Primary Male Appendiceal Choriocarcinoma With Background of Low-Grade Appendiceal Mucinous Neoplasm: Diagnostic Challenges and Poor Prognosis.
4 affiliations
- 1Rio Americano High School, Sacramento, CA, U.S.A.
- 2School of Nursing, Pacific Union College, Angwin, CA, U.S.A.
- 3Department of Biochemistry and Molecular Medicine, University of California, Davis Medical Center, Sacramento, CA, U.S.A.
- 4Department of Pathology and Laboratory Medicine, Kaiser Permanente Sacramento Medical Center, Sacramento, CA, U.S.A.
Abstract
Background/aim
Choriocarcinoma is a highly aggressive trophoblastic malignancy that is most commonly gestational in origin. Non-gestational choriocarcinoma in the male gastrointestinal tract is extremely rare, and primary appendiceal involvement is exceptional.Case report
We report such a case of a 75-year-old male who presented to the emergency room with severe abdominal pain. Physical examination indicated possible acute appendicitis suspicious for perforation, and computed tomography (CT) scans revealed a 7.5 cm mass in the lower right quadrant as well as numerous liver hypoechoic nodules. No lesions/nodules were identified in his testicles or mediastinum. Emergency surgery with right hemicolectomy and concurrent liver biopsy was performed. Pathology gross exam revealed a 7.5 cm appendiceal mass with associated appendiceal perforation. Histologically, the tumor showed a poorly differentiated carcinoma morphologically composed of syncytiotrophoblasts, cytotrophoblasts and intermediate trophoblasts with adjacent low-grade appendiceal mucinous neoplasm (LAMN). The carcinoma cells were positive for cytokeratin 7 (CK7), beta-human chorionic gonadotropin (β-hCG), GATA-binding protein 3 (GATA 3) and Sal-like protein 4 (SALL4). These features supported a primary appendiceal choriocarcinoma and the liver biopsy demonstrated metastasis of choriocarcinoma with extensive tumor necrosis. Post-surgery lab tests also revealed an exceptionally high serum β-hCG titer of 289,308 mIU/ml. The patient underwent hospice care after surgery and expired one month later.Conclusion
This is the first documented case of male primary choriocarcinoma with background of LAMN and poor prognosis. Recognition of the three carcinoma components in combination with the corresponding tumor immunohistochemical markers is the key for the precise diagnosis.
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BibTeX
@article{feng2026primary,
title = {{Primary Male Appendiceal Choriocarcinoma With Background of Low-Grade Appendiceal Mucinous Neoplasm: Diagnostic Challenges and Poor Prognosis.}},
author = {Alicia Feng and Katherine Y Bai and Ying Gao and Ruohao Fan and Yuanpei Li and Jinping Lai},
journal = {Cancer diagnosis \& prognosis},
year = {2026},
month = nov,
doi = {10.21873/cdp.10606},
url = {https://europepmc.org/article/MED/42968948},
note = {CC BY-NC-ND},
}
RIS
TY - JOUR
TI - Primary Male Appendiceal Choriocarcinoma With Background of Low-Grade Appendiceal Mucinous Neoplasm: Diagnostic Challenges and Poor Prognosis.
AU - Alicia Feng
AU - Katherine Y Bai
AU - Ying Gao
AU - Ruohao Fan
AU - Yuanpei Li
AU - Jinping Lai
PY - 2026
DA - 2026/11/01
JO - Cancer diagnosis & prognosis
DO - 10.21873/cdp.10606
UR - https://europepmc.org/article/MED/42968948
AB - Background/aim Choriocarcinoma is a highly aggressive trophoblastic malignancy that is most commonly gestational in origin. Non-gestational choriocarcinoma in the male gastrointestinal tract is extremely rare, and primary appendiceal involvement is exceptional.Case report We report such a case of a 75-year-old male who presented to the emergency room with severe abdominal pain. Physical examination indicated possible acute appendicitis suspicious for perforation, and computed tomography (CT) scans revealed a 7.5 cm mass in the lower right quadrant as well as numerous liver hypoechoic nodules. No lesions/nodules were identified in his testicles or mediastinum. Emergency surgery with right hemicolectomy and concurrent liver biopsy was performed. Pathology gross exam revealed a 7.5 cm appendiceal mass with associated appendiceal perforation. Histologically, the tumor showed a poorly differentiated carcinoma morphologically composed of syncytiotrophoblasts, cytotrophoblasts and intermediate trophoblasts with adjacent low-grade appendiceal mucinous neoplasm (LAMN). The carcinoma cells were positive for cytokeratin 7 (CK7), beta-human chorionic gonadotropin (β-hCG), GATA-binding protein 3 (GATA 3) and Sal-like protein 4 (SALL4). These features supported a primary appendiceal choriocarcinoma and the liver biopsy demonstrated metastasis of choriocarcinoma with extensive tumor necrosis. Post-surgery lab tests also revealed an exceptionally high serum β-hCG titer of 289,308 mIU/ml. The patient underwent hospice care after surgery and expired one month later.Conclusion This is the first documented case of male primary choriocarcinoma with background of LAMN and poor prognosis. Recognition of the three carcinoma components in combination with the corresponding tumor immunohistochemical markers is the key for the precise diagnosis.
SN - 2732-7787
ER -