Journal of Pathology and Translational Medicine

Case Study

Encapsulated Papillary Thyroid Tumor with Delicate Nuclear Changes and a KRAS Mutation as a Possible Novel Subtype of Borderline Tumor: Kenji Ohba, Norisato Mitsutake, Michiko Matsuse, Tatiana Rogounovitch, Nobuhiko Nishino, Yutaka Oki, Yoshie Goto, Kennichi Kakudo; J Pathol Transl Med. 2019;53(2):136-141. Published online January 14, 2019; DOI: https://doi.org/10.4132/jptm.2018.12.07

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Although papillary thyroid carcinoma (PTC)–type nuclear changes are the most reliable morphological feature in the diagnosis of PTC, the nuclear assessment used to identify these changes is highly subjective. Here, we report a noninvasive encapsulated thyroid tumor with a papillary growth pattern measuring 23 mm at its largest diameter with a nuclear score of 2 in a 26-year-old man. After undergoing left lobectomy, the patient was diagnosed with an encapsulated PTC. However, a second opinion consultation suggested an alternative diagnosis of follicular adenoma with papillary hyperplasia. When providing a third opinion, we identified a low MIB-1 labeling index and a heterozygous point mutation in the KRAS gene but not the BRAF gene. We speculated that this case is an example of a novel borderline tumor with a papillary structure. Introduction of the new terminology “noninvasive encapsulated papillary RAS-like thyroid tumor (NEPRAS)” without the word “cancer” might relieve the psychological burden of patients in a way similar to the phrase “noninvasive follicular thyroid neoplasm with papillary-like nuclear features (NIFTP).”

Citations

Citations to this article as recorded by

The Asian Thyroid Working Group, from 2017 to 2023
Kennichi Kakudo, Chan Kwon Jung, Zhiyan Liu, Mitsuyoshi Hirokawa, Andrey Bychkov, Huy Gia Vuong, Somboon Keelawat, Radhika Srinivasan, Jen-Fan Hang, Chiung-Ru Lai
Journal of Pathology and Translational Medicine.2023; 57(6): 289. CrossRef
Whole Tumor Capsule Is Prognostic of Very Good Outcome in the Classical Variant of Papillary Thyroid Cancer
Carlotta Giani, Liborio Torregrossa, Teresa Ramone, Cristina Romei, Antonio Matrone, Eleonora Molinaro, Laura Agate, Gabriele Materazzi, Paolo Piaggi, Clara Ugolini, Fulvio Basolo, Raffaele Ciampi, Rossella Elisei
The Journal of Clinical Endocrinology & Metabolism.2021; 106(10): e4072. CrossRef
Evidence of Cooperation between Hippo Pathway and RAS Mutation in Thyroid Carcinomas
Thaise Nayane Ribeiro Carneiro, Larissa Valdemarin Bim, Vanessa Candiotti Buzatto, Vanessa Galdeno, Paula Fontes Asprino, Eunjung Alice Lee, Pedro Alexandre Favoretto Galante, Janete Maria Cerutti
Cancers.2021; 13(10): 2306. CrossRef
Capsular Invasion Matters Also in “Papillary Patterned” Tumors: A Study on 121 Cases of Encapsulated Conventional Variant of Papillary Thyroid Carcinoma
Dilara Akbulut, Ezgi Dicle Kuz, Nazmiye Kursun, Serpil Dizbay Sak
Endocrine Pathology.2021; 32(3): 357. CrossRef
Noninvasive encapsulated papillary RAS-like thyroid tumor (NEPRAS) or encapsulated papillary thyroid carcinoma (PTC)
Pedro Weslley Rosario
Journal of Pathology and Translational Medicine.2020; 54(3): 263. CrossRef
Updates in the Pathologic Classification of Thyroid Neoplasms: A Review of the World Health Organization Classification
Yanhua Bai, Kennichi Kakudo, Chan Kwon Jung
Endocrinology and Metabolism.2020; 35(4): 696. CrossRef

Original Article

A Serous Papillary Cystadenoma of Low Malignant Potential in Paratesticular Tissue.: Keum Min Park, Nam Bok Cho, Kye Yong Song; Korean J Pathol. 1996;30(5):463-465.

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Abstract PDF: The serous papillary cystadenoma of low malignant potential in the paratesticular tissue is a tumor characterized by its resemblance to ovarian serous papillary tumor of low malignant potential. Several possible origins have been proposed but it is thought to originate from the Mullerian-type epithelium just like ovarian serous tumor of low grade malignancy. Because of the rarity of this tumor, the natural history and treatment have not been established. Herein we report on a serous papillary cystadenoma arising in the paratesticular tissue of a 12 year old . To our knowledge, this is the first reported case in the Korean literature. The size of this cystic tumor in this patient was 0.9x0.9x0.8 cm and had a sponge-like cut surface. The papillary structure was lined by both ciliated and nonciliated secretory cells, the same cellular structure as the salpinx. Areas with just a few mitoses and stratification of epithelial cells were noted, but invasion into the paratesticular tissue was absent, suggesting borderline malignancy. He is doing well after 5 months follow up. Prognosis of this scrotal tumor is thought to be better than the ovarian tumor of similar histology due to a better possibility of early detection.

Case Reports

Papillary Serous Cystadenoma of Borderline Malignancy Arising from a Paramesonephric Parovarian Cyst: A case report.: Ji Han Jung, Youn Soo Lee, Seok Jin Kang, Byung Kee Kim, Sang In Shim; Korean J Pathol. 1999;33(6):457-459.

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Abstract PDF: Primary malignant parovarian epithelial tumors are extremely rare, with only 56 cases previously reported in the world literature. Most parovarian epithelial tumors are of paramesonephric (Mullerian) origin. In this article, we report the first case in the Korean literature of papillary serous cystadenoma of borderline malignancy from paramesonephric parovarian cyst. This case presented here is of particular interest since this neoplasm is rare. A 48-year-old female underwent a hysterectomy with bilateral adnexectomy and pelvic lymph node dissection for a cystic tumor of the right parovarian area. The tumor, measuring 13 cm in diameter, was apart from the ovaries, fallopian tubes and uterus. The cyst wall had well-developed smooth muscle layers. The cyst was lined with mildly atypical ciliated and non-ciliated low columnar cells compatible with those of paramesonephric origin. From the inner surface of the cyst several cauliflower-like structures projected into the lumen. They were of a predominantly papillary architecture covered by atypical epithelial cells with piling-up and occasional glandular growth. No destructive stromal invasion was identified.

Borderline Clear Cell Adenofibromatous Tumors of the Ovary: Two Case Reports.: Heejeong Lee, Tae Jung Kim, Jeana Kim, Eun Joo Seo, Kyo Young Lee; Korean J Pathol. 2007;41(6):420-423.

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Abstract PDF: Borderline clear cell adenofibromatous tumors are rare with only 26 cases reported in the English literature. Five of these cases exhibited microinvasion and 4 demonstrated intraepithelial carcinoma. We report 2 cases, one typical case and the other with microinvasion. The histological findings revealed widely spaced and focally crowded, variably-sized atypical glands or tubules lined by clear, eosinophilic or hobnail cells set in a dense fibrous stroma. One of the two cases had small solid nests or single cells in the stroma around the proliferative glands less than 1 mm in length that was considered to be a microinvasion.

Original Article

Ovarian Borderline Epithelial Tumors.: Geunghwan Ahn; Korean J Pathol. 2005;39(5):291-300.

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Abstract PDF: Ovarian borderline epithelial tumors are abnormal proliferative epithelial lesions without obvious invasion of the stroma of the ovary, a finding distinguishing between borderline tumors and carcinoma. There have been controversies regarding the terminology and diagnostic feature of the tumors, even though these tumors have been accepted as a distinct entity in WHO classification of ovarian epithelial tumors. This review is limited to serous and mucinous borderline tumors which are the most common and about which many clinicopathological studies have been undertaken. It has been agreed that "micropapillary carcinoma" espoused by a group of pathologists is a micropapillary variant of serous borderline tumor in the borderline ovarian tumor workshop. Diagnostic criteria of invasive implants needs further study but invasion of underlying normal tissue was reported to be correlated well with prognosis. Other issues such as diagnostic criteria of microinvasion and multiplicity of serous borderline tumors have been presented. The sole diagnostic criteria agreed upon for the diagnosis of intraepithelial carcinoma in the mucinous borderline tumor was the presence of severe cytological atypia. It was also agreed that the ovarian tumors associated with pseudomyxoma peritonei are almost invariably from gastrointestinal tract, usually appendix. Stratification and complex intracystic growth without severe cytological atypia are considered to be characteristics of mucinous borderline tumors. Diagnostic criteria of microinvasion and two types of invasion, expansile and infiltrative invasion, have also been discussed.

In Vitro

Ovarian Serous Borderline Tumors with Peritoneal Implants: A clinicopathologic and flow cytometric DNA analysis of 5 cases.: Kyu Rae Kim, Kwang Yul Cha, Soon Hee Jung, Woo Hee Jung, Dong Hee Choi, Jong Wook Kim; Korean J Pathol. 1993;27(2):143-151.

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Abstract PDF: Five cases of ovarian serous borderline tumor associated with multiple peritoneal implants were analysed. The age of 5 patients ranged from 34 to 45(mean: 39) years at the time of diagnosis. Two patients presented with secondary infertility underwent hyperstimulation of the ovary under the in vitro fertilization program. One patient was presented with abdominal pain and the remaining two with lower abdominal mass accompanied by abdominal pain. Serous surface papilloma of borderline malignancy, involving one or both ovaries, was present in all 5 cases and papillary serous cystic tumor of borderline malignancy was associated simultaneously in one or both ovaries in all cases. Marked adhesions between the pelvic organs, and multiple granularities and nodularities of the omentum and peritoneum were noted. Microscopically, the peritoneal lesions were composed of noninvasive implants of tumor cells and pasmmoma bodies on the surface of entire pelvic and abdominal organs. Flow cytometric analysis of nuclear DNA content from paraffin-embedded tissue fo primary ovarian tumor classified as aneuploidy in 3 cases and as diploidy in 2 cases. The DNA index of the aneuploid tumors ranged from 1.21 to 1.37. Four patients underwent hysterectomy, bilateral salpingo-oophorectomy and partial omentectomy in which two were followed by chemotherapy and one case underwent bilateral salpingo-oophorectomy.

Case Report

Retroperitoneal Mucinous Tumor: Report of two Cases.: Eun Kyung Kim, Seong Ran Hong, Hy Sook Kim, Young Hyeh Ko, Jung Dal Lee; Korean J Pathol. 1992;26(6):632-634.

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Abstract PDF: Primary retroperitoneal mucinous tumor is very rare. In the worldwide literature, only 14 cases have been reported. Herein, we report two cases of retroperitoneal mucinous tumor found at laparotomy. The patients were females, aged 24 and 21 years, respectively. They had bilateral normal ovaries. Histologically, one was mucinous cystadenoma and the other showed borderline histology. The connective tissue wall resembling ovarian stroma was noted in one case. The pathogenesis was discussed.

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