原发子宫肝样腺癌2例临床病理分析及文献复习

doi:10.19723/j.issn.1671-167X.2024.06.030

摘要/Abstract

Abstract:

Primary hepatoid adenocarcinoma (HAC) of the uterus is a particular tumour that bears high similarity to hepatocellular carcinoma histologically, and may easily be misdiagnosed because it is rare if you don' t remember it. In this report, we describe two cases of alpha-fetoprotein (AFP)-producing HAC of the uterus. Case 1 was a 69-year-old postmenopausal woman who was presented to the hospital for a medical examination. Positron emission computed tomography and gross examination revealed an invasive mass on the cervix. Microscopically, the tumor cells grew in trabecularand and solid patterns with heteromorphic nuclei and abundant eosinophilic cytoplasm, and were stained positively for AFP, spalt-like transcription factor 4 (SALL-4), cytokeratin 7 (CK7), hepatocyte paraffin 1 (Hep Par 1), Glypican 3 and p16. The paired box protein 8 (PAX8), Vimentin, CK20, estrogen receptor (ER), progesterone receptor (PR) were negative. P53 protein was strongly diffuse staining, suggesting the possibility of potential mutation in the TP53 gene. The final pathological diagnosis was cervical HAC combined with endocervical adenocarcinoma and endocervical adenocarcinoma in situ. To the best of our knowledge, however, it is the third case confined to the uterine cervix reported in Chinese and English literature. Case 2 was a 57-year-old postmenopausal woman with abnormal vaginal bleeding for 4 months. Biopsy was considered as poorly differentiated endometrial carcinoma. Finally, pure HAC in endometrium was diagnosed in postoperative specimens. The histological features and immunohistochemical results were similar to those in case 1. A total abdominal hysterectomy, bilateral salpingo-oophorectomy, pelvic lymphadenectomy and pelvic adhesiolysis were carried out in both cases. Serum levels of AFP were increased remarkably in both cases pre-operation and decreased after surgery, which was proved to be closely related to tumor progression, recurrence, and also the patient' s response to treatment. The diagnosis of HAC is mainly based on the histological features, and immunohistochemistry is a good assistant, but it needs to be differentiated from metastatic hepatocellular carcinoma (HCC), germ cell tumors, and yolk sac tumor. Following surgery, both patients received chemotherapy, and case 1 also received radiotherapy, and has been free of disease for 25 months and 5 months, respectively.

Key words: Hepatoid adenocarcinoma, Uterus, Cervix, Clinicopathological feature

中图分类号:

R737.33

罗丹, 黄海建, 陈新, 陈小岩. 原发子宫肝样腺癌2例临床病理分析及文献复习[J]. 北京大学学报（医学版）, 2024, 56(6): 1126-1131.

Dan LUO, Haijian HUANG, Xin CHEN, Xiaoyan CHEN. Primary uterine hepatoid adenocarcinoma: Clinicopathological analysis of 2 cases and literature review[J]. Journal of Peking University (Health Sciences), 2024, 56(6): 1126-1131.

图/表 7

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表1

本组病例及以往文献报道的共20例原发性子宫肝样腺癌的临床病理信息"

Reports	Age/years	Preoperative AFP value/(μg/L)	Location	Histological type	Stage	Surgery, stage	Adjuvant therapy	Disease outcome
Kato, et al^[1]	65	105	Cervix	Endometrioid+hepatoid adenoca	ⅠB2	TAH BSO	Radiotherapy	Recurrence at 12 months Disease free for 22 months after radiotherapy
Liu, et al^[2]	59	>20 000	Cervix	Endocervical adenoca+hepatoid adenoca	ⅠB3	TAH BSO+BPLND	Paclitaxel+cisplatin	Disease free for 4 months
Hoshida, et al^[3]	66	16 170	Endometrium	Endometrioid+hepatoid adenoca	ⅢC1	TAH BSO+BPLND	Radiotherapy	Died 12 months after surgery for disease
Yamamoto, et al^[4]	62	280.3	Endometrium	Endometrioid+hepatoid adenoca	ⅣB	TAH BSO+liver BX	Cyclophosphamide+doxorubicin+cisplatin	Died 3 months after surgery with lung metastasis
Sun, et al^[5]	46	117	Endometrium	Hepatoid adenoca	NA	TAH BSO+BPLND	NA	Disease free for 8 months
Toyoda, et al^[6]	60	31 950	Endometrium	Endometrioid+hepatoid adenoca	ⅢC2	TAH BSO+BPPALND	Cyclophosphamide+doxorubicin+cisplatin	Died 12 months after surgery with lung metastasis
Adams, et al^[7]	66	351	Endometrium	Endometrioid+hepatoid adenoca	ⅠB	TAH BSO+BPPALND+PW	Cyclophosphamide+doxorubicin+cisplatin	Disease free for 96 months
Takano, et al^[8]	63	5 060	Endometrium	Carcinosarcoma+hepatoid adenoca	ⅠA	TAH BSO+BPPALND	Paclitaxel+carbop-latin	Disease free for 12 months
Takahashi, et al^[9]	68	2 800	Endometrium	Carcinosarcoma+hepatoid adenoca	ⅡB	TAH BSO	NA	NA
Takeuchi, et al^[10]	61	453	Endometrium	Endometrioid+hepatoid adenoca	ⅣB	TAH BSO+BPLND+OMM+PW	Paclitaxel+carbop-latin	Disease free for 12 months
Sun, et al^[11]	56	>3 000	Endometrium	Endometrioid+hepatoid adenoca	ⅠB	TAH BSO+BPLND	Cyclophosphamide+epirubicin+cisplatin	Disease free for 30 months
Wu, et al^[12]	63	NA	Endometrium	Hepatoid adenoca	NA	NA	Radiotherapy+chemotherapy (the details were unknown)	Disease free for 16 months
Kawaguchi, et al^[13]	63	10 131	Endometrium	Endometrioid+hepatoid adenoca	ⅡB	TAH BSO+BPLND	Paclitaxel+carboplatin	Disease free for 12 months
Kawaguchi, et al^[13]	82	401	Endometrium	Carcinosarcoma+hepatoid adenoca	ⅠB	TAH BSO+PW	Non	Died 12 months after surgery with lung metastasis
Hwang, et al^[14]	75	90 508	Endometrium	NA	NA	TAH BSO+BPPALND+PW	NA	NA
Wu, et al^[15]	61	253.3	Endometrium	Endometrioid+hepatoid adenoca	ⅢC1	TAH BSO+BPPALND+OMM+PW	Radiotherapy+paclitaxel+cisplatin (1 cycle)+pacli-taxel+carboplatin (2 cycles)+doxoru-bicin+carboplatin (1 cycle)	Died 16 months after surgery with lung metastasis
Altn, et al^[16]	72	>54 000	Endometrium	Endometrioid+hepatoid adenoca	ⅣB	TAH BSO+BPLND+OMM+ileal resection with anastomosis+metastasectomy+PW	5-FU	Died 2 months after surgery
Cai, et al^[17]	63	543.91	Endometrium	Hepatoid adenoca	ⅠB	TAH BSO+BPLND	NA	NA
Case 1 in this study	69	1 528	Cervix	Endocervical adenoca+hepatoid adenoca	ⅠB2	TAH BSO+BPPALND	Paclitaxel+cisplatin+radiotherapy	Disease free for 22 months
Case 2 in this study	57	172.8	Endometrium	Hepatoid adenoca	ⅢC1p	TAH BSO+BPPALND+OMM+PW	Paclitaxel+carboplatin	Disease free for 5 months

表1

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