Journal of Pathology and Translational Medicine

Case Study

Ovarian Gynandroblastoma with a Juvenile Granulosa Cell Tumor Component in a Postmenopausal Woman: A Case Report and Literature Review: Nu Ri Jang, Dae Hyung Lee, Eun Jung Jang, Young Kyung Bae, Jina Baek, Min Hye Jang; J Pathol Transl Med. 2018;52(5):344-348. Published online July 17, 2018; DOI: https://doi.org/10.4132/jptm.2018.06.28

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Gynandroblastoma is an extremely rare sex cord-stromal tumor with both female (granulosa cell tumor) and male (Sertoli-Leydig cell tumor) elements. Juvenile granulosa cell tumors are also very rare and are so named because they usually occur in children and adolescents. A 71-year-old woman with right upper quadrant abdominal pain visited our hospital. Pelvic computed tomography showed a large multilocular cystic mass, suspected to be of ovarian origin. We performed a total abdominal hysterectomy (total abdominal hysterectomy was performed) with bilateral salpingooophorectomy. A 13-cm multilocular cystic mass with serous fluid was observed in her right ovary. Upon microscopic examination, the solid component of the mass showed both Sertoli-Leydig cell and juvenile granulosa cell differentiation, which we diagnosed as gynandroblastoma. Gynandroblastoma with a juvenile granulosa cell tumor component is extremely rare and, until now, only six cases have been reported in the English literature. We report the first gynandroblastoma with a juvenile granulosa cell tumor component diagnosed in an elderly patient, along with a literature review.

Citations

Citations to this article as recorded by

Case of Gynandroblastoma of the Ovary with Raised AFP and Associated DICER 1 Mutation
Dipak Limbachiya, Rajnish Tiwari, Rashmi Kumari, Priti Trivedi
The Journal of Obstetrics and Gynecology of India.2024;[Epub] CrossRef
Ovarian cancer in children and adolescents: A unique clinical challenge
Marina Jakimovska Stefanovska, Aleksandar Celebic, Jean Calleja-Agius, Kristina Drusany Staric
European Journal of Surgical Oncology.2024; : 108785. CrossRef
Ovarian Gynandroblastoma with a Juvenile Granulosa Cell Tumor Component in a Postmenopausal Woman
Soohyun Hwang, Byoung-Gie Kim, Sang Yong Song, Hyun-Soo Kim
Diagnostics.2020; 10(8): 537. CrossRef
Clinical and histological criteria for sex cord ovarian stromal tumors
A. М. Beishembaev, K. I. Zhordania
Obstetrics, Gynecology and Reproduction.2020; 14(3): 261. CrossRef

Case Reports

Secretory Carcinoma of the Breast: A case report.: Kyu Rae Kim, Jung Hyun Yang, Yeon Lim Seo, Howe Jung Ree; Korean J Pathol. 1996;30(4):347-350.

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Abstract PDF: We report a case of secretory carcinoma with axillary lymph node metastasis in a 21-year old woman. She was aware of a mass in her breast for 10 years and noticed a rapid growth of the preexisting mass during the last years. Histologically, the tumor was composed of micropapillary and microcystic or cribriform glandular structures which contained eosinophilic, mucinous, intraluminal secretions. The center had a dense hyalinized strama with a solid infiltrative growth of tumor cells with intracytoplasmic secretory vacuoles at the periphery. In addition, marked intraductal papillary epithelial proliferations were present at the superficial portions of the tumor near the nipple. Prognostic factors and their relationship to juvenile papillomatosis are discussed with a review of the literature.

Familial Juvenile Polyposis.: Sun Hee Chang, Shi Nae Lee, Hea Soo Koo, Ok Kyung Kim, Sun Sub Jung, Eung Bum Park; Korean J Pathol. 1997;31(2):185-188.

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Abstract PDF: Familial juvenile polyposis is a rare intestinal polyposis characterized by the occurrence of multiple juvenile polyps in the gastrointestinal tract. We report a case of familial juvenile polyposis in a 17-year-old man with a review of the literature. This patient underwent total colectomy due to a 6 years history of rectal bleeding. Grossly, the colon showed 36 variable sized pedunculated polyps, measuring 2.5cm x 2cm from the largest size and 0.2cm x 0.2cm to the smallest size. Histologically, the polyps consisted of cystically dilated glands, lined by normal colonic epithelial cells, scattered in loose, edematous stroma containing inflammatory cell infiltration. There were no areas of tubular adenoma or malignancy in any of the polyp.

Original Article

Gastrointestinal Polyposis in Koreans: A Nationwide Survey of Clinicopathologic Analysis of 112 Surgically Resected Cases.: Mee Soo Chang, Hoguen Kim, Woo Ho Kim, Chan Il Park, Eun Kyung Hong, Han Kyeom Kim, In Soo Suh, Byung Kee Kim, Ja June Jang, Woon Sub Han, Hyung Sik Shin, So Young Jin, Dae Young Kang, Yong Il Kim; Korean J Pathol. 1998;32(6):404-412.

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Abstract: Gastrointestinal polyposis (GIP) is a rare disease characterized by formation of the numerous polyps in the gastrointestinal tract and presenting several extraintestinal manifestations. Most of the diseases are transmitted in an autosomal dominant pattern. In Korea, the epidemiological study as well as the pathological analysis of the GIP is not well established. We therefore analysed 38 items of GIP using surgically resected specimens. The materials in this study were collected from the 12 institutions and case reports in Korean literature between 1980 and 1991. The clinicopathologic findings were reevaluated by several members of the study group for gastrointestinal pathology. The results are as follows: (1) A total of 112 cases were included in this study: 83 cases were collected from 12 institutions and 29 cases were collected from Korean literature. The cases were classified as familial adenomatous polyposis (FAP), 59 cases; Gardner's syndrome, 3 cases; juvenile polyposis, 12 cases; Peutz-Jeghers syndrome, 35 cases; multiple colonic adenomas, 3 cases. (2) Among 59 cases of FAP, the range of age at operation was 14 to 61 years, and a family history was positive in 25 cases. The number of polyps in colorectum was 100~8,000. Of the 37 cases in which the examination of polyp density was available, 16 cases (43%) showed the highest density in the rectum and the sigmoid colon. The carcinomatous change within polyp(s) was present in 18 cases (31%), and associated advanced single or multiple colonic carcinomas existed in 37 cases (63%). Twenty-six (45%) tumors out of total 58 carcinomas were in the rectum. Twenty-five patients were evaluated for the upper gastrointestinal lesions, and 11 patients (44%) had pathologic lesions; multiple fundic gland polyps in 3 cases (12%), gastric and duodenal adenomas in 2 cases (8%), gastric adenomas in 2 cases (8%), duodenal adenomas in 2 cases (8%), gastric carcinoma and adenoma in 1 case (4%), gastric carcinoma in 1 case (4%). (3) Among 3 cases of Gardner's syndrome, the range of age at operation was 25 to 31 years, a family history was identified in 2 cases. One case was associated with an advanced colonic carcinoma and carcinomatous change within polyp. Extra gastrointestinal lesions were sebaceous cyst, epidermal cyst, osteoma and desmoid tumor. (4) Among 12 juvenile polyposis, the range of age at operation was 8 to 51 years and 5 patients had a family history. The carcinomatous change within polyp was found in 2 cases (17%) and associated advanced colonic carcinoma was in 4 cases (33%). The associated different type of polyps was tubular adenomas in 9 cases (75%), hyperplastic polyps in 4 cases (33%) and villous adenomas in 2 cases (17%). (5) Among 35 Peutz-Jeghers syndrome, the range of age at first operation was 6 to 42 years, family history was positive in 11 cases. The carcinomatous change within polyp was found in 1 case (3%), and associated advanced colonic carcinoma in 1 case (3%). The epithelial misplacement was observed in 4 cases (11%), and tubular or villous adenomatous feature in 4 cases (11%). In summary, the most frequent GIP for the surgical resection in Korea is FAP and the FAP is associated with high incidence of coexisting advanced and intramucosal carcinomas. Hamartomatous polyposis syndromes, such as juvenile polyposis and Peutz-Jeghers syndrome are another frequent disease for the surgical resection and are also associated with an increased risk of cancer.

Case Report

Juvenile Granulosa Cell Tumor Arising in Undescended Testis: A case report.: So Ya Paik, Hae Youn Kang, Jae Ho Han, Woo Ick Yang, Seok Joo Han; Korean J Pathol. 1999;33(5):376-379.

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Abstract PDF: Juvenile granulosa cell tumor is rare but one of the common congenital testicular neoplasms. Although histological features are typical of its ovarian counterpart, testicular juvenile granulosa cell tumor has a distinctly different clinical presentation. We report a case of juvenile granulosa cell tumor arising in the cryptochid testis of a 4-day-old newborn. A 6 5 5 cm sized multilocular cyst containing thick, mucinous fluid was found in the peritoneal cavity. The external surface of the cyst was smooth and the septae were relatively thin. The cyst consisted of numerous mucin-filled, cystic follicles lined by cells having vacuolated cytoplasm and round to oval dark nuclei without grooves. Cells resembling granulosa cells of an ovarian follicle were also observed in the intervening stroma forming irregular solid nests.

Original Articles

Juvenile Hyaline Fibromatosis in an Adult.: Young A Kim, Seoung Wan Chae, Chong Jai Kim, Je G Chi; Korean J Pathol. 2000;34(3):239-242.

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Abstract PDF: Juvenile hyaline fibromatosis is a rare disorder probably inherited as an autosomal recessive trait. It is characterized by multiple slowly growing subcutaneous nodules, hypertrophy of gingiva, flexion contracture, and radiolucent bone destruction. The histological features of the tumor-like lesions are characterized by the deposition of amorphous hyaline material in which spindle shaped cells are embedded. We report a case of juvenile hyaline fibromatosis in a 26 year-old-woman. She had multiple subcutaneous nodules in scalp, ear, forearms, right knee, and back. Surgical excision of the tumors in the scalp and ear was done. The largest one measured 13 9 6 cm, and had homogeneous, grayish yellow cut surface with calcification. Light microscopic examination showed abundant eosinophilic hyaline material with extensive calcification and metaplastic bone formation. Spindle cells were rarely observed at the periphery of the tumor. Hyaline matrix was PAS positive, diastase resistant, and alcian blue negative. Scattered spindle cells were positive for vimentin but negative for S-100 protein and smooth muscle actin. There were many reports regarding early lesions of juvenile hyaline fibromatosis; however in this patient, tumor existed for more than 20 years and the histology was somewhat different from the early lesions reported in the literature.

Juvenile Ossifying Fibroma: A Clinicopathologic Study of 8 Cases and Comparison with Craniofacial Fibro-osseous Lesions.: Sohyung Park, Bong Jae Lee, Jeong Hyun Lee, Kyung Ja Cho; Korean J Pathol. 2007;41(6):373-379.

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Abstract PDF: BACKGROUND
Juvenile ossifying fibroma (JOF) is defined as a variant of the ossifying fibroma, and the latter includes juvenile trabecular ossifying fibroma (JTOF) and juvenile psammomatoid ossifying fibroma (JPOF). JOF can be distinguished from other craniofacial fibroosseous lesions by its tendency to recur and its clinical mimicry of malignant bone tumors, but some clinical and histological features of JOF overlap with the other fibro-osseous lesions as well. We aimed to identify the clinicopathologic definition of JOF.
METHODS
Forty-two cases of fibro-osseous lesions were reviewed and they were classified into JOF, fibrous dysplasia (FD) and ossifying fibroma (OF).
RESULTS
JTOF had long, slender and anastomosing trabeculae of osteoid in a fibrocellular stroma, and JPOF had small ossicles resembling psammoma bodies with a thick collagenous rim in the fibrous stroma, which are features that differ from those of FD and OF. Radiologically, JOF and OF showed a well-defined lesion but FD exhibited an ill-defined lesion. Clinically, the average age of the JOF patients was the youngest, followed by OF and FD. For JOF, three cases had rapid growth and two others showed recurrences. JOF mainly occurred in the paranasal sinuses, OF in the mandible and FD in any craniofacial bone.
CONCLUSION
We demonstrated the distinct characteristics of JOF and these features may be helpful for the diagnosis and management of this malady.

Case Reports

Juvenile Xanthogranuloma of the Nasal Cavity: A Case Report and Review of the Literature.: Jung Suk An, Sun Hee Han, Ju Han Lee, Eung Seok Lee, Heum Rye Park, Young Sik Kim; Korean J Pathol. 2006;40(3):241-244.

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Abstract PDF: Juvenile xanthogranuloma (JXG) is a benign and self-limiting non-Langerhans-cell histiocytosis that generally occurs during infancy and childhood. It develops frequently in the head and neck but is very rare in the nasal cavity. To date, only five cases of JXG in the nasal cavity have been reported. Here, we report the second case of JXG in the nasal cavity in Korea. A 19-year-old male patient presented with a protruding 1.1 cm mass in the left nasal vestibule. Histologically, a dense dermal infiltrate of histiocytes with Touton giant cells was observed. Immunohistochemically, the histiocytes tested positive for CD68 and the S-100 protein but negative for CD1a. This shows that a S-100-positive histiocytic lesion dose not exclude a diagnosis of JXG.

Juvenile Granulosa Cell Tumor of the Ovary: Report of a Case of Malignant Form with Unusual Pleomorphism.: Chong Jai Kim, Jin Suk Suh, Sung Hye Park, Je G Chi; Korean J Pathol. 1990;24(3):316-320.

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Abstract PDF: A case of Juvenile Granulosa Cell Tumor (JGCT) of the ovary with unusual pleomorphic histologic and malignant biologic behaviour is described. The tumor occurred in a 10-year-old girl and was associated with clinical features of isosexual pseudoprecosity and a marked elevation of serum estradiol. The mass manifested initially in the right ovary and subsequently involved the contralateral ovary. A multi-organ metastasis developed during a 6-month-interval despite chemotherapy. She received two operations at 6-month interval, and tissues were obtained from the tumor mass. A marked histologic difference was observed between these two samples. The second biopsy showed profound cellular pleomorphism with numerous multinucleated tumor giant cell formation and hyaline bodies. The differential diagnosis from germ cell tumor and the possible factors for the pleomorphism are discussed.

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