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Hydatid of Morgagni: a possible underestimated cause of unexplained infertility
European Journal of Obstetrics & Gynecology and Reproductive Biology 158 (2011) 62–66
Contents lists available at ScienceDirect
European Journal of Obstetrics & Gynecology and Reproductive Biology journal homepage: www.elsevier.com/locate/ejogrb
Hydatid of Morgagni: a possible underestimated cause of unexplained infertility Salah M. Rasheed *, Allam M. Abdelmonem Department of Obstetrics and Gynecology, Sohag Faculty of Medicine, Sohag University, Sohag, Egypt
A R T I C L E I N F O
A B S T R A C T
Article history: Received 8 October 2010 Received in revised form 4 March 2011 Accepted 26 April 2011
Objective: To evaluate the possible role of hydatid of Morgagni in patients with unexplained infertility. Study design: This was a non-randomized controlled trial conducted at the Department of Obstetrics and Gynecology of Sohag University, Egypt. Two hundred and thirteen patients with unexplained infertility and hydatid of Morgagni diagnosed at laparoscopy were included. The laterality (bilateral vs unilateral), location (fimbrial vs juxta-fimbrial), number (single vs multiple) and diameter of the hydatids of Morgagni were recorded. Patients were allocated to a study group (n = 127) who underwent laparoscopic excision of hydatid of Morgagni and a control group (n = 86) who underwent no intervention. Patients were followed for six months without any infertility or hormonal treatment to detect spontaneous pregnancy. Patients missed during the follow-up or who received infertility treatment were excluded. Statistical analysis was done using Chi-square test and Student’s t-test. To find the most important character of hydatid of Morgagni which impedes pregnancy, logistic regression analysis of the dependent variable (no pregnancy) and independent variables (different characters of hydatid of Morgagni) was carried out in the control group. Results: Hydatid of Morgagni was detected in 52.1% of patients with unexplained infertility compared to 25.6% of those with explained infertility (p < 0.001). The pregnancy rate was higher in the study group than the control group (58.7% vs 20.6%, p < 0.001). The pregnancy rate was significantly higher in the study group than the control group if the hydatid cyst was bilateral (85.7% vs 5.3%, p < 0.001), fimbrial (85.6% and 9.1%, p < 0.001), single (57.6% and 30.3%, p < 0.001) or 1–2 cm in diameter (58.1% and 25.5%, p < 0.001). Logistic analysis showed that the bilaterality and fimbrial location of the hydatid of Morgagni were the most significant characteristics impeding pregnancy (odds ratio = 7.27 and 3.67 respectively). Conclusions: Hydatid of Morgagni is a possible underestimated cause of unexplained infertility. Laparoscopic removal of hydatid of Morgagni in patients with unexplained infertility was followed with a high spontaneous pregnancy rate. This is particularly obvious with bilateral and fimbrial hydatid of Morgagni. ß 2011 Elsevier Ireland Ltd. All rights reserved.
Keywords: Hydatid of Morgagni Unexplained infertility Pregnancy
1. Introduction Infertility is a rather important health problem with a reported prevalence of about 8–10% worldwide [1,2]. Unexplained infertility, which has been reported to constitute about 15–30% of all causes of infertility, is a frustrating diagnosis both for the couple and the gynecologist [3]. Customarily, the diagnosis of unexplained infertility is assigned if the basic infertility investigations are normal. These investigations include semen analysis, detection of ovulation, hysterosalpingography, assessment of ovarian reserve, if indicated, and laparoscopy [4]. The diagnosis of unexplained infertility may suggest either deficiency of the currently available
* Corresponding author at: Obstetrics and Gynecology Department, Faculty of Medicine, Sohag University, University Street, Sohag, Egypt. Tel.: +20 932320071; fax: +20 932603963; mobile: +20 0124653702. E-mail address: [email protected] (S.M. Rasheed). 0301-2115/$ – see front matter ß 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ejogrb.2011.04.018
diagnostic tools to detect a hidden cause or the presence of unknown factors controlling the delicate process of human fertility. Hydatid cysts of Morgagni are vestigial remnants of the paramesonephric ducts which appear as sessile or pedunculated cystic structures at or near the fimbriae of the fallopian tubes [5]. Although hydatid cysts of Morgagni are frequently seen as an asymptomatic incidental finding during laparoscopy [6], their relation to infertility is still elusive. Not only is there considerable paucity of literature about the impact of hydatid of Morgagni on fertility, but also there is a lack of any scientifically sound consensus amongst gynecologists as to removing or leaving these cysts during laparoscopy done for infertile women (personal communication). The delicate and close fimbrio-ovarian relationship is a crucial factor for the process of ovum pick-up and hence conception [7]. Any factor, including cysts of Morgagni, which disrupts this intimate relationship may consequently hinder the process of
S.M. Rasheed, A.M. Abdelmonem / European Journal of Obstetrics & Gynecology and Reproductive Biology 158 (2011) 62–66
conception [8]. Accordingly, the aim of this study was to evaluate the effect of hydatid of Morgagni on the pregnancy rate in patients with unexplained infertility. 2. Materials and methods The present nonrandomized controlled trial was conducted between January 15, 2006 and January 15, 2010 at the Obstetrics and Gynecology Department of Sohag University Hospital, Egypt. During the study period, all patients with either primary or secondary infertility who were candidates for laparoscopy were invited to participate (n = 1141). During laparoscopy (Storz, Germany), patients with unexplained infertility and hydatid of Morgagni constituted the study group (n = 213). The laterality (bilateral vs unilateral), location (fimbrial vs juxta-fimbrial), number (single vs multiple), and diameter (measured in cm using the graded laparoscopic probe) of hydatids of Morgagni were assessed and recorded in all patients. The diagnostic criteria of unexplained infertility included infertility >1 year despite regular marital life, normal husband semen analysis according to WHO criteria [9], normal hysterosalpingography, regular ovulation for at least 3 consecutive cycles (documented by serial folliculometry and midluteal serum progesterone >10 ng/dl) and normal laparoscopic findings. These investigations were documented and conducted either at Sohag University Hospital or at other licensed hospitals or infertility units. The inclusion criteria were the diagnostic criteria of unexplained infertility, age <30 years, regular cycles, uneventful clinical examination and normal basal hormonal profile (serum FSH, LH, T3, T4 and prolactin). The exclusion criteria were refusal to participate, obesity (BMI >30 kg/m2), uterine or adnexal pathology detected either clinically or by trans-vaginal sonography, previous pelvic inflammatory disease, previous pelvic surgery and the presence of galactorrhea even with normal serum prolactin level. The institutional ethics committee provided approval and informed consent was obtained from all participants. The eligible participants were allocated into two groups; group I (study group; n = 127) underwent laparoscopic excision of hydatid cysts of Morgagni and group II (control group; n = 86) underwent no intervention. The cyst was easily excised using scissors if it was pedunculated. Otherwise the cyst was dissected from its bed using grasping, non-traumatic forceps and scissors. Electro-cauterization was not used to fulgurate the cysts, but bipolar diathermy was used, only if needed, to control bleeding from the cyst’s bed. A report on the laparoscopy was given to all patients and they were thereafter followed for 6 months without any infertility or hormonal treatment. The patients were asked to contact the Obstetrics and Gynecology Department of Sohag University Hospital once they had a missed period or positive pregnancy test, where serum human chorionic gonadotropin assay and transvaginal sonography were done to diagnose pregnancy. The remaining patients were asked to contact the department every 2 months just for assurance and answering any enquiries. Any patient who dropped-out during the 6-month follow-up period, or who received infertility or hormonal treatment or was enrolled into assisted reproduction, was excluded from the study. At the end of the study period, the pregnancy rate, either chemical or clinical, was calculated and compared between the 2 arms of the study using the Chi-square test (p < 0.05 was considered significant). Real variables were compared using the Student’s t-test. To find the most significant character of hydatid of Morgagni which impedes pregnancy, logistic regression analysis of the dependent variable (no pregnancy) and independent variables (different characters of hydatid of Morgagni) was carried out in the control group. The data were analyzed using SPSS version (SPSS, Chicago, USA).
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3. Results During the 4-year study period, a total of 1141 infertile patients underwent laparoscopy at the Department of Obstetrics and Gynecology of Sohag University Hospital as a part of infertility work-up. After laparoscopy, 409 patients (35.8%) were diagnosed as having unexplained infertility while 732 patients (64.2%) were assigned as having explained infertility. At laparoscopy, 213 patients (52.1%) with unexplained infertility were found to have hydatid of Morgagni compared to 188 patients (25.6%) of those with explained infertility (p < 0.001). One hundred and ninety six (48.8%) of the patients with unexplained infertility were therefore excluded and the remaining 213 patients were allocated into the study (n = 127) and control (n = 86) groups (Fig. 1). Laparoscopic extirpation of hydatid of Morgagni was successful in 119 patients (93.7%) while in 8 patients (6.3%) accidental rupture of the cyst occurred followed by incomplete removal (de-roofing). Bipolar diathermy was necessary to control bleeding from the bed of the cyst in one patient (0.8%). At the end of the 6-month follow-up period, 82 patients (64 patients in group I and 18 patients in group II) were excluded (37.3%). In group II, 11 patients dropped out during the follow-up and 7 patients received infertility treatment. The patients in group I were excluded due to diverse causes; 29 patients dropped out during the follow-up, 28 received infertility treatment, 4 underwent assisted reproduction, two were divorced and one patient was operated on due to appendicitis. The remaining 131 patients (63 in group I and 68 in group II) constituted the final study group. There was no statistically significant difference between the 2 groups regarding the mean age (26.2 3.1 vs 27.6 1.7 years, p > 0.05), duration of infertility (5.1 1.6 vs 6.2 2.4 years, p > 0.05) and body mass index (23.5 1.5 vs 25.1 1.9 kg/m2, p > 0.05) (data not shown). The laterality, location, number and diameter of hydatids of Morgagni were comparable between the 2 groups (Table 1). In the vast majority of patients in both groups the hydatid cysts were unilateral (66.7% vs 72.1%, p = 0.50), single (82.5% vs 79.4%, p = 0.64), located juxta-fimbrial (77.8% vs 83.8%, p < 0.37) and had diameters of 1–2 cm (68.3% vs 57.3%, p = 0.43). In group I, 37 patients (58.7%) conceived, compared to 14 patients (20.6%) in group II (p < 0.001). The pregnancy rate was higher in group I than group II irrespective of the laterality, location, number and diameter of the cysts of Morgagni. The pregnancy rate was higher in group I than group II whether the cyst was bilateral (85.7% and 5.1% respectively, p < 0.001) or unilateral (45.2% and 26.5%, p = 0.05). The pregnancy rate was higher in group I than group II whether the cyst was fimbrial (85.6% and 9.1% respectively, p < 0.001) or juxta-fimbrial (51.0% and 22.8%, p = 0.003). Regarding the number of the cysts, the pregnancy rate was higher in group I than group II whether the cyst was single (57.6% and 20.3% respectively, p < 0.001) or multiple (63.6% and 21.4% respectively, p = 0.05). The pregnancy rate was higher in group I than in group II regardless of their diameters, but the most significant difference was found if the diameter was 1–2 cm (58.1% and 20.5% respectively, p < 0.001). The results of the logistic regression analysis are displayed in Table 2. The regression model showed that bilaterality and fimbrial location of the hydatid of Morgagni were the most significant factors impeding with pregnancy (odds ratios = 7.27 and 3.67, respectively). Patients with bilateral and fimbrial hydatids of Morgagni reached their maximal cumulative pregnancy rate (85.7% and 85.6% respectively) at the end of the second and third months after removal of the cysts respectively. This is in contrast to patients with other characters of hydatid of Morgagni where the maximal
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S.M. Rasheed, A.M. Abdelmonem / European Journal of Obstetrics & Gynecology and Reproductive Biology 158 (2011) 62–66
Patients assessed for eligibility (n=1141)
Total excluded (n=196)
* Did have hydatid of Morgagni (n=93) * Did not meet inclusion criteria (n=44) * Refusal to participate (n=59)
Unexplained infertility (n=409)
Explained infertility (n=732)
Enrollment (n=213)
Excluded
Allocation (n=213)
Group I (n=127) Laparoscopic removal of hydatid of Morgagni * Dropped out during follow-up (n=29) * Received infertility treatment (n=28) * Underwent assisted reproduction (n=4) * Divorced (n=2) * Explored due to appendicitis (n=1)
Final analysis (n=63)
Pregnancy rate (58.7%)
Group II (n=86) No intervention
Excluded during follow-up (n=82)
Follow up and final analysis (n=131)
Main final outcome
* Dropped out during follow-up (n=11) * Received infertility treatment (n=7)
Final analysis (n=68)
Pregnancy rate (20.6%)
Fig. 1. Flow chart of the study.
cumulative pregnancy rate was reached over a longer time (6 months) (Fig. 2). 4. Comments Unexplained infertility is one of the most enigmatic problems of human reproduction. Although the diagnosis is reached by default, suggesting that the probable cause is still unclear, many causes such as luteal phase defect, [10] and antisperm antibodies [11] are postulated. Although hydatid cysts of Morgagni are amongst the most frequent benign conditions affecting the fallopian tubes [5], they have received negligible attention regarding their role in infertility. Moreover, to our knowledge there is no evidence-based approach concerned with the management of these cysts when
detected in infertile patients. The decision whether to remove or leave these cysts is dependent mainly on the gynecologists own opinion and judgment rather than being a scientifically based one. Some gynecologists elect to remove the cyst whatever its location or size while others consider it an incidental finding and pass over it unheeded. To the best of our knowledge, this is the first study which addresses the issue of the role of hydatid of Morgagni in unexplained infertility in a non-randomized controlled trial. The results of the present study provide evidence about the incriminating role of hydatid of Morgagni in unexplained infertility. The high prevalence of these cysts in patients with unexplained infertility (52.1%) compared with those with explained infertility (25.6%) may suggest a possible role.
S.M. Rasheed, A.M. Abdelmonem / European Journal of Obstetrics & Gynecology and Reproductive Biology 158 (2011) 62–66
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Table 1 Characters of hydatid of Morgangni and the pregnancy rates in the 2 groups of patients. Characters of hydatid of Morgagni
Laterality Bilateral Unilateral Location Fimbrial Juxtafimbrial Number Single Multiple Diameter (cm) <1 1–2 >2
Pregnancy rate
Group I n (%)
Group II n (%)
p Value
Group I n (%)
Group II n (%)
p Value
21 (33.3) 42 (66.7)
19 (27.9) 49 (72.1)
0.50
18 (85.7) 19 (45.2)
1 (5.3) 13 (26.5)
<0.001 0.05
14 (22.2) 49 (77.8)
11 (16.1) 57 (83.8)
0.37
12 (85.6) 25 (51.0)
1 (9.1) 13 (22.8)
<0.001 0.003
52 (82.5) 11 (17.5)
54 (79.4) 14 (20.6)
0.64
30 (57.6) 7 (63.6)
11 (30.3) 3 (21.4)
<0.001 0.05
13 (20.6) 43 (68.3) 7 (11.1)
19 (27.9) 39 (57.3) 10 (14.7)
0.43
7 (53.8) 25 (58.1) 5 (71.4)
3 (15.8) 8 (20.5) 3 (30.0)
0.02 <0.001 0.09
All data were given as number (percentage) unless otherwise indicated.
Table 2 Logistic regression analysis of the dependent variable (no pregnancy) indepenindependent variable (characters of hydatic of Morgagni) in the control group. Variables Laterality Bilateral Unilateral Location Fimbrial Juxtafimbrial Number Single Multiple Diameter (cm) <1 1–2 >2
Unadjusted OR (95% CI)
p Value
Adjusted OR (95% CI)
p Value
6.5 (0.78–53.68) 1 (reference)
0.82
7.27 (0.83–63.09) 1 (reference)
0.07
2.95 (0.34–25.3) 1 (reference)
0.32
3.67 (0.38–34.75) 1 (reference)
0.26
1 (reference) 1.07 (0.25–4.49)
0.09
1 (reference) 0.61 (0.12–3.04)
0.55
1 (reference) 0.73 (0.16–3.12) 0.43 (0.07–2.73)
0.67 0.37
1 (reference) 0.85 (0.18–3.98) 0.41 (0.06–3.01)
0.84 0.38
bilaterality and the fimbrial location of hydatid of Morgagni were the most important characters interfering with pregnancy. This finding can not only be considered as important evidence about the implication of hydatid of Morgagni in the problem of infertility but also it may support the previous assumption that hydatid of Morgagni impedes fertility through interfering with the process of ovum pick-up. The close fimbrio-ovarian relationship is a crucial event for the process of ovum pick-up. Any factor, even short fimbria, interfering with ovum pick-up can lead to infertility [7]. Owing to its weight, hydatid of Morgagni may displace the fimbriae away from the ovulatory side, leading to mechanical infertility. Moreover, using hydrolaparoscopy, it has been shown that the fimbriae at the time of ovulation were congested, elongated and erected with pulsatile movements attempting to suck the ovum [14]. Whether the
OR, odds ratio; CI, confidence interval.
The considerably higher pregnancy rate (58.7%) after laparoscopic extirpation of these cysts compared to the control group (20.6%) is another finding supporting the probable impact of hydatid of Morgagni on fertility. In this context, a study involving 3 patients with unexplained infertility who underwent laparoscopic removal of these cysts has been conducted [12]. The author reported a high pregnancy rate after removal of the cysts and concluded that hydatid of Morgagni as a single pelvic pathology might hinder fertility. Moreover, he advocated the policy of laparoscopic extirpation of these cysts in infertile patients. The prevalence of hydatid cysts of Morgagni in a setting of 240 fertile patients undergoing cesarean section was compared with that reported in 204 infertile patients undergoing laparoscopy [13]. The authors reported that the frequency of hydatid of Morgagni was significantly higher (17.8%) in infertile patients compared to fertile women (6.4%). They concluded that hydatid of Morgagni, through impeding the process of ovum pick-up, may be a new factor in infertility. Although the characteristics of the patients and the study design were completely different from those reported in our study, our data strongly support and reinforce these preliminary reports. The most evident point of the current study is the remarkable difference in pregnancy rates between the study and control groups amongst patients with bilateral (85.7% and 5.3% respectively, p < 0.001) and fimbrially located hydatid of Morgagni (85.6% and 9.1% respectively, p < 0.001). This finding was further supported by the regression model which revealed that both the
Fig. 2. Cummulative pregnancy rate in relation to the characters of hydatid of Morgagni.
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S.M. Rasheed, A.M. Abdelmonem / European Journal of Obstetrics & Gynecology and Reproductive Biology 158 (2011) 62–66
fimbrially located hydatid of Morgagni may disrupt these physiologic and vascular changes necessary for ovum pick-up is a question that remains unanswered. One of the most interesting findings in the present study is the high cumulative pregnancy rate in patients with bilateral and fimbrially located hydatid of Morgagni during the first 2 months of removal of the cysts. Once again, this finding seems to provide important evidence that hydatid of Morgagni may be an important cause of unexplained infertility. In conclusion, hydatid cysts of Morgagni are an important underestimated cause of unexplained infertility. Laparoscopic removal of these cysts in patients with unexplained infertility was followed by a high spontaneous pregnancy rate. This is particularly evident if the hydatid cyst was bilateral and/or fimbrial. More studies including larger number of patients are needed, however, to reach more significant conclusions. There are certain points of concern regarding the present study. The most evident of these is the ethical consideration concerned with leaving hydatid of Morgagni in a setting of infertile patients (control group). The authors thought that it was not unethical. The aim of the study was adequately explained to all patients and any patient who refused to participate in the study received her full clinical care. Moreover, as mentioned before, up till now there is no general consensus regarding the management of these cysts. Admittedly, considering the conclusions of the present study, the policy of our department nowadays is to remove any cyst of Morgagni irrespective of its character during laparoscopy conducted for infertile patients. Another point of limitation of the present study is the nonrandomization of the study design. At the beginning of the study the authors designed the study as a randomized one using a serially numbered closed envelopes. However, this randomization was not possible because a large number of patients refused to be included in the control group as they did not accept the policy of conserving the hydatid cysts. A third point of concern is the possible reformation of the cyst in the setting of patients who underwent de-roofing of the cysts.
Moreover, the possible formation of tubal adhesions following laparoscopic removal of these cysts is another concern. These points are difficult to assess because another laparoscopy is needed. However, the high pregnancy rate in the study group, the low risk of adhesions following laparoscopic surgery and the negligible use of electrocauterization, all together point to the assumption that the risk of adhesion formation, if present, is negligible.
References [1] Inborn MC. Global infertility and the globalization of new reproductive technologies: illustration from Egypt. Soc Sci Med 2003;56(9):1837–51. [2] Mohsen G, El Awady M, Abd El-Azeem O. Prevelance of infertility in rural areas of Kafr El Sheikh-Egypt: a community based study. J Egypt Public Health Assoc 2001;67(6):469–86. [3] The practice committee of the American society for reproductive medicine: effectiveness and treatment of unexplained infertility. Fertil Steril 2006;86(5):111–4. [4] The practice committee of the American society for reproductive medicine: optimal evaluation of the infertile female. Fertil Steril 2006;86(5):264–7. [5] Perlman S, Hertweck P, Fallt M. Paratubal and tubal abnormalities. Semin Pediatr Surg 2005;14(2):124–34. [6] Sunoo C, Terada K, Kamemoto L, Hale R. Adnexal masses in pregnancy: occurrence by ethnic group. Obstet Gynecol 1990;75(1):38–40. [7] Roy KK, Hedge P, Banerjee K, et al. Fimbrio-ovarian relationship in unexplained infertility. Gynecol Obstet Invest 2005;60(3):128–32. [8] Daiter E. Pelvic factor tutorial. Distal fallopian tube. Available at: http// www.infertilitytutorials.com. [9] Laboratory manual of the WHO for the examination of human semen and sperm-cervical mucus interaction. Ann 1st Super Sanita 2001; 1–123. 1-XII. [10] Blackert C, Ginsburg K, Leach R, Randolph J, Moghissi K. Unexplained infertility: evaluation of the luteal phase; results of the national center at Michigan. Fertil Steril 1997;67(3):437–42. [11] Helmerhorst F, Finken M, Erwich J. Antisperm antibodies: detection assays for antisperm antibodies: what do they test? Hum Reprod 1999;14(7): 1669–71. [12] Abd-El Maeboud KH. Hydatid of Morgagni: any impact on fertility. J Obstet Gynecol Res 1997;23(5):427–31. [13] Cebesoy FB, Kutlar I, Dikensoy E, Yazicioglu C, Kalayci H. Morgagni hydatids: a new factor in infertility? Arch Gynecol Obstet 2010;281(2):1015–7. [14] Stephan G, Rudi C, Luk R, Ivo B. Endoscopic visualization of the process of fimbrial ovum retrieval in the human. Hum Reprod 1998;13(6):1425–8.
Contents lists available at ScienceDirect
European Journal of Obstetrics & Gynecology and Reproductive Biology journal homepage: www.elsevier.com/locate/ejogrb
Hydatid of Morgagni: a possible underestimated cause of unexplained infertility Salah M. Rasheed *, Allam M. Abdelmonem Department of Obstetrics and Gynecology, Sohag Faculty of Medicine, Sohag University, Sohag, Egypt
A R T I C L E I N F O
A B S T R A C T
Article history: Received 8 October 2010 Received in revised form 4 March 2011 Accepted 26 April 2011
Objective: To evaluate the possible role of hydatid of Morgagni in patients with unexplained infertility. Study design: This was a non-randomized controlled trial conducted at the Department of Obstetrics and Gynecology of Sohag University, Egypt. Two hundred and thirteen patients with unexplained infertility and hydatid of Morgagni diagnosed at laparoscopy were included. The laterality (bilateral vs unilateral), location (fimbrial vs juxta-fimbrial), number (single vs multiple) and diameter of the hydatids of Morgagni were recorded. Patients were allocated to a study group (n = 127) who underwent laparoscopic excision of hydatid of Morgagni and a control group (n = 86) who underwent no intervention. Patients were followed for six months without any infertility or hormonal treatment to detect spontaneous pregnancy. Patients missed during the follow-up or who received infertility treatment were excluded. Statistical analysis was done using Chi-square test and Student’s t-test. To find the most important character of hydatid of Morgagni which impedes pregnancy, logistic regression analysis of the dependent variable (no pregnancy) and independent variables (different characters of hydatid of Morgagni) was carried out in the control group. Results: Hydatid of Morgagni was detected in 52.1% of patients with unexplained infertility compared to 25.6% of those with explained infertility (p < 0.001). The pregnancy rate was higher in the study group than the control group (58.7% vs 20.6%, p < 0.001). The pregnancy rate was significantly higher in the study group than the control group if the hydatid cyst was bilateral (85.7% vs 5.3%, p < 0.001), fimbrial (85.6% and 9.1%, p < 0.001), single (57.6% and 30.3%, p < 0.001) or 1–2 cm in diameter (58.1% and 25.5%, p < 0.001). Logistic analysis showed that the bilaterality and fimbrial location of the hydatid of Morgagni were the most significant characteristics impeding pregnancy (odds ratio = 7.27 and 3.67 respectively). Conclusions: Hydatid of Morgagni is a possible underestimated cause of unexplained infertility. Laparoscopic removal of hydatid of Morgagni in patients with unexplained infertility was followed with a high spontaneous pregnancy rate. This is particularly obvious with bilateral and fimbrial hydatid of Morgagni. ß 2011 Elsevier Ireland Ltd. All rights reserved.
Keywords: Hydatid of Morgagni Unexplained infertility Pregnancy
1. Introduction Infertility is a rather important health problem with a reported prevalence of about 8–10% worldwide [1,2]. Unexplained infertility, which has been reported to constitute about 15–30% of all causes of infertility, is a frustrating diagnosis both for the couple and the gynecologist [3]. Customarily, the diagnosis of unexplained infertility is assigned if the basic infertility investigations are normal. These investigations include semen analysis, detection of ovulation, hysterosalpingography, assessment of ovarian reserve, if indicated, and laparoscopy [4]. The diagnosis of unexplained infertility may suggest either deficiency of the currently available
* Corresponding author at: Obstetrics and Gynecology Department, Faculty of Medicine, Sohag University, University Street, Sohag, Egypt. Tel.: +20 932320071; fax: +20 932603963; mobile: +20 0124653702. E-mail address: [email protected] (S.M. Rasheed). 0301-2115/$ – see front matter ß 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ejogrb.2011.04.018
diagnostic tools to detect a hidden cause or the presence of unknown factors controlling the delicate process of human fertility. Hydatid cysts of Morgagni are vestigial remnants of the paramesonephric ducts which appear as sessile or pedunculated cystic structures at or near the fimbriae of the fallopian tubes [5]. Although hydatid cysts of Morgagni are frequently seen as an asymptomatic incidental finding during laparoscopy [6], their relation to infertility is still elusive. Not only is there considerable paucity of literature about the impact of hydatid of Morgagni on fertility, but also there is a lack of any scientifically sound consensus amongst gynecologists as to removing or leaving these cysts during laparoscopy done for infertile women (personal communication). The delicate and close fimbrio-ovarian relationship is a crucial factor for the process of ovum pick-up and hence conception [7]. Any factor, including cysts of Morgagni, which disrupts this intimate relationship may consequently hinder the process of
S.M. Rasheed, A.M. Abdelmonem / European Journal of Obstetrics & Gynecology and Reproductive Biology 158 (2011) 62–66
conception [8]. Accordingly, the aim of this study was to evaluate the effect of hydatid of Morgagni on the pregnancy rate in patients with unexplained infertility. 2. Materials and methods The present nonrandomized controlled trial was conducted between January 15, 2006 and January 15, 2010 at the Obstetrics and Gynecology Department of Sohag University Hospital, Egypt. During the study period, all patients with either primary or secondary infertility who were candidates for laparoscopy were invited to participate (n = 1141). During laparoscopy (Storz, Germany), patients with unexplained infertility and hydatid of Morgagni constituted the study group (n = 213). The laterality (bilateral vs unilateral), location (fimbrial vs juxta-fimbrial), number (single vs multiple), and diameter (measured in cm using the graded laparoscopic probe) of hydatids of Morgagni were assessed and recorded in all patients. The diagnostic criteria of unexplained infertility included infertility >1 year despite regular marital life, normal husband semen analysis according to WHO criteria [9], normal hysterosalpingography, regular ovulation for at least 3 consecutive cycles (documented by serial folliculometry and midluteal serum progesterone >10 ng/dl) and normal laparoscopic findings. These investigations were documented and conducted either at Sohag University Hospital or at other licensed hospitals or infertility units. The inclusion criteria were the diagnostic criteria of unexplained infertility, age <30 years, regular cycles, uneventful clinical examination and normal basal hormonal profile (serum FSH, LH, T3, T4 and prolactin). The exclusion criteria were refusal to participate, obesity (BMI >30 kg/m2), uterine or adnexal pathology detected either clinically or by trans-vaginal sonography, previous pelvic inflammatory disease, previous pelvic surgery and the presence of galactorrhea even with normal serum prolactin level. The institutional ethics committee provided approval and informed consent was obtained from all participants. The eligible participants were allocated into two groups; group I (study group; n = 127) underwent laparoscopic excision of hydatid cysts of Morgagni and group II (control group; n = 86) underwent no intervention. The cyst was easily excised using scissors if it was pedunculated. Otherwise the cyst was dissected from its bed using grasping, non-traumatic forceps and scissors. Electro-cauterization was not used to fulgurate the cysts, but bipolar diathermy was used, only if needed, to control bleeding from the cyst’s bed. A report on the laparoscopy was given to all patients and they were thereafter followed for 6 months without any infertility or hormonal treatment. The patients were asked to contact the Obstetrics and Gynecology Department of Sohag University Hospital once they had a missed period or positive pregnancy test, where serum human chorionic gonadotropin assay and transvaginal sonography were done to diagnose pregnancy. The remaining patients were asked to contact the department every 2 months just for assurance and answering any enquiries. Any patient who dropped-out during the 6-month follow-up period, or who received infertility or hormonal treatment or was enrolled into assisted reproduction, was excluded from the study. At the end of the study period, the pregnancy rate, either chemical or clinical, was calculated and compared between the 2 arms of the study using the Chi-square test (p < 0.05 was considered significant). Real variables were compared using the Student’s t-test. To find the most significant character of hydatid of Morgagni which impedes pregnancy, logistic regression analysis of the dependent variable (no pregnancy) and independent variables (different characters of hydatid of Morgagni) was carried out in the control group. The data were analyzed using SPSS version (SPSS, Chicago, USA).
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3. Results During the 4-year study period, a total of 1141 infertile patients underwent laparoscopy at the Department of Obstetrics and Gynecology of Sohag University Hospital as a part of infertility work-up. After laparoscopy, 409 patients (35.8%) were diagnosed as having unexplained infertility while 732 patients (64.2%) were assigned as having explained infertility. At laparoscopy, 213 patients (52.1%) with unexplained infertility were found to have hydatid of Morgagni compared to 188 patients (25.6%) of those with explained infertility (p < 0.001). One hundred and ninety six (48.8%) of the patients with unexplained infertility were therefore excluded and the remaining 213 patients were allocated into the study (n = 127) and control (n = 86) groups (Fig. 1). Laparoscopic extirpation of hydatid of Morgagni was successful in 119 patients (93.7%) while in 8 patients (6.3%) accidental rupture of the cyst occurred followed by incomplete removal (de-roofing). Bipolar diathermy was necessary to control bleeding from the bed of the cyst in one patient (0.8%). At the end of the 6-month follow-up period, 82 patients (64 patients in group I and 18 patients in group II) were excluded (37.3%). In group II, 11 patients dropped out during the follow-up and 7 patients received infertility treatment. The patients in group I were excluded due to diverse causes; 29 patients dropped out during the follow-up, 28 received infertility treatment, 4 underwent assisted reproduction, two were divorced and one patient was operated on due to appendicitis. The remaining 131 patients (63 in group I and 68 in group II) constituted the final study group. There was no statistically significant difference between the 2 groups regarding the mean age (26.2 3.1 vs 27.6 1.7 years, p > 0.05), duration of infertility (5.1 1.6 vs 6.2 2.4 years, p > 0.05) and body mass index (23.5 1.5 vs 25.1 1.9 kg/m2, p > 0.05) (data not shown). The laterality, location, number and diameter of hydatids of Morgagni were comparable between the 2 groups (Table 1). In the vast majority of patients in both groups the hydatid cysts were unilateral (66.7% vs 72.1%, p = 0.50), single (82.5% vs 79.4%, p = 0.64), located juxta-fimbrial (77.8% vs 83.8%, p < 0.37) and had diameters of 1–2 cm (68.3% vs 57.3%, p = 0.43). In group I, 37 patients (58.7%) conceived, compared to 14 patients (20.6%) in group II (p < 0.001). The pregnancy rate was higher in group I than group II irrespective of the laterality, location, number and diameter of the cysts of Morgagni. The pregnancy rate was higher in group I than group II whether the cyst was bilateral (85.7% and 5.1% respectively, p < 0.001) or unilateral (45.2% and 26.5%, p = 0.05). The pregnancy rate was higher in group I than group II whether the cyst was fimbrial (85.6% and 9.1% respectively, p < 0.001) or juxta-fimbrial (51.0% and 22.8%, p = 0.003). Regarding the number of the cysts, the pregnancy rate was higher in group I than group II whether the cyst was single (57.6% and 20.3% respectively, p < 0.001) or multiple (63.6% and 21.4% respectively, p = 0.05). The pregnancy rate was higher in group I than in group II regardless of their diameters, but the most significant difference was found if the diameter was 1–2 cm (58.1% and 20.5% respectively, p < 0.001). The results of the logistic regression analysis are displayed in Table 2. The regression model showed that bilaterality and fimbrial location of the hydatid of Morgagni were the most significant factors impeding with pregnancy (odds ratios = 7.27 and 3.67, respectively). Patients with bilateral and fimbrial hydatids of Morgagni reached their maximal cumulative pregnancy rate (85.7% and 85.6% respectively) at the end of the second and third months after removal of the cysts respectively. This is in contrast to patients with other characters of hydatid of Morgagni where the maximal
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Patients assessed for eligibility (n=1141)
Total excluded (n=196)
* Did have hydatid of Morgagni (n=93) * Did not meet inclusion criteria (n=44) * Refusal to participate (n=59)
Unexplained infertility (n=409)
Explained infertility (n=732)
Enrollment (n=213)
Excluded
Allocation (n=213)
Group I (n=127) Laparoscopic removal of hydatid of Morgagni * Dropped out during follow-up (n=29) * Received infertility treatment (n=28) * Underwent assisted reproduction (n=4) * Divorced (n=2) * Explored due to appendicitis (n=1)
Final analysis (n=63)
Pregnancy rate (58.7%)
Group II (n=86) No intervention
Excluded during follow-up (n=82)
Follow up and final analysis (n=131)
Main final outcome
* Dropped out during follow-up (n=11) * Received infertility treatment (n=7)
Final analysis (n=68)
Pregnancy rate (20.6%)
Fig. 1. Flow chart of the study.
cumulative pregnancy rate was reached over a longer time (6 months) (Fig. 2). 4. Comments Unexplained infertility is one of the most enigmatic problems of human reproduction. Although the diagnosis is reached by default, suggesting that the probable cause is still unclear, many causes such as luteal phase defect, [10] and antisperm antibodies [11] are postulated. Although hydatid cysts of Morgagni are amongst the most frequent benign conditions affecting the fallopian tubes [5], they have received negligible attention regarding their role in infertility. Moreover, to our knowledge there is no evidence-based approach concerned with the management of these cysts when
detected in infertile patients. The decision whether to remove or leave these cysts is dependent mainly on the gynecologists own opinion and judgment rather than being a scientifically based one. Some gynecologists elect to remove the cyst whatever its location or size while others consider it an incidental finding and pass over it unheeded. To the best of our knowledge, this is the first study which addresses the issue of the role of hydatid of Morgagni in unexplained infertility in a non-randomized controlled trial. The results of the present study provide evidence about the incriminating role of hydatid of Morgagni in unexplained infertility. The high prevalence of these cysts in patients with unexplained infertility (52.1%) compared with those with explained infertility (25.6%) may suggest a possible role.
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Table 1 Characters of hydatid of Morgangni and the pregnancy rates in the 2 groups of patients. Characters of hydatid of Morgagni
Laterality Bilateral Unilateral Location Fimbrial Juxtafimbrial Number Single Multiple Diameter (cm) <1 1–2 >2
Pregnancy rate
Group I n (%)
Group II n (%)
p Value
Group I n (%)
Group II n (%)
p Value
21 (33.3) 42 (66.7)
19 (27.9) 49 (72.1)
0.50
18 (85.7) 19 (45.2)
1 (5.3) 13 (26.5)
<0.001 0.05
14 (22.2) 49 (77.8)
11 (16.1) 57 (83.8)
0.37
12 (85.6) 25 (51.0)
1 (9.1) 13 (22.8)
<0.001 0.003
52 (82.5) 11 (17.5)
54 (79.4) 14 (20.6)
0.64
30 (57.6) 7 (63.6)
11 (30.3) 3 (21.4)
<0.001 0.05
13 (20.6) 43 (68.3) 7 (11.1)
19 (27.9) 39 (57.3) 10 (14.7)
0.43
7 (53.8) 25 (58.1) 5 (71.4)
3 (15.8) 8 (20.5) 3 (30.0)
0.02 <0.001 0.09
All data were given as number (percentage) unless otherwise indicated.
Table 2 Logistic regression analysis of the dependent variable (no pregnancy) indepenindependent variable (characters of hydatic of Morgagni) in the control group. Variables Laterality Bilateral Unilateral Location Fimbrial Juxtafimbrial Number Single Multiple Diameter (cm) <1 1–2 >2
Unadjusted OR (95% CI)
p Value
Adjusted OR (95% CI)
p Value
6.5 (0.78–53.68) 1 (reference)
0.82
7.27 (0.83–63.09) 1 (reference)
0.07
2.95 (0.34–25.3) 1 (reference)
0.32
3.67 (0.38–34.75) 1 (reference)
0.26
1 (reference) 1.07 (0.25–4.49)
0.09
1 (reference) 0.61 (0.12–3.04)
0.55
1 (reference) 0.73 (0.16–3.12) 0.43 (0.07–2.73)
0.67 0.37
1 (reference) 0.85 (0.18–3.98) 0.41 (0.06–3.01)
0.84 0.38
bilaterality and the fimbrial location of hydatid of Morgagni were the most important characters interfering with pregnancy. This finding can not only be considered as important evidence about the implication of hydatid of Morgagni in the problem of infertility but also it may support the previous assumption that hydatid of Morgagni impedes fertility through interfering with the process of ovum pick-up. The close fimbrio-ovarian relationship is a crucial event for the process of ovum pick-up. Any factor, even short fimbria, interfering with ovum pick-up can lead to infertility [7]. Owing to its weight, hydatid of Morgagni may displace the fimbriae away from the ovulatory side, leading to mechanical infertility. Moreover, using hydrolaparoscopy, it has been shown that the fimbriae at the time of ovulation were congested, elongated and erected with pulsatile movements attempting to suck the ovum [14]. Whether the
OR, odds ratio; CI, confidence interval.
The considerably higher pregnancy rate (58.7%) after laparoscopic extirpation of these cysts compared to the control group (20.6%) is another finding supporting the probable impact of hydatid of Morgagni on fertility. In this context, a study involving 3 patients with unexplained infertility who underwent laparoscopic removal of these cysts has been conducted [12]. The author reported a high pregnancy rate after removal of the cysts and concluded that hydatid of Morgagni as a single pelvic pathology might hinder fertility. Moreover, he advocated the policy of laparoscopic extirpation of these cysts in infertile patients. The prevalence of hydatid cysts of Morgagni in a setting of 240 fertile patients undergoing cesarean section was compared with that reported in 204 infertile patients undergoing laparoscopy [13]. The authors reported that the frequency of hydatid of Morgagni was significantly higher (17.8%) in infertile patients compared to fertile women (6.4%). They concluded that hydatid of Morgagni, through impeding the process of ovum pick-up, may be a new factor in infertility. Although the characteristics of the patients and the study design were completely different from those reported in our study, our data strongly support and reinforce these preliminary reports. The most evident point of the current study is the remarkable difference in pregnancy rates between the study and control groups amongst patients with bilateral (85.7% and 5.3% respectively, p < 0.001) and fimbrially located hydatid of Morgagni (85.6% and 9.1% respectively, p < 0.001). This finding was further supported by the regression model which revealed that both the
Fig. 2. Cummulative pregnancy rate in relation to the characters of hydatid of Morgagni.
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fimbrially located hydatid of Morgagni may disrupt these physiologic and vascular changes necessary for ovum pick-up is a question that remains unanswered. One of the most interesting findings in the present study is the high cumulative pregnancy rate in patients with bilateral and fimbrially located hydatid of Morgagni during the first 2 months of removal of the cysts. Once again, this finding seems to provide important evidence that hydatid of Morgagni may be an important cause of unexplained infertility. In conclusion, hydatid cysts of Morgagni are an important underestimated cause of unexplained infertility. Laparoscopic removal of these cysts in patients with unexplained infertility was followed by a high spontaneous pregnancy rate. This is particularly evident if the hydatid cyst was bilateral and/or fimbrial. More studies including larger number of patients are needed, however, to reach more significant conclusions. There are certain points of concern regarding the present study. The most evident of these is the ethical consideration concerned with leaving hydatid of Morgagni in a setting of infertile patients (control group). The authors thought that it was not unethical. The aim of the study was adequately explained to all patients and any patient who refused to participate in the study received her full clinical care. Moreover, as mentioned before, up till now there is no general consensus regarding the management of these cysts. Admittedly, considering the conclusions of the present study, the policy of our department nowadays is to remove any cyst of Morgagni irrespective of its character during laparoscopy conducted for infertile patients. Another point of limitation of the present study is the nonrandomization of the study design. At the beginning of the study the authors designed the study as a randomized one using a serially numbered closed envelopes. However, this randomization was not possible because a large number of patients refused to be included in the control group as they did not accept the policy of conserving the hydatid cysts. A third point of concern is the possible reformation of the cyst in the setting of patients who underwent de-roofing of the cysts.
Moreover, the possible formation of tubal adhesions following laparoscopic removal of these cysts is another concern. These points are difficult to assess because another laparoscopy is needed. However, the high pregnancy rate in the study group, the low risk of adhesions following laparoscopic surgery and the negligible use of electrocauterization, all together point to the assumption that the risk of adhesion formation, if present, is negligible.
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