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Pregnancy complicated by spontaneous suprarenal and ovarian venous hemorrhage
PREGNANCY COMPLICATED BY SPONTANEOUS SUPRARENAL AND OVARIAN VENOUS HEMORRHAGE A Case Report A. W. DIDDLE, M.D., K. A. O’CONNOR, S. J. PLATT, M.D., KNOXVILLE, TENN. (From
East Tennessee
Baptist
M.D., AND
Hospital)
T
HIS discussion concerns an instance of pregnancy complicated by unusual, spontaneous, catastrophic hemorrhage from the suprarenal and ovarian veins. The combination presented a problem in diagnosis and treatment. An 1%year-old primigravida was hospitalized in the thirty-eighth week of pregnancy at 9:30 A.M., Sept. 30, 1956. The prenatal course had been uneventful until September29. Then the patient experienced transitory, sharp pain in the right costovertebral angle. Since it disappeared quickly, she disregarded it. Approximately 24 hours later the excruciating pain reappeared suddenly as she sat down in a chair. Nearly two hours later, when hospitalized, the blood pressure was 110/90, the pulse 60 per minute, and the temperature 98.6” F. The skin was dry with goocl turgor. The mucous membranes were moderately pale. The head, neck, chest, heart, and lungs appeared essentially normal. The uterine fundus was soft, nontender, and within 4 cm. of the xiphoid process. The baby presented by the vertex. One observer thought the fetal heartbeat was faint for a short time. Light pressure over the right kitlney posteriorly reproducecl much discomfort. The cervix was long, uneffaced, and closed. Rectal examination added no information. There was no etlema of the extremities. The tentative diagnosis was renal colic with obstruction due to a stone, versus torsion or rupture of a viacus as a seconll choice. Hospitnl
Course.-
Ante parturn : The patient did feel faint when placed in the erect position. This sign raised the question of internal bleeding or a neurogenic effect from pain. In anticipation of operative intervention a blood transfusion was started on the basis of a low hematocrit. Symptoms and signs diminished during the next 3 hours. The blood pressure remained fairly stable and the pulse fluctuated between 70 and 80 per minute at different times. Undoubtedly the transfusion camouflaged the process of internal hemorrhage found later. In the meantime 2 plus albuminuria and an abundance of white blood cells were found in a catheterized urine specimen. This observation supported the added diagnosis of acute pyelonephritis. On the other hand, it confused the interpretation of the situation at the time. A hematocrit taken nearly 3 hours after the blood transfusion was started was only slightly higher than that obtained before the transfusion. The insignificant change indicated there was blood loss occurring The uterus was still soft and nontender, while the pain had Shortly thereafter retrograde pyelograms were made because of the original receded. diagnosis and because severe, colicky pain recurred. The urinary tract proved to be normal. It was now 2:30 P.M. Five physicians experienced especially either in gynecic problems, urology, general surgery, or anesthesiology gathered to evaluate the situation. There was a difference of opinion as to the advisability of doing an exploratory procedure at this 207
DIDDLE,
208
O’COIVNOR,
4KD
PLATT
Am.
J. Oler. 6 C;y~ir~~ .l.
19iF
time because localizing signs were nil and because the best route of exploration was in question. It was debatable whether the pathologic process was extraperitoneal and Tellal or intraperitoneal. The abdominal route was selected later when the right costovertebral pain recurred, epigastric discomfort appeared for the first time, the abdomen was equivocally “fuller” and the fetal heart rate dropped to near 100. Sow the diagnosis entertained was concealed abruptio placentae associated with some other undetermined tylje of intraabdominal pathology. Intra partum : A low cervical cesarean section was done under epidura1 anesthesia. A very markedly enlarged right uteroovarian venous plexus was noted on first entering the abdomen. Free blood was present in the abdominal cavity. Initially this was suspected to have regurgitated from the oviducts. The uterus was bluer in color than normal. il concealed partial separation of the placenta occupied the right side of the uterine cavity. The amniotic fluid was clear, indicating that little, if any, blood had penetrated into the amniotic sac. Not until the baby and placenta were removed and the uterus was closed was it possible to see a large retroperitoneal hematoma in the right abdominal gutter. It extended from the right infundibulopelvic ligament to the diaphragm and medially beneath the first portion of the duodenum. As the hematoma was evacuated bloody ooze came from the ovarian plexus. This was excised. Still ooze continued to come from the region of the renal vessels. This bleeding ceased when a vein above the main right renal vessels was tied. Acute hypotension developed after the baby was extracted but the blood Up to this time 2,500 c.c. of blood pressure returned to normal by the end of the operation. had
been
given.
Postoperatively : Approximately one hour postoperatively the patient precipitiouslp went into shock. The operative team theorized that bleeding was coming from elsewhere above the right kidney. An operative approach was made through an incision over the twelfth rib, which was removed. An estimated 500 to 800 C.C. of clotted blood was found extraperitoneally above the kidney. Bleeding came from multiple points surrounding the right adrenal gland and from the main venous channel leading from the suprarenal to the vena cava. It was necessary to remove most of the gland as well as to ligate the principal venous channel to the vena cava. Gelfoam and a pack were left in the pocket previously Thirty-five hundred cubic centimeters of blood was given during this occupied by clot. phase of treatment, bringing the total blood given to 6,000 C.C. Two hundred milligrams of cortisone daily were given three and six days, respectively. The temperature of 100 to 101” F., mild hypertension, 5 postoperative days. Thereafter each was within patient is well. from
The the
final diagnoses suprarenal and
included: concealed ovarian venous plexuses,
and 1 Gm. Combiotic every eight hours pack was removed by the third day. -1 and pulse over 100 persisted for the first normal range. Eight months later the abruptio placentae, and pyelonephritis.
multiple
hemorrhages
Baby: The baby weighed nearly 3,600 grams. It was born alive but died shortly after birth. Clinically the infant drowned from a waterlogged pulmonary system. Aspiration of the trachea by direct vision proved to be a futile procedure. Unfortunately, a postmortem examination was not granted but it is believed that the baby aspirated much amniotic fluid because of chronic intrauterine anoxia. Laboratory with a red cell The hematocrit after operation were tive
and roentgenographic stzldies: The initial hematocrit was 30 vol. per cent count of 3.09 million, a white cell count of 14,250, and a normal differential. taken 3 hours after the transfusion was started was 31 vol. per cent; the day it was 39 per cent.
Albuminuris and pus were found no microscopic elements or other day. The specific gravity ranged
before operation as previously mentioned. There abnormalities in the urine after the fifth postoperafrom 1.015 to 1.031 at various times.
F”‘;;z~:’ u
SUPRARENAL
Roentgenographic in pregnancy. Tissues removed
AND studies were
OVARIAN showed
histologically
VEIN
RUPTURE
IN
the normal
physiologic
dilatation
normal
artery,
veins,
and
209
PREGNANCY of the ureters adrenal
seen
gland.
Comment Keele and Keele5 are quoted as being the first to differentiate between adrenal hemorrhage of the Waterhouse-Friderichsen syndrome and hemorrhage of a noninfectious variety. Other91 6 collected 36 instances of suprarenal hemorrhage of a noninfectious kind complicating pregnancy or the puerperium. Most of the hemorrhages occurred on the right side, in primigravidas, and nearly all died before a correct diagnosis was suspected. None survived. Usually the complication developed during the course of a pathologic gestation. The combination suggests more than a coincidental event. Menaker and Cauble* collected 73 cases of rupture of the ovarian vessels in pregnant or puerperal patients. One half of them died. Some women required more than 10,000 C.C. of blood given as an emergency measure for survival. Hodgkinson’P physiologic studies show that the quantity of blood carried by the ovarian veins increases more than 60 times by the thirty-sixth week of pregnancy as compared to the nonpregnant state. Concomitantly the venous pressure in these channels nearly trebles. Under stress of labor, manipulation, or disease one or more of the vessels may rupture. According to anatomic studies made by Notkovich,4 the adrenal-renal venous blood supply may intercommunicate with one another as well as with the ovarian veins. We are of the opinion this was the situation in our patient. The abruptio placentae in our case was probably secondary to the venous pressure built up distal to the vein ruptures. We presume that the mechanism is similar to the situation produced by compressing the vena cava directly and causing retroplacental hemorrhage.3 Summary
and Conclusions
A case history is given of a gravid patient who had multiple hemorrhages from the right ovarian and suprarenal venous plexuses. A collective review from the literature shows that ruptures of the suprarenal or ovarian veins or both during pregnancy or the puerperium are accompanied by acute pain or shock or both, frequently on the right side, often among primigravidas, and frequently associated with a pathologic gestation. The complication is so catastrophic that surgical treatment is mandatory. Untreated, death will usually occur from internal hemorrhage. References 1. Hodgkinson, C. P.: Obst. & Gynec. 1: 26, 1953. 2. Menaker, J. S., and Cauble, W. G.: Obst. & Gynec. 2: 92, 1953. 3. Mengert, W. F., Goodson, J. H., Campbell, R. G., and Haynes, D. M.: AM. GYNEC. 66: 1104, 1953. 4. Notkovich, H.: Surg., Gynec. & Obst. 103: 487, 1956. 5. Peterson, W. F., and Goldzieher, J.: AM. J. OBST. & GYNEC. 66: 648, 1953. 6. Vielens, G.: Acta obst. et gynec. scandinav. 32: 440, 1953.
J. 0~s~.
&
East Tennessee
Baptist
M.D., AND
Hospital)
T
HIS discussion concerns an instance of pregnancy complicated by unusual, spontaneous, catastrophic hemorrhage from the suprarenal and ovarian veins. The combination presented a problem in diagnosis and treatment. An 1%year-old primigravida was hospitalized in the thirty-eighth week of pregnancy at 9:30 A.M., Sept. 30, 1956. The prenatal course had been uneventful until September29. Then the patient experienced transitory, sharp pain in the right costovertebral angle. Since it disappeared quickly, she disregarded it. Approximately 24 hours later the excruciating pain reappeared suddenly as she sat down in a chair. Nearly two hours later, when hospitalized, the blood pressure was 110/90, the pulse 60 per minute, and the temperature 98.6” F. The skin was dry with goocl turgor. The mucous membranes were moderately pale. The head, neck, chest, heart, and lungs appeared essentially normal. The uterine fundus was soft, nontender, and within 4 cm. of the xiphoid process. The baby presented by the vertex. One observer thought the fetal heartbeat was faint for a short time. Light pressure over the right kitlney posteriorly reproducecl much discomfort. The cervix was long, uneffaced, and closed. Rectal examination added no information. There was no etlema of the extremities. The tentative diagnosis was renal colic with obstruction due to a stone, versus torsion or rupture of a viacus as a seconll choice. Hospitnl
Course.-
Ante parturn : The patient did feel faint when placed in the erect position. This sign raised the question of internal bleeding or a neurogenic effect from pain. In anticipation of operative intervention a blood transfusion was started on the basis of a low hematocrit. Symptoms and signs diminished during the next 3 hours. The blood pressure remained fairly stable and the pulse fluctuated between 70 and 80 per minute at different times. Undoubtedly the transfusion camouflaged the process of internal hemorrhage found later. In the meantime 2 plus albuminuria and an abundance of white blood cells were found in a catheterized urine specimen. This observation supported the added diagnosis of acute pyelonephritis. On the other hand, it confused the interpretation of the situation at the time. A hematocrit taken nearly 3 hours after the blood transfusion was started was only slightly higher than that obtained before the transfusion. The insignificant change indicated there was blood loss occurring The uterus was still soft and nontender, while the pain had Shortly thereafter retrograde pyelograms were made because of the original receded. diagnosis and because severe, colicky pain recurred. The urinary tract proved to be normal. It was now 2:30 P.M. Five physicians experienced especially either in gynecic problems, urology, general surgery, or anesthesiology gathered to evaluate the situation. There was a difference of opinion as to the advisability of doing an exploratory procedure at this 207
DIDDLE,
208
O’COIVNOR,
4KD
PLATT
Am.
J. Oler. 6 C;y~ir~~ .l.
19iF
time because localizing signs were nil and because the best route of exploration was in question. It was debatable whether the pathologic process was extraperitoneal and Tellal or intraperitoneal. The abdominal route was selected later when the right costovertebral pain recurred, epigastric discomfort appeared for the first time, the abdomen was equivocally “fuller” and the fetal heart rate dropped to near 100. Sow the diagnosis entertained was concealed abruptio placentae associated with some other undetermined tylje of intraabdominal pathology. Intra partum : A low cervical cesarean section was done under epidura1 anesthesia. A very markedly enlarged right uteroovarian venous plexus was noted on first entering the abdomen. Free blood was present in the abdominal cavity. Initially this was suspected to have regurgitated from the oviducts. The uterus was bluer in color than normal. il concealed partial separation of the placenta occupied the right side of the uterine cavity. The amniotic fluid was clear, indicating that little, if any, blood had penetrated into the amniotic sac. Not until the baby and placenta were removed and the uterus was closed was it possible to see a large retroperitoneal hematoma in the right abdominal gutter. It extended from the right infundibulopelvic ligament to the diaphragm and medially beneath the first portion of the duodenum. As the hematoma was evacuated bloody ooze came from the ovarian plexus. This was excised. Still ooze continued to come from the region of the renal vessels. This bleeding ceased when a vein above the main right renal vessels was tied. Acute hypotension developed after the baby was extracted but the blood Up to this time 2,500 c.c. of blood pressure returned to normal by the end of the operation. had
been
given.
Postoperatively : Approximately one hour postoperatively the patient precipitiouslp went into shock. The operative team theorized that bleeding was coming from elsewhere above the right kidney. An operative approach was made through an incision over the twelfth rib, which was removed. An estimated 500 to 800 C.C. of clotted blood was found extraperitoneally above the kidney. Bleeding came from multiple points surrounding the right adrenal gland and from the main venous channel leading from the suprarenal to the vena cava. It was necessary to remove most of the gland as well as to ligate the principal venous channel to the vena cava. Gelfoam and a pack were left in the pocket previously Thirty-five hundred cubic centimeters of blood was given during this occupied by clot. phase of treatment, bringing the total blood given to 6,000 C.C. Two hundred milligrams of cortisone daily were given three and six days, respectively. The temperature of 100 to 101” F., mild hypertension, 5 postoperative days. Thereafter each was within patient is well. from
The the
final diagnoses suprarenal and
included: concealed ovarian venous plexuses,
and 1 Gm. Combiotic every eight hours pack was removed by the third day. -1 and pulse over 100 persisted for the first normal range. Eight months later the abruptio placentae, and pyelonephritis.
multiple
hemorrhages
Baby: The baby weighed nearly 3,600 grams. It was born alive but died shortly after birth. Clinically the infant drowned from a waterlogged pulmonary system. Aspiration of the trachea by direct vision proved to be a futile procedure. Unfortunately, a postmortem examination was not granted but it is believed that the baby aspirated much amniotic fluid because of chronic intrauterine anoxia. Laboratory with a red cell The hematocrit after operation were tive
and roentgenographic stzldies: The initial hematocrit was 30 vol. per cent count of 3.09 million, a white cell count of 14,250, and a normal differential. taken 3 hours after the transfusion was started was 31 vol. per cent; the day it was 39 per cent.
Albuminuris and pus were found no microscopic elements or other day. The specific gravity ranged
before operation as previously mentioned. There abnormalities in the urine after the fifth postoperafrom 1.015 to 1.031 at various times.
F”‘;;z~:’ u
SUPRARENAL
Roentgenographic in pregnancy. Tissues removed
AND studies were
OVARIAN showed
histologically
VEIN
RUPTURE
IN
the normal
physiologic
dilatation
normal
artery,
veins,
and
209
PREGNANCY of the ureters adrenal
seen
gland.
Comment Keele and Keele5 are quoted as being the first to differentiate between adrenal hemorrhage of the Waterhouse-Friderichsen syndrome and hemorrhage of a noninfectious variety. Other91 6 collected 36 instances of suprarenal hemorrhage of a noninfectious kind complicating pregnancy or the puerperium. Most of the hemorrhages occurred on the right side, in primigravidas, and nearly all died before a correct diagnosis was suspected. None survived. Usually the complication developed during the course of a pathologic gestation. The combination suggests more than a coincidental event. Menaker and Cauble* collected 73 cases of rupture of the ovarian vessels in pregnant or puerperal patients. One half of them died. Some women required more than 10,000 C.C. of blood given as an emergency measure for survival. Hodgkinson’P physiologic studies show that the quantity of blood carried by the ovarian veins increases more than 60 times by the thirty-sixth week of pregnancy as compared to the nonpregnant state. Concomitantly the venous pressure in these channels nearly trebles. Under stress of labor, manipulation, or disease one or more of the vessels may rupture. According to anatomic studies made by Notkovich,4 the adrenal-renal venous blood supply may intercommunicate with one another as well as with the ovarian veins. We are of the opinion this was the situation in our patient. The abruptio placentae in our case was probably secondary to the venous pressure built up distal to the vein ruptures. We presume that the mechanism is similar to the situation produced by compressing the vena cava directly and causing retroplacental hemorrhage.3 Summary
and Conclusions
A case history is given of a gravid patient who had multiple hemorrhages from the right ovarian and suprarenal venous plexuses. A collective review from the literature shows that ruptures of the suprarenal or ovarian veins or both during pregnancy or the puerperium are accompanied by acute pain or shock or both, frequently on the right side, often among primigravidas, and frequently associated with a pathologic gestation. The complication is so catastrophic that surgical treatment is mandatory. Untreated, death will usually occur from internal hemorrhage. References 1. Hodgkinson, C. P.: Obst. & Gynec. 1: 26, 1953. 2. Menaker, J. S., and Cauble, W. G.: Obst. & Gynec. 2: 92, 1953. 3. Mengert, W. F., Goodson, J. H., Campbell, R. G., and Haynes, D. M.: AM. GYNEC. 66: 1104, 1953. 4. Notkovich, H.: Surg., Gynec. & Obst. 103: 487, 1956. 5. Peterson, W. F., and Goldzieher, J.: AM. J. OBST. & GYNEC. 66: 648, 1953. 6. Vielens, G.: Acta obst. et gynec. scandinav. 32: 440, 1953.
J. 0~s~.
&