CASE R E P O R T
Ruptured ovarian artery aneurysm: A case report William L. M. King, M D , F R C S ( C ) , F R C S , Toronto, Ontario, Canada Ruptured aneurysms o f the ovarian artery are exceedingly rare: Five cases have been previously repo~ted in the English-language literature, all occurred in the early postpartum period. The present report details another such case, the first, to my knowledge, to have been demonstrated by angiography or to have been treated nonoperatively. (J VASe Sting 1990;12:190-3.)
A ruptured ovarian artery aneurysm is an exceedingly rare lesion, seemingly occurring only in the early postpartum period. Only five cases have been reported in the English-language literature, ls with one o f the articles 1 quoting two other reports from the first half o f this century, one in the German and one in the Italian-language literature. The following case report adds a sixth case. CASE REPORT The patient, a 36-year-old woman, came to our emergency department on April 14, 1989, 2 to 3 days before the expected date of confinement of her sixth pregnancy, complaining of pain in her right flank. In addition to tenderness in the region of her right kidney, she had a temperature of 39° C, and white cells and bacteria were seen on a microscopic examination of her urine (subsequent urine cultures were negative). She gave a history of having had a kidney infection in June 1988 but could not remember the side. She was admitted to the hospital with a diagnosis of pyelonephritis in pregnancy and treated with broad-spectrum antibiotics. On the following day (April 15) she delivered a healthy infant without incident, and the postpartum period was uneventful until April 19 when more severe flank pain recurred. Abdominal ultrasonography performed the same day showed a collection of fluid around the lower pole of the right kidney, which in light of the previous diagnosis was interpreted as a perinephric abscess. However, when needle aspiration of the fluid was performed under CT
From the Department of Surgery, North York GeneralHospital, Toronto, Ontario, Canada. Reprint requests: W. L. M. King, MD, North York MedicalArts Building, 1333 Sheppard Ave. East, Suite 207, Willowdale, Ontario M2J 1V1, Canada. 24/37/20845 190
control (Fig. 1), the fluid was discovered to be old blood. At about the same time it was reported that the patient's hemoglobin level, which had been 110 gm/dl on admission, had fallen to 78 gm/dl. Angiography was performed on April 24. Transfemoral catheter aortography (Fig. 2) showed a clear abnormality of the right ovarian artery and also what was believed to be a less dramatic enlargement of the left ovarian artery. A selective right ovarian angiograrn (Fig. 3) showed an aneurysm in the upper third of the artery, the presumed source of bleeding, and coils were introduced via the angiocatheter to occlude the ovarian artery above the level of the lesion. The patient made an uneventful postprocedural recovery and has had no recurrence of abdominal or flank pain. A recheck angiogram (Fig. 4) was obtained on June 22. The right ovarian artery was shown to be OCcluded. The left ovarian artery was not demonstrated on an aortic injection, and its abnormalities were presumed to have regressed. The patient has agreed to undergo ele~"~, rive tubal ligation. DISCUSSION In their review o f pregnancy-related ruptured arterial aneurysms, Barrett et al. 6 list, in decreasing order o f frequency, intracranial arteries, aorta (usually dissecting) with or without involvement o f the coronary arteries, splenic artery, renal artery, and ovarian artery as sites where aneurysmal rupture has occurred with more than single-case frequency. O f these the ovarian artery location is by far the least frequent, with only five cases documented in the Englishlanguage literature. The pathogenesis o f these lesions is poorly understood. T w o classes o f factors may be important, hemodynamic and hormonal. The systemic hemodynamic changes that occur in pregnancy include increased cardiac output and blood volume. Systemic
Volume 12 Number 2 Au~x~st1990
Ruptured ovarian aneurysm
Fig. 1. CT scan at the level o f the lower pole of the right kidney. The arrows indicate the perinephric fluid collection.
Fig. 2. Transfemoral catheter aortogram. The arrows indicate the right ovarian aneurysm and the dilated left ovarian artery.
191
192
Journal of VASCULAR SURGERY
K/ng
Fig. 3. Selective right ovarian angiogram. Note the aneurysm in the upper third and the markedly-dilated distal ovarian artery.
Fig. 4. Follow-up transfemoral catheter aortogram obtained 2 months later. The coils are seen occluding the right ovarian artery. The left ovarian artery is no longer demonstrated.
Volume 12 Number 2 August 1990
hypertension is common, and significant swings in "local blood pressure may result from compression of the abdominal aorta by the gravid uterus when the patient lies in the supine position. With enlargement of the uterus, dilation of the pelvic arteries occurs with correspondingly increased uterine blood flow. Burnett and Carfrae 5 have postulated that during the iaormal process of involution, which occurs during the postpartum period, a segment or segments of the ovarian circulation may fail to involute, predisposing to aneurysm formation in a subsequent pregnancy. It is likely more than coincidental that all reported cases of ovarian artery rupture have occurred ha mul~tiparous women. A combination of animal and human studies suggest that the pregnancy-related changes in steroid :rmones may bring about a variety of arterial changes including intimal hyperplasia, thickening of the media associated with smooth muscle hyperpla"sia, fragmentation ofreticnlum fibers, decrease in acid mucopolysaccharides, and loss of normal corrugation of elastic fibers. 6 The only report to include detailed histologic studies of the arterial specimen is that of B~trnett and Carfrae. ~ Both specimens they reviewed showed similar changes of deendothelialization, greatly thinned and relatively acellular media with only a few degenerate smooth muscle cells remaining and fragmentation of the internal elastic lamina.
Ruptured ovarian aneu~ysm 193
All reported cases had flank pain associated with some degree of hemodynamic instability. All had symptoms early in the puerperium, although in some cases definitive surgical diagnosis was delayed as long as 2 months. With the advantages of modern diagnostic techniques, such cases should be ideal for radiologic diagnosis and nonoperative management unless there is continued hemodynamaic instability, in which case early surgical intervention is necessary. Although oophorectomy has been advocated when the ovarian arterial supply is interrupted, 6 the rich collateral supply from the uterine circulation would appear to make this measure unnecessary. REFERENCES 1. Caillouette JC, Owen HW. Postpamun spontaneous rupture of an ovarian-artery aneurysm. Obstet Gynecol 1963;21: 510-1. 2. Tsoutsoplides GC. Post-parmm spontaneous rupture of a branch of the ovarian artery. Scot Med J I967;12:289-90. 3. Riley PM. Rupture of right ovarian artery aneurysm during defivery. S Afr Med J 1975;49:729. 4. Jafari K, Saleh I. Postpartum spontaneous rupture of ovarian artery aneurysm. Obstet Gynecol 1977;49:493-5. 5. Burnett RA, Carfrae DC. Spontaneous rupture of ovarian artery aneurysm in the puerperium: two case reports and a review of the literature. Br J Obstet Gynaecol 1976;83:744-50. 6. Barrett JM, Van Hooydonk IE, Boehm FH. Pregnancyrelated rupture of arterial aneurysms. Obstet Gynecol Surv 1982;37:557-60.
Ruptured ovarian artery aneurysm: A case report William L. M. King, M D , F R C S ( C ) , F R C S , Toronto, Ontario, Canada Ruptured aneurysms o f the ovarian artery are exceedingly rare: Five cases have been previously repo~ted in the English-language literature, all occurred in the early postpartum period. The present report details another such case, the first, to my knowledge, to have been demonstrated by angiography or to have been treated nonoperatively. (J VASe Sting 1990;12:190-3.)
A ruptured ovarian artery aneurysm is an exceedingly rare lesion, seemingly occurring only in the early postpartum period. Only five cases have been reported in the English-language literature, ls with one o f the articles 1 quoting two other reports from the first half o f this century, one in the German and one in the Italian-language literature. The following case report adds a sixth case. CASE REPORT The patient, a 36-year-old woman, came to our emergency department on April 14, 1989, 2 to 3 days before the expected date of confinement of her sixth pregnancy, complaining of pain in her right flank. In addition to tenderness in the region of her right kidney, she had a temperature of 39° C, and white cells and bacteria were seen on a microscopic examination of her urine (subsequent urine cultures were negative). She gave a history of having had a kidney infection in June 1988 but could not remember the side. She was admitted to the hospital with a diagnosis of pyelonephritis in pregnancy and treated with broad-spectrum antibiotics. On the following day (April 15) she delivered a healthy infant without incident, and the postpartum period was uneventful until April 19 when more severe flank pain recurred. Abdominal ultrasonography performed the same day showed a collection of fluid around the lower pole of the right kidney, which in light of the previous diagnosis was interpreted as a perinephric abscess. However, when needle aspiration of the fluid was performed under CT
From the Department of Surgery, North York GeneralHospital, Toronto, Ontario, Canada. Reprint requests: W. L. M. King, MD, North York MedicalArts Building, 1333 Sheppard Ave. East, Suite 207, Willowdale, Ontario M2J 1V1, Canada. 24/37/20845 190
control (Fig. 1), the fluid was discovered to be old blood. At about the same time it was reported that the patient's hemoglobin level, which had been 110 gm/dl on admission, had fallen to 78 gm/dl. Angiography was performed on April 24. Transfemoral catheter aortography (Fig. 2) showed a clear abnormality of the right ovarian artery and also what was believed to be a less dramatic enlargement of the left ovarian artery. A selective right ovarian angiograrn (Fig. 3) showed an aneurysm in the upper third of the artery, the presumed source of bleeding, and coils were introduced via the angiocatheter to occlude the ovarian artery above the level of the lesion. The patient made an uneventful postprocedural recovery and has had no recurrence of abdominal or flank pain. A recheck angiogram (Fig. 4) was obtained on June 22. The right ovarian artery was shown to be OCcluded. The left ovarian artery was not demonstrated on an aortic injection, and its abnormalities were presumed to have regressed. The patient has agreed to undergo ele~"~, rive tubal ligation. DISCUSSION In their review o f pregnancy-related ruptured arterial aneurysms, Barrett et al. 6 list, in decreasing order o f frequency, intracranial arteries, aorta (usually dissecting) with or without involvement o f the coronary arteries, splenic artery, renal artery, and ovarian artery as sites where aneurysmal rupture has occurred with more than single-case frequency. O f these the ovarian artery location is by far the least frequent, with only five cases documented in the Englishlanguage literature. The pathogenesis o f these lesions is poorly understood. T w o classes o f factors may be important, hemodynamic and hormonal. The systemic hemodynamic changes that occur in pregnancy include increased cardiac output and blood volume. Systemic
Volume 12 Number 2 Au~x~st1990
Ruptured ovarian aneurysm
Fig. 1. CT scan at the level o f the lower pole of the right kidney. The arrows indicate the perinephric fluid collection.
Fig. 2. Transfemoral catheter aortogram. The arrows indicate the right ovarian aneurysm and the dilated left ovarian artery.
191
192
Journal of VASCULAR SURGERY
K/ng
Fig. 3. Selective right ovarian angiogram. Note the aneurysm in the upper third and the markedly-dilated distal ovarian artery.
Fig. 4. Follow-up transfemoral catheter aortogram obtained 2 months later. The coils are seen occluding the right ovarian artery. The left ovarian artery is no longer demonstrated.
Volume 12 Number 2 August 1990
hypertension is common, and significant swings in "local blood pressure may result from compression of the abdominal aorta by the gravid uterus when the patient lies in the supine position. With enlargement of the uterus, dilation of the pelvic arteries occurs with correspondingly increased uterine blood flow. Burnett and Carfrae 5 have postulated that during the iaormal process of involution, which occurs during the postpartum period, a segment or segments of the ovarian circulation may fail to involute, predisposing to aneurysm formation in a subsequent pregnancy. It is likely more than coincidental that all reported cases of ovarian artery rupture have occurred ha mul~tiparous women. A combination of animal and human studies suggest that the pregnancy-related changes in steroid :rmones may bring about a variety of arterial changes including intimal hyperplasia, thickening of the media associated with smooth muscle hyperpla"sia, fragmentation ofreticnlum fibers, decrease in acid mucopolysaccharides, and loss of normal corrugation of elastic fibers. 6 The only report to include detailed histologic studies of the arterial specimen is that of B~trnett and Carfrae. ~ Both specimens they reviewed showed similar changes of deendothelialization, greatly thinned and relatively acellular media with only a few degenerate smooth muscle cells remaining and fragmentation of the internal elastic lamina.
Ruptured ovarian aneu~ysm 193
All reported cases had flank pain associated with some degree of hemodynamic instability. All had symptoms early in the puerperium, although in some cases definitive surgical diagnosis was delayed as long as 2 months. With the advantages of modern diagnostic techniques, such cases should be ideal for radiologic diagnosis and nonoperative management unless there is continued hemodynamaic instability, in which case early surgical intervention is necessary. Although oophorectomy has been advocated when the ovarian arterial supply is interrupted, 6 the rich collateral supply from the uterine circulation would appear to make this measure unnecessary. REFERENCES 1. Caillouette JC, Owen HW. Postpamun spontaneous rupture of an ovarian-artery aneurysm. Obstet Gynecol 1963;21: 510-1. 2. Tsoutsoplides GC. Post-parmm spontaneous rupture of a branch of the ovarian artery. Scot Med J I967;12:289-90. 3. Riley PM. Rupture of right ovarian artery aneurysm during defivery. S Afr Med J 1975;49:729. 4. Jafari K, Saleh I. Postpartum spontaneous rupture of ovarian artery aneurysm. Obstet Gynecol 1977;49:493-5. 5. Burnett RA, Carfrae DC. Spontaneous rupture of ovarian artery aneurysm in the puerperium: two case reports and a review of the literature. Br J Obstet Gynaecol 1976;83:744-50. 6. Barrett JM, Van Hooydonk IE, Boehm FH. Pregnancyrelated rupture of arterial aneurysms. Obstet Gynecol Surv 1982;37:557-60.
