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.

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.

Papillary cystadenoma of the major salivary gland: A case report and review of literature.

Papillary cystadenoma (PC) of the salivary gland is an uncommon benign epithelial neoplasm that shows predominantly multicystic growth pattern with intraluminal papillary proliferation and areas of oncocytic differentiation. We report a case of papillary cystadenoma of the parotid gland in a 44-years-old female. The patient presented with painful nodular swelling in the right parotid region for two months. Ultrasonography revealed a well marginated oval lesion with altered signal intensity involving the superficial lobe. The excision specimen showed a neoplasm with multicystic spaces having papillary projections lined by benign low-grade epithelium and supported by fibrovascular cores. No significant cytological atypia or mitosis was observed. The cells were immunoreactive for Keratin, Keratin 7, and were negative for Keratin 20, AR, HeR2/neu, TTF1, CDX2, and GATA3. p63 and Keratin 5/6 highlighted the myoepithelial cell layer lining the cystic spaces as well as the papillary projections. The Ki-67 proliferation index was 6%. The patient is on close clinical and imaging follow-up for the last 1year and 8 months without any evidence of disease recurrence or metastasis. Rarity of the lesion and distinct histomorphology warrants appropriate knowledge and discussion of the subject.

Gastrointestinal stromal tumour: presenting as an ovarian cystadenoma.

A gastrointestinal stromal tumortumour (GIST) is an uncommon gastrointestinal neoplasm that can arise from any part of the gastrointestinal tract. They can rarely present as a pelvic mass, which might result in a gynaecological condition being misdiagnosed in a female patient. A woman in her early 70s presented with a huge pelvic mass. Abdomen-pelvis CT scan showed a significant cystic mass in the left-sided pelvis with a mass effect on adjacent structures, which suggested a possibility of an ovarian cystadenoma. Her CA-125 was normal. She underwent an exploratory laparotomy with pelvic mass excision. A diagnosis of a gastrointestinal stromal tumour (GIST) arising from the ileum was made on a histopathology study.

Coexisting ovarian serous cystadenoma with fibroma: A very unusual combination.

Surface epithelial neoplasms are the most common ovarian tumors, constituting around 60% of all ovarian malignancies. They are classified as benign, borderline, and malignant. Ovarian cystadenomas are common benign epithelial neoplasms which carry an excellent prognosis. Ovarian thecoma-fibroma groups are uncommon sex cord-stromal neoplasms, constituting 1.0%-4.0% of all ovarian tumors. Most of them are benign and often found in postmenopausal patients. Combination tumors in the ovary are known. The most common combination is mucinous cystadenoma which occurs in association with Brenner tumor, mature cystic teratoma, Sertoli-Leydig cell tumor, or even a serous cystadenoma. A combination of surface epithelial and thecoma-fibroma group is very rarely encountered. A case of one such combination of serous cystadenoma and fibroma of the ovary is being presented here in a postmenopausal woman.

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.

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.

Management of Mucinous Cystic Neoplasms of the Liver.

BACKGROUND: Mucinous cystic neoplasms of the liver (MCN-L) including biliary cystadenomas (BCA) and biliary cystadenocarcinomas (BCAC) are rare cystic lesions that comprise less than 5% of all liver cysts and affect only a small subset of individuals. We herein review the current evidence regarding the clinical presentation, imaging characteristics, tumor markers, pathological findings, clinical management, and prognosis of MCN-L. METHODS: A comprehensive review of the literature was performed using MEDLINE/Pubmed and Web of Science databases. In PubMed, the terms "biliary cystadenoma," "biliary cystadenocarcinoma," and "non parasitic hepatic cysts" were queried to identify the most recent data on MCN-L. RESULTS: US imaging, CT, and MRI, as well as consideration of clinicopathological features, are required to appropriately characterize and diagnose hepatic cystic tumors. BCA are premalignant lesions and cannot be reliably differentiated from BCAC based on imaging alone. As such, both types of lesions should be treated with margin-negative surgical resection. Following surgical resection, recurrence is fairly low among patients with BCA and BCAC. Despite having worse long-term outcomes than BCA, the prognosis following surgical resection of BCAC still remains more favorable than other primary malignant liver tumors. CONCLUSION: MCN-L are rare cystic liver tumors that include BCA and BCAC, which can be difficult to differentiate based on imaging alone. Surgical resection remains the mainstay of management for MCN-L with recurrence being generally uncommon. Future multi-institutional studies are still required to better understand the biology behind BCA and BCAC to improve the care of patients with MCN-L.

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.

Cistoadenoma mucinoso apendicular / Appendiceal mucinous cystadenoma

El cistoadenoma apendicular es una neoplasia poco frecuente, que tiene una incidencia de 0,2 % a 0,3 % en todas las apendicetomías; esta afección predomina en pacientes féminas y su presentación es poco específica en cuanto a los síntomas, los cuales pueden compararse a un cuadro de apendicitis aguda, una masa abdominal, un cuadro obstructivo o ginecológico, o manifestaciones urológicas, que son las menos frecuentes. Se presentó una paciente femenina de 59 años de edad con dolor abdominal localizado en la fosa ilíaca derecha de 4 meses de evolución; se realizaron varios exámenes complementarios, una laparoscopia diagnóstica, una laparotomía exploratoria y también una apendicetomía. Después de estos exámenes se realizó un diagnóstico histológico de cistoadenoma mucinoso apendicular.

Appendiceal cystadenoma is a rare neoplasm, with an incidence of 0.2% to 0.3% among all appendectomies; this condition predominates in female patients and its presentation is unspecific in terms of symptoms, which can be compared to acute appendicitis, an abdominal mass, obstructive or gynecological symptoms, or urological manifestations, which are the least frequent. We present a 59-year-old female patient with an abdominal pain over 4 months located in the right iliac fossa; several complementary tests were performed such as a diagnostic laparoscopy, an exploratory laparotomy and an appendectomy. After these examinations, a histological diagnosis of appendiceal mucinous cystadenoma was made.

Genetic analysis of heterogeneous subsets of circulating tumour cells from high grade serous ovarian carcinoma patients.

Circulating tumour cells (CTCs) are heterogenous and contain genetic information from the tumour of origin. They bear specific intra- and extra-cellular protein markers aiding in their detection. However, since these markers may be shared with other rare cells in the blood, only genetic testing can confirm their malignancy. Herein, we analyse different CTC subsets using single cell whole genome DNA sequencing to validate their malignant origin. We randomly selected putative CTCs identified by immunostaining that were isolated from 4 patients with high grade serous ovarian cancer (HGSOC) and one with benign cystadenoma. We specifically targeted CTCs positive for epithelial (CK/EpCAMpos), mesenchymal (vimentinpos), and pseudoendothelial (CK/EpCAMpos plus CD31pos) markers. We isolated these cells and performed whole genome amplification (WGA) and low-pass whole-genome sequencing (LP-WGS) for analysis of copy number alterations (CNA). Of the CK/EpCAMpos cells analysed from the HGSOC patients, 2 of 3 cells showed diverse chromosomal CNAs. However, the 4 pseudoendothelial cells (CK/EpCAMpos plus CD31pos) observed in the HGSOC cases did not carry any CNA. Lastly, two of the clusters of vimentin positive cells sequenced from those found in the benign cystadenoma case had CNA. Despite the low number of cells analysed, our results underscore the importance of genetic analysis of putative CTCs to confirm their neoplastic origin. In particular, it highlights the presence of a population of CK/EpCAMpos cells that are not tumour cells in patients with HGSOC, which otherwise would be counted as CTCs.

Radiologic Reporting of Simple Hepatic Cyst Versus Biliary Cystadenoma May Lead to Unnecessary Surgery.

BACKGROUND AND AIMS: Simple liver cyst (SHC) is a benign condition with no malignant potential. They are typically discovered incidentally due to the increased use of abdominal imaging, but some patients may present with abdominal pain. A radiologist's differential diagnosis in cases of SHC will often include "rule out biliary cystadenoma." Under these circumstances, patients and surgeons are more likely to pursue surgical options even in asymptomatic cases. The aim of this study is to conduct a retrospective analysis of presentation, radiologic reporting, management plan, and histopathology of patients referred to a tertiary hospital in order to determine the correlation between radiology and histology. METHODS: We retrospectively analyzed the clinical, radiological, and histopathological data of 20 patients operated for a diagnosis of a cystic lesion in the liver. RESULT: The CT/MRI of 6 (30%) patients was reported as a biliary cystadenoma, 13 (65%) were reported as a simple hepatic cyst and 1 patient (5%) had hepatocellular carcinoma (HCC) with the additional diagnosis of multiple hepatic cysts. The lesion reported as HCC on the scan was separate from the cystic lesions. The modality of imaging for these cysts was evenly split, 50% of patients had a CT scan, and 50% had an MRI performed. All imaging studies were interpreted by an attending radiologist and most of them were discussed in multidisciplinary meetings. Nineteen patients (95%) had an intraoperative diagnosis of a simple liver cyst based on its visual appearance and clear fluid within the cyst. These patients underwent cyst wall fenestration and de-roofing with the cyst wall sent for histopathology. One patient (5%) with HCC underwent a non-anatomical liver resection. Histopathology was conclusive for a benign hepatic cystic lesion from the cyst wall biopsy. All 20 patients in this study underwent surgery, either due to symptoms or due to radiologic diagnosis of BCA. Four of the 20 cases (20%) were asymptomatic and out of these four cases, 3 (75%) were diagnosed as cystadenoma on the preoperative imaging studies. All 19 cases were diagnosed as a simple liver cyst on pathology. CONCLUSION: In summary, there is a growing trend of "ruling-out the diagnosis of biliary cystadenoma" in patients who present with liver cysts. Patients are appropriately more anxious after this preoperative diagnosis and the treating surgeons have medico-legal concerns regarding conservative management in asymptomatic patients diagnosed as BCA. This single center experience draws attention to the radiology criteria utilized for diagnosing a biliary cystadenoma and suggests that it is time to revisit the imaging interpretation and differential diagnosis.

[Salivary papillary cystic low-grade mucoepidermoid carcinoma and cystadenoma: a comparison of clinicopathological and genetic features].

Objective: To study the clinicopathologic and genetic features of papillary cystic low-grade mucoepidermoid carcinoma (LG-MEC) and cystadenoma. Methods: A retrospective review was performed on salivary gland tumor patients with papillary cystic architecture who presented to department of oral pathology, Peking University School and Hospital of Stomatology between January 2010 and June 2022. Among this cohort, there were 17 males and 17 females with a range age of 23-82 years [(55.6±14.6) years]. Diagnosis was confirmed by histological, immunohistochemistry (IHC) and fluorescence in situ hybridization (FISH) analysis. Finally, 15 papillary cystic LG-MEC and 19 cystadenoma patients were included in the present study. All patients were followed clinically and radiologically, and the duration of follow-up ranged from 1 to 141 months. Results: All neoplasms showed papillary proliferation with multilocular or giant cystic tumors. Papillary cystic LG-MEC was characterized by epidermoid cells, intermediate cell and mucous cells with multiple lining-layers. Papillary cystic LG-MEC had mild cellular atypia and a pushing infiltration. Cystadenoma was characterized by cuboidal, columnar and ciliated pseudostratified columnar lining epithelium. Squamous metaplasia, mucinous metaplasia and acidophilic degeneration could also be observed focally in cystadenoma. For IHC staining, papillary cystic LG-MEC showed diffusely and strongly positive for mucin 4 (MUC4) (15/15) and mucin 5 Subtype AC (MUC5AC) (4/15) in the epidermoid cells, intermediate cell and mucous cells. The epidermoid cells and intermediate cells were diffusely positive for p40 and p63. The Ki-67 index was about 10%-15% in LG-MEC. As a contrast, p40 (17/19) and p63 (14/15) were only detected in the basal cells of cystadenoma. Cystadenoma showed focal MUC5AC (4/19)expression and MUC4 (19/19)diffuse expression. In addition, the Ki-67 index was 5%-10% in cystadenoma. The MAML2 gene translocation was detected in 11 LG-MEC patients, but none in cystadenoma. Conclusions: The differential diagnosis points between papillary cystic LG-MEC and cystadenoma included the specific epidermoid cells, intermediate cells and mucus cells in LG-MEC, cell atypia, the pushing-infiltration pattern, diffuse expression of p40 and p63 in the lining epithelium, and a MAML2 gene rearrangement. The molecular test of MAML2 should be recommended to reduce missed LG-MEC diagnoses.