Journal of Pathology and Translational Medicine

Case Studies

Intraoperative frozen cytology of intraosseous cystic meningioma in the sphenoid bone: Na Rae Kim, Gie-Taek Yie; J Pathol Transl Med. 2020;54(6):508-512. Published online July 1, 2020; DOI: https://doi.org/10.4132/jptm.2020.05.21

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Meningiomas in bone are rarely subjected to fine-needle aspiration diagnosis, and those arising in the skull bone with a cystic presentation are rare. A 24-year-old woman presented with subdural hemorrhage, and subsequent radiology depicted an osteolytic mass-like lesion in the sphenoid bone. Intraoperatively, a solid and cystic hemorrhagic lesion mimicking an aneurysmal bone cyst was observed in the sphenoid bone with dural tearing. Frozen cytology showed singly scattered or epithelioid clusters of round to elongated cells intermixed with many neutrophils. Tumor cells had bland-looking round nuclei with rare prominent nucleoli and nuclear inclusions and eosinophilic granular to globoid cytoplasm in capillary-rich fragments. Histology revealed intraosseous meningothelial and microcystic meningioma (World Health Organization grade 1) in right lesser wing of the sphenoid bone. Considering its unusual location and cytologic findings, differential diagnoses included chordoma, chondroma, chondrosarcoma, and aneurysmal bone cyst. The present case posed a diagnostic challenge due to possible confusion with these entities.

Citations

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Middle ear adenoma: Cytohistologic features and differential diagnosis
Abdullah Almajnooni, Matthew Vega, Lin Cheng, Paolo Gattuso, Mary K. Allen‐Proctor
Diagnostic Cytopathology.2023;[Epub] CrossRef
Exploring the role of epidermal growth factor receptor variant III in meningeal tumors
Rashmi Rana, Vaishnavi Rathi, Kirti Chauhan, Kriti Jain, Satnam Singh Chhabra, Rajesh Acharya, Samir Kumar Kalra, Anshul Gupta, Sunila Jain, Nirmal Kumar Ganguly, Dharmendra Kumar Yadav, Timir Tripathi
PLOS ONE.2021; 16(9): e0255133. CrossRef

Cystic Benign Phyllodes Tumor in the Inguinal Region: Jai Hyang Go; Korean J Pathol. 2013;47(6):583-586. Published online December 24, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.6.583

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Abstract PDF

The present lesion was the first reported case of a benign intracystic phyllodes tumor in the inguinal region. We report the case of a 51-year-old female patient who presented with an inguinal mass. A clinical diagnosis of malignant lymphoma was considered in this case. The resected tumor was well-circumscribed and showed numerous papillary nodular protrusions into a central cystic cavity (3.5×2.5 cm). The microscopic findings showed hyperplastic epithelium-lined cysts with leaf-like intraluminal epithelium-lined bland stromal projections. The epithelial cell linings were strongly positive for estrogen and progesterone receptors.

Citations

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Computed tomography and magnetic resonance imaging in diagnosis of metastatic pleural lesion with pleural effusion in patients with breast carcinoma
P. M. Kotlyarov, I. D. Lagkueva, N. I. Sergeev
Russian Pulmonology.2019; 29(1): 112. CrossRef
Mama ectópica en la región inguinal
V.Y. Presas, L.M. Mastronardi, S. Saucedo, E. Rojas Bilbao
Clínica e Investigación en Ginecología y Obstetricia.2017; 44(2): 89. CrossRef

Heterotopic Intestinal Cyst of the Submandibular Gland: A Case Study: Mi Jung Kwon, Dong Hoon Kim, Hye-Rim Park, Soo Kee Min, Jinwon Seo, Eun Soo Kim, Si Whan Kim, Bumjung Park; Korean J Pathol. 2013;47(3):279-283. Published online June 25, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.3.279

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Abstract PDF

Heterotopic gastrointestinal cysts are rarely found in the oral cavity. Most of these cysts are lined with gastric mucosa and involve the tongue. There have been no reported heterotopic intestinal cysts of the submandibular gland that are completely lined with colonic mucosa. An 8-year-old girl presented with an enlarging swelling in the left submandibular area, and a 4-cm unilocular cyst was fully excised. The cyst was completely lined with colonic mucosa that was surrounded by smooth muscle layer, and the lining cells were positive for CDX-2, an intestinal marker, indicating a high degree of differentiation. The pathogenesis remains unclear, but it may be related to the misplacement of embryonic rests within the oral cavity during early fetal development. Although heterotopic intestinal cysts rarely occur in the submandibular gland, they should be considered in the differential diagnosis of facial swellings in the pediatric population.

Citations

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Heterotopic gastrointestinal cyst in the floor of mouth: a case report
Naoaki SAITO, Satoshi MARUYAMA, Yusuke KATO, Ryoko TAKEUCHI, Jun-ichi TANUMA, Tadaharu KOBAYASHI
Japanese Journal of Oral and Maxillofacial Surgery.2023; 69(1): 27. CrossRef
A case report of oral heterotopic gastrointestinal cysts (HGIC) and review of the literature
Gursimran Kaur Bains, Richard Pilkington, Joanna Stafford, Sunil Bhatia
Oral Surgery.2022; 15(1): 71. CrossRef
A Rare Case of Ectopic Colonic Mucosa Presenting With Airway Compromise in a Neonate
Justin Hall, Fatima Z Aly, Julia Comer, Michael P Gebhard, Thomas Schrepfer
Cureus.2022;[Epub] CrossRef
Ultrasonic Features of Uncommon Congenital Heterotopic Colon and Pancreas in the Neck: An Extremely Rare Case Report
Yingli Wei, Zhihao Pan, Xiaoling Kang, Cuiqing Huang, Dan Chen
Frontiers in Pediatrics.2021;[Epub] CrossRef
Quiste gastrointestinal heterotópico en la cavidad oral
Beatriz Arango de Samper, Eliana Elisa Muñoz López, Estefanía Morales González
Latin American Journal of Oral and Maxillofacial Surgery.2021; 1(1): 40. CrossRef

Multicystic Biliary Hamartoma of the Liver: Ji Soo Song, Sang Jae Noh, Baik Hwan Cho, Woo Sung Moon; Korean J Pathol. 2013;47(3):275-278. Published online June 25, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.3.275

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Abstract PDF

Multicystic biliary hamartoma (MCBH) is a very rare hamartomatous cystic nodule of the liver, which has recently been described as a new entity of a hepatic nodular lesion. We report a unique case of MCBH with a review of the literatures. A hepatic multicystic mass of segment 3 was detected in a 52-year-old male by abdominal computed tomography, and resection of this lesion was performed. Macroscopic examination revealed a 2.7×2.0 cm nodular mass with a multicystic honeycomb cut surface. Histologically, this lesion consisted of multiple dilated cystic ducts lined by biliary type epithelial cells, periductal glands and connective tissue, which included small amounts of hepatic parenchyma and blood vessels. Recognition of this unusual lesion is essential to avoid confusion with other cystic tumors of the liver, and to learn more about its natural history and response to treatment.

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Benjamin H. Brinkmann
Journal of Clinical Neurophysiology.2024; 41(1): 2. CrossRef
A Case of Multicystic Biliary Hamartoma with a Marked Peribiliary Gland Component Successfully Treated by Purely Laparoscopic Anatomical Liver Resection
Keita Kai, Takao Ide, Tomokazu Tanaka, Kumpei Yukimoto, Hiroyuki Irie, Hirokazu Noshiro, Shinichi Aishima
Journal of Gastrointestinal Cancer.2023; 54(3): 996. CrossRef
Characteristics of multicystic biliary hamartoma: A case report
Jia Lian, Lixia Sun, Yankai Yang, Jun Li, Ye Zhang, Guiqiu Liu, Weijuan Hu
Frontiers in Surgery.2023;[Epub] CrossRef
Hamartoma multiquístico de vías biliares
Victoria Carmona, Iago Justo, Yolanda Rodríguez-Gil, Alberto Marcacuzco, Carmelo Loinaz, Carlos Jiménez
Cirugía Española.2022; 100(12): 800. CrossRef
Multicystic Biliary Hamartoma With Xanthogranulomatous Inflammation on 18F-FDG PET/CT
Nahomi Shono, Yoichi Otomi, Hideki Otsuka, Takayoshi Shinya, Masafumi Harada
Clinical Nuclear Medicine.2022; 47(10): 882. CrossRef
Intrahepatic multicystic biliary hamartoma: A case report
Chen-Yu Wang, Fu-Yang Shi, Wei-Feng Huang, Yan Tang, Ting Li, Guo-Lin He
World Journal of Clinical Cases.2022; 10(26): 9361. CrossRef
A Case of Multicystic Biliary Hamartoma Treated with Left Medial Sectionectomy
Naomi KUROKI, Tomoaki TANAKA, Takanobu SUGASE, Syoji TANIGUCHI, Takashi GOTO, Rintaro KOGA, Takumi KIWAKI, Hiroyuki TANAKA
Nihon Rinsho Geka Gakkai Zasshi (Journal of Japan Surgical Association).2022; 83(2): 395. CrossRef
Multicystic biliary hamartoma
Victoria Carmona, Iago Justo, Yolanda Rodríguez-Gil, Alberto Marcacuzco, Carmelo Loinaz, Carlos Jiménez
Cirugía Española (English Edition).2022; 100(12): 800. CrossRef
Case Report: Incidentally Discovered a Rare Cystic Lesion of Liver: Multicystic Biliary Hamartoma
Wentao Mu, Peng Su, Shanglei Ning
Pathology and Oncology Research.2021;[Epub] CrossRef
Bile Duct Hamartoma Mimicking Liver Metastasis in Suspected Porcelain Gallbladder: a Case Report
Gautham Krishnamurthy, Harjeet Singh, Sravya Deepika Ganti, Ganga Ram Verma
Journal of Gastrointestinal Cancer.2019; 50(4): 1022. CrossRef
A variant of multicystic biliary hamartoma presenting as an intrahepatic cystic neoplasm
Tetsuro Tominaga, Takafumi Abo, Naoe Kinoshita, Tomonori Murakami, Yasunori Sato, Yasuni Nakanuma, Kenich Harada, Junichi Masuda, Takeshi Nagayasu, Atsushi Nanashima
Clinical Journal of Gastroenterology.2015; 8(3): 162. CrossRef
Hamartoma biliar multiquístico intrahepático: presentación de un caso clínico
María Jezabel Fernández-Carrión, Ricardo Robles Campos, Asunción López Conesa, Roberto Brusadín, Pascual Parrilla Paricio
Cirugía Española.2015; 93(9): e103. CrossRef
Intrahepatic Multicystic Biliary Hamartoma: Presentation of a Case Report
María Jezabel Fernández-Carrión, Ricardo Robles Campos, Asunción López Conesa, Roberto Brusadín, Pascual Parrilla Paricio
Cirugía Española (English Edition).2015; 93(9): e103. CrossRef
Multicystic biliary hamartoma: A report of a rare entity and a review of the literature
Rachel E. Beard, Eric U. Yee, Koenraad J. Mortele, Khalid Khwaja
International Journal of Surgery Case Reports.2014; 5(12): 919. CrossRef
Multicystic biliary hamartoma mimicking intrahepatic cholangiocarcinoma: report of a case
Tomoaki Yoh, Ryuji Okamura, Hiroyuki Nakayama, Xue Lin, Yuya Nakamura, Tatsushi Kato
Clinical Journal of Gastroenterology.2014; 7(5): 418. CrossRef

Review

Pathologic Review of Cystic and Cavitary Lung Diseases: Na Rae Kim, Joungho Han; Korean J Pathol. 2012;46(5):407-414. Published online October 25, 2012; DOI: https://doi.org/10.4132/KoreanJPathol.2012.46.5.407

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Abstract PDF

Pulmonary cystic and cavitary lesions caused by diverse etiologies are commonly encountered in chest imaging. The terms "cyst" and "cavity" are used to describe air-filled regions in the center of a nodule or consolidation of the lung. To date, only radiologic aspects of these lesions have been addressed. The morphologies of pulmonary cystic and cavitary lesions exhibit a broad spectrum, ranging from benign to malignant pulmonary diseases of acquired or congenital origin, including variable infectious diseases. In this review, we summarized the differential diagnosis of pathological entities to provide pathologists and radiologists with an overview of the diseases most commonly associated with pulmonary cystic and cavitary lesions in adults and children. The results showed slightly different patterns in the distribution of the diseases in the two groups. The most common causes of cavitary lesions include malignancy and infection in adults, and congenital malformation in children. Therefore, identification of pathologic entities correlating with the radiologic findings, clinical course, and location of the lesion is important in the evaluation of cystic and cavitary lung lesions in order to avoid unnecessary surgical procedures or delayed treatment.

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High-Resolution Computed Tomography of Cystic Lung Disease
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Seminars in Respiratory and Critical Care Medicine.2022; 43(06): 792. CrossRef
Miliary tuberculosis in a paediatric patient with psoriasis
Jacob Kilgore, Jonathon Pelletier, Bradford Becken, Stephen Kenny, Samrat Das, Lisa Parnell
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Atypical pulmonary metastases in children: the spectrum of radiologic findings
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Pediatric Radiology.2021; 51(10): 1907. CrossRef
Radiographic and CT appearance of cavitary pulmonary lesions in a lamb
J Kan, J Bauquier, D Tyrrell, K O'Byrne, AW Stent, B Brosnan
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Community-acquired Achromobacter xylosoxidans infection presenting as a cavitary lung disease in an immunocompetent patient
Chan Hee Hwang, Woo Jin Kim, Hye Young Jwa, Sung Heon Song
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Dong Wang, Lei Cui, Zhi-Gang Li, Li Zhang, Hong-Yun Lian, Hong-Hao Ma, Yun-Ze Zhao, Xiao-Xi Zhao, Tian-You Wang, Rui Zhang
Chinese Medical Journal.2018; 131(15): 1793. CrossRef
Benign features of infection‐related tumor‐like lesions of the lung: A retrospective imaging review study
Chun‐Chao Huang, Sho‐Ting Hung, Wei‐Chin Chang, Chin‐Yin Sheu
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Case Report

Sporadic Cutaneous Keratocyst without Nevoid Basal Cell Carcinoma Syndrome: Report of 1 Case.: Hye Won Lee, Ji Young Park, Sun Hee Kang, Misun Choe; Korean J Pathol. 2011;45(3):322-325.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.3.322

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Abstract PDF

A cutaneous keratocyst is very rare and is ordinarily associated with nevoid basal cell carcinoma syndrome (NBCCS), also known as Gorlin syndrome. NBCCS is a rare autosomal-dominant disorder that results from the mutation in the patched homologue 1 (PTCH1) gene located on chromosome 9q22.3, with high penetrance and variable expressivity. NBCCS demonstrates multisystem manifestations such as multiple basal cell carcinomas in early age, jaw cysts and pits of the hands and feet. Cutaneous keratocysts are characteristically lined by festooned keratinized squamous epithelium with parakeratosis. The cystic wall contains neither granular cell layer nor skin appendages. To the best of our knowledge, only two cases of cutaneous keratocysts not associated with NBCCS have been reported to date. We report one another case of a histologically confirmed cutaneous keratocyst in a 50-year-old female without a family history and clinical features of NBCCS.

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Sporadic Cutaneous Keratocyst of the Scalp: A Report of an Extremely Rare Lesion
Rana S AL-Zaidi, Eyad Tantawi, Rahaf AL-Radadi, Asrar Banjar
Cureus.2021;[Epub] CrossRef
A Large Extragnathic Keratocystic Odontogenic Tumour
Soumya Makarla, Radhika M. Bavle, Sudhakara Muniswamappa, Srinath Narasimhamurthy
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Original Article

Pathologic Characteristics of Ovarian Hemorrhagic Polycyst in Estrogen Receptor-alpha (ERalpha) Knockout Mice and Roles of ERalpha in Hemorrhagic Polycyst.: Hyun Jin Son, Joo Heon Kim, Hye Kyung Lee, Mee Ja Park, Dong Wook Kang, Che Myong Ko; Korean J Pathol. 2010;44(4):376-383.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.4.376

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Abstract PDF: BACKGROUND
Polycystic ovary syndrome (PCOS) is the most common endocrinopathy causing anovulation in women of childbearing age. It has been well established that estrogen receptor-alpha knockout (ERalphaKO) mice display several pathologic ovarian phenotypes of PCOS. The aims of this study were to determine ovarian pathology in new ERalphaKO mice using a CreloxP approach and intra-ovarian ERalpha function as regulating key aspects of PCOS.
METHODS
ERalphaKO mice, which were deficient in exon 3 of the ERalpha gene, were used. Immunohistochemical studies were done on ovaries of control and ERalphaKO mice using antibodies specific to ERalpha, ERbeta, inhibin-alpha, and alpha-smooth muscle actin (SMA), as well as histochemical staining using Sudan black-B.
RESULTS
All ovaries of ERalphaKO mice were larger than control mouse ovaries and displayed a disrupted theca-interstitial tissue organization, multiple atretic follicles and multiple hemorrhagic cysts. None of the ERalphaKO mouse ovaries showed a corpus luteum. In addition, heavy deposition of Sudan black-B positive foamy cells was seen. The theca externa of preantral immature follicles and hemorrhagic cysts showed strong expression of alpha-SMA.
CONCLUSIONS
ERalphaKO mice show hemorrhagic polycystic ovaries and hyperplasia of the theca externa. This study demonstrates that the ERalpha is the functional key to the pathogenesis of PCOS.

Case Report

A Case of Paraduodenal Pancreatitis and Immunohistochemical Analysis.: Mi Jung Kwon, Eun Sook Nam, Seong Jin Cho, Hyung Sik Shin, Joo Seop Kim, Doo Jin Kim; Korean J Pathol. 2010;44(2):199-203.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.2.199

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Abstract PDF: Paraduodenal pancreatitis (PP) is a rare, distinct form of chronic pancreatitis, and it is related to alcohol abuse in middle-aged men. A 36-year-old man with a history of chronic recurrent pancreatitis for 4 years and alcohol abuse for 15 years presented with abdominal pain. Computed tomography revealed a multilocular cystic mass 3.2 x 3 x 3 cm in size and it was located within the muscular layer of the duodenal wall. The cysts were lined by a single layer of eosinophilic cuboidal epithelial cells that stained positively for mucin (MUC)1, MUC6, cytokeratin (CK)7 and CK19 and they stained negatively for MUC2, MUC5AC and CK5/6. Mild, chronic inflammatory reaction around the cystic wall, Brunner's gland hyperplasia and several clusters of heterotopic pancreatic tissue were noted. We report here on a case of PP and we demonstrated that the pancreatitis was of pancreatic ductal cell origin according to the MUC and CK expression patterns we observed on the immunohistochemical analysis.

Original Article

Congenital Cystic Disease of the Kidney overview and a classification.: Mee Joo, Yeon Mee Kim, Chong Jai Kim, Yeon Lim Suh, Jeong Wook Seo, Je Geun Chi; Korean J Pathol. 1997;31(3):233-243.

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Abstract PDF: The congenital renal cystic disease encompasses a complex group of pathologic and clinical entities. We retrospectively reviewed 42 cases of congenital renal cystic lesions classified into four Potter types in a series of 2,063 consecutive autopsies from 1981 to 1996. According to our study based on morphologic, clinical, genetic features and associated anomalies, type I and III are relatively compatible with Potter's original definition. However, it was reasonable that type II and IV are classified to the same group because of: 1) very similar histologic findings representing dysplastic kidney, 2) many associated anomalies, 3) no evidence of inheritance, and 4) presence of a combined type. Syndrome associated cysts, such as Meckel-Gruber syndrome, were also separately classified. If the dysplastic evidence was insufficient for diagnosis to the dysplastic kidney in type II and IV, then these cases would be better classified into a cystic disease associated with congenital hydronephrosis. We propose a classification of the congenital cystic disease of the kidney to be: 1) dysplastic kidney, 2) cystic disease associated with congenital hydronephrosis, 3) polycystic kidney, and 4) syndromic cystic disease.

Case Reports

Giant Cystic Adenomatoid Tumor of the Uterus: A Case Report.: Mi Jin Kim, Mi Jin Gu; Korean J Pathol. 2004;38(6):415-418.

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Abstract PDF: Adenomatoid tumor of the uterus is a rare benign tumor of the uterine serosa and myometrium. It usually appears as a small nodular lesion having a distinctive histologic appearance. We describe a case of 67-year old female with a large, grossly cystic adenomatoid tumor 7x7 cm in dimension that was located at the right uterine cornus. Microscopically the tumor had unusually extensive solid proliferation of signet ring cells with large vacuoles in their cytoplasm. Also, adenoid and honeycomb-like cystic patterns were observed. The immunohistochemical and ultrastructural studies demonstrated the mesothelial origin.

Granulosa Cell Tumor of the Unilocular Cystic Type: A Case Report.: Kyu Yun Jang, Myoung Ja Chung, Woo Sung Moon, Myoung Jae Kang, Dong Geun Lee; Korean J Pathol. 2004;38(4):284-287.

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Abstract PDF: Unilocular cystic granulosa cell tumors (UCG) are extremely rare. Due to the relatively small mass of the tumor available for histologic examination, diagnosis of UCG is not easy. Here we present a case of UCG in a 54-year old female. A 12x10 cm unilocular cystic mass was identified in the right ovary. The tumor was thin-walled and consisted of a single large cavity with a smooth internal surface. In most areas there were no cells lining the cyst, however, in focal areas the unilocular cyst was lined by one or more layers of uniform granulosa cells, forming Call-Exner bodies. A diagnosis of UCG was made after multiple sections were examined. A careful histologic examination is required for the correct diagnosis of UCG.

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