Abstract
We report a case of alpha-fetoprotein-producing endometrioid carcinoma (AFP-EC) that originated within an adenomyoma of the uterine corpus. A 76-year-old Japanese woman was incidentally discovered to have a uterine tumor along with multiple lung nodules. Upon surgical removal of the uterus, it was revealed that the tumor was situated within the adenomyoma. The tumor exhibited microfollicular structures and solid growth patterns, with hyaline globules, clear cell glands, and primitive tumor cells. Immunohistochemical analysis indicated the presence of germ cell markers, including AFP, SALL4, and glypican3, leading to final diagnosis of AFP-EC. Histopathologically, AFP-ECs exhibit characteristics similar to those of AFP-producing neoplasms in other organs. Furthermore, a nomenclature issue arises when distinguishing AFP-ECs from yolk sac tumors of the endometrium in older patients due to their shared features. The concept of retrodifferentiation or neometaplasia suggests that “endometrioid carcinoma with yolk sac tumor differentiation” or “endometrioid carcinoma with a primitive phenotype” may serve as more fitting terms for the diverse spectrum of AFP-producing neoplasms in the endometrium. In conclusion, this case underscores the diagnostic challenges posed by AFP-ECs arising from adenomyomas and emphasizes the need for refining the nomenclature and classification of AFP-producing neoplasms within the endometrium.
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The data that supports the findings reported in this study are available from the corresponding author, HN, upon reasonable request.
References
McIntire KR, Waldmann TA, Moertel CG, Go VL (1975) Serum alpha-fetoprotein in patients with neoplasms of the gastrointestinal tract. Cancer Res 35:991–996
Wilson D, Bordoni B (2023) Embryology, mullerian ducts (paramesonephric ducts). In: StatPearls. StatPearls Publishing. https://www.ncbi.nlm.nih.gov/books/NBK557727/. Accessed 20 Jan 2024
Kawagoe K (1985) A case of mixed mesodermal tumor of the uterus with alpha-fetoprotein production. Jpn J Clin Oncol 15:577–583
Otani T, Murakami K, Shiraishi N, Hagiyama M, Satou T, Matsuki M, Matsumura N, Ito A (2021) α-fetoprotein-producing endometrial carcinoma is associated with fetal gut-like and/or hepatoid morphology, lymphovascular infiltration, TP53 abnormalities, and poor prognosis: five cases and literature review. Front Med (Lausanne) 8:799163
Kinjo T, Taniguchi H, Kushima R, Sekine S, Oda I, Saka M, Gotoda T, Kinjo F, Fujita J, Shimoda T (2012) Histologic and immunohistochemical analyses of α-fetoprotein—producing cancer of the stomach. Am J Surg Pathol 36:56–65
Papadimitriou JC, Drachenberg CB, Brenner DS, Newkirk C, Trump BF, Silverberg SG (2000) ‘Thanatosomes’: a unifying morphogenetic concept for tumor hyaline globules related to apoptosis. Hum Pathol 31:1455–1465
Tran TA, Ortiz HB, Holloway RW, Bigsby GE, Finkler NJ (2007) Alpha-fetoprotein-producing serous carcinoma of the uterus metastasizing to the ovaries, mimicking primary ovarian yolk sac tumor: a case report and review of the literature. Int J Gynecol Pathol 26:66–70
Wu QY, Wan XY, Xie X, Lu BJ (2014) Endometrial hepatoid adenocarcinoma: a rare cause of elevated serum α-fetoprotein. J Obstet Gynaecol Res 40:873–877
Nogales FF, Preda O, Nicolae A (2012) Yolk sac tumours revisited. A review of their many faces and names. Histopathology 60:1023–1033
McNamee T, Damato S, McCluggage WG (2016) Yolk sac tumours of the female genital tract in older adults derive commonly from somatic epithelial neoplasms: somatically derived yolk sac tumours. Histopathology 69:739–751
Ravishankar S, Malpica A, Ramalingam P, Euscher ED (2017) Yolk sac tumor in extragonadal pelvic sites: still a diagnostic challenge. Am J Surg Pathol 41:1–11
Ramalingam P, Masand RP, Euscher ED, Malpica A (2016) Undifferentiated carcinoma of the endometrium: an expanded immunohistochemical analysis including PAX-8 and basal-like carcinoma surrogate markers. Int J Gynecol Pathol 35:410–418
Yamazawa S, Ushiku T, Shinozaki-Ushiku A, Hayashi A, Iwasaki A, Abe H, Tagashira A, Yamashita H, Seto Y, Aburatani H, Fukayama M (2017) Gastric cancer with primitive enterocyte phenotype: an aggressive subgroup of intestinal-type adenocarcinoma. Am J Surg Pathol 41:989–997
Sampson JA (1925) Endometrial carcinoma of the ovary, arising in endometrial tissue in that organ. Arch Surg 10:1–72
Machida H, Maeda M, Cahoon SS, Scannell CA, Garcia-Sayre J, Roman LD, Matsuo K (2012) Endometrial cancer arising in adenomyosis versus endometrial cancer coexisting with adenomyosis: are these two different entities? Arch Gynecol Obstet 295:1459–1468
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Oyama, Y., Kusaba, T., Takao, K. et al. Alpha-fetoprotein producing endometrioid carcinoma arising in an adenomyoma of the uterus. Med Mol Morphol (2024). https://doi.org/10.1007/s00795-024-00384-1
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DOI: https://doi.org/10.1007/s00795-024-00384-1