Correspondence
points to TGF-b signalling and endothelial-to-mechenchymal transdifferentiation in the onset and progression of cerebral cavernous haemangioma.4 As CD109 directly modulates the TGF-b signalling cascade, it may regulate EC differentiation in cavernous malformations of the skin. Further studies on CD109 should enhance our understanding of the pathogenesis of cutaneous cavernous haemangioma. ACKNOWLEDGEMENT
This study was supported by a Shandong Taishan Scholarship from Shandong Provincial Government.
Author contributions F. Dong, Y. Cheng, Q. Sun, WL, G. Zhang and L. Li performed the experiments and analysed data. J. Liu and T. D. Allen designed the research study, analysed the data, and wrote the paper. All authors read and approved the final manuscript.
Conflict of interest The authors declare no conflicts of interest. Fengyun Dong Yuxia Cheng Qing Sun Wei Lu Guoxia Zhang Liqun Li Thaddeus D Allen1 Ju Liu Shandong Provincial Qianfoshan Hospital, Shandong University, Jinan, Shandong, China, and 1G. W. Hooper Research Foundation, University of California at San Francisco, San Francisco, CA, USA 1. Hunt SJ, Santa Cruz DJ. Vascular tumors of the skin: a selective review. Semin. Diagn. Pathol. 2004; 21; 166–218. 2. Goh SG, Calonje E. Cutaneous vascular tumours: an update. Histopathology 2008; 52; 661–673. 3. Greenberger S, Bischoff J. Pathogenesis of infantile haemangioma. Br. J. Dermatol. 2013; 169; 12–19. 4. Maddaluno L, Rudini N, Cuttano R et al. ENDMT contributes to the onset and progression of cerebral cavernous malformations. Nature 2013; 498; 492–496. 5. Finnson KW, Tam BY, Liu K et al. Identification of CD109 as part of the TGF-beta receptor system in human keratinocytes. FASEB J. 2006; 20; 1525–1527. 6. Murray LJ, Bruno E, Uchida N et al. CD109 is expressed on a subpopulation of CD34+ cells enriched in hematopoietic stem and progenitor cells. Exp. Hematol. 1999; 27; 1282–1294. Histopathology, 67, 130–144.
135
Combined hepatoid and serous carcinoma of the uterine corpus: an undescribed phenomenon DOI: 10.1111/his.12614 © 2014 John Wiley & Sons Ltd
Sir: We report here the first case, to our knowledge, of combined hepatoid and serous adenocarcinoma arising in the uterine corpus. The patient is a 63-year-old Japanese woman. She presented with vaginal abnormal bleeding, and subsequent imaging analysis disclosed a mass in the uterine corpus. Gynaecologists suspected uterine corpus cancer. The pre-operative serum alpha-fetoprotein (AFP) value was elevated to 151 ng/ml. In the pre-operative biopsy, a diagnosis of endometrioid adenocarcinoma was suspected. Hysterectomy, bilateral salpingo-oophorectomy, omentectomy and pelvic lymphadenectomy were performed. Macroscopically, the exophytic polypoid mass measuring 40 9 35 mm was identified in the uterine body. Histologically, the tumour was composed predominantly of thick trabecular and solid growth of neoplastic cells with clear to eosinophilic cytoplasm and occasional hyaline globules (Figure 1A). Sinusoidlike blood vessels and complex canalicular structures were occasionally seen. This hepatoid component comprised 70% of total neoplasm. Additionally, serous adenocarcinoma was observed as a minor component, accounting for 30% of total neoplasm (Figure 1B). These two components were observed separately. Immunohistochemically, the former component showed diffuse positivity for AFP (Figure 2A), glypican-3 and hepatocyte (Hep-Par1). AFP was also positive in the hyaline globules. Considering the elevation of serum AFP and these histological findings, the former component was considered to be AFP-producing hepatoid adenocarcinoma. The serous carcinoma component demonstrated the diffuse immunolabelling for p53 (Figure 2B) and p16. Finally, we diagnosed this tumour as combined hepatoid and serous adenocarcinoma. The final stage was FIGO IA and the tumour was confined to the inner half of the myometrium; there was no cervical, omental or nodal involvement. The postoperative serum AFP level recovered within normal limits. However, adjuvant chemotherapy started because of the combined presence of hepatoid and serous carcinoma. She was uneventful 2 months postoperatively. Previously, 11 cases of hepatoid adenocarcinoma arising in the uterine corpus have been reported.1–10 Clinicopathological features of these cases are summarized in Table 1. It is crucial for gynaecologists
136
Correspondence
A
B
Figure 1. Microscopic findings. A, The tumour consists of thick trabecular and solid architectures of neoplastic cells with clear cytoplasm. B, As a minor component, serous adenocarcinoma is observed.
A
B
Figure 2. Immunohistochemical findings. A, The hepatoid component is diffusely positive for AFP. B, Tumour cells of serous adenocarcinoma show the diffuse positivity for p53.
Table 1. Summary of previously reported cases of hepatoid adenocarcinoma arising in the uterine corpus Case
Age (years)
Other subtypes
Adjuvant therapy
Follow-up
Outcome
Reporter
1
66
Endometrioid
RT
32 months
DOD
Hoshida2
2
62
Endometrioid
CH
4 months
DOD
Yamamoto1
3
60
Endometrioid
CH
12 months
DOD
Toyoda3
4
66
Endometrioid
CH
8 months
AWOD
Adams4
5
63
Endometrioid + sarcoma
CH
12 months
DOD
Takano5
6
68
Endometrioid + sarcoma
NI
NI
NI
Takahashi6
7
61
Endometrioid
CH
12 months
AWOD
Takeuchi7
8
75
Endometrioid
CH, RT
NI
NI
Huang8
9
86
Endometrioid
CH, RT
36 months
AWD
Ishibashi9
10
63
Sarcoma
CH
24 months
AWOD
Kawaguchi10
11
82
Endometrioid
None
12 months
DOD
Kawaguchi10
12
63
Serous
CH
2 months
AWOD
Kuroda
RT, Radiation therapy; CH, chemotherapy; NI, not informative; DOD, died of disease; AWOD, alive without disease; AWD, alive with disease.
and pathologists to diagnose this tumour correctly, because this tumour often behaves in an aggressive fashion despite multimodal therapy, including surgical
resection, chemotherapy and radiation.1–3,5,10 Hepatoid adenocarcinoma can also occur in the uterine cervix.11 In the majority of previously described cases Histopathology, 67, 130–144.
Correspondence
that have occurred in the uterine corpus, hepatoid adenocarcinoma was associated with endometrioid adenocarcinoma.1–10 On one hand, two cases of AFPproducing endometrioid adenocarcinoma without an obvious hepatoid component have been described.12,13 On the other hand, two cases of AFPproducing papillary adenocarcinoma or serous adenocarcinoma in the uterine corpus without a hepatoid component have been presented.14,15 However, we could not find any cases of combined hepatoid and serous adenocarcinoma of the uterine corpus dispaying AFP production. The histological differential diagnosis of the present case is clear cell adenocarcinoma and yolk sac tumour. Clear cell adenocarcinoma is one of the Type II endometrial adenocarcinomas and is often associated with endometrial intraepithelial carcinoma (EIC).16 Although, focally, the present case showed solid growth of clear neoplastic cells, complex canalicular structures, sinusoid-like blood vessels and AFP-positivity are not the features of clear cell adenocarcinoma. Several cases of endometrial yolk sac tumour associated with endometrial carcinoma have been reported.17–19 Yolk sac tumour component is considered to arise as a result of metaplastic or transdifferentiation processes. The patient’s age and AFP positivity are both comparable to endometrial adenocarcinoma with yolk sac tumour differentiation. However, the characteristic histological patterns, such as Schiller–Duval body and reticular pattern, were not identified in the present case. Accordingly, this is the first case of combined hepatoid and serous adenocarcinoma arising in the uterine corpus. Gynaecologists and pathologists should bear in mind that such a tumour may occur in the uterine corpus.
Acknowledgements The authors are grateful to Dr Takako Kawami and Yuka Kai, Department of Gynaecology. Kochi Red Cross Hospital for supplying clinical information. Naoto Kuroda Suzuko Moritani1 Shu Ichihara1 Department of Diagnostic Pathology, Kochi Red Cross Hospital, Kochi, Japan, and 1Department of Advanced Diagnosis, Division of Pathology, Nagoya Medical Center, Nagoya, Japan 1. Yamamoto R, Ishikura H, Azuma M et al. Alpha-fetoprotein production by a hepatoid adenocarcinoma of the uterus. J. Clin. Pathol. 1996; 49; 420–422. Histopathology, 67, 130–144.
137
2. Hoshida Y, Nakagawa T, Mano S, Taguchi T, Aozasa K. Hepatoid adenocarcinoma of the endometrium associated with alpha-fetoprotein production. Int. J. Gynecol. Pathol. 1999; 15; 266–269. 3. Toyoda H, Hirai T, Ishii E. Alpha-fetoprotein producing uterine corpus carcinoma: a hepatoid adenocarcinoma of the endometrium. Pathol. Int. 2000; 50; 847–852. 4. Adams SF, Yamada SD, Montag A, Rotmensch JR. An alphafetoprotein-producing hepatoid adenocarcinoma of the endometrium. Gynecol. Oncol. 2001; 83; 418–421. 5. Takano M, Shibasaki T, Sato K, Aida S, Kikuchi Y. Malignant mixed Mullerian tumor of the uterine corpus with alpha-fetoprotein-producing hepatoid adenocarcinoma component. Gynecol. Oncol. 2003; 91; 444–448. 6. Takahashi Y, Inoue T. Hepatoid carcinoma of the uterus that collided with carcinosarcoma. Pathol. Int. 2003; 53; 323–326. 7. Takeuchi K, Kitazawa R, Hamanishi S, Inagaki M, Murata K. A case of alpha-fetoprotein-producing adenocarcinoma of the endometrium with a hepatoid component as a potential source for alpha-fetoprotein in a postmenopausal woman. Int. J. Gynecol. Cancer 2006; 16; 1439–1478. 8. Hwang JH, Song SH, Kim YH et al. Primary hepatoid adenocarcinoma of the endometrium with a high alphafetoprotein level. Scot. Med. J. 2011; 56; 1–4. 9. Ishibashi K, Kishimoto T, Yonemori Y, Hirahashi K, Hiroshima K, Nakatani Y. Primary hepatoid adenocarcinoma of the uterine corpus: a case report with immunohistochemical study for expression liver-rich nuclear factors. Pathol. Res. Pract. 2011; 207; 332–336. 10. Kawaguchi R, Furukawa N, Yamada Y, Ooi H, Kobayashi H. Carcinosarcoma of the uterine corpus with alpha-fetoproteinproducing hepatoid adenocarcinoma: a report of two cases. Case Rep. Oncol. 2011; 4; 358–362. 11. Kato K, Suzuka K, Osaki T, Itami M, Tanaka N. Primary hepatoid adenocarcinoma of the uterine cervix. Int. J. Gynecol. Cancer 2007; 17; 1131–1171. 12. Kodama J, Seki N, Yanai H et al. a-fetoprotein-producing endometrial adenocarcinoma without an obvious hepatoid component. Oncol. Rep. 2010; 1; 243–245. 13. Akhavan A, Zarchi MK, Tafti MA, Navabii H. a-Fetoprotein produced by endometrial adenocarcinoma of uterus. BMJ Case Rep. 2012; doi.10.1136/bcr.02.2012.5830. 14. Kubo K, Lee GH, Yamaguchi K, Kitagawa T. Alpha-fetoproteinproducing papillary adenocarcinoma originating from a uterine body. A case report. Acta Pathol. Jpn 1991; 41; 399–403. 15. Tran TA, Ortiz HB, Holloway RW, Bigsby GE, Finkler NJ. Alpha-fetoprotein-producing serous carcinoma of the uterus metastasizing to ovaries, mimicking primary ovarian yolk sac tumor: a case report and review of the literature. Int. J. Gynecol. Pathol. 2007; 26; 66–70. 16. Fadare O, Liang SX, Ulukus EC, Chambers SK, Zheng W. Precursors of endometrial clear cell carcinoma. Am. J. Surg. Pathol. 2006; 30; 1519–1530. 17. Oguri H, Sumitomo R, Maeda N, Fukaya T, Moriki T. Primary yolk sac tumor concomitant with carcinosarcoma originating from the endometrium: case report. Gynecol. Oncol. 2006; 103; 368–371. 18. Patsner B. Primary endodermal sinus tumor of the endometrium presenting as ‘recurrent’ endometrial adenocarcinoma. Gynecol. Oncol. 2001; 80; 93–95. 19. Spatz A, Bouron D, Pautier P, Castaigne D, Duvillard P. Primary yolk sac tumor of the endometrium: a case report and review of the literature. Gynecol. Oncol. 1998; 70; 285–288.
points to TGF-b signalling and endothelial-to-mechenchymal transdifferentiation in the onset and progression of cerebral cavernous haemangioma.4 As CD109 directly modulates the TGF-b signalling cascade, it may regulate EC differentiation in cavernous malformations of the skin. Further studies on CD109 should enhance our understanding of the pathogenesis of cutaneous cavernous haemangioma. ACKNOWLEDGEMENT
This study was supported by a Shandong Taishan Scholarship from Shandong Provincial Government.
Author contributions F. Dong, Y. Cheng, Q. Sun, WL, G. Zhang and L. Li performed the experiments and analysed data. J. Liu and T. D. Allen designed the research study, analysed the data, and wrote the paper. All authors read and approved the final manuscript.
Conflict of interest The authors declare no conflicts of interest. Fengyun Dong Yuxia Cheng Qing Sun Wei Lu Guoxia Zhang Liqun Li Thaddeus D Allen1 Ju Liu Shandong Provincial Qianfoshan Hospital, Shandong University, Jinan, Shandong, China, and 1G. W. Hooper Research Foundation, University of California at San Francisco, San Francisco, CA, USA 1. Hunt SJ, Santa Cruz DJ. Vascular tumors of the skin: a selective review. Semin. Diagn. Pathol. 2004; 21; 166–218. 2. Goh SG, Calonje E. Cutaneous vascular tumours: an update. Histopathology 2008; 52; 661–673. 3. Greenberger S, Bischoff J. Pathogenesis of infantile haemangioma. Br. J. Dermatol. 2013; 169; 12–19. 4. Maddaluno L, Rudini N, Cuttano R et al. ENDMT contributes to the onset and progression of cerebral cavernous malformations. Nature 2013; 498; 492–496. 5. Finnson KW, Tam BY, Liu K et al. Identification of CD109 as part of the TGF-beta receptor system in human keratinocytes. FASEB J. 2006; 20; 1525–1527. 6. Murray LJ, Bruno E, Uchida N et al. CD109 is expressed on a subpopulation of CD34+ cells enriched in hematopoietic stem and progenitor cells. Exp. Hematol. 1999; 27; 1282–1294. Histopathology, 67, 130–144.
135
Combined hepatoid and serous carcinoma of the uterine corpus: an undescribed phenomenon DOI: 10.1111/his.12614 © 2014 John Wiley & Sons Ltd
Sir: We report here the first case, to our knowledge, of combined hepatoid and serous adenocarcinoma arising in the uterine corpus. The patient is a 63-year-old Japanese woman. She presented with vaginal abnormal bleeding, and subsequent imaging analysis disclosed a mass in the uterine corpus. Gynaecologists suspected uterine corpus cancer. The pre-operative serum alpha-fetoprotein (AFP) value was elevated to 151 ng/ml. In the pre-operative biopsy, a diagnosis of endometrioid adenocarcinoma was suspected. Hysterectomy, bilateral salpingo-oophorectomy, omentectomy and pelvic lymphadenectomy were performed. Macroscopically, the exophytic polypoid mass measuring 40 9 35 mm was identified in the uterine body. Histologically, the tumour was composed predominantly of thick trabecular and solid growth of neoplastic cells with clear to eosinophilic cytoplasm and occasional hyaline globules (Figure 1A). Sinusoidlike blood vessels and complex canalicular structures were occasionally seen. This hepatoid component comprised 70% of total neoplasm. Additionally, serous adenocarcinoma was observed as a minor component, accounting for 30% of total neoplasm (Figure 1B). These two components were observed separately. Immunohistochemically, the former component showed diffuse positivity for AFP (Figure 2A), glypican-3 and hepatocyte (Hep-Par1). AFP was also positive in the hyaline globules. Considering the elevation of serum AFP and these histological findings, the former component was considered to be AFP-producing hepatoid adenocarcinoma. The serous carcinoma component demonstrated the diffuse immunolabelling for p53 (Figure 2B) and p16. Finally, we diagnosed this tumour as combined hepatoid and serous adenocarcinoma. The final stage was FIGO IA and the tumour was confined to the inner half of the myometrium; there was no cervical, omental or nodal involvement. The postoperative serum AFP level recovered within normal limits. However, adjuvant chemotherapy started because of the combined presence of hepatoid and serous carcinoma. She was uneventful 2 months postoperatively. Previously, 11 cases of hepatoid adenocarcinoma arising in the uterine corpus have been reported.1–10 Clinicopathological features of these cases are summarized in Table 1. It is crucial for gynaecologists
136
Correspondence
A
B
Figure 1. Microscopic findings. A, The tumour consists of thick trabecular and solid architectures of neoplastic cells with clear cytoplasm. B, As a minor component, serous adenocarcinoma is observed.
A
B
Figure 2. Immunohistochemical findings. A, The hepatoid component is diffusely positive for AFP. B, Tumour cells of serous adenocarcinoma show the diffuse positivity for p53.
Table 1. Summary of previously reported cases of hepatoid adenocarcinoma arising in the uterine corpus Case
Age (years)
Other subtypes
Adjuvant therapy
Follow-up
Outcome
Reporter
1
66
Endometrioid
RT
32 months
DOD
Hoshida2
2
62
Endometrioid
CH
4 months
DOD
Yamamoto1
3
60
Endometrioid
CH
12 months
DOD
Toyoda3
4
66
Endometrioid
CH
8 months
AWOD
Adams4
5
63
Endometrioid + sarcoma
CH
12 months
DOD
Takano5
6
68
Endometrioid + sarcoma
NI
NI
NI
Takahashi6
7
61
Endometrioid
CH
12 months
AWOD
Takeuchi7
8
75
Endometrioid
CH, RT
NI
NI
Huang8
9
86
Endometrioid
CH, RT
36 months
AWD
Ishibashi9
10
63
Sarcoma
CH
24 months
AWOD
Kawaguchi10
11
82
Endometrioid
None
12 months
DOD
Kawaguchi10
12
63
Serous
CH
2 months
AWOD
Kuroda
RT, Radiation therapy; CH, chemotherapy; NI, not informative; DOD, died of disease; AWOD, alive without disease; AWD, alive with disease.
and pathologists to diagnose this tumour correctly, because this tumour often behaves in an aggressive fashion despite multimodal therapy, including surgical
resection, chemotherapy and radiation.1–3,5,10 Hepatoid adenocarcinoma can also occur in the uterine cervix.11 In the majority of previously described cases Histopathology, 67, 130–144.
Correspondence
that have occurred in the uterine corpus, hepatoid adenocarcinoma was associated with endometrioid adenocarcinoma.1–10 On one hand, two cases of AFPproducing endometrioid adenocarcinoma without an obvious hepatoid component have been described.12,13 On the other hand, two cases of AFPproducing papillary adenocarcinoma or serous adenocarcinoma in the uterine corpus without a hepatoid component have been presented.14,15 However, we could not find any cases of combined hepatoid and serous adenocarcinoma of the uterine corpus dispaying AFP production. The histological differential diagnosis of the present case is clear cell adenocarcinoma and yolk sac tumour. Clear cell adenocarcinoma is one of the Type II endometrial adenocarcinomas and is often associated with endometrial intraepithelial carcinoma (EIC).16 Although, focally, the present case showed solid growth of clear neoplastic cells, complex canalicular structures, sinusoid-like blood vessels and AFP-positivity are not the features of clear cell adenocarcinoma. Several cases of endometrial yolk sac tumour associated with endometrial carcinoma have been reported.17–19 Yolk sac tumour component is considered to arise as a result of metaplastic or transdifferentiation processes. The patient’s age and AFP positivity are both comparable to endometrial adenocarcinoma with yolk sac tumour differentiation. However, the characteristic histological patterns, such as Schiller–Duval body and reticular pattern, were not identified in the present case. Accordingly, this is the first case of combined hepatoid and serous adenocarcinoma arising in the uterine corpus. Gynaecologists and pathologists should bear in mind that such a tumour may occur in the uterine corpus.
Acknowledgements The authors are grateful to Dr Takako Kawami and Yuka Kai, Department of Gynaecology. Kochi Red Cross Hospital for supplying clinical information. Naoto Kuroda Suzuko Moritani1 Shu Ichihara1 Department of Diagnostic Pathology, Kochi Red Cross Hospital, Kochi, Japan, and 1Department of Advanced Diagnosis, Division of Pathology, Nagoya Medical Center, Nagoya, Japan 1. Yamamoto R, Ishikura H, Azuma M et al. Alpha-fetoprotein production by a hepatoid adenocarcinoma of the uterus. J. Clin. Pathol. 1996; 49; 420–422. Histopathology, 67, 130–144.
137
2. Hoshida Y, Nakagawa T, Mano S, Taguchi T, Aozasa K. Hepatoid adenocarcinoma of the endometrium associated with alpha-fetoprotein production. Int. J. Gynecol. Pathol. 1999; 15; 266–269. 3. Toyoda H, Hirai T, Ishii E. Alpha-fetoprotein producing uterine corpus carcinoma: a hepatoid adenocarcinoma of the endometrium. Pathol. Int. 2000; 50; 847–852. 4. Adams SF, Yamada SD, Montag A, Rotmensch JR. An alphafetoprotein-producing hepatoid adenocarcinoma of the endometrium. Gynecol. Oncol. 2001; 83; 418–421. 5. Takano M, Shibasaki T, Sato K, Aida S, Kikuchi Y. Malignant mixed Mullerian tumor of the uterine corpus with alpha-fetoprotein-producing hepatoid adenocarcinoma component. Gynecol. Oncol. 2003; 91; 444–448. 6. Takahashi Y, Inoue T. Hepatoid carcinoma of the uterus that collided with carcinosarcoma. Pathol. Int. 2003; 53; 323–326. 7. Takeuchi K, Kitazawa R, Hamanishi S, Inagaki M, Murata K. A case of alpha-fetoprotein-producing adenocarcinoma of the endometrium with a hepatoid component as a potential source for alpha-fetoprotein in a postmenopausal woman. Int. J. Gynecol. Cancer 2006; 16; 1439–1478. 8. Hwang JH, Song SH, Kim YH et al. Primary hepatoid adenocarcinoma of the endometrium with a high alphafetoprotein level. Scot. Med. J. 2011; 56; 1–4. 9. Ishibashi K, Kishimoto T, Yonemori Y, Hirahashi K, Hiroshima K, Nakatani Y. Primary hepatoid adenocarcinoma of the uterine corpus: a case report with immunohistochemical study for expression liver-rich nuclear factors. Pathol. Res. Pract. 2011; 207; 332–336. 10. Kawaguchi R, Furukawa N, Yamada Y, Ooi H, Kobayashi H. Carcinosarcoma of the uterine corpus with alpha-fetoproteinproducing hepatoid adenocarcinoma: a report of two cases. Case Rep. Oncol. 2011; 4; 358–362. 11. Kato K, Suzuka K, Osaki T, Itami M, Tanaka N. Primary hepatoid adenocarcinoma of the uterine cervix. Int. J. Gynecol. Cancer 2007; 17; 1131–1171. 12. Kodama J, Seki N, Yanai H et al. a-fetoprotein-producing endometrial adenocarcinoma without an obvious hepatoid component. Oncol. Rep. 2010; 1; 243–245. 13. Akhavan A, Zarchi MK, Tafti MA, Navabii H. a-Fetoprotein produced by endometrial adenocarcinoma of uterus. BMJ Case Rep. 2012; doi.10.1136/bcr.02.2012.5830. 14. Kubo K, Lee GH, Yamaguchi K, Kitagawa T. Alpha-fetoproteinproducing papillary adenocarcinoma originating from a uterine body. A case report. Acta Pathol. Jpn 1991; 41; 399–403. 15. Tran TA, Ortiz HB, Holloway RW, Bigsby GE, Finkler NJ. Alpha-fetoprotein-producing serous carcinoma of the uterus metastasizing to ovaries, mimicking primary ovarian yolk sac tumor: a case report and review of the literature. Int. J. Gynecol. Pathol. 2007; 26; 66–70. 16. Fadare O, Liang SX, Ulukus EC, Chambers SK, Zheng W. Precursors of endometrial clear cell carcinoma. Am. J. Surg. Pathol. 2006; 30; 1519–1530. 17. Oguri H, Sumitomo R, Maeda N, Fukaya T, Moriki T. Primary yolk sac tumor concomitant with carcinosarcoma originating from the endometrium: case report. Gynecol. Oncol. 2006; 103; 368–371. 18. Patsner B. Primary endodermal sinus tumor of the endometrium presenting as ‘recurrent’ endometrial adenocarcinoma. Gynecol. Oncol. 2001; 80; 93–95. 19. Spatz A, Bouron D, Pautier P, Castaigne D, Duvillard P. Primary yolk sac tumor of the endometrium: a case report and review of the literature. Gynecol. Oncol. 1998; 70; 285–288.
