Repeated laparoscopic complete resection was performed without any complications and further recurrence. Giant multilocular prostatic cystadenoma has the risk of recurrence in case of incomplete resection. Surgical treatment should be performed with the goal of complete removal following the same principles as cancer surgery.

Histopathological evaluation of minor salivary gland papillary-cystic tumours: focus on genetic alterations in sialadenoma papilliferum and intraductal papillary mucinous neoplasm.

AIMS: Minor salivary gland tumours showing a predominant papillary-cystic structure are rare, and constitute a mixture of various types of neoplasm; thus, the histopathological assessment of these tumours poses a significant diagnostic challenge. We aimed to delineate the histological characteristics of these tumours and further mutational aspects with a particular focus on sialadenoma papilliferum (SP) and intraductal papillary mucinous neoplasm (IPMN). METHODS AND RESULTS: We retrieved 28 papillary-cystic tumours of the minor salivary glands, and performed histological re-evaluation and mutation analyses of several key oncogenes. The histological classifications were as follows: SP (n = 10), SP-like intraductal papillary tumour (SP-IPT) (n = 2), IPMN (n = 9), intraductal papilloma, cystadenoma, and cystadenocarcinoma (two, three and two respectively). Whereas SP typically consisted of a combination of exophytic squamous epithelium and endophytic intraductal papillary infoldings, SP-IPT lacked the exophytic component. SP and SP-IPT frequently harboured BRAF V600E mutations (75.0%), which were identified in both squamous and ductal components. IPMN was characterised by a well-demarcated cystic lesion filled exclusively with a papillary proliferation of mucinous cells and a high rate of AKT1 E17K mutations (88.9%). Intraductal papillomas were unilocular cystic lesions with intraluminal papillary growth of bland columnar cells. In contrast, both cystadenomas and cystadenocarcinomas showed a multicystic appearance with a papillary configuration. Cystadenocarcinomas invaded the surrounding tissue and were composed of markedly atypical tumour cells. CONCLUSION: The appropriate interpretation of histological findings and specific genetic alterations (e.g. BRAF V600E and AKT1 E17K in SP and IPMN) would be useful for the correct diagnosis of minor salivary gland papillary-cystic tumours.