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Autopsy & Case Reports 2022Papillary cystadenoma is a rare benign neoplasm of the epididymis. It may occur sporadically or in association with von Hippel-Lindau disease (VHLD). Papillary...
BACKGROUND
Papillary cystadenoma is a rare benign neoplasm of the epididymis. It may occur sporadically or in association with von Hippel-Lindau disease (VHLD). Papillary cystadenoma of the epididymis (PCE) is a benign mimic of metastatic clear cell renal cell carcinoma (CCRCC) given their histologic similarities.
CASE PRESENTATION
Herein, we present the case of a 40-year-old man with a four-year history of microhematuria and a recently detected right paratesticular mass. A testicular sonogram revealed a hypoechoic, hypervascular solid mass in the right epididymal head treated by surgical excision. Histopathological examination demonstrated a 1.1 cm papillary cystadenoma of the epididymis. Genetic testing performed later showed no signs of VHLD. However, heterozygous mutations in three genes - , , and - were found which have never been reported in PCE before.
CONCLUSIONS
Papillary cystadenoma of the epididymis should always be considered in the differential diagnosis of epididymal lesions, especially those that are cystic. The mainstay of treatment remains surgical excision, which provides an excellent prognosis.
PubMed: 35496736
DOI: 10.4322/acr.2021.374 -
Archives of Pathology & Laboratory... Apr 2010Papillary cystadenoma is a rare benign neoplasm of the epididymis, occurring mainly in young adult males. More than one-third of the cases reported in the literature... (Review)
Review
Papillary cystadenoma is a rare benign neoplasm of the epididymis, occurring mainly in young adult males. More than one-third of the cases reported in the literature have occurred in patients with von Hippel-Lindau disease. Conversely, epididymal nodules presumed to be papillary cystadenomas are found in one-third of males with von Hippel-Lindau disease. The association is stronger for bilateral tumors. The pathogenesis involves loss of the von Hippel-Lindau gene resulting in overexpression of the angiogenic protein "hypoxia-inducible factor." Papillary cystadenoma is of mesonephric derivation. It originates in the efferent ductules of the head of the epididymis in the form of tiny precursor lesions. Histologically, papillary cystadenoma is characterized by cystic spaces with intracystic papillary projections lined by clear cells, with a resultant resemblance to renal cell carcinoma. Immunohistochemical markers may facilitate the distinction between the 2 tumors. Treatment consists of surgical excision and the prognosis is excellent.
Topics: Adolescent; Adult; Aged; Carcinoma, Renal Cell; Cystadenoma, Papillary; Diagnosis, Differential; Epididymis; Humans; Male; Middle Aged; Testicular Neoplasms; Young Adult; von Hippel-Lindau Disease
PubMed: 20367315
DOI: 10.5858/134.4.630 -
Pathology, Research and Practice Nov 2023Papillary cystadenoma (PC) of the salivary gland is an uncommon benign epithelial neoplasm that shows predominantly multicystic growth pattern with intraluminal... (Review)
Review
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.
Topics: Humans; Female; Adult; Cystadenoma, Papillary; Neoplasm Recurrence, Local; Parotid Gland; Oxyphil Cells; Epithelium; Cystadenoma
PubMed: 37913638
DOI: 10.1016/j.prp.2023.154884 -
British Dental Journal Feb 2024
Topics: Humans; Cystadenoma, Papillary
PubMed: 38332080
DOI: 10.1038/s41415-024-7093-5 -
European Archives of... Sep 2021Oncocytic papillary cystadenomas (OPCs) of the larynx are rare benign cystic lesions that usually present as supraglottic masses arising from the laryngeal ventricles.... (Review)
Review
OBJECTIVE
Oncocytic papillary cystadenomas (OPCs) of the larynx are rare benign cystic lesions that usually present as supraglottic masses arising from the laryngeal ventricles. OPCs are found in patients older than 60 years, with a female predominance. Symptoms vary from asymptomatic to hoarseness, dyspnea, and dysphagia; often, they mimic a laryngocele. The treatment is surgical. Diagnosis is based on histopathologic examination.
MATERIALS AND METHODS
Surgical records for laryngeal masses diagnosed between 2005 and 2020 were searched retrospectively.
RESULTS
Ten patients were identified and included in the study. OPCs predominantly occurred in women (9/10), and the mean age at presentation was 73 years. Most patients (8/10) presented with hoarseness and were smokers. OPCs were localized in the ventricle in eight out of ten patients. Surgical treatment was performed in all cases, mostly using transoral endolaryngeal approach (9/10). Histopathologic examination revealed oncocytic cyst or oncocytic papillary cystadenoma (the former term being the older synonym for OPC).
CONCLUSION
OPCs present a separate clinicopathologic entity, distinct from other cystic laryngeal lesions. They have a characteristic location, age and sex group, microscopic appearance, and potential for local recurrence.
Topics: Cystadenoma, Papillary; Female; Humans; Laryngeal Neoplasms; Larynx; Male; Neoplasm Recurrence, Local; Retrospective Studies
PubMed: 33909144
DOI: 10.1007/s00405-021-06841-2 -
International Journal of Oral Surgery Dec 1985A rare case of a papillary cystadenoma, mucous-cell type, in the lower lip of a 73-year-old male is presented. The tumor consisted of many small cysts with multiple...
A rare case of a papillary cystadenoma, mucous-cell type, in the lower lip of a 73-year-old male is presented. The tumor consisted of many small cysts with multiple papillary projections into their cystic spaces. The projections and cyst walls were lined with pseudostratified mucous-cell epithelium. Clinically and histologically, the present papillary cystadenoma was considered as a true tumor originated from the ducts of minor salivary glands. There exists only one case report of a papillary cystadenoma with adequate description and photomicrographs in the Japanese dental literature.
Topics: Aged; Cystadenoma; Humans; Lip Neoplasms; Male
PubMed: 3936807
DOI: 10.1016/s0300-9785(85)80065-x -
Oral and Maxillofacial Surgery Sep 2013Salivary gland tumors are uncommon and constitute 2-6.5 % of all head and neck neoplasms. Tumors of minor salivary gland origin account for less than 25 % of all... (Review)
Review
BACKGROUND
Salivary gland tumors are uncommon and constitute 2-6.5 % of all head and neck neoplasms. Tumors of minor salivary gland origin account for less than 25 % of all salivary gland neoplasms. Papillary cystadenoma of salivary glands is a rare benign epithelial neoplasm characterized by multicystic growth in which the epithelium exhibits adenomatous proliferation. Papillary cystadenoma of minor salivary glands most frequently involves the lip, buccal mucosa, and palate. This tumor typically presents as a slow-growing, painless mass, usually with diameter of less than 1 cm and clinical resemblance to a mucocele. Although most papillary cystadenomas are predominantly of one cell type, a regional variability may be present.
CASE REPORT
We present a case of papillary cystadenoma of the minor salivary glands in a 58-year-old patient exhibiting an upper respiratory tract epithelium, a profoundly atypical benign tumor.
DISCUSSION
This type of minor salivary gland tumor epithelium in the lower lip may be the result of a metaplastic process or simply another neoplastic manifestation of papillary cystadenoma. As far as the differential diagnosis of this entity is concerned, it is important to distinguish it from papillary cystadenoma lymphomatosum (Warthin's tumor), low-grade mucoepidermoid carcinoma, the papillary-cystic variant of acinic cell carcinoma, and cystadenocarcinoma Recognition of this lesion is important for the clinician since the differential diagnosis includes lesions with similar clinical appearance and infiltrative behavior.
Topics: Cystadenoma, Papillary; Diagnosis, Differential; Female; Humans; Lip Neoplasms; Middle Aged; Mouth Mucosa; Salivary Gland Neoplasms; Salivary Glands, Minor
PubMed: 22933035
DOI: 10.1007/s10006-012-0357-2 -
International Journal of Gynecological... May 2021Clear cell papillary cystadenoma of the epididymis is an uncommon benign neoplasm, usually seen in patients with von Hippel-Lindau disease. Morphologic and... (Review)
Review
Clear cell papillary cystadenoma of the epididymis is an uncommon benign neoplasm, usually seen in patients with von Hippel-Lindau disease. Morphologic and immunohistochemical examination aid in distinguishing clear cell papillary cystadenoma from malignant histologic mimics including low-grade mesothelial proliferations and metastatic clear cell renal cell carcinomas. Analogous lesions have been described in the female genital tract, often posing diagnostic challenges due to their low incidence. Here, we present the difficult diagnostic aspects of the first case of clear cell papillary cystadenoma involving the ovary, including the salient immunohistochemical, ultrastructural, and molecular characteristics.
Topics: Carcinoma, Renal Cell; Cystadenoma, Papillary; Diagnosis, Differential; Female; High-Throughput Nucleotide Sequencing; Humans; Immunohistochemistry; Microscopy, Electron; Middle Aged; Ovarian Neoplasms; Ovary; Point Mutation; Sequence Analysis, DNA; Von Hippel-Lindau Tumor Suppressor Protein
PubMed: 31985579
DOI: 10.1097/PGP.0000000000000666 -
Zhonghua Nan Ke Xue = National Journal... Feb 2015To study the clinicopathological characteristics of papillary cystadenoma of the epididymis. (Review)
Review
OBJECTIVE
To study the clinicopathological characteristics of papillary cystadenoma of the epididymis.
METHODS
Using routine pathology and immunohistochemistry, we observed the surgically obtained samples from 2 cases of papillary cystadenoma of the epididymis, analyzed their pathological features and clinical presentations, and reviewed the related literature.
RESULTS
The 2 patients were both adult males. The tumors typically manifested as painless swelling in the epididymis, with occasionally dull pain and tenesmus in 1 of the cases. Pathologically, the lesions exhibited three morphological features, i. e., dilated ducts and small cysts surrounded by fibrous connective tissue, adenoid papillary hyperplasia into the cysts embraced by fibrovascular stroma, and acidophil substance present in the cysts. Immunohistochemistry showed that the tumors were strongly positive for CK8/18, CK7, and EMA, but negative for CK20, CEA, MC, Calretenin, P53, P63, SMA, VHL, and CD10, with the positive rate of Ki-67 <1%. Follow-up visits revealed good prognosis in both cases.
CONCLUSION
Papillary cystadenoma of the epididymis is a rare benign tumor in the male urogenital system, which may be accompanied by the VHL syndrome. Surgery is the first choice for its treatment.
Topics: Adult; Cystadenoma, Papillary; Epididymis; Genital Neoplasms, Male; Humans; Immunohistochemistry; Male; von Hippel-Lindau Disease
PubMed: 25796691
DOI: No ID Found -
Oral Surgery, Oral Medicine, Oral... Jan 2008A 91-year-old man presented with an asymptomatic swelling in the roof of his mouth. Clinically the lesion was nontender and appeared cystic. A CT scan showed a soft... (Review)
Review
A 91-year-old man presented with an asymptomatic swelling in the roof of his mouth. Clinically the lesion was nontender and appeared cystic. A CT scan showed a soft tissue swelling of his palate with no bony involvement. An orthopantogram and blood tests were noncontributory. A fine-needle aspiration biopsy was suggestive of a minor salivary gland neoplasm. An excision biopsy showed papillary cystadenoma of the minor salivary gland, with numbers of psammoma bodies. A stepwise approach is demonstrated in the diagnosis of this rare minor salivary gland neoplasm. To the authors' best knowledge, this is the 12th case of this tumor seen in the palate. Furthermore, no previous case has included a fine-needle aspiration biopsy in the work-up, the cytological features of which will be described.
Topics: Aged, 80 and over; Biopsy, Fine-Needle; Cystadenoma, Papillary; Diagnosis, Differential; Humans; Male; Palatal Neoplasms; Salivary Gland Neoplasms; Salivary Glands, Minor
PubMed: 18155598
DOI: 10.1016/j.tripleo.2007.07.019