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Case Report

Ruptured Hemorrhagic Corpus Luteum Cyst in an Undescended


Ovary: A Rare Cause of Acute Abdomen
Dong Soo Suh MD, PhD 1, Si Eun Han MD 1, Ka Yeong Yun MD 1, Nam Kyung Lee MD, PhD 2,
Ki Hyung Kim MD, PhD 1,*, Man Soo Yoon MD, PhD 1
1
Department of Obstetrics and Gynecology, Pusan National University School of Medicine and Biomedical Research Institute, Pusan National University Hospital, Busan,
Republic of Korea
2
Department of Radiology, Pusan National University School of Medicine and Biomedical Research Institute, Pusan National University Hospital, Busan, Republic of Korea

a b s t r a c t
Background: Undescended ovaries are typically detected during infertility evaluations and are frequently associated with uterine mal-
formations. Ruptured hemorrhagic corpus luteum cyst of an undescended ovary is an unusual cause of acute abdomen in an adolescent.
Case: A 15-year-old girl presented with right lower quadrant pain, nausea, and vomiting, and transabdominal sonography and magnetic
resonance imaging of the pelvis showed a 10 cm  5 cm sized cystic mass at the level of the pelvic brim, anterior to the psoas muscle
suggestive of a retroperitoneal hemorrhagic cyst. At surgery, the uterus and left adnexa appeared normal, but the right ovary was not
visible within the pelvic cavity, and the right pelvic retroperitoneum was distended. After opening the retroperitoneum and aspirating
blood clots, the undescended ovary with a ruptured cyst was visualized within the retroperitoneum. Right ovarian wedge resection was
performed and the right ovary was repositioned in the pelvic cavity.
Summary and Conclusion: Rupture of a corpus luteum cyst in an undescended ovary should be included in the differential diagnosis of acute
abdomen in adolescents.
Key Words: Undescended ovary, Corpus luteum cyst, Acute abdomen

Introduction the RLQ area containing a hemorrhagic portion. Magnetic


resonance imaging (MRI) of the pelvis depicted a 10  5 cm
Undescended ovaries and tubes are a relatively uncom- cystic lesion at the level of the pelvic brim, anterior to the
mon congenital disorder. Ovaries and testes have similar psoas muscle (Figure 1). On T2-weighted images, the lesion
developmental descending pathways, but ovaries descend presented as a 4  2.7 cm unilocular hypointense cyst that
from the posterior abdominal wall and end in the pelvic was entrapped within the hyperintense fluid-filled cystic
cavity. Furthermore, abnormalities in the descent of ovaries lesion. The central unilocular cyst showed fluid-fluid level
and tubes are less frequent.1 Common causes of acute on T2-weighted images, hyperintensity on T1-weighted
abdomen in adolescents, such as, acute appendicitis, torsion images, and thick wall with enhancement on contrast-
of an ovarian cyst, rupture of a corpus luteum cyst, and enhanced images, findings indicative of hemorrhagic
ectopic pregnancy should be included in the differential corpus luteum cyst. The peripheral cystic lesion was sus-
diagnosis. Here, we report a rare cause of acute abdomen in pected to be a peritoneal inclusion cyst. The uterus, vagina,
a patient who presented with a retroperitoneal unde- right ovary, and both kidneys were normal in appearance on
scended ovary with corpus luteum hemorrhage. MRI. The RLQ pain, nausea and vomiting persisted after the
patient received painkillers and antiemetics. The patient
Case was taken to the operating room for diagnostic laparoscopy.
The uterus and left adnexa appeared normal but the right
A 15-year-old girl presented with aggravating right lower ovary was not visible within the pelvic cavity, and the right
quadrant (RLQ) pain, nausea, and vomiting. Her last men- pelvic retroperitoneum was distended. The retro-
strual period had occurred 3 weeks previously. She had a peritoneum was opened and hemorrhage and blood clots
history of surgery for a right inguinal hernia at 9 months of were aspirated. The right ovary with a ruptured cyst was
age. Physical examination revealed tenderness in the RLQ visualized. The right tube was not in communication with
but no peritoneal signs. Her vital signs were stable, her urine the uterus, and showed some swelling and a hydrosalpinx
pregnancy test was negative, and laboratory examinations pattern. Right ovarian wedge resection and salpingectomy
were within normal limits. Transabdominal sonography were performed. The retroperitoneum was repaired, and
showed a 9.5  4.5 cm, well circumscribed, cystic mass in then the right ovary was repositioned at the end of the
repaired peritoneum at the level of the contralateral ovary.
The authors indicate no conflicts of interest. No other associated malformations were found.
* Address correspondence to: Ki Hyung Kim, MD, PhD, Department of Obstetrics Her postoperative recovery was uneventful, and she was
and Gynecology, Pusan National University School of Medicine, 179, Gudeok-Ro,
Seo-Gu, Busan 602-739, Korea; Phone 82-51-240-7287:
discharged 3 days after surgery. Postoperative pathological
E-mail address: ghkim@pusan.ac.kr (K.H. Kim). findings revealed a corpus luteal cyst of the ovary.
1083-3188/$ - see front matter Ó 2016 North American Society for Pediatric and Adolescent Gynecology. Published by Elsevier Inc.
http://dx.doi.org/10.1016/j.jpag.2015.09.004
e22 D.S. Suh et al. / J Pediatr Adolesc Gynecol 29 (2016) e21ee24

Figure 1. Magnetic resonance imaging findings of ruptured hemorrhagic corpus luteum cyst of an undescended ovary. (A) Coronal and (B) sagittal T2-weighted images of the pelvis
show a unilocular hemorrhagic cyst (arrows) in the right undescended ovary at the level of the pelvic brim, anterior to the psoas muscle. The hemorrhagic cyst of the right ovary is
entrapped within the fluid-filled peritoneal inclusion cyst (*). (C) On axial T1-weighted image, the right undescended ovary appears as a hyperintense hemorrhagic cyst (arrow). (D)
Axial contrast-enhanced fat-suppressed T1-weighted image shows a peripheral thick wall with enhancement (arrowhead) in the hemorrhagic cyst. (E) Axial T2-weighted image
shows a normal uterus and left-sided ovary. (F) Axial T1-weighted image of the upper abdomen shows normal kidneys.

Karyotype analysis showed a normal female chromosome. occur unilaterally or bilaterally, and might occur in patients
During follow-up, the patient reported regular menstrua- with a normal uterus, but its incidence is significantly
tion and no recurrence of abdominal pain, nausea, or greater in women with uterine anomalies.5 In cases of
vomiting. unicornuate uterus, the incidence of ectopic or unde-
scended ovary has been reported to be as high as 42%.5 In
Summary and Conclusion addition, unilateral undescended ovary has been reported
to be more common on the right side and more likely to be
We report a rare cause of acute abdomen that presented located in the retroperitoneum.6 Although undescended
with a ruptured hemorrhagic corpus luteum cyst of an un- ovary is associated with infertility, spontaneous pregnancy
descended ovary in an adolescent girl. The reported inci- could be possible when a patient possesses at least 1 normal
dence of undescended ovary is 0.3%-2%,2 and the condition tube and 1 normal ovary.6
can occur when the gubernaculum fails to attach to the Undescended ovary is uncommonly diagnosed in isola-
lower pole of the gonad.3 Defects in the developments of tion, and might be more frequent in infertile individuals or
the Mu€ llerian structures can be accompanied by renal in association with developmental anomalies of the uterus
anatomical defects, such as ectopic location, aberrant or renal collecting system.2 In a previous report, 77% of
anatomy, or complete agenesis.4 Undescended ovary can reported cases were asymptomatic and diagnosed
D.S. Suh et al. / J Pediatr Adolesc Gynecol 29 (2016) e21ee24 e23

Table 1
Symptomatic Cases of Undescended Ovary

Source Cases Age, years Mu€ llerian Renal Anomalies Method of Preoperative Postoperative Affected
Anomalies Diagnosis Diagnosis Diagnosis Side

Kives et al8 1 13 Bicornuate uterus None CT, laparoscopy Congenital intestinal Ruptured hemorrhagic Both
duplication cyst of right ovary
Gabriel et al* 1 29 Left unicornuate Right renal agenesis IVP, laparoscopy, Ectopic pregnancy Right tubal pregnancy Right
uterus USG
Cohen et al* 1 18 None None CT, laparoscopy abdominal wall Functional cyst of left Left
hemangioma ovary
Pokoly et al* 1 26 Right unicornuate None HSG, laparoscopy Ectopic pregnancy Primary amenorrhea Left
uterus
Seoud et al* 1 28 Right unicornuate Left pelvic kidney Laparotomy Ectopic pregnancy Left tubal pregnancy Left
uterus
Brown et al* 1 24 Right unicornuate Left renal agenesis IVP, laparotomy, Ectopic pregnancy Left tubal pregnancy Left
uterus USG
Granat et al* 1 23 Right unicornuate Left renal agenesis HSG, IVP, laparotomy Ectopic pregnancy Left tubal pregnancy Left
uterus
Dabby et al* 1 34 None Right renal agenesis IVP, laparotomy None Right tubal pregnancy Right
Adnopoz et al* 1 22 None Not listed Laparotomy Torsion of left Torsion of left ovarian Left
ovarian cyst cyst
Nichols et al* 1 36 None Left renal agenesis Laparotomy Acute cholecystitis Hemorrhagic cyst of Left
right ovary
Present case 1 14 Left unicornuate None Laparoscopy, MRI, Pseudocyst Ruptured hemorrhagic Right
(2015) uterus USG cyst of right ovary

CT, computed tomography; HSG, hysterosalpingography; IVP, intravenous pyelography; MRI, magnetic resonance imaging; USG, ultrasonography
* Dietrich et al.7

incidentally during infertility evaluation.2 Furthermore, oocytes from an undescended ovary for in vitro fertilization
ectopic pregnancy rates are increased in these populations, were reported.2 In addition, some authors have concluded
with a prevalence of 4%,2 especially when an undescended that ovarian stimulation offers a useful approach to facili-
ovary has a rudimentary or ectopic tube or undescended tate the diagnosis of undescended ovary, especially in pa-
ovary is bilateral. If there are additional uterine anomalies, tients with uterine anomalies.5 Sometimes, an
such as unicornuate uterus, the risk of miscarriage in- undescended ovary becomes symptomatic and might cause
creases.2 More than 10 cases of successful retrieval of an acute surgical abdomen. In symptomatic cases, the

Table 2
Asymptomatic Cases of Undescended Ovary

Source Case Age, Mu€ llerian Renal Method of Diagnosis Diagnosis during Chief Problem Affected
years Anomalies Anomalies Infertility Workup Side
(yes or no)

Uyar et al1 1 26 Left unicornuate Right renal HSG, laparoscopy, MRI Yes Primary infertility Right
uterus agenesis
Halil et al* 1 27 None None Laparoscopy, USG Yes Secondary infertility Right
Trinidad et al2 1 20 None None HSG, MRI, USG No Menstrual irregularity Right
2 14 MRKH syndrome Left renal MRI, USG No Primary amenorrhea Left
agenesis
Ombelet et al3 1 Not listed Right unicornuate None HSG, laparoscopy, MRI Yes Primary infertility Left
uterus
2 26 Right unicornuate None HSG, hysteroscopy, Yes Primary infertility Left
uterus laparoscopy, MRI
3 Not listed Right unicornuate Left renal HSG, laparoscopy, MRI Yes Primary infertility Left
uterus agenesis
4 Not listed Left unicornuate Right renal HSG, laparoscopy, MRI Yes Primary infertility Right
uterus agenesis
4
Ombelet et al 1 28 Right unicornuate None HSG, hysteroscopy, IVP, Yes Secondary infertility Left
uterus laparoscopy, MRI
Ombelet et al* 1 Not listed Right unicornuate Left renal HSG, laparoscopy, MRI Yes Primary infertility Left
uterus agenesis
2 Not listed Left unicornuate None MRI, USG Yes Primary infertility Right
uterus
5
Gorgen et al 1 19 Bicornuate uterus None HSG, laparoscopy, USG Yes Primary infertility Both
Van Voorhis et al6 1 35 Bicornuate uterus None HSG, laparoscopy Yes Secondary infertility Both
Verkauf et al* 1 27 Bicornuate uterus None HSG, laparoscopy Yes Primary infertility Both
2 31 None None HSG, hysteroscopy, Yes Secondary infertility Left
laparoscopy
3 33 Left unicornuate None HSG, laparoscopy Yes Primary infertility Right
uterus
4 32 None None HSG, laparoscopy Yes Secondary infertility Right
5 35 None None HSG, laparoscopy Yes Secondary infertility Right
€ ster-Hauser; USG, ultrasonography.
HSG, hysterosalpingography; IVP, intravenous pyelography; MRI, magnetic resonance imaging; MRKH, Mayer-Rokitansky-Ku
* Dietrich et al.7
e24 D.S. Suh et al. / J Pediatr Adolesc Gynecol 29 (2016) e21ee24

Table 3 postoperatively (Table 1).7,8 Furthermore, a comparison of


Comparison Between Asymptomatic Type and Symptomatic Type of Undescended
Ovary
these 18 asymptomatic and symptomatic cases (Table 3)
revealed mean age at diagnosis of 26.5  5.93 and
Variable Asymptomatic Type Symptomatic Type
24.3  7.07 years, respectively. The symptomatic type was
Number 18 cases 11 cases
usually diagnosed after MRI and surgery, and the asymp-
Mean age  SD, years 26.5  5.93 24.3  7.07
Method of diagnosis Infertility workup, ovarian MRI, surgery tomatic type was diagnosed during infertility workup and
hyperstimulation ovarian hyperstimulation. Mu € llerian anomaly and renal
Mullerian anomaly 10 of 17 (55.6%) 7 of 11 (63.4%)
anomaly were more common for the symptomatic type.
Renal anomaly 5 of 17 (29.4%) 5 of 10 (50%)
Affected site Affected sites were not different for the asymptomatic type.
Right 7 of 17 (41.2%) 2 of 11 (18.2%) However, the symptomatic type usually affected the left
Left 7 of 17 (41.2%) 7 of 11 (63.6%)
side, and a history of inguinal hernia was more common for
Both 3 of 17 (17.6%) 1 of 11 (9.1%)
History of Inguinal hernia 1 case 3 cases the symptomatic type (three cases vs one case). However,
MRI, magnetic resonance imaging.
small case numbers obscure the relation between unde-
scended ovary and inguinal hernia.
We report herein on a case of retroperitoneal unde-
common symptom is abdominal pain secondary to a scended ovary with corpus luteum hemorrhage as a rare
ruptured ovarian cyst or ovarian torsion.6 Furthermore, the cause of acute abdomen. In addition, we included a com-
condition might easily be mischaracterized because of parison of the features of asymptomatic and symptomatic
diverse preoperative impressions (Table 1).7 In all reported undescended ovary. Notably, the asymptomatic type is
cases, a definite diagnosis was made after surgery. Many usually diagnosed during infertility workup and the
authors have reported that MRI is the best diagnostic im- symptomatic type is usually diagnosed after surgery
aging modality for undescended ovary, because it is because of abdominal pain.
noninvasive, highly sensitive, and specific for the diagnoses
of genital tract and combined renal anomalies.2,5
Conservative management is preferentially considered References
for undescended ovary. Morbid ovaries or tubes are not
always indicated for resection because no association has 1. Uyar I, Gulhan I, Sipahi M, et al: Ectopic ovary confirmed by ovarian stimulation
in a case of unicornuate uterus. Fertil Steril 2011; 96:e122
been found between it and premature ovarian failure, 2. Trinidad C, Tardaguila F, Fernandez GC, et al: Ovarian maldescent. Eur Radiol
polycystic ovaries, endometriosis, or other gynecological 2004; 14:805
3. Ombelet W, Grieten M, DeNeubourg P, et al: Undescended ovary and
conditions.1,2 However, a noncommunicating fallopian tube unicornuate uterus: simplified diagnosis by the use of clomiphene citrate
should be removed immediately to prevent ectopic preg- ovarian stimulation and magnetic resonance imaging (MRI). Hum Reprod
nancy when recognized.6 Undescended ovary might be 2003; 18:858
4. Ombelet W, Deblaere K, Grieten M, et al: Intrauterine pregnancy following
associated with a previous history of ipsilateral inguinal transperitoneal oocyte and/or sperm migration in a woman with an ectopic
hernia,5,7 as was shown in our case. (undescended) ovary. Reprod Biomed Online 2003; 7:110
5. Gorgen H, Api M, Delikara N: Undescended fallopian tubes and ovaries: a rare
Twenty-nine cases (including our case) of asymptomatic incidental finding during an infertility investigation work up. Acta Obstet
or symptomatic undescended ovary have been reported. Of Gynecol Scand 2002; 81:371
6. Van Voorhis BJ, Dokras A, Syrop CH: Bilateral undescended ovaries: association
these, 18 cases (62%) were asymptomatic and 11 (38%) were with infertility and treatment with IVF. Fertil Steril 2000; 74:1041
symptomatic. Sixteen of the asymptomatic cases were 7. Dietrich JE, Hertweck SP, Bond S: Undescended ovaries: a clinical review.
J Pediatr Adolesc Gynecol 2007; 20:57
diagnosed at the time of infertility work-up (Table 2).1e6 8. Kives SL, Perlman S, Bond S: Ruptured hemorrhagic cyst in an undescended
However, all reported symptomatic cases were diagnosed ovary. J Pediatr Surg 2004; 39:e4

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