Ultrasound Obstet Gynecol 2015; 46: 243–246 Published online 2 July 2015 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/uog.14853
Ultrasound-guided percutaneous aspiration of hyperreactio luteinalis avoids laparoscopic untwisting of ovarian torsion C. SAKAE, Y. SATO, A. TAGA, Y. SATAKE, I. EMOTO, S. MARUYAMA and T. KIM Department of Obstetrics and Gynecology, Otsu Red Cross Hospital, Nagara, Otsu, Shiga, Japan
K E Y W O R D S: acute abdomen; detorsion; functional ovarian cyst; hydrops fetalis; laparoscopy
ABSTRACT Hyperreactio luteinalis (HL) is characterized by multicystic bilateral enlargement of the ovaries and is a selflimiting benign condition associated with pregnancy or trophoblastic disease. Since HL regresses spontaneously over time, it should be managed conservatively as long as the patient’s condition permits; torsion of the enlarged ovaries is believed to be the only exception that mandates surgical intervention. Here, we describe a case of HL complicated by ovarian torsion that was treated successfully without surgical intervention. A 33-year-old woman was admitted to our hospital owing to acute abdomen. Nine days previously, she had had a stillbirth caused by hydrops fetalis at 24 weeks’ gestation. The characteristic findings observed on magnetic resonance imaging (MRI) led to the diagnosis of HL complicated by torsion of the enlarged left ovary. Emergency laparoscopic detorsion of the ischemic left ovary was planned. Aiming to reduce the risk of cystic injury and bleeding at the trocar insertion site, volume reduction of the left ovarian cyst was performed by percutaneous aspiration. The patient’s pain diminished rapidly and laparoscopic surgery was deferred. Subsequent MRI revealed that gadolinium enhancement of the left ovarian tumor had been restored, indicating spontaneous detorsion of the left ovary. The patient remained asymptomatic and was discharged from the hospital 12 days after aspiration of the cyst. From this experience, we propose that, in cases of ovarian torsion occurring in large functional cysts, including HL, volume reduction by percutaneous cyst aspiration should be considered before performing emergency laparoscopic surgery. Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
CASE REPORT A 33-year-old woman, gravida 3 para 2, presented at the emergency room of our hospital owing to acute abdomen. Transabdominal ultrasound examination (TAS) revealed
bilateral multicystic tumors occupying the pelvic cavity, and the patient was subsequently admitted to the obstetrics and gynecology department. The patient’s third pregnancy had ended, 9 days previously, in fetal demise at 24 weeks’ gestation caused by hydrops fetalis associated with cystic hygroma (karyotype was normal 46,XX). On the 2nd day postpartum, she had been discharged from the hospital with unremarkable transvaginal ultrasound findings. On admission to the obstetrics and gynecology department, the patient’s lower abdomen was slightly distended and she had severe pain and tenderness, which were dominant on the left side. Subsequent blood testing found elevated white blood cell (WBC) count (12 900/μL), C-reactive protein (CRP) (0.7 mg/dL) and D-dimer (15.8 μg/mL) levels. Magnetic resonance imaging (MRI) confirmed bilateral ovarian multicystic enlargement, reaching more than 10 cm in diameter (Figure 1a). Aggregation of coiled tubular structures, which was considered to represent twisted and congested blood vessels, was observed between the uterus and the left ovarian tumor (Figure 1a). Some of these blood vessels contained nodules of low signal intensity, suggestive of intravenous clot formation. On T1-weighted MRI, the septa and surface of the left ovarian tumor were poorly enhanced by gadolinium, as compared with the right side (Figure 1b). From these observations, the patient was diagnosed with hyperreactio luteinalis (HL) complicated by torsion of the enlarged left ovary. Serum levels of β-human chorionic gonadotropin (β-hCG) and testosterone were 1550 mIU/mL and 3.22 ng/mL, respectively, both of which were consistent with the values recorded 9 days postpartum. Emergency laparoscopic detorsion of the ischemic left ovary was planned. Aiming to reduce the risk of cystic injury and bleeding at the trocar insertion site, volume reduction of the left ovarian tumor was performed by ultrasound-guided percutaneous transabdominal aspiration of the cyst. About 200 mL of thin yellow serous fluid was removed from three cystic components through
Correspondence to: Dr Y. Sato, Department of Obstetrics and Gynecology, Otsu Red Cross Hospital, 1-1-35 Nagara, Otsu 520-8511, Shiga, Japan (e-mail: [email protected]) Accepted: 17 March 2015
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
CASE REPORT
Sakae et al.
244
T2WI (coronal)
T1 CE (axial)
Figure 1 T2-weighted coronal (a) and gadolinium-enhanced T1-weighted axial (b) pelvic magnetic resonance images in a case of hyperreactio luteinalis on day of admission, before percutaneous aspiration of left ovarian cyst. (a) Bilateral large ovarian multicystic tumors (arrows) occupy pelvic cavity. Aggregation of coiled tubular structures (arrowhead) represents twisted and congested blood vessels. Low-signal-intensity nodules (∗ ) can be seen in some of the blood vessels, suggesting intravenous clot formation. (b) Septa and surface of left ovarian tumor (dashed circle) are poorly enhanced by gadolinium as compared with right counterpart.
T2WI (coronal)
CE-T1WI (axial)
Figure 2 T2-weighted coronal (a) and gadolinium-enhanced T1-weighted axial (b) pelvic magnetic resonance images in a case of hyperreactio luteinalis on day after cyst aspiration. (a) Aggregation of coiled tubular structures (arrowhead) is shrunken as compared with that in Figure 1a and gadolinium enhancement of septa and surface of left ovarian tumor (dashed circle) is comparable to that of right counterpart.
a 23-gauge needle. Cytology of the aspirated fluid was unremarkable. The time elapsed between the onset of acute abdomen and cyst aspiration was about 10 h. Unexpectedly, the patient’s pain diminished rapidly following aspiration and, as a result, laparoscopic surgery was deferred.
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
MRI performed on the next day revealed that the aggregation of coiled tubular structures between the uterus and the left ovarian tumor had reduced in size (Figure 2a) and the gadolinium-enhanced septa and surface of the left ovarian tumor resembled those of the contralateral ovary (Figure 2b). These findings indicated
Ultrasound Obstet Gynecol 2015; 46: 243–246.
Ovarian torsion in hyperreactio luteinalis
245
CE-CT (axial)
∗
Right ovary
Left ovary
Figure 3 Color Doppler transabdominal ultrasound images in a case of hyperreactio luteinalis 7 days after percutaneous cyst aspiration (a) and contrast-enhanced computed tomogram 59 days after percutaneous cyst aspiration (b). (a) Blood circulation in left ovary is equivalent to that in right ovary (dotted lines). (b) Normal-sized bilateral ovaries (dashed circles) are seen with contrast enhancement that is equivalent to that of uterine myometrium (∗ ). Table 1 Blood-test results and size of ovaries in a woman with hyperreactio luteinalis complicated by ovarian torsion according to time interval following treatment by percutaneous cyst aspiration Days from aspiration 0 Parameter WBC (n × 100/μL) CRP (mg/dL) D-dimer (μg/mL) β-hCG (mIU/mL) Testosterone (ng/mL) Size of ovary Left (mm) Right (mm)
Before aspiration
After aspiration
1
2
3
5
7
9
11
26
59
129 0.7 15.8 1550 3.22
149 0.7 — — —
109 1.3 — 860 —
123 5.2 — — —
109 5.5 3.2 — —
85 2.8 2.6 — —
84 2.0 5.5 — —
96 1.3 4.7 — —
90 0.7 4.6 — —
53 0.1 1.0 0.9 1.41
52 0.0 0.5 0.4 0.55
113 × 92 103 × 71
100 × 61 —
94 × 70 106 × 83
100 × 75 103 × 65
83 × 45 114 × 67
79 × 44 112 × 62
98 × 47 101 × 60
61 × 43 89 × 50
— —
31 × 26 39 × 30
β-hCG, β-human chorionic gonadotropin; CRP, C-reactive protein; WBC, white blood cell count.
that the left ovarian torsion had untwisted spontaneously and blood perfusion in the left ovary had been restored. Continuous heparin infusion (20 000 units/day) was instituted to prevent possible thromboembolic complications. Color Doppler TAS 7 days after cyst aspiration showed blood flow to be similar in both ovaries (Figure 3a). The patient remained asymptomatic and was discharged from hospital 12 days after cyst aspiration. After discharge, warfarin (2 mg/day) and aspirin (100 mg/day) were administered orally for 2 months until the level of blood D-dimer returned to within the normal range. The size of the bilateral ovarian tumors decreased gradually. Computed tomography performed 59 days after cyst aspiration revealed normal-sized bilateral ovaries with contrast enhancement equivalent to that of the myometrium (Figure 3b). Temporal changes in blood-test results (WBC, CRP, D-dimer, β-hCG and testosterone levels) and size of the bilateral ovaries are shown in Table 1.
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
DISCUSSION HL is a rare benign condition characterized by bilaterally-enlarged ovaries with multiple theca lutein cysts. It is typically associated with pregnancy or trophoblastic disease and regresses spontaneously as serum levels of β-hCG decline. It is believed that increased levels of β-hCG and/or an exaggerated ovarian response to β-hCG is responsible for the development of HL. Considering the self-limiting nature of HL, conservative management has become the mainstay of treatment in recent years1 . Nevertheless, according to a recent review, surgery was performed in 31/65 cases of HL, including 5/18 asymptomatic cases2 . A precise preoperative diagnosis of HL is required to prevent unnecessary surgical intervention. In the present case, we confidently made a diagnosis of HL based on the sudden appearance of large tumors and the bilateral multicystic nature of the ovarian tumors. Ovarian torsion is a relatively common cause of gynecological emergency3 and usually develops in ovaries
Ultrasound Obstet Gynecol 2015; 46: 243–246.
Sakae et al.
246
that are enlarged by more than 4 cm. Ovarian enlargement results from the formation of either a pathological tumor, such as a dermoid cyst, or a functional cyst, such as a follicular cyst, corpus luteum cyst, ovarian hyperstimulation syndrome or HL. Whatever the cause may be, surgical intervention – preferably performed by laparoscopy4 – is mandated when ovarian torsion is suspected. Indeed, ovarian torsion has been reported in four cases of HL, all of which were treated with unilateral adnexectomy5 – 8 . At surgery, preservation of ovarian function should be considered for women of reproductive age and for prepubertal girls. Although the duration of ischemia that causes irreversible damage to the twisted ovaries is unknown, Oelsner and Shashar9 demonstrated that ovarian function could be preserved in more than 90% of cases after untwisting the ovaries. Since the theoretical possibility of thromboembolism secondary to untwisting of ischemic ovary has been reported as no more than 0.2%10 , and the challenge of cystectomy when the ovary is ischemic could cause additional damage to remaining viable follicles, Oelsner and Shashar concluded that detorsion is the only procedure option at initial laparoscopy. When pathological ovarian tumor persists, elective laparoscopic cystectomy should be performed 4–6 weeks after the initial laparoscopy9 . In the present case, we first planned emergency laparoscopy to untwist the ischemic left ovary. However, volume reduction of the twisted ovarian cyst by percutaneous cyst aspiration led to spontaneous detorsion, as evidenced by rapid amelioration of the pain symptoms and by restoration of contrast enhancement in the ischemic ovary. As a result, subsequent laparoscopic surgery became unnecessary. Restoration of ovarian mobility by volume reduction was likely to have contributed to the spontaneous detorsion. Aspiration of the contralateral untwisted
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
ovarian tumor was not offered because we considered that the possible risks of bleeding, chemical peritonitis and newly formed ovarian torsion, due to increased mobility from volume reduction, outweighed the benefit. From this experience, we propose that, in cases of ovarian torsion occurring in large functional cysts such as HL, percutaneous cyst aspiration to reduce the ovarian volume should be considered before deciding on emergency laparoscopic surgery. Cyst aspiration should be carried out in a facility in which ultrasound expertise is available, and only after malignancy or dermoid cyst has been confidently excluded using various imaging modalities and tumor markers, such as CA 125, carcinoembryonic antigen, alpha-fetoprotein and CA19-9.
REFERENCES 1. Lynn KN, Steinkeler JA, Wilkins-Haug LE, Benson CB. Hyperreactio luteinalis (enlarged ovaries) during the second and third trimesters of pregnancy: common clinical associations. J Ultrasound Med 2013; 32: 1285–1289. 2. Cavoretto P, Giorgione V, Sigismondi C, Mangili G, Serafini A, Dallagiovanna C, Candiani M. Hyperreactio luteinalis: timely diagnosis minimizes the risk of oophorectomy and alerts clinicians to the associated risk of placental insufficiency. Eur J Obstet Gynecol Reprod Biol 2014; 176: 10–16. 3. Huchon C, Fauconnier A. Adnexal torsion: a literature review. Eur J Obstet Gynecol Reprod Biol 2010; 150: 8–12. 4. Cohen SB, Wattiez A, Seidman DS, Goldenberg M, Admon D, Mashiach S, Oelsner G. Laparoscopy versus laparotomy for detorsion and sparing of twisted ischemic adnexa. JSLS 2003; 7: 295–299. 5. Wajda KJ, Lucas JG, Marsh WL Jr. Hyperreactio luteinalis. Benign disorder masquerading as an ovarian neoplasm. Arch Pathol Lab Med 1989; 113: 921–925. 6. Upadhyaya G, Goswami A, Babu S. Bilateral theca lutein cysts: a rare cause of acute abdomen in pregnancy. Emerg Med Australas 2004; 16: 476–477. 7. Grgic O, Radakovic B, Barisic D. Hyperreactio luteinalis could be a risk factor for development of HELLP syndrome: case report. Fertil Steril 2008; 90: 2008.e13–16. 8. Germeyer A, Kommoss F, Strowitzki T, von Wolff M. [Hyperreactive luteomas during pregnancy – symptoms and complications: a case report]. Gynakol Geburtshilfliche Rundsch 2009; 49: 331–334. 9. Oelsner G, Shashar D. Adnexal torsion. Clin Obstet Gynecol 2006; 49: 459–463. 10. McGovern PG, Noah R, Koenigsberg R, Little AB. Adnexal torsion and pulmonary embolism: case report and review of the literature. Obstet Gynecol Surv 1999; 54: 601–608.
Ultrasound Obstet Gynecol 2015; 46: 243–246.
Ultrasound-guided percutaneous aspiration of hyperreactio luteinalis avoids laparoscopic untwisting of ovarian torsion C. SAKAE, Y. SATO, A. TAGA, Y. SATAKE, I. EMOTO, S. MARUYAMA and T. KIM Department of Obstetrics and Gynecology, Otsu Red Cross Hospital, Nagara, Otsu, Shiga, Japan
K E Y W O R D S: acute abdomen; detorsion; functional ovarian cyst; hydrops fetalis; laparoscopy
ABSTRACT Hyperreactio luteinalis (HL) is characterized by multicystic bilateral enlargement of the ovaries and is a selflimiting benign condition associated with pregnancy or trophoblastic disease. Since HL regresses spontaneously over time, it should be managed conservatively as long as the patient’s condition permits; torsion of the enlarged ovaries is believed to be the only exception that mandates surgical intervention. Here, we describe a case of HL complicated by ovarian torsion that was treated successfully without surgical intervention. A 33-year-old woman was admitted to our hospital owing to acute abdomen. Nine days previously, she had had a stillbirth caused by hydrops fetalis at 24 weeks’ gestation. The characteristic findings observed on magnetic resonance imaging (MRI) led to the diagnosis of HL complicated by torsion of the enlarged left ovary. Emergency laparoscopic detorsion of the ischemic left ovary was planned. Aiming to reduce the risk of cystic injury and bleeding at the trocar insertion site, volume reduction of the left ovarian cyst was performed by percutaneous aspiration. The patient’s pain diminished rapidly and laparoscopic surgery was deferred. Subsequent MRI revealed that gadolinium enhancement of the left ovarian tumor had been restored, indicating spontaneous detorsion of the left ovary. The patient remained asymptomatic and was discharged from the hospital 12 days after aspiration of the cyst. From this experience, we propose that, in cases of ovarian torsion occurring in large functional cysts, including HL, volume reduction by percutaneous cyst aspiration should be considered before performing emergency laparoscopic surgery. Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
CASE REPORT A 33-year-old woman, gravida 3 para 2, presented at the emergency room of our hospital owing to acute abdomen. Transabdominal ultrasound examination (TAS) revealed
bilateral multicystic tumors occupying the pelvic cavity, and the patient was subsequently admitted to the obstetrics and gynecology department. The patient’s third pregnancy had ended, 9 days previously, in fetal demise at 24 weeks’ gestation caused by hydrops fetalis associated with cystic hygroma (karyotype was normal 46,XX). On the 2nd day postpartum, she had been discharged from the hospital with unremarkable transvaginal ultrasound findings. On admission to the obstetrics and gynecology department, the patient’s lower abdomen was slightly distended and she had severe pain and tenderness, which were dominant on the left side. Subsequent blood testing found elevated white blood cell (WBC) count (12 900/μL), C-reactive protein (CRP) (0.7 mg/dL) and D-dimer (15.8 μg/mL) levels. Magnetic resonance imaging (MRI) confirmed bilateral ovarian multicystic enlargement, reaching more than 10 cm in diameter (Figure 1a). Aggregation of coiled tubular structures, which was considered to represent twisted and congested blood vessels, was observed between the uterus and the left ovarian tumor (Figure 1a). Some of these blood vessels contained nodules of low signal intensity, suggestive of intravenous clot formation. On T1-weighted MRI, the septa and surface of the left ovarian tumor were poorly enhanced by gadolinium, as compared with the right side (Figure 1b). From these observations, the patient was diagnosed with hyperreactio luteinalis (HL) complicated by torsion of the enlarged left ovary. Serum levels of β-human chorionic gonadotropin (β-hCG) and testosterone were 1550 mIU/mL and 3.22 ng/mL, respectively, both of which were consistent with the values recorded 9 days postpartum. Emergency laparoscopic detorsion of the ischemic left ovary was planned. Aiming to reduce the risk of cystic injury and bleeding at the trocar insertion site, volume reduction of the left ovarian tumor was performed by ultrasound-guided percutaneous transabdominal aspiration of the cyst. About 200 mL of thin yellow serous fluid was removed from three cystic components through
Correspondence to: Dr Y. Sato, Department of Obstetrics and Gynecology, Otsu Red Cross Hospital, 1-1-35 Nagara, Otsu 520-8511, Shiga, Japan (e-mail: [email protected]) Accepted: 17 March 2015
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
CASE REPORT
Sakae et al.
244
T2WI (coronal)
T1 CE (axial)
Figure 1 T2-weighted coronal (a) and gadolinium-enhanced T1-weighted axial (b) pelvic magnetic resonance images in a case of hyperreactio luteinalis on day of admission, before percutaneous aspiration of left ovarian cyst. (a) Bilateral large ovarian multicystic tumors (arrows) occupy pelvic cavity. Aggregation of coiled tubular structures (arrowhead) represents twisted and congested blood vessels. Low-signal-intensity nodules (∗ ) can be seen in some of the blood vessels, suggesting intravenous clot formation. (b) Septa and surface of left ovarian tumor (dashed circle) are poorly enhanced by gadolinium as compared with right counterpart.
T2WI (coronal)
CE-T1WI (axial)
Figure 2 T2-weighted coronal (a) and gadolinium-enhanced T1-weighted axial (b) pelvic magnetic resonance images in a case of hyperreactio luteinalis on day after cyst aspiration. (a) Aggregation of coiled tubular structures (arrowhead) is shrunken as compared with that in Figure 1a and gadolinium enhancement of septa and surface of left ovarian tumor (dashed circle) is comparable to that of right counterpart.
a 23-gauge needle. Cytology of the aspirated fluid was unremarkable. The time elapsed between the onset of acute abdomen and cyst aspiration was about 10 h. Unexpectedly, the patient’s pain diminished rapidly following aspiration and, as a result, laparoscopic surgery was deferred.
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
MRI performed on the next day revealed that the aggregation of coiled tubular structures between the uterus and the left ovarian tumor had reduced in size (Figure 2a) and the gadolinium-enhanced septa and surface of the left ovarian tumor resembled those of the contralateral ovary (Figure 2b). These findings indicated
Ultrasound Obstet Gynecol 2015; 46: 243–246.
Ovarian torsion in hyperreactio luteinalis
245
CE-CT (axial)
∗
Right ovary
Left ovary
Figure 3 Color Doppler transabdominal ultrasound images in a case of hyperreactio luteinalis 7 days after percutaneous cyst aspiration (a) and contrast-enhanced computed tomogram 59 days after percutaneous cyst aspiration (b). (a) Blood circulation in left ovary is equivalent to that in right ovary (dotted lines). (b) Normal-sized bilateral ovaries (dashed circles) are seen with contrast enhancement that is equivalent to that of uterine myometrium (∗ ). Table 1 Blood-test results and size of ovaries in a woman with hyperreactio luteinalis complicated by ovarian torsion according to time interval following treatment by percutaneous cyst aspiration Days from aspiration 0 Parameter WBC (n × 100/μL) CRP (mg/dL) D-dimer (μg/mL) β-hCG (mIU/mL) Testosterone (ng/mL) Size of ovary Left (mm) Right (mm)
Before aspiration
After aspiration
1
2
3
5
7
9
11
26
59
129 0.7 15.8 1550 3.22
149 0.7 — — —
109 1.3 — 860 —
123 5.2 — — —
109 5.5 3.2 — —
85 2.8 2.6 — —
84 2.0 5.5 — —
96 1.3 4.7 — —
90 0.7 4.6 — —
53 0.1 1.0 0.9 1.41
52 0.0 0.5 0.4 0.55
113 × 92 103 × 71
100 × 61 —
94 × 70 106 × 83
100 × 75 103 × 65
83 × 45 114 × 67
79 × 44 112 × 62
98 × 47 101 × 60
61 × 43 89 × 50
— —
31 × 26 39 × 30
β-hCG, β-human chorionic gonadotropin; CRP, C-reactive protein; WBC, white blood cell count.
that the left ovarian torsion had untwisted spontaneously and blood perfusion in the left ovary had been restored. Continuous heparin infusion (20 000 units/day) was instituted to prevent possible thromboembolic complications. Color Doppler TAS 7 days after cyst aspiration showed blood flow to be similar in both ovaries (Figure 3a). The patient remained asymptomatic and was discharged from hospital 12 days after cyst aspiration. After discharge, warfarin (2 mg/day) and aspirin (100 mg/day) were administered orally for 2 months until the level of blood D-dimer returned to within the normal range. The size of the bilateral ovarian tumors decreased gradually. Computed tomography performed 59 days after cyst aspiration revealed normal-sized bilateral ovaries with contrast enhancement equivalent to that of the myometrium (Figure 3b). Temporal changes in blood-test results (WBC, CRP, D-dimer, β-hCG and testosterone levels) and size of the bilateral ovaries are shown in Table 1.
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
DISCUSSION HL is a rare benign condition characterized by bilaterally-enlarged ovaries with multiple theca lutein cysts. It is typically associated with pregnancy or trophoblastic disease and regresses spontaneously as serum levels of β-hCG decline. It is believed that increased levels of β-hCG and/or an exaggerated ovarian response to β-hCG is responsible for the development of HL. Considering the self-limiting nature of HL, conservative management has become the mainstay of treatment in recent years1 . Nevertheless, according to a recent review, surgery was performed in 31/65 cases of HL, including 5/18 asymptomatic cases2 . A precise preoperative diagnosis of HL is required to prevent unnecessary surgical intervention. In the present case, we confidently made a diagnosis of HL based on the sudden appearance of large tumors and the bilateral multicystic nature of the ovarian tumors. Ovarian torsion is a relatively common cause of gynecological emergency3 and usually develops in ovaries
Ultrasound Obstet Gynecol 2015; 46: 243–246.
Sakae et al.
246
that are enlarged by more than 4 cm. Ovarian enlargement results from the formation of either a pathological tumor, such as a dermoid cyst, or a functional cyst, such as a follicular cyst, corpus luteum cyst, ovarian hyperstimulation syndrome or HL. Whatever the cause may be, surgical intervention – preferably performed by laparoscopy4 – is mandated when ovarian torsion is suspected. Indeed, ovarian torsion has been reported in four cases of HL, all of which were treated with unilateral adnexectomy5 – 8 . At surgery, preservation of ovarian function should be considered for women of reproductive age and for prepubertal girls. Although the duration of ischemia that causes irreversible damage to the twisted ovaries is unknown, Oelsner and Shashar9 demonstrated that ovarian function could be preserved in more than 90% of cases after untwisting the ovaries. Since the theoretical possibility of thromboembolism secondary to untwisting of ischemic ovary has been reported as no more than 0.2%10 , and the challenge of cystectomy when the ovary is ischemic could cause additional damage to remaining viable follicles, Oelsner and Shashar concluded that detorsion is the only procedure option at initial laparoscopy. When pathological ovarian tumor persists, elective laparoscopic cystectomy should be performed 4–6 weeks after the initial laparoscopy9 . In the present case, we first planned emergency laparoscopy to untwist the ischemic left ovary. However, volume reduction of the twisted ovarian cyst by percutaneous cyst aspiration led to spontaneous detorsion, as evidenced by rapid amelioration of the pain symptoms and by restoration of contrast enhancement in the ischemic ovary. As a result, subsequent laparoscopic surgery became unnecessary. Restoration of ovarian mobility by volume reduction was likely to have contributed to the spontaneous detorsion. Aspiration of the contralateral untwisted
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
ovarian tumor was not offered because we considered that the possible risks of bleeding, chemical peritonitis and newly formed ovarian torsion, due to increased mobility from volume reduction, outweighed the benefit. From this experience, we propose that, in cases of ovarian torsion occurring in large functional cysts such as HL, percutaneous cyst aspiration to reduce the ovarian volume should be considered before deciding on emergency laparoscopic surgery. Cyst aspiration should be carried out in a facility in which ultrasound expertise is available, and only after malignancy or dermoid cyst has been confidently excluded using various imaging modalities and tumor markers, such as CA 125, carcinoembryonic antigen, alpha-fetoprotein and CA19-9.
REFERENCES 1. Lynn KN, Steinkeler JA, Wilkins-Haug LE, Benson CB. Hyperreactio luteinalis (enlarged ovaries) during the second and third trimesters of pregnancy: common clinical associations. J Ultrasound Med 2013; 32: 1285–1289. 2. Cavoretto P, Giorgione V, Sigismondi C, Mangili G, Serafini A, Dallagiovanna C, Candiani M. Hyperreactio luteinalis: timely diagnosis minimizes the risk of oophorectomy and alerts clinicians to the associated risk of placental insufficiency. Eur J Obstet Gynecol Reprod Biol 2014; 176: 10–16. 3. Huchon C, Fauconnier A. Adnexal torsion: a literature review. Eur J Obstet Gynecol Reprod Biol 2010; 150: 8–12. 4. Cohen SB, Wattiez A, Seidman DS, Goldenberg M, Admon D, Mashiach S, Oelsner G. Laparoscopy versus laparotomy for detorsion and sparing of twisted ischemic adnexa. JSLS 2003; 7: 295–299. 5. Wajda KJ, Lucas JG, Marsh WL Jr. Hyperreactio luteinalis. Benign disorder masquerading as an ovarian neoplasm. Arch Pathol Lab Med 1989; 113: 921–925. 6. Upadhyaya G, Goswami A, Babu S. Bilateral theca lutein cysts: a rare cause of acute abdomen in pregnancy. Emerg Med Australas 2004; 16: 476–477. 7. Grgic O, Radakovic B, Barisic D. Hyperreactio luteinalis could be a risk factor for development of HELLP syndrome: case report. Fertil Steril 2008; 90: 2008.e13–16. 8. Germeyer A, Kommoss F, Strowitzki T, von Wolff M. [Hyperreactive luteomas during pregnancy – symptoms and complications: a case report]. Gynakol Geburtshilfliche Rundsch 2009; 49: 331–334. 9. Oelsner G, Shashar D. Adnexal torsion. Clin Obstet Gynecol 2006; 49: 459–463. 10. McGovern PG, Noah R, Koenigsberg R, Little AB. Adnexal torsion and pulmonary embolism: case report and review of the literature. Obstet Gynecol Surv 1999; 54: 601–608.
Ultrasound Obstet Gynecol 2015; 46: 243–246.
