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Ruptured hemorrhagic cyst of undescended ovary mimicking mucocele: A rare pediatric case.

Abstract

Undescended ovary is a rare entity and usually presentedas a case report. It is associated with urinary and uterine anomalies. Symptomatic patients are diagnosed during surgery. Most of the patients are asymptomatic and treatment is unnecessary. They are incidentally diagnosed during infertility evaluation and treatment such as ovarian hyperstimulation studies. A 15-year-old female patient presented with the diagnosis of renal cystic massas identifiedduring ultrasonography in another hospital. Abdominal computedtomography image was requested. A cystic lesion of about 48x34 mm with well-defined borders associated with the appendix and probably with mucocele of the appendix was reported in the lower right abdominal quadrant close to the cecum. During exploration, the right ovary was seen to be attached to the cecum and was higher in position as well as a right ovary originated cystic structure of 5x5 cm. Using needle aspiration,intraovarian hemorrhage was confirmed and partial cystectomy was performed. The present study reports on an undescended ovary that hadacute abdomen symptoms imitating mucocele. In girls referring to the hospital with abdominal pain, although quite rare, undescended ovaries are to be also considered. As the incidence of renal and uterine anomalies is higher in suchpatients, in symptomatic cases relevant organs are to be investigated carefully during surgical intervention.

Please cite this article as: Sekmenli T, Gunduz M, Ciftci I. Ruptured Hemorrhagic Cyst of Undescended Ovary Mimicking Mucocele: A Rare Pediatric Case. Iran J Med Sci. 2017;42(1):98-101.

Keywords * Abdominal pain * Mucocele * Endometriosis * Ovarian neoplasms

Introduction

Undescended ovaries and tubes are rare congenital disorders. While they develop and descend like testicles, ovaries differ as they descend from the posterior abdominal wall and end in the pelvic cavity. There are fewer abnormalities in the ovaries and tubular descent. (1)

In the differential diagnosis of cases with acute abdomen symptoms in adolescents, the underlying causes such as acute appendicitis, ruptured corpus luteum cyst, torsion of an ovarian cyst, and ectopic pregnancy are to be considered by surgeons. Although quite rare and clinically not considered in the initial diagnosis, herein we report a case of undescended ovary mimicking mucocele of the appendix that was presented to our clinic as acute abdomen.

Case Presentation

A 15-year-old female patient was referred to our clinic with continuous abdominal pain for the last four days. Initial diagnosis of renal cystic mass was made in another hospital based on ultrasonography(USG). Physical examination revealed tenderness in the right lower quadrant. The last menstrual period of the patient was 2 weeks prior to admission.

Patient's hemogram and hormonal profiles did not reveal any pathology. Abdominal computed tomography (CT) image was requested. A cystic lesion of about 48x34 mm with well-defined borders associated with the appendix and probably with mucocele of the appendix was reported in the lower right abdominal quadrant close to the cecum based on the tomography image (figure 1). Opting for surgical intervention and upon obtaining informed consent for surgery, the patient was admitted to our pediatric surgery clinic in 2015. Using an infraumbilical right transverse incision, abdominal access was realized. During surgical exploration, the right ovary was seen to be attached to the cecum and was higher in position as well as a right ovary originated cystic structure of 5x5 cm. Using needle aspiration, intraovarian hemorrhage was confirmed and partial cystectomy was performed. Current clinical condition was decided to be the result of hemorrhagic cyst rupture. The uterus and the other ovary were considered as normal. Similarly, her appendix was normal and did not reveal any pathology. The patient was discharged without any postoperative problems the next day. Three weeks later, during the USGcontrols, the right ovary was considered as normal and no pathologies could be determined.

Discussion

During intrauterine growth period of the fetus, the ovarian tissue descents from the medial of the urogenital folds down to the pelvic cavity. If there are any interruptions during this descent, the ovary is seen above the normal ovarian location and is hence called undescended ovary. In utero 5th month, the ovaries are in the iliac fossa and at that term at the pelvic ridge. During the postpartum period, they are supposed to have moved to their normal location. (2,3)

The term "ectopic ovary" is often used for abnormal localized excess ovarian tissue in addition to the normal ovary. (4) Undescended ovary and ectopic ovary are among the extremely rare cases without a clear incidence. Undescended ovary can be unilateral or bilateral, even in patients with normal uterus. However, its incidence is significantly higher in the presence of uterine anomalies. (5)

Anomalies such as Mullerian agenesis or unicornuate uterus are commonly seen in undescended ovary cases. Anomalies such as renal agenesis are also witnessed in patients. In patients with congenital uterine, anomalies ovarian malposition (ovarian maldescent) are more common. (6,7) To the best of our knowledge, there is no established definitive association between undescended ovary and infertility or malignancy. Undescended ovary is generally associated with urinary and uterine anomalies. Most of the patients for whom treatment is considered unnecessary are asymptomatic and diagnosed during infertility evaluation and treatment. Including the present report, as far as we know, there have been only 30cases of asymptomatic or symptomatic undescended ovary reports. Among these, 18 (60%) cases were asymptomatic and 12 (40%) were symptomatic (table 1). (4,8-14) All symptomatic cases were similar to the present case and diagnosed during surgical intervention. Asymptomatic cases were diagnosed after infertility and ovarian hyper stimulation studies. (8) Moreover, unilateral undescended ovaries are reported to be more common on the right side and more likely to be located in the retro peritoneum. (15) Mullerianand renal anomalies were more common in symptomatic undescended ovary cases.

Undescended ovary can become symptomatic and lead to acute surgical abdomen. The most common symptoms are abdominal pain secondary to a ruptured ovarian cyst or ovarian torsion. (15) In our patient, due to the high preoperative localization of the ovary in the abdominal CT-scan, mucocele was considered during the primary examination. In the diagnosis of similar cases, magnetic resonance imaging(MRI) is preferred by many clinicians due to its high sensitivity and as a noninvasive method for renal and genital tract anomaly diagnoses. (5) If undescended ovaries are determined incidentally, there is no need for any kind of radical intervention unless they are symptomatic. (8)

Cystic formations of the undescended ovary, if located on the right, can be confused with acute appendicitis, plastrone appendicitis, paracolic abscess, or mucocele. Although quite rare, in young female patients with severe abdominal pain, the possibility of undescended ovaries should also be considered. In symptomatic cases, during surgical intervention, relevant neighboring tissues are to be investigated carefully.

We have reported a case that had undescended ovary presenting with acute abdomen symptoms mimicking mucocele of the appendix. Although rare, but it should be part of a differential diagnosis in patients with peritoneal irritation symptoms. Surgical exploration must be carefully made in suspected cases.

Conclusion

In women, especially underagecases, painful acute abdominal symptoms in the right lower quadrantmight be related to hemorrhagic cystic complications of the undescended ovary. In the absence of surgical intervention and solely based on radiological analysis, it can be confused with the mucocele of the appendix. As these patients are often diagnosed during the operation, a thorough surgical exploration of the uterus for the presence gonadal anomalies is deemed necessary due to inherent infertility risk.

Conflict of Interest: None declared.

References

(1.) Uyar I, Gulhan I, Sipahi M, Hanhan HM, Ozeren M. Ectopic ovary confirmed by ovarian stimulation in a case of unicornuate uterus. Fertil Steril. 2011;96:e122-4. doi: 10.1016/j.fertnstert.2011.05.020. PubMed PMID: 21640342.

(2.) Yoshinaga K, Hess DL, Hendrickx AG, Zamboni L. The development of the sexually indifferent gonad in the prosimian, Galago crassicaudatus crassicaudatus. Am J Anat. 1988;181:89-105. doi: 10.1002/aja.1001810110. PubMed PMID: 3348150.

(3.) Speroff L, Fritz MA. The ovary-embryology and development. Philadelphia: Clinical gynecologic endocrinology and infertility Lippincott Williams & Wilkins; 2005. p. 97-111.

(4.) Cohen JA, Holzman. A giant ectopic ovary. J Laparoendosc Adv Surg Tech A. 2001;11:31-5. doi: 10.1089/10926420150502913. PubMed PMID: 11444321.

(5.) Gorgen H, Api M, Delikara N. Undescended fallopian tubes and ovaries: a rare incidental finding during an infertility investigation work up. Acta Obstet Gynecol Scand. 2002;81:371-4. PubMed PMID: 11952473.

(6.) Dabirashrafi H, Mohammad K, Moghadami-Tabrizi N. Ovarian malposition in women with uterine anomalies. Obstet Gynecol. 1994;83:293-4. PubMed PMID: 8290197.

(7.) Allen JW, Cardall S, Kittijarukhajorn M, Siegel CL. Incidence of ovarian maldescent in women with mullerian duct anomalies: evaluation by MRI. AJR Am J Roentgenol. 2012;198:W381-5. doi: 10.2214/AJR.11.6595. PubMed PMID: 22451577.

(8.) Suh DS, Han SE, Yun KY, Lee NK, Kim KH, Yoon MS. Ruptured Hemorrhagic Corpus Luteum Cyst in an Undescended Ovary: A Rare Cause of Acute Abdomen. J Pediatr Adolesc Gynecol. 2016;29:e21-4. doi: 10.1016/j.jpag.2015.09.004. PubMed PMID: 26403474.

(9.) Kives SL, Perlman S, Bond S. Ruptured hemorrhagic cyst in an undescended ovary. J Pediatr Surg. 2004;39:e4-6. PubMed PMID: 15547819.

(10.) Gabriel B, Fischer DC, Sergius G. Unruptured pregnancy in a non-communicating heterotopic right fallopian tube associated with left unicornuate uterus: evidence for transperitoneal sperm and oocyte migration. Acta Obstet Gynecol Scand. 2002;81:91-2. PubMed PMID: 11942895.

(11.) Pokoly TB. Ectopic pregnancy in a noncommunicating tube of a unicornuate uterus. A case report. J Reprod Med. 1989;34:994-5. PubMed PMID: 2621743.

(12.) Kim SJ, Cho DJ, Song CH. Ovarian transposition with subsequent intrauterine pregnancy. Fertil Steril. 1993;59:468-9. PubMed PMID: 8425652.

(13.) Granat M, Evron S, Navot D. Pregnancy in heterotopic fallopian tube and unilateral ovarian hyperstimulation. Acta Obstet Gynecol Scand. 1981;60:215-7. PubMed PMID: 7246088.

(14.) Nichols C, Koong D, Faulkner K, Thompson G. A hepatic ectopic pregnancy treated with direct methotrexate injection. Aust N Z J Obstet Gynaecol. 1995;35:221-3. PubMed PMID: 7677699.

(15.) Van Voorhis BJ, Dokras A, Syrop CH. Bilateral undescended ovaries: association with infertility and treatment with IVF. Fertil Steril. 2000;74:1041-3. PubMed PMID: 11056257.

Tamer Sekmenli, MD; Metin Gunduz, MD; Ilhan Ciftci, MD

Department of Pediatric Surgery, Selcuk University, Faculty of Medicine Konya, Turkey

Correspondence:

Tamer Sekmenli, MD; Department of Pediatric Surgery, Faculty of Medicine, Selcuk University Alaeddin Keykubat Yerleskesi, Selcuklu Konya 42131, Turkey.

Tel: +90 332 2412181-83

Fax: +90 332 2412184

Email: dr_sekmenli@hotmail.com

Received: 20 February 2016

Revised: 26 April 2016

Accepted: 8 May 2016

What's Known

* Undescended ovary is a rare entity. It is associated with urinary and uterine anomalies.

* Most patients in whom treatment is unnecessary are asymptomatic and diagnosed during infertility evaluation and treatment such as ovarian hyperstimulation studies.

What's New

* The present study reports on an undescended ovary that had acute abdomen symptoms imitating mucosal.

* In girls referring to a hospital with abdominal pain, although quite rare, undescended ovaries are also to be considered.

* In symptomatic cases, relevant organs are to be investigated carefully during surgical intervention.

Table 1: All reported cases of symptomatic undescended ovary

Source       Age,     Affected  Method of     Preoperative
             cases    side      diagnosis     diagnosis
             (years,
             number)

Cohen        18,1     Left      CT,           Abdominal
et al. (4)                      Laparoscopy   wall
                                              hemangioma
Suh          14,1     Right     Laparoscopy,  Pseudocyst
et al. (8)                      MRI, USG

Adnopoz      22,1     Left      Laparotomy    Torsion of left
et al. (8)                                    ovarian cyst
Brown        24,1     Left      IVP,          Ectopic
et al. (8)                      Laparotomy,   pregnancy
                                USG
Dabby        34,1     Right     IVP,          None
et al. (8)                      Laparotomy
Kives        13,1     Both      CT,           Congenital
et al. (9)                      Laparoscopy   intestinal
                                              duplication

Gabriel      29,1     Right     IVP,          Ectopic
et al. (10)                     Laparoscopy,  pregnancy
                                USG
Pokoly (11)  26,1     Left      HSG,          Ectopic
                                Laparoscopy   pregnancy
Kim (12)     28,1     Left      Laparotomy    Ectopic
                                              pregnancy
Granat       23,1     Left      HSG, IVP,     Ectopic
et al. (13)                     Laparotomy    pregnancy
Nichols      36,1     Left      Laparotomy    Acute
et al. (14)                                   cholecystitis

Present      15,1     Right     USG, CT,      Mucocele
case                            Laparatomy

Source       Postoperative    Renal        Mulleriananomalies
             diagnosis        anomalies

Cohen        Functional       None         None
et al. (4)   cyst of left
             ovary
Suh          Ruptured         None         Left unicornuate
et al. (8)   hemorrhagic                   uterus
             cyst of right
             ovary
Adnopoz      Torsion of left  Not listed   None
et al. (8)   ovarian cyst
Brown        Left tubal       Left renal   Right unicornuate
et al. (8)   pregnancy        agenesis     uterus

Dabby        Right tubal      Right renal  None
et al. (8)   pregnancy        agenesis
Kives        Ruptured         None         Bicornuate uterus
et al. (9)   hemorrhagic
             cyst of right
             ovary
Gabriel      Right tubal      Right renal  Left unicornuate
et al. (10)  pregnancy        agenesis     uterus

Pokoly (11)  Primary          None         Right unicornuate
             amenorrhea                    uterus
Kim (12)     Left tubal       Left renal   Right unicornuate
             pregnancy        agenesis     uterus
Granat       Left tubal       Left renal   Right unicornuate
et al. (13)  pregnancy        agenesis     uterus
Nichols      Hemorrhagic      Left renal   None
et al. (14)  cyst of right    agenesis
             ovary
Present      Ruptured         None         None
case         hemorrhagic
             cyst of right
             ovary
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Article Details
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Title Annotation:Case Report
Author:Sekmenli, Tamer; Gunduz, Metin; Ciftci, Ilhan
Publication:Iranian Journal of Medical Sciences
Article Type:Report
Geographic Code:7IRAN
Date:Jan 1, 2017
Words:2125
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