Journal of Human Reproductive Science
Home Ahead of Print Current Issue Archives
   Bookmark this page Print this page Email this page Small font sizeDefault font size Increase font size    Users online: 1313


 
CASE REPORT Table of Contents   
Year : 2008  |  Volume : 1  |  Issue : 2  |  Page : 90-92
 

Successful pregnancy in a patient with pseudomyxoma peritonei following in-vitro fertilization using donor eggs


Bangalore Assisted Conception Center, #6/7, Kumara Krupa, High Grounds, Bangalore - 560 001, India

Date of Web Publication19-Nov-2008

Correspondence Address:
S A Narvekar
Fellow in Reproductive Medicine, Bangalore Assisted Conception Center, #6/7, Kumara Krupa, High Grounds, Bangalore- 560 001
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0974-1208.44119

Rights and Permissions

 

   Abstract 

Pseudomyxoma peritonei is a rare, chronic relapsing disease with a guarded prognosis. Here, we describe such a case of a young patient presenting with primary infertility, who conceived following in-vitro fertilization with donor egg and had a successful pregnancy outcome. Literature regarding fertility and pregnancy outcome in this condition is reviewed.


Keywords: Donor egg, in-vitro fertilization, mucinous cystadenoma, pregnancy, pseudomyxoma peritonei


How to cite this article:
Narvekar S A, VijayKumar P K, Shetty N, Srinivas M S, Rao K A. Successful pregnancy in a patient with pseudomyxoma peritonei following in-vitro fertilization using donor eggs. J Hum Reprod Sci 2008;1:90-2

How to cite this URL:
Narvekar S A, VijayKumar P K, Shetty N, Srinivas M S, Rao K A. Successful pregnancy in a patient with pseudomyxoma peritonei following in-vitro fertilization using donor eggs. J Hum Reprod Sci [serial online] 2008 [cited 2020 Feb 26];1:90-2. Available from: http://www.jhrsonline.org/text.asp?2008/1/2/90/44119



   Introduction Top


Pseudomyxoma peritonei (PMP) is a rare, chronic and poorly understood disease. It is characterized by mucinous ascites and a protracted clinical course with multiple recurrences despite surgery and/or chemotherapy. There is limited data available addressing the reproductive prognosis in these patients. Also, the course of pregnancy in these patients is largely unknown. Only three pregnancies in patients with PMP have been reported to date. [1],[2]

Here, we present a case of a young girl, with PMP secondary to a mucinous cystadenoma of the ovaries, who presented with primary infertility and had a successful pregnancy outcome, having conceived with in-vitro fertilization (IVF), using donor eggs.


   Case Report Top


A 25-year-old patient, presented to us with primary infertility since four years. She had undergone an appendectomy at the age of 14 years and a laparotomy at the age of 20 years for acute abdomen secondary to intestinal obstruction. There were no records available of the first surgery. The intraoperative findings of the second surgery revealed jelly-like material filling the peritoneal cavity suggestive of pseudomucinous peritonei. Both ovaries were replaced by huge multiloculated cystic masses 10 x 10 cm each, densely adhered to the omentum and the sigmoid colon. There was a constriction band at the ileocaecal junction that was resected and intestinal continuity reestablished. The ovarian masses were suboptimally debulked. Both tubes were reported to be distorted and atrophic. She did not receive intraperitoneal chemotherapy as there were no facilities available. Histology revealed a benign mucinous cystadenoma of both ovaries. After the surgery, she was lost for follow-up.

At presentation to us, she had regular menstrual cycles, her FSH and LH levels being 12.1IU/L and 10.8 IU/L respectively. Other than her fertility concerns, she was asymptomatic. Transvaginal scan revealed a bulky uterus with heterogeneous multiloculated cystic masses 10 x 10 cm each, in both adnexae. Normal ovarian tissue could not be demonstrated. CA125 and CEA were normal. The patient was referred to a gynecological oncologist, who advised revision laparotomy in view of probable recurrence of the disease, but she refused. Hysterosalpingogram confirmed the bilateral tubal block. Her husband's semen parameters were normal.

Options of controlled ovarian hyperstimulation could not be considered due to the presence of persistent large cysts in both ovaries and the inability to visualize normal ovarian tissue. In such a situation, to fulfill her desire to have a child, we counseled the couple to opt for an IVF with donor oocyte. The endometrial preparation consisted of luteal downregulation with 0.5 ml of leuprolide acetate (Lupride® Sun Pharmaceutical Halol, Gujarat India Ltd), followed by sequentially increasing doses of oral estradiol valerate (Progynova®; Schering, Berlin, Germany), for a period of twelve days. Injectable progesterone 50 mg (Gestone® Ferring Pharmaceuticals Pvt Ltd, Powai, Mumbai, India) was administered daily from the day of the oocyte pickup. She had two, grade A, 8-cell stage embryos transferred. The dose of progesterone was increased to 100 mg on the day of embryo transfer and continued till 12 weeks of pregnancy. She conceived in the first cycle. The antenatal period was uneventful. The masses were persistent during the pregnancy and did not increase in size. She underwent a caesarean section for failed induction and delivered a healthy baby weighing 3 kg. Peroperatively, there were dense adhesions between the anterior abdominal wall and the uterus. The bladder was pulled up and was densely adherent to the anterior uterine wall.

There was an inadvertent injury to the bladder, which was repaired and she had an uneventful recovery. She is now 3-months postnatal. On rescan, the ovarian cysts were persistent. She has been referred to a gynecological oncologist for further management.


   Discussion Top


The term PMP was coined by Werth in 1884 [3] to describe massive intraperitoneal accumulation of gelatinous pseudomucin. [4] This condition may be associated with benign or malignant lesions of the appendix or ovary and rarely pancreas,  Fallopian tube More Detailss and intestines. It preferentially affects women in the age range of 29-76 years with a peak at 53 years. [2] The patients with PMP usually present with nonspecific complaints namely - nausea, fatigue, abdominal pain, distension or a mass in the abdomen - and the diagnosis is usually made accidentally, at surgery. In advanced stages, it may lead to fistula formation and adhesions, with partial or complete bowel obstruction. [1]

The treatment of PMP primarily remains aggressive surgical debulking, which may have to be done repeatedly, due to its recurrent nature. Postoperative adjuvant therapy includes the use of intraperitoneal 5-fluorouracil, mitomycin-C and cisplatin. [5] The prognosis is extremely guarded with 5 and 10-year survival rates of 50% and 20%, respectively. [2]

A pubmed search for the terms 'infertility' and 'pregnancy in PMP', revealed only four case reports, [6],[7],[8],[9] probably because of its rarity and the poor prognosis. Hales et al. , [6] reported a case of secondary infertility attributed to PMP caused by ruptured mucocele of the appendix and resection of the tumor and visible mucinous ascites resulted in spontaneous conception. They hypothesized that secondary infertility was caused by significant peritoneal inflammation and inhibition of sperm-oocyte interaction from the ascites. Bocca et al. , reported a case of primary infertility, scheduled for a laparoscopic cornual detachment of the hydrosalpinx prior to IVF. On laparoscopy, besides the hydrosalpinx, a thin layer of mucus was found covering the uterus and the tube, which on biopsy proved to be PMP, the origin of which was later found to be a low-grade mucinous appendiceal neoplasm. They concluded that malignancies, although rare, should always be part of the differential diagnosis of external causes of tubal disease. [7] Niwa et al. , reported a case of PMP secondary to ovarian mucinous cystadenocarcinoma, who conceived naturally, twice, after surgical clearance and postoperative adjuvant chemotherapy. [8] A case of acute abdomen secondary to small bowel obstruction in a patient at 34 weeks of pregnancy with PMP secondary to mucinous cystadenoma of the appendix has also been reported. [9]

In this case report, we wish to highlight that although patients with PMP have a guarded long-term prognosis, their desire to have a child needs to be addressed. Presence of the pseudomucin in the peritoneum and the distorted tubo-ovarian relation, secondary to the dense adhesions, may impair fertility. Natural conception can occur after surgical clearance and chemotherapy. IVF may be the only option wherein corrective surgery is not possible due to dense adhesions. In our case, the presence of persistent multiloculated cysts in both ovaries and the absence of demonstratable normal ovarian tissue made controlled ovarian hyperstimulation impossible. Hence, we counseled her to opt for donor eggs.

During pregnancy, complications related to PMP, like bowel obstruction, fistula formation may occur. Dense adhesions should be anticipated at caesarean section.

To the best of our knowledge, this is the first case report of a patient with PMP, who conceived with IVF using donor eggs. The case emphasizes that pregnancies are possible in impossible situations like PMP, through ART.

 
   References Top

1.Sherer DM, Abulafia O, Eliakim R. Pseudomyxoma peritonei: A review of current literature. Gynecol Obstet Invest 2001;5:73-80.  Back to cited text no. 1    
2.Jivan S, Bahal V. Pseudomyxoma peritonei. Postgrad Med J 2002;78:170-2.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Werth R. Pseudomyxoma peritonei. Arch Gynakol 1884;24:100.  Back to cited text no. 3    
4.Esquivel J, Sugarbaker PH. Clinical presentation of Pseudomyxoma peritonei syndrome. Br J Surg 2000;87:1414-8.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.Beller FK, Zimmerman RE, Nienhaus H. Biochemical identification of mucus of pseudo-myxoma peritonei as the basis of mucolytic treatment. Am J Obstet Gynecol 1986;155:970-3.  Back to cited text no. 5  [PUBMED]  
6.Hales HA, Peterson CM, Jolles CJ, Varner MW, Keye WR. Pseudomyxoma peritonei associated with secondary infertility. Fertil Steril 1992;58:425-6.  Back to cited text no. 6  [PUBMED]  
7.Bocca SM, Perry R, Oehninger S. Pseudomyxoma peritonei: A rare differential diagnosis of hydrosalpinx. Fertil Steril 2005;84:1017.   Back to cited text no. 7    
8.Niwa K, Morishita S, Murase T, Kawabata I, Imai A, Shimokawa K, et al. Successful pregnancy in a patient with pseudomyxoma peritonei arising from ovarian mucinous cystadenocarcinoma treated with cisplatin. Gynecol Oncol 1995;59:398-400.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Masakazui I, Hideyuki M, Michihiko M, Masaki K, Motohiro H, Kazumasa F, et al. A case of pseudomyxoma peritonei arising from mucinous cystadenoma of the appendix during pregnancy. J Jpn Coll Surg 2006;31:91-5.  Back to cited text no. 9    



This article has been cited by
1 Successful pregnancy following transfer of frozen-thawed embryos in a patient with pseudomyxoma peritonei who underwent peritonectomy and bilateral oophorectomy
Ido Laskov, Nadav Michaan, Ariel Many, Ami Amit, Foad Azem
Journal of Gynecologic Oncology. 2012; 23(2): 129
[VIEW] | [DOI]
2 Childbearing After Hyperthermic Intraperitoneal Chemotherapy: Results From an International Survey
Pablo Ortega-Deballon, Olivier Glehen, Edward Levine, Pompiliu Piso, Paul H. Sugarbaker, Andrea Hayes-Jordan, Audrey Facy, Naoual Bakrin, Patrick Rat
Annals of Surgical Oncology. 2011;
[VIEW] | [DOI]



 

Top
Print this article  Email this article
                 

    

 
   Search
 
  
    Similar in PUBMED
    Search Pubmed for
    Search in Google Scholar for
  Related articles
    Article in PDF (142 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
    Introduction
    Case Report
    Discussion
    References

 Article Access Statistics
    Viewed2409    
    Printed194    
    Emailed0    
    PDF Downloaded169    
    Comments [Add]    
    Cited by others 2    

Recommend this journal