Copyright C Acta Obstet Gynecol Scand 1999

Acta Obstet Gynecol Scand 1999; 78: 737–741 Printed in Denmark ¡ All rights reserved

Acta Obstetricia et Gynecologica Scandinavica ISSN 0001-6349

CASE REPORTS

Recurrent incarceration of the retroverted gravid uterus – a case report BO JACOBSSON1 AND DAG WIDE-SWENSSON2 From the Departments of Obstetrics and Gynecology, 1 Sahlgrenska University Hospital, Go¨teborg and the 2 University Hospital of Lund, Lund, Sweden Acta Obstet Gynecol Scand 1999; 78: 737. C Acta Obstet Gynecol Scand 1999 Key words: case report; human; incarcerated; obstetrics; pregnancy; retroverted; uterus Submitted 4 January, 1999 Accepted 10 April, 1999

If a pregnancy begins in a retroverted uterus it spontaneously reverts to an upward position before the 14th week. Only in a few cases does the uterus persist in the retroverted position and it will be wedged in the pelvic cavity as it grows. We present the third case report in the literature where incarceration of the retroverted gravid uterus occurred twice in the same woman (1, 2).

Case report The patient, a 32-year-old gravida 3, para 1, was sent to the antenatal ward of the University Hospital of Lund from a district maternal unit in gestational week 29 because she was thought to have an incarcerated gravid retroverted uterus. Seven years earlier, in the first trimester of her first pregnancy she experienced periods of abdominal pain. Her obstetrician found a soft resistance in the lateral part of the pelvis and an extra-uterine pregnancy was suspected. Ultrasound showed an intrauterine pregnancy. Throughout the pregnancy she had tenderness in the abdomen. At 28 weeks of gestation, she consulted her obstetrician due to uterine contractions. Terbutaline injections were given with prompt effect. A vaginal examination was performed, but the cervix was not reached. She was put on tocolytic medication (terbutaline tablets, 5 mg, three times a day). Five weeks later, in the 33th week of the pregnancy the obstetrician suspected sacculation or an abdominal pregnancy. The following night she developed severe abdominal pain and a cesarean section was performed. During the operation an incarcerated retroverted uterus was found. There was no other apparent gross pathology . The incision was at first made in the cervix. The mistake was discovered and a new incision was made higher up in the lower uterine segment. The child, a healthy boy, was admitted to the neonatal ward due to prematurity. Two years later she became pregnant again. This time there was an extrauterine pregnancy in the left tube and a tubotomy was made. The surgeon found and divided adhesions from the uterine fundus to the abdominal wall. After two years of secondary infertility a diagnostic laparos-

copy was performed. The surgeon found new adhesions from the abdominal wall to the left tube and the sigmoid colon. When checked with methylene blue, no passage through the left tube was seen. At these two laparoscopies the uterus was inspected and no pathology noted. One year later she became pregnant spontaneously. At a check-up made by an obstetrician at 11 weeks of gestation a retroverted tender uterus was found. Check-ups according to the maternal health program were planned. During the second trimester the woman suffered from abdominal pain similar to those she had experienced in her first pregnancy. She was therefore, in gestational week 23, admitted to a district hospital where they suspected placenta previa. Four weeks later the diagnosis was revised to a recurrent incarcerated gravid uterus. In gestational week 29 the abdominal pain increased and she developed uterine contractions. With tocolytic drugs and analgesics the pregnancy was prolonged to gestational age 34 weeks plus 6 days. Due to increasing abdominal pain, a cesarean section was performed under general anesthesia. A long midline incision was made in the skin. The vagina and the urinary bladder were drawn up on the front of the uterus, which confirmed the suspected diagnosis. Without going through the urinary bladder or the vagina, a high hysterotomy was performed in the lower segment of the uterus. A healthy child was taken care of by the neonatalogists. A normal contraction of the uterus was obtained. The abdominal organs were inspected. We found nothing to explain the condition of the incarcerated retroverted gravid uterus.

Discussion When a cesarean section is performed it is often recommended that you should replace the uterus to an upward position before making the incision. Our patient had requested general anesthesia and therefore, in order to avoid negative effects of the anasthetic drugs on the baby, we wanted to take it out as fast as posible. In our case the uterus was not anteverted before the hysterotomy was made. However, we were conscious of the condition and of the risks. Therefore it was possible to place the incision accordingly. Our case shows that this condition can happen twice in the same patient and we recommend that women with a history of incarceration of a gravid uterus should be examined early in the second trimester of her following pregnancies. A review of the condition is published in this issue (3).

References 1. McGann KP, Griffin WT. Recurrent classical sacculation of the pregnant uterus. J Fam Pract 1988; 26: 339–41. 2. Wood PA. Posterior sacculation of the uterus in a patient with a double uterus. Am J Obstet Gynecol 1967; 99: 907– 8. 3. Jacobsson B, Wide-Swensson D. Incarceration of the retroverted gravid uterus – a review of the literature. Acta Obstet Gynecol Scand 1999; 78: 665–8. Address for correspondence: Bo Jacobsson, M.D. Department of Obstetrics and Gynecology Sahlgrenska University Hospital/East Hospital S-416 85 Go¨teborg, Sweden bo.jacobsson/bigfoot.com C Acta Obstet Gynecol Scand 78 (1999)

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Case Reports

Successful management of malignant hyperthermia susceptibility during cesarean hysterectomy for postpartum hemorrhage YI-CHENG WU1, CHIU-MING HO2, MEI-YUNG TSOU2, JENG-HSIU HUNG1, CHIOU-CHUNG-YUAN1, TAK-YU LEE2 AND HEUNG-TAT NG1 From the Departments of 1Obstetrics and Gynecology, and 2 Anesthesiology, Veterans General Hospital-Taipei, National Yang-Ming University, School of Medicine, Taipei, Taiwan, Republic of China Acta Obstet Gynecol Scand 1999; 78: 738–739. C Acta Obstet Gynecol Scand 1999 Key words: cesarean hysterectomy; malignant hyperthermia; malignant hyperthermia susceptibility Submitted 15 July, 1998 Accepted 19 April, 1999

If neglected, malignant hyperthermia susceptibility, in response to the administration of general anesthesia, characterized by hypermetabolism with varying clinical signs and symptoms, can result in death. Its occurrence during cesarean hysterectomy is rare but the mortality is high. Malignant hyperthermia susceptibility is an inherited pharmacogenetic disorder that occurs in susceptible subjects after exposure to inhaled anesthetics and depolarizing muscle relaxants. This case report points out that, given the unavailability of a muscle biopsy, the immediate diagnosis of malignant hyperthermia, utilizing a system of measurable variables, may very well save patients from the life-threatening or debilitating effects of this disorder.

Case report A 33-year old Chinese woman, gravida 3, para 3, was presented at 29 weeks’ gestation with painless vaginal bleeding, and was admitted for further obstetric evaluation. No other obvious anomaly in the previous prenatal work-up, other than the ultrasonographic examination on first admission, revealed a total type of placenta previa. There was no family history of illness or anesthetic problems and routine blood investigations were normal. The patient was administered oral nifedipine and an intravenous injection of ritodrine for tocolysis. Prior to this admission, the patient had undergone an appendectomy with spinal anesthesia (0.5% bupivacaine) and received laparoscopic surgery with spinal anesthesia (0.5% bupivacaine) seven years earlier. The patient’s first pregnancy was complicated by a cesarean delivery with spinal anesthesia (0.5% bupivacaine), in our hospital, for severe preeclampsia, acute fetal distress, and breech presentation at 38 weeks’ gestation. A cesarean section was repeated in the second pregnancy, with spinal anesthesia (0.5% bupivacaine) at term because of a failure to induce labor.

Abbreviations: CK: creatine kinase; Et CO2: end-tidal carbon dioxide; MH: malignant hyperthermia; MHS: malignant hyperthermia susceptibility; SaO2: oxygen saturation in arterial blood. C Acta Obstet Gynecol Scand 78 (1999)

A subsequent review of hospital and anesthesia departmental records concerning the above procedures disclosed no evidence of a malignant hyperthermia episode. Massive antepartum hemorrhage developed on the patient’s second admission and ultrasound revealed a singleton fetus, grade IV placenta previa and biometry consistent with 35 1/7 weeks’ gestation. The patient was prescribed absolute bed-rest and started on an intravenous MgSO4 solution with a maintenance dosage of 2 g per hour for two days. Two units of packed red cells were administered after the patient’s hemoglobin count fell to 8.2 g/dL. On hospital day two, with no uterine contractions detected, the patient was then switched to intravenous ritodrine at a dose of 0.1 mg per min, and oral nifedipine at a dose of 5 mg every 6 hours. The tocolytic therapy was discontinued on hospital day eleven and cesarean section was scheduled for hospital day fifteen. The patient underwent the cesarean section with spinal anesthesia (0.5% bupivacaine) at 8.30 a.m., and then changed to general anesthesia with isoflurane, succinylcholine (100 mg) for massive intraoperative bleeding. At the time of cesarean section, a 3090 g female infant was delivered with Apgar scores of 7 and 9, at 1 and 5 minutes intervals, respectively. The surgical blood loss of 4500 mL was replaced by transfusion with 8 units of packed blood cells, 6 units of fresh frozen plasma, and 12 units of platelets. After the operation, uncontrollable postpartum vaginal bleeding occurred, in spite of an intravenous injection of oxytocin and the placement of rolled gauze in the uterus. Persistent, unstable maternal vital signs soon developed despite conservative management of the transfusion, and an emergency exploratory laparotomy with general anesthesia (isoflurane, succinylcholine 100 mg) was performed 12 hours after the cesarean section. To limit further intraoperative blood loss, the uterus and cervix were then removed. In the beginning, the vital signs were BP 134/80 mmHg, HR 76 bpm, PR 16 bpm, BT 37.5æC, SaO2 96%, and Et CO2 36 mmHg at 10.30 p.m. Two hours after induction the heart rate suddenly increased to 140 bpm, the patient was warm peripherally and oral temperature showed a rapid elevation from 37.8æC to 41.5æC within a 20minute period. End-tidal carbon dioxide increased to more than 60 mmHg, serum potassium levels increased from 3.0 to 5.7 mmol/L, and a drop in blood pressure to 70/50 mmHg with severe acidosis of PaCO2 89 mmHg, pHa 7.04, BE ª12 mEq/ L, PaO2 518 mmHg was revealed by arterial blood gas data; the serum CK was 7568 U/L. Muscle rigidity was not a feature at any time. Diagnosing probable malignant hyperthermia, the respirator was checked and inhaled anesthetics were discontinued with replacement by narcotic intravenous anesthetics. Nasogastric and urinary bladder lavage with ice water was performed and a dose of 2 mg/kg IV of dantrolene (68 kg, total 140 mg) was administered at 1.00 a.m. The blood pressure returned to normal within 10 minutes and body temperature lowered to 38.3æC and acidosis improved (PaCO2 35 mmHg, pHa 7.396, PaO2 531 mmHg) over the next half an hour. The operative blood loss of 1600 mL was replaced by transfusion with 6 units of packed red blood cells, the temperature lowered again to 37.8æC and arterial blood gas data continued to improve when the hysterectomy was closed at 2.00 a.m. Throughout this period, active cooling was continued. The patient developed mild hypotension with tachycardia, prolonged prothrombin time and activated partial thromboplastin time were revealed by disseminated intravascular coagulation testing, the serum CK was 4320 U/L (normal range, 52–336) with a serum lactate of 4.90 mmol/L (normal range, 0.93–1.65). The patient was then transferred to the surgical intensive care unit for close observation. After conservative treatment, including ventilator support and transfusion was completed, the patient was extubated the following morning. The patient left the hospital after nine days without complication.

Case Reports Discussion Malignant hyperthermia is estimated to occur in approximately one in 100,000 cases of general anesthesia for adults (1), and it also occurs without a positive family history. According to a report by Strazis and Fox (2), about 1.5% of all MH cases involved complicated obstetrical surgery and one tenth of all MH cases were Asian. Therefore, its occurrence during cesarean hysterectomy is rare in Asian populations. Isoflurane and succinylcholine are both anesthetic agents known to trigger MH. In the above case, it appears that the second dose of succinylcholine (total dose of 200 mg) administered within a one-day period, was a significant factor in triggering the acute episode of MH. Documentation shows that its use will usually delay an episode of MH for several hours, increasing the incidence of unexplained sudden death (3) in susceptible patients. Given the lack of a personal and family history concerning adverse reactions to anesthesia, and the unavailability of muscle biopsy, a clinical grading scoring system should be used to improve the prediction of MHS. The system should depend on muscle rigidity, muscle breakdown, respiratory acidosis, temperature increase, cardiac involvement, family history and other indicators (4). In the above case (total score of 58 points), the patient was classified as a 6 (the highest rank for MH), indicating that the onset of MH was almost certain. The in vitro Caffeine Halothane Contracture Test is the gold standard for diagnosis (5), however, a fulminant episode with marked hypermetabolism must be treated promptly before a confirmed diagnosis can be made. Elevated serum CK levels represent increased membrane permeability of cells, primary skeletal muscle, containing CK. The patient in the above case exhibited increased serum CK level of 7568 U/L during operation, without a history of muscular trauma, ischemia, or other inflammation. The serum CK level decreased to 153 U/L on post-operation hospital day eight. Previous reports concluded that serum CK is of no value as a screening test for MH in patients with no clinical or family history of MH (5). However, high serum CK level could also be adjuvant clinical evidence of MH and a follow up test if the level returns to normal range after dantrolene treatment. The earliest signs indicating the development of an attack of MH were tachycardia with an increase in arterial carbon dioxide tension, an increase in serum potassium concentration and a fall in arterial pH (6). In the above case, the first warning signs were tachycardia with a rapid elevation of end-tidal CO2 and oral temperature (an increase 3.7æC within a 20-minute period). If these signs were missed or neglected during this short period of time, the patient might still have survived the crisis, yet may have suffered muscle damage with myoglobinuria and neurological complications. The primary pharmacological action attributed to dantro-

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lene is the relaxation of skeletal muscle by interference with excitation-contraction coupling and reduced sacroplasmic reticulum calcium release. The patient in the above case received a total dose of 140 mg, administered by intravenous injection, which rapidly and dramatically controlled the episode within a 10-minute period. No recurrence was noted during the postanesthetic period. The standard recommendation for treatment of MH is an initial 2.5 mg/kg dose, followed by additional doses of up to 10 mg/kg if symptoms persist, followed by intravenous maintenance doses of 1 mg/kg/6 h for 24–48 hours (7). Based on the experience of the above case, we suggest early detection via heat rate, end-tidal carbon dioxide and body temperature monitoring. When an attack of MH is predicted, and conditions that mimic MH are excluded, such as sepsis, thyrotoxicosis, phaeochromocytoma, myotonia, and hypoxic brain damage. All inhaled anesthetics should be discontinued and immediately dantrolene should be administered, with an ice water lavage to reduce the possibility of maternal mortality.

References 1. Britt BA, Kalow W. Malignant hyperthermia: a statistical review. Can Anaesth Soc J 1970; 17: 293–315. 2. Strazis KP, Fox AW. Malignant hyperthermia: a review of published cases. Anesth Analg 1993; 77: 297–304. 3. Denborough MA, Galloway GJ, Hopkinson KC. Malignant hyperpyrexia and sudden infant death. Lancet 1982; 2: 1068–9. 4. Larach MG, Localio AR, Allen GC, Denborough MA, Ellis FR, Gronert GA et al. A clinical grading scale to predict malignant hyperthermia susceptibility. Anesthesiology 1994; 80: 771–9. 5. Weglinski MR, Wedel DJ, Engel AG. Malignant hyperthermia testing in patients with persistently increased serum creatine kinase levels. Anesth Analg 1997; 84: 1038– 41. 6. Willatts SM. Malignant hyperthermia susceptibility (management during pregnancy and labour). Anaesthesia 1979; 34: 41–6. 7. DeRuyter ML, Wedel DJ, Berge KH. Hyperthermia requiring prolonged administration of high-dose dantroline in the postoperative period. Anesth Analg 1995; 80: 834–6.

Address for correspondence: Chiu-Ming Ho, M.D. Department of Anesthesiology Veterans General Hospital-Taipei Shih-Pai Road, Section 2, No 201, Taipei Taiwan 11217, Republic of China

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Case Reports

Anti-shock trousers (MAST) and transcatheter embolization in the management of massive obstetric hemorrhage A report of two cases BENGT ANDRAE1, LARS GUNNAR ERIKSSON2 AND GUNNAR SKOOG3 From the Departments of 1Obstetrics & Gynecology, 2 Radiology, and 3Anesthesiology, County Hospital, Ga¨vle, Sweden Acta Obstet Gynecol Scand 1999; 78: 740–741. C Acta Obstet Gynecol Scand 1999 Key words: embolization therapeutic; G suits; hemostatic techniques; human; placenta diseases; postpartum hemorrhage; pregnancy complications; puerperal disorders; uterus Submitted 31 July, 1998 Accepted 15 April, 1999

The safe use of embolization as a first line treatment of massive obstetrical hemorrhage requires simple routines to enable patient preparation as well as allowing time for an experienced radiologist to arrive so the procedure can be performed under optimal conditions. We present two cases where the use of pneumatic military anti-shock trousers (MAST) made it possible to reverse the shock and stabilize the circulation before successful interventional radiology.

Case reports Case 1: A 27-year-old woman with a previous cesarean section due to cephalopelvic disproportion underwent an elective repeat cesarean section with a healthy child, but a placenta accreta was found attached to the uterine isthmus. As much trophoblast tissue as possible was scraped out and several stitches were put in the uterine wall. The hemorrhage was reduced but not stopped. The abdomen was closed and MAST were applied. Quick diagnosis of a coagulation disorder was made with Sonoclot indicating platelet dysfunction and hypofibrinogenemia. Routine coagulation tests were taken simultaneously. Blood loss was treated and coagulation factors were corrected. When the MAST were deflated after four hours substantial bleeding resumed. The patient was brought to the department of radiology where angiographic embolization was performed. She was conscious and actively participating the whole time. Recovery was uneventful and breast-feeding was established. At her postoperative check up the patient was in a good psychological condition and menstruation had resumed. Case 2: Late one evening a 25 year-old woman with a previous normal delivery presented at term with intrauterine death. Spontaneous rupture of the membranes and a rapid vaginal delivery took place. The placenta followed uneventfully but after forty minutes a massive hemorrhage began. Peripheral shock evolved and hemoglobin declined to 50 mg/ml. Uterine massage, oxytocin and carboprost were given. MAST were applied and the bleeding stopped. Desmopressine, tranexamic acid and antithrombin III were given. Several units of erythrocytes and plasma were transfused. The circulation was stabilized but the bleeding started again when the pressure of the MAST was released. Inspection of the cervix showed no laceration but there C Acta Obstet Gynecol Scand 78 (1999)

was profuse bleeding from the uterine cavity. The MAST were reapplied and the patient was transferred to the interventional radiology laboratory where trans-catheter embolization was performed. Recovery was uneventful and the same afternoon the patient was back in the ordinary ward. Sixteen months later she delivered a healthy baby boy by elective cesarean section.

Discussion Massive obstetrical hemorrhage may evolve very rapidly and can become life threatening within a few minutes. Laparotomy and even emergency hysterectomy on a circulatory unstable patient may be a consideration. In order to avoid this, time is required to stop bleeding, substitute blood loss, evaluate and treat the coagulation disorder, identify the source of the hemorrhage and achieve permanent hemostasis as conservatively and non-invasively as possible. Surgical ligature of the hypogastric artery does not stop all bleeding as there are many collaterals and it prohibits subsequent angiography (1, 2). Emergency hysterectomy is a major trauma to a young woman and is a risky procedure (2, 3). The use of transcatheter embolization (2, 4–6) may therefore be considered but few hospitals have immediate access to interventional radiology after office hours. The critical condition of the patient may make the anesthesiologist and the obstetrician reluctant to move her outside of the intensive care unit or operating room. The use of anti-shock trousers in hemorrhagic shock is well documented (7–9) but few obstetricians have tried it and they hesitate to rely on the method. We have established the following emergency procedures used in our hospital with the resources available to us: Anti Shock Trousers (MAST) MAST is the acronym for military anti-shock trousers and there are currently at least two manufacturers (David Clark Inc., Worcester, MA and Life Support Products Inc., St. Louis, MO). We have the MAST at hand in the Emergency room next to the delivery ward. Emergency room nurses are familiar with their use. They can be applied in three minutes with the patient in her bed or on the OR table after a urine catheter has been inserted. The MAST stop virtually all bleeding (even arterial) below the renal arteries even in the presence of coagulopathy. In most cases inflation to 25–35 mm Hg is sufficient. The pressure can be maintained for 4 – 8 hours (up to 24h), and deflation should be gradual to avoid blood pressure drop. Inflated MAST redistribute the remaining blood volume to the vital organs from kidney level and above and also facilitate venous puncture. When the bleeding has stopped and blood pressure is resumed, the panic in the room calms down and there is time to assess the situation. The anesthesiologist can concentrate on replacement of blood loss. Quick methods like Sonoclot (Sienco Inc., Morrison, CO) (10) can be used to assess the coagulation disorder. There is no longer need for emergency surgery, instead the interventional radiologist can be summoned and has several hours for preparation. The patient and her husband can be taken care of psychologically. Transcatheter embolization The femoral artery is punctured unilaterally. Via an introducer sheath the hypogastric and uterine arteries on both sides are catheterized with a standard 5F catheter, to locate the bleeding vessel. A piece of gelfoam is scraped with a scalpel and suspended in a saline solution mixed with contrast 50–50 to 20 cc. The suspension is injected as close to the source of bleeding as possible. If necessary, grains of gelfoam can be added. Embolization material is injected until hemostasis is achieved, (no more angiographically visible extravasation). Introducer

Case Reports sheaths can be left in place for 24 hours in case additional treatment should become necessary. Organization If a major hemorrhage after a delivery cannot be sufficiently controlled using oxytocin and prostaglandins or by means of surgical ligation of identified bleeding vessels vaginally or via the cesarean incision, then the MAST are brought from the emergency room and immediately applied. The interventional radiologist on duty is alerted. The blood loss and coagulation disorder are corrected after diagnostic tests including Sonoclot which are available at the intensive care unit. If bleeding resumes when the MAST are deflated after a couple of hours the patient can be transferred, with stable circulation, to the radiologist who should be prepared for immediate embolization under local anesthesia. The leg compartments of the MAST can remain inflated during the procedure. This approach is also used in cases of postoperative hemorrhage. One of three interventional radiologists is available within two hours around the clock in our hospital.

References 1. Gilbert WM, Moore TR, Resnik R, Doemeny J, Chin H, Bookstein JJ. Angiographic embolisation in the management of hemorrhagic complications of pregnancy. Am J Obstet Gynecol 1992; 166: 493–7. 2. Vedantham S, Goodwin SC, McLucas B, Mohr G. Uterine artery embolization: an underused method of controlling pelvic hemorrhage. Am J Obstet Gynecol 1997; 176: 938– 48.

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3. Zelop CM, Harlow BL, Frigoletto FD Jr, Safon LE, Saltzman DH. Emergency peripartum hysterectomy. Am J Obstet Gynecol 1993; 168: 1443–8. 4. Yamashita Y, Harada M, Yamamoto H, Miyazaki T, Takahashi M, Miyazaki K et al. Transcatheter arterial embolization of obstetric and gynaecological bleeding: efficacy and clinical outcome. Br J Radiol 1994; 67: 530–4. 5. Hsu YR, Wan YL. Successful management of intractable puerperal hematoma and severe postpartum hemorrhage with DIC through transcatheter arterial embolization – two cases. Acta Obstet Gynecol Scand 1998; 77: 129–31. 6. Svendsen P, Wikholm G. Embolisering fo¨rsta åtga¨rd vid svåra blo¨dningar. (In Swedish). La¨kartidningen 1998; 95: 3290–4. 7. McSwain NE. Pneumatic anti-shock garment: state of the art 1988. Ann Emerg Med 1988; 17: 506–25. 8. Pelligra R, Sandberg EC. Control of intractable abdominal bleeding by external counterpressure. JAMA 1979; 241: 708–13. 9. Strandell A, Skoog G. Antichockbyxa – terapeutiskt komplement vid svåra obstetriska blo¨dningar. (In Swedish). La¨kartidningen 1992; 89: 2853–4. 10. Steer PL, Krantz HB. Thromboelastography and Sonoclot analysis in the healthy parturient. J Clin Anesth 1993; 5: 419–24. Address for correspondence: Bengt Andrae, M.D. Department of Obstetrics and Gynecology County Hospital S-801 87 Gävle Sweden

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