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Journal of Peking University (Health Sciences) ›› 2024, Vol. 56 ›› Issue (6): 1126-1131. doi: 10.19723/j.issn.1671-167X.2024.06.030

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Primary uterine hepatoid adenocarcinoma: Clinicopathological analysis of 2 cases and literature review

Dan LUO1, Haijian HUANG2,*(), Xin CHEN2, Xiaoyan CHEN2   

  1. 1. Department of Pathology, Fujian Maternity and Child Health Hospital, College of Clinical Medicine for Obstetrics & Gynecology and Pediatrics, Fujian Medical University, Fuzhou 350005, China
    2. Department of Pathology, Fujian Provincial Hospital, Provincial Clinical Medical College of Fujian Medical University, Fuzhou 350001, China
  • Received:2023-03-17 Online:2024-12-18 Published:2024-12-18
  • Contact: Haijian HUANG E-mail:huanghaijian2008@sina.com

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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

CLC Number: 

  • R737.33

Figure 1

MRI showed annular swelling of the cervix with irregular infiltrating abnormal signal, the range was about 2.9 cm×2.1 cm in the sagittal plane"

Figure 2

The tumor forms a combination of medullary (right) and glandular components (left) (HE ×100)"

Figure 3

Polygonal tumor cells with abundant cytoplasm and round nuclei resemble hepatocellular carcinoma at high-power magnification HE ×200)"

Figure 4

Periodic acid-Schiff-positive hyaline globules were recognized in the hepatoid areas (HE ×400)"

Figure 5

The hepatoid component AFP+(EnVision ×100) AFP, alpha-fetoprotein."

Figure 6

Strongly positive for SALL-4 (EnVision ×100) SALL-4, spalt-like transcription factor 4."

Table 1

Clinicopathological information of 20 cases of primary uterine hepatoid adenocarcinoma in this study and previous literature reports"

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
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