Open Access

The long-term survival in primary retroperitoneal mucinous cystadenocarcinoma: a case report

  • Hirotaka Tokai1Email authorView ORCID ID profile,
  • Yasuhiro Nagata3,
  • Ken Taniguchi1,
  • Naomi Matsumura1,
  • Amane Kitasato1,
  • Takayuki Tokunaga1,
  • Hiroaki Takeshita1,
  • Tamotsu Kuroki1,
  • Shigeto Maeda1,
  • Masahiro Ito2 and
  • Hikaru Fujioka1
Surgical Case Reports20173:117

https://doi.org/10.1186/s40792-017-0394-z

Received: 17 August 2017

Accepted: 15 November 2017

Published: 25 November 2017

Abstract

Background

Primary retroperitoneal mucinous cystadenocarcinoma (PRMC) is extremely rare, and its biological behavior, pathogenesis, optimum treatments, and prognosis remain to be elucidated. We herein report a case of PRMC with an 80-month follow-up.

Case presentation

A 29-year-old woman was diagnosed with unknown retroperitoneal tumor with benign right ovarian cyst and uterine fibroids, and she underwent laparotomy. The tumor was completely resected with a subsequent histopathological diagnosis of primary retroperitoneal mucinous cystadenocarcinoma (PRMC). Eighty months after surgery, she remains recurrence-free.

Conclusion

PRMC is an extremely rare tumor. Only around 60 cases have so far been published in the literature. The preoperative diagnosis of PRMC is difficult, and a definitive diagnosis can usually only be made based on the findings of histopathological examinations after surgery. Presently, only radical resection is useful for both diagnostic and therapeutic purposes. The optimal long-term management after surgery is still not well established. Further studies on PRMC are therefore needed to elucidate the etiology and establish effective treatments.

Keywords

RetroperitonealCystadenocarcinomaMucinous

Background

Primary retroperitoneal mucinous cystadenocarcinoma (PRMC) is extremely rare, with the first case was reported in 1965 [1]. Since then, only 61 cases have been reported in the English literature, to our knowledge. Little is known about the biological behavior and pathogenesis of this disease. The diagnosis is often confusing preoperatively. In addition, the optimum treatments and prognosis of PRMCs remain to be uncertain. We herein report a case of PRMC with 80 months of follow-up.

Case presentation

A 29-year-old woman was admitted to our hospital for further examination due to abdominal pain and a cystic mass in the right lower abdomen. A physical examination revealed no tumor in her abdomen on palpation. Contrast-enhanced computed tomography (CT) revealed a cystic and well-defined tumor of 8.5 cm in diameter behind the ascending colon. The tumor had enhanced and segmented components (Fig. 1). Magnetic resonance imaging (MRI) demonstrated a mass behind the ascending colon of iso-intensity on T1-weighted and high intensity on T2-weighted images. Other bi-cystic masses of 8 cm in diameter at the right ovary and uterine fibroids were also observed (Fig. 2). There was no evidence of invasion to adjacent tissues or distant metastasis. Based on imaging findings, the differential diagnosis of this tumor was thought to be neurogenic tumor, leiomyosarcoma, malignant fibrous histiocytoma, and primary retroperitoneal tumor. The serum levels of carcinoembryonic antigen (CEA) were within normal limits, while those of cancer antigen (CA) 19-9 and 125 (CA125) were elevated. She was diagnosed with unknown retroperitoneal tumor with benign right ovarian cyst and uterine fibroids.
Fig. 1

(a) Horizontal and (b) sagittal image of CT reveals a cystic and well-defined tumor 8.5 cm in diameter behind the ascending colon (arrow heads) with enhanced and segmented components (arrow)

Fig. 2

MRI demonstrates a cystic mass of iso-intensity on T1-weighted and high intensity on T2-weighted images (arrow heads), uterine fibroids (arrow), and cystic masses at the right ovary (broken arrow)

Laparotomy was performed, revealing a tumor capsulated with a thin wall behind the ascending colon, an ovarian cyst, and uterine fibroids. The tumor was completely removed without injury of its capsule. The ovarian cyst was enucleated, but the uterine fibroids were not removed in accordance with the preoperative informed consent.

A histopathological examination showed that the tumor was unilocular with a large mural solid nodule. In the nodule, there were multi-cystic lesions covered with papillovillous hyperchromatic cells made of intestinal type epithelium ranging from adenoma- to invasive adenocarcinoma type. In addition, ovarian-like stroma was seen in the cyst wall, but ovarian tissue and teratomatous elements were not (Fig. 3). An immuno-histopathological examination revealed positivity for CK 7 and CEA but negativity for CK 20 in the tumor cells (Fig. 4), and negativity for estrogen and progesterone receptors in the ovarian-like stromal cells. Considering the histopathological features and the location, the tumor was diagnosed as PRMC without sarcomatous change.
Fig. 3

Gross appearance of the specimen (a). Microscopic images show that the tumor consists of a unilocular cyst and a large mural solid nodule (b). In the nodule, multi-cystic lesions covered by papillovillous hyperchromatic cells are observed. They comprise intestinal-type epithelium with goblet cells (c). Some of them are invasive adenocarcinoma cells (d) with stromal invasion (e). Ovarian-like stroma is also observed (f)

Fig. 4

An immuno-histopathological examination revealed positivity for CK 7 (a) and CEA (b) but negativity for CK 20 in the tumor cells

The surgical margin was free from tumor cells. The right ovarian cyst was diagnosed as an endometrial cyst (chocolate cyst). After the surgery, she had no major complications and left our hospital 8 days later. During the follow-up period of 80 months, neither local recurrence nor distant metastasis was found. Her other ovarian cysts, however, gradually enlarged, so she underwent surgery to remove them 4 years after the initial surgery. A histopathological examination revealed that they were benign chocolate cysts. The serum levels of CA 125 and CA 19-9, which had been elevated before the second surgery, normalized after surgery.

Discussion

Retroperitoneal mucinous cystic neoplasms, including cystadenoma and cystadenocarcinoma, are so rare that the accurate incidence is not available. We searched the PubMed database for published English studies using the terms “primary” and “retroperitoneal” and “mucinous” and “cystadenocarcinoma” or “adenocarcinoma.” Cases of adenomas, borderline tumors, and metastasis to the retroperitoneum were excluded, but mixed-type tumors were included. Our present case is only the 62nd case (Table 1) [146]. The mean age is 44.7 years (range 18–86 years), and the mean tumor size is 13.6 cm (range 3 to 26 cm). Only five cases were reported in males. To our knowledge, among the 51 cases in which the follow-up period was mentioned (range 1–130 months; median 16 months), only 7 cases had been followed for over 5 years. They are all alive without recurrence of disease. Although most PRMCs tend not to develop further disease, some with sarcoma-like or anaplastic components have a very aggressive character and can metastasize. Myriokefalitaki [9] reported that the 5-year overall survival was 75.4%.
Table 1

Published English studies searched in PubMed

Case no.

Author

Sex

Age

Diameter(cm)

Adjuvant chemotherapy

Follow-up (month)

Status

1

Douglas 1965 [1]

F

18

5

No

N/R

DOD

2

Tykkä 1975 [2]

F

23

10

No

11

DOD

3

Roth 1977 [3]

F

48

N/R

No

6

DOD

4

Fujii 1986 [4]

F

69

23

No

36

NED

5

Nelson 1988 [5]

F

35

20

No

22

NED

6

Chida 1990 [6]

F

42

N/R

No

N/R

N/R

7

Seki 1990 [7]

F

42

11

No

N/R

N/R

8

Park 1990 [8]

F

40

24

Yes

3

NED

9

Jorgensen 1991 [9]

F

38

8

No

9

NED

10

Søndergaard 1991 [10]

F

37

13

No

18

NED

11

Gotoh 1992 [11]

F

44

12.5

Yes

4

DOD

12

Tenti 1994 [12]

F

46

20

Yes

33

NED

13

 

F

45

20

No

19

NED

14

Motoyama 1994 [13]

F

42

11

N/R

N/R

N/R

15

Carabias 1995 [14]

F

43

15

No

24

NED

16

Lee 1996 [15]

F

55

20

No

30

NED

17

 

F

45

17

No

15

NED

18

Dore 1996 [16]

F

45

20

No

16

NED

19

Uematsu 2000 [17]

F

86

23

No

72

NED

20

Suzuki 2001 [18]

F

40

15

No

15

NED

21

Tangjitgamol 2002 [19]

F

41

12

Yes

18

NED

22

Kessler 2002 [20]

F

38

11.5

N/R

60

NED

23

Mikami 2003 [21]

F

38

16

Yes

18

DOD

24

Song 2005 [22]

F

72

12

No

4

DOD

25

Sonntag 2005 [23]

F

60

5

No

12

NED

26

Thamboo 2006 [24]

M

64

24

No

18

NED

27

Fan 2006 [25]

F

68

17

No

N/R

N/R

28

Law 2006 [26]

F

35

11

No

60

NED

29

de Leon 2007 [27]

F

36

19

Yes

8

AWD

30

 

F

21

26

No

6

NED

31

Kashima 2007 [28]

F

28

17

No

13

NED

32

Lee 2007 [29]

F

32

15

Yes

42

NED

33

Green 2007 [30]

M

83

26

No

6

NED

34

Tjalma 2007 [31]

F

74

3

Yes

31

DOD

35

Moral 2008 [32]

F

47

24

No

8

NED

36

Youssef 2008 [33]

F

70

10

Yes

24

NED

37

Roma 2009 [34]

F

35

13

No

13

NED

38

 

F

47

21

No

1

NED

39

 

F

24

18

No

2

NED

40

 

F

43

10

No

5

DOD

41

 

F

40

11

No

9

DOD

42

 

F

27

8

No

11

NED

43

 

F

63

7.5

No

14

AWD

44

 

F

31

18

No

26

AWD

45

 

F

48

26

No

58

AWD

46

 

F

40

15

No

58

NED

47

 

F

35

N/R

No

91

NED

48

 

F

49

11

No

130

NED

49

 

F

20

N/R

No

N/R

N/R

50

Hrora 2009 [35]

M

42

5

No

6

NED

51

Dierickx 2010 [36]

F

50

13

Yes

58

NED

52

Jian 2011 [37]

F

21

14.6

Yes

6

AWD

53

Kanayama 2012 [38]

F

40

25

No

6

AWD

54

Feng 2013 [39]

M

63

4

No

13

NED

55

Hanhan 2014 [40]

F

37

22

No

N/R

N/R

56

Shiau 2013 [41]

M

59

7.5

No

79

NED

57

Kurita 2014 [42]

F

30

19

No

32

AWD

58

Kamiyama 2015 [43]

F

62

10

No

15

DOD

59

Cupp 2015 [44]

F

39

20

Yes

N/R

N/R

60

Dong 2014 [45]

F

52

3.8

No

N/R

N/R

61

Myriokefalitaki 2016 [46]

F

56

24

No

17

NED

62

Present case

F

28

8.5

No

80

NED

N/R not recorded, DOD dead of disease, NED no evidence of disease, AWD alive with disease

The etiology and biological behavior of PRMCs are still unclear; however, some hypotheses have been proposed to explain the genesis of these tumors as follows: (1) heterotopic ovarian tissue [3, 11, 47], (2) monodermal variant of teratomas [22, 48], (3) intestinal duplication [49], and (4) coelomic metaplasia [4, 8, 12, 50]. In our case, ovarian-like stroma was histopathologically found in the tumor, although no definitive evidence of ovarian tissue was observed, which was also supported by the results of an immunohistochemical examination of the estrogen and progesterone receptors. These findings exclude the hypothesis of heterotopic ovarian tissue. In addition, the hypotheses of teratoma and intestinal duplication can also be excluded because of the lack of structures of teratoma or well-developed intestinal mucosa and smooth muscle. The fourth hypothesis, which is most well-described in the previous literature, is that PMRCs occur from invaginations of the peritoneal epithelium during embryogenesis. Those invaginated coelomic epithelial cells form cysts that may act like epithelial ovarian tissue and undergo the process of Müllerian differentiation. Eventually, the coelomic epithelia of these cysts undergo metaplasia and develop a spectrum of histological cells in different stages. In our case, the ovarian-like stroma and intestinal type epithelium ranged from adenoma-type mucinous cells to invasive adenocarcinoma cells, which may be explained by the last theory. In addition, the pattern of immunohistochemical expression of CK 7 and CK 20 is similar to those seen in ovarian and pancreatic mucinous neoplasms.

There are no pathognomonic clinical or radiological findings for PRMC, making the preoperative diagnosis of this disease challenging. PRMCs usually present as a multi- or unilocular cystic mass, varying in size and localized anywhere in the retroperitoneal space. Regarding the imaging findings, the diagnostic value of computed tomography and MRI is similar, but MRI can further characterize these lesions and identify their mucinous component. Notable radiographic findings may include thickening and calcification of the cyst wall or mural nodules on imaging that may suggest malignant lesions [27]. Aspiration cytology and/or a biopsy may help with the diagnosis, although they carry risks of recurrence and dissemination in cystic tumors. Serological investigations provide limited diagnostic utility. Tumor markers, including CEA, CA 125, and CA 19-9, are also not very helpful for differentiating from other benign tumors, including ovarian cyst, cystic lymphangioma, and cystic methothelioma. Indeed, in our case, the elevation of serum levels of CA19-9 and CA 125 was also observed during the follow-up period. However, these levels normalized after she underwent a second surgery to resect the benign ovarian cysts. Therefore, imaging examinations such as CT or ultrasonography are the most effective tools for performing follow-up for PRMC.

The management of PRMC is not well established. There is currently no significant chemotherapy for PRMC. The commonly used chemotherapeutic regimes were cyclophosmide and adriamycin, cyclophosmide, adriamycin and cisplatin, cisplatin alone, carboplatin and paclitaxel, or carboplatin alone. Of the 12 patients who received adjuvant chemotherapy, 5 had recurrence (41.7%). Therefore, radical tumor excision is clearly mandatory. Radical resection without rupture is the standard therapy and the most important prognostic tool [46], but whether lymphadenectomy or adjuvant chemotherapy provide benefit is still controversial [11, 12, 19, 21, 27, 29, 31, 36].

Conclusions

PRMC is a rare tumor that can have an aggressive potential for recurrence. The diagnosis remains difficult preoperatively, and surgeons should be aware of this disease as a differential diagnosis of large retroperitoneal cystic masses with indolent symptoms. The long-term management after surgery is not well established yet. Further studies about PRMC are needed to elucidate the etiology and effective treatments.

Abbreviations

CA: 

Cancer antigen

CEA: 

Carcinoembryonic antigen

CT: 

Computed tomography

MRI: 

Magnetic resonance imaging

PRMC: 

Primary retroperitoneal mucinous cystadenocarcinoma

Declarations

Funding

The authors declare no financial or any other type of support.

Authors’ contributions

HT drafted the manuscript. YN participated in the surgery and supervised the writing of the manuscript. KT, NM, AK, TT, HT, TK, and SM performed perioperative management on the patient and helped to draft the manuscript. HF is a chairperson of our department who supervised the entire process. MI helped to draft the manuscript of the pathological findings. All authors read and approved the final manuscript.

Ethics approval and consent to participate

This article does not contain any studies involving human participants, human data, or human tissue, so our manuscript is not applicable in this section.

Consent for publication

The patient has provided permission to publish these features of the cases, and the identity of the patient has been protected.

Competing interests

The authors declare that they have no competing interests.

Publisher’s Note

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Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.

Authors’ Affiliations

(1)
Department of Surgery, National Hospital Organization Nagasaki Medical Center, Nagasaki, Japan
(2)
Department of pathology, National Hospital Organization Nagasaki Medical Center, Nagasaki, Japan
(3)
Center for Comprehensive Community Care Education Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan

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Copyright

© The Author(s). 2017