No cardiac measurements could be obtained from earlier ultrasounds. The findings and outcome of this case indicate the importance of third-trimester echocardiographic monitoring of heart function in fetuses of mothers with diabetes. Although the incidence of fatal hypertrophic cardiomyopathy in infants of women with diabetes is believed to be rare, treatment, including early delivery and propranolol treatment of the neonate, might have improved the outcome in this case.3 Indeed it is possible that the unexplained fetal deaths described in previous reports6,7 might be attributable to hypertrophic cardiomyopathy. Diligent monitoring of fetal progress with special consideration of the possibility of hypertrophic cardiomyopathy could reveal additional cases, and could indicate more aggressive intervention than previously seemed appropriate.
3. Weintrob N, Karp M, Hod M. Short- and long-range complications in offspring of diabetic mothers. J Diabetes Complications 1996;10:294 –301. 4. Leslie J, Shen C, Strauss L. Hypertrophic cardiomyopathy in a midtrimester fetus born to a diabetic mother. J Pediatr 1982;100:631–2. 5. Maron BJ, Edwards JE, Henry WL, Clark CE, Bingle GJ, Epstein SE. Asymmetric septal hypertrophy (ASH) in infancy. Circulation 1974;50:809 –20. 6. Girz BA, Divon MY, Merkatz IR. Sudden fetal death in women with well-controlled, intensively monitored gestational diabetes. J Perinatol 1992;12:229 –33. 7. Gutgesell HP, Speer ME, Rosenberg HS. Characterization of the cardiomyopathy in infants of diabetic mothers. Circulation 1980;61:441–50.
REFERENCES
8. McMahon JN, Berry PJ, Joffe HS. Fatal hypertrophic cardiomyopathy in an infant of a diabetic mother. Pediatr Cardiol 1990;11:211–2.
1. Pildes, RS. Infants of diabetic mothers. N Engl J Med 1973;289:902– 4. 2. Tyrala EE. The infant of the diabetic mother. Obstet Gynecol Clin North Am 1996;23:221– 41.
Received December 15, 2000. Received in revised form March 9, 2001. Accepted April 19, 2001.
Placenta Percreta With Spontaneous Rupture of an Unscarred Uterus in the Second Trimester William J. LeMaire, MD, Claire Louisy, MD, Kathie Dalessandri, MD, and Frederick Muschenheim, MD Department of Obstetrics and Gynaecology, St. Jude Hospital, St. Lucia, West Indies; the Department of Obstetrics and Gynecology, University of Miami School of Medicine, Miami, Florida; and the Department of Surgery, Stanford University, Point Reyes Station, California
BACKGROUND: Rupture of a pregnant uterus occurs most often in a scarred uterus, and spontaneous rupture of a non-scarred uterus in the early second trimester is rare. CASE: A woman with two previous normal vaginal deliveries and no prior trauma to the uterus presented at 16 weeks’ gestation with an acute abdomen due to intraperitoneal hemorrhage. A large rupture of the fundus of the uterus was found. A supracervical hysterectomy was carAddress reprint requests to: William J. LeMaire, MD, PO Box 1672, Sitka, Alaska 99835; E-mail:
[email protected].
ried out, with subsequent good recovery. The specimen showed placenta percreta. CONCLUSION: Spontaneous rupture of an unscarred uterus, due to placenta percreta, should be considered in cases of acute intraperitoneal hemorrhage, even in early pregnancy. (Obstet Gynecol 2001;98:927–9. © 2001 by the American College of Obstetricians and Gynecologists.)
Spontaneous rupture of a non-scarred uterus is a relatively rare occurrence. If it happens, it usually happens in the third trimester of pregnancy, during the process of labor and delivery. It’s occurrence in the early second trimester, without any predating injury or trauma, is very rare indeed. We report a case of spontaneous rupture of an unscarred uterus in the early second trimester of an otherwise uncomplicated pregnancy. CASE REPORT The patient is a gravida 3 with two previous healthy vaginal deliveries. She was seen in the emergency room at approximately 13 weeks’ gestation, calculated from her last menstrual period. She had a weeklong history of left lower abdominal pain. This pain had progressively increased, becoming diffuse throughout the abdomen, and was associated with vomiting. She had no vaginal
VOL. 98, NO. 5, PART 2, NOVEMBER 2001 © 2001 by The American College of Obstetricians and Gynecologists. Published by Elsevier Science Inc.
0029-7844/01/$20.00 PII S0029-7844(01)01580-0
927
bleeding, no fainting, and no urinary symptoms. There was no history of previous surgery, uterine evacuation or termination of pregnancy, and she denied any trauma to the abdomen. Her past medical history was uncomplicated. On admission, the patient looked ill, in pain, and dehydrated. Her blood pressure was 135/65 mmHg, and her pulse was 112 beats per minute. Her body temperature was 98.6F. Her abdomen was tense, distended, and diffusely tender in all quadrants, with rebound tenderness and guarding. The bowel sounds were decreased. No distinct mass could be palpated in her abdomen. On pelvic examination, the appearance of the cervix was consistent with pregnancy. The os was closed, without bleeding. There was cervical tenderness, and the uterus was enlarged but difficult to outline. The pouch of Douglas was tender. There was no blood on rectal exam. The patient’s hematocrit was 26%, with a white blood cell count of 7600/mm3. Blood chemistries were normal, including a serum amylase. An ultrasound examination in the emergency room showed an intrauterine pregnancy with a live fetus of approximately 16 weeks’ gestation. No adnexal masses were seen. Fluid was present in the cul-de-sac. A chest x-ray was normal. A diagnosis was made of an intrauterine pregnancy with an acute surgical abdomen. Several possible diagnoses were entertained, including a ruptured viscus with intraperitoneal hemorrhage. A concomitant ruptured ectopic pregnancy was considered. The patient was taken immediately to the operating room. Under general anesthesia, a laparotomy was performed through a lower abdominal midline incision. Upon entry of the abdomen, there was a large amount of blood and clots. The uterus was 16 –18 weeks in size, with a large (⬃8 cm) ragged opening in the fundus. Placental tissue and membranes were protruding. When the membranes were ruptured, a live, immature fetus was extruded. The clinical impression was that of a ruptured uterus due to placenta percreta. There was a 4-cm fibroma in the right lateral aspect of the uterus beneath, but not involved with, the rupture. Both cornua of the uterus were clearly identified separately from the rupture, ruling out a ruptured cornual pregnancy. As the patient’s condition was unstable, a subtotal hysterectomy with preservation of both ovaries was carried out without difficulty. All other intraperitoneal organs were intact and normal. The operative procedure was relatively bloodless as soon as the uterine arteries were clamped. The total blood loss was estimated at 1.5 L, and 1 L of matched blood was transfused intra- and postoperatively. The patient received four doses of a cephalosporin antibiotic (one dose pre-operatively). The postoperative hemoglobin was 6.7 g/dL, with a
928
LeMaire et al
Rupture of Uterus in the Second Trimester
hematocrit of 19.3%. Except for a low-grade fever during the first 36 hours after surgery, the patient did well and was discharged on the third postoperative day on iron tablets. The pathology report showed chorionic villi invading the myometrium, without intervening basal plate, consistent with the diagnosis of placenta percreta. In addition, there was a 3.5-cm leiomyoma with necrosis in the wall of the uterus. The fetus weighed 120 g and measured 12 cm from crown to rump. This was consistent with approximately 17 weeks’ gestation.
COMMENT Spontaneous rupture of an unscarred uterus during pregnancy is rare. Placenta percreta has been recognized as a possible cause.1 Uterine rupture due to placenta percreta is reported to occur primarily during the late second and the third trimester of pregnancy.2 There have been isolated case reports in the first and early second trimester.2,3 The case reported here draws attention to the possibility of catastrophic intraperitoneal hemorrhage due to spontaneous rupture of the uterus associated with placenta percreta. This diagnosis should be considered in cases of an acute intra-abdominal event as early as the late first and early second trimester, even in the absence of previous trauma to the uterus (cesarean delivery, myomectomy, perforation, etc). The diagnosis of abnormal placentation (placenta accreta, increta, or percreta) before delivery or the occurrence of acute hemorrhage is probably not feasible on clinical grounds, in the absence of any risk factors (eg, placenta previa and previous cesarean delivery). Unexplained elevation of maternal serum alpha-fetoprotein4 and elevated creatinine kinase5 have recently been reported in association with such abnormal placentation. Ultrasonographic studies and/or magnetic resonance imaging may confirm this diagnosis.6 An early diagnosis, before the acute presentation, would be invaluable for the management of such cases.6 Once presented with an acute intraperitoneal hemorrhage, prompt surgical intervention is essential. Although hysterectomy, total or subtotal, would be the management choice in most cases, conservative management could be considered in selected cases.6 – 8
REFERENCES 1. Imseis H, Murtha A, Alexander K, Barnett B. Spontaneous rupture of a primigravid uterus secondary to placenta percreta. J Reprod Med 1998;43:233– 6. 2. Woolcott R, Nicholl M, Gibson J. A case of placenta per-
OBSTETRICS & GYNECOLOGY
3. 4.
5.
6.
creta presenting in the first trimester of pregnancy. Aust NZ J Obstet Gynaecol 1964;4:258 – 60. Heider P. Placenta percreta in early gestation. J Pak Med Assoc 1990;40:274 –5. McDuffy R, Harkness L, McVay R, Haverkamp A. Midtrimester hemoperitoneum caused by placenta percreta in association with elevated maternal serum alpha-fetoprotein level. Am J Obstet Gynecol 1994;171:565– 6. Ophir E, Tendler R, Odeh M, Khoury S, Oettinger M. Creatinine kinase as a biochemical marker in diagnosis of placenta increta and percreta. Am J Obstet Gynecol 1999; 180:1039 – 40. Hudon L, Belfort M, Broome D. Diagnosis and manage-
Placenta Percreta and Uterine Rupture Associated With Prior Whole Body Radiation Therapy Errol R. Norwitz, MD, PhD, Howard M. Stern, MD, PhD, Holcombe Grier, MD, and Aviva Lee-Parritz, MD Departments of Obstetrics and Gynecology and Pathology, Brigham and Women’s Hospital, and Department of Pediatric Oncology, Dana Farber Cancer Institute, Harvard Medical School, Boston, Massachusetts
BACKGROUND: Injury to reproductive organs including the uterus is a known complication of ionizing radiation, but the risks to the mother and fetus during subsequent pregnancies are not well defined. CASE: A young woman with a remote history of whole body irradiation for childhood leukemia had uterine rupture at 17 weeks’ gestation. Pathologic examination of the supracervical hysterectomy specimen revealed a posteriorfundal placenta percreta with a diffusely thinned myometrium (1– 6 mm). The clinicopathologic findings were consistent with prior radiation injury. CONCLUSION: Uterine irradiation may predispose to abnormal placentation and uterine rupture in a subsequent pregnancy. (Obstet Gynecol 2001;98:929 –31. © 2001 by the American College of Obstetricians and Gynecologists.)
Injury to reproductive organs is a known complication of ionizing radiation. The potential adverse effects of radiAddress reprint requests to: Errol R. Norwitz, MD, PhD, Department of Obstetrics and Gynecology, Harvard Medical School, Brigham and Women’s Hospital, 75 Francis Street, Boston, MA 02115, E-mail:
[email protected].
ment of placenta percreta: A review. Obstet Gynecol Surv 1998;53:509 –17. 7. Yeshaya A, Ofer L, Sorina G-G, Ori S, Yoram D. Conservative surgical management of acute abdomen caused by placenta percreta in the second trimester. Am J Obstet Gynecol 1994;170:1388 –9. 8. Cox S, Carpenter R, Cotton D. Placenta percreta: Ultrasound diagnosis and conservative management. Obstet Gynecol 1988;71:454 – 65.
Received February 20, 2001. Received in revised form May 29, 2001. Accepted July 5, 2001.
ation on the fetus include spontaneous abortion, microcephaly, mental retardation, growth restriction, germ cell mutations, and possibly an increased risk of malignancy later in life.1 However, little is known about the long-term effects of pelvic radiation on subsequent pregnancy outcome. We present a case of placenta percreta and uterine rupture at 17 weeks’ gestation in a primigravida with a remote history of whole body irradiation for childhood leukemia. CASE A 23-year-old woman, gravida 1, presented at 17 weeks’ gestation with acute abdominal pain referred to the shoulders bilaterally. She was stable hemodynamically, and the fetal heart rate was 140 beats per minute. Her medical history was remarkable for successful treatment for chronic myelogenous leukemia. At age 5 years, she was treated initially with vincristine, prednisone, methotrexate, doxorubicin, and mercaptopurine chemotherapy. She then received a bone marrow transplant from her brother, who had identical human leukocyte antigens. Her preoperative regimen included high-dose cytarabine, cyclophosphamide, and 875-rad total body irradiation given in a single fraction. She developed chronic graft-versus-host disease which was treated with imuran and prednisone and resolved by age 9 years. She had delayed puberty, achieving a final adult height of 4 feet 11 inches with Tanner stage 2 breast development. This pregnancy was a spontaneous conception. Her antenatal course was uncomplicated, including a normal sonographic examination of a singleton intrauterine pregnancy at 11.6 weeks’ gestation. Immediate surgical exploration found hemoperitoneum and a posterior-fundal uterine rupture without evidence of cornual ectopic pregnancy. There was no identifiable ovarian tissue on the right and a small streak
VOL. 98, NO. 5, PART 2, NOVEMBER 2001 © 2001 by The American College of Obstetricians and Gynecologists. Published by Elsevier Science Inc.
0029-7844/01/$20.00 PII S0029-7844(01)01435-1
929