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Fetus papyraceous: an unusual cause of obstructed labour
European Journal of Obstetrics & Gynecology and Reproductive Biology 86 (1999) 109–111
Case Report
Fetus papyraceous: an unusual cause of obstructed labour W.C. Lau*, M.S. Rogers Department of Obstetrics and Gynaecology, Prince of Wales Hospital, The Chinese University of Hong Kong, Shatin, NT, Hong Kong Received 2 November 1998; received in revised form 6 January 1999; accepted 22 February 1999
Abstract Fetus papyraceous is a relatively rare complication in twin pregnancy. The occurrence of fetus papyraceous is frequently associated with perinatal morbidity in the other twin, making antenatal diagnosis of this condition desirable. Ultrasound detection is not always possible due to anatomical position and technical difficulties. A case of fetus papyraceous, found during Caesarean section for obstructed labour is reported and the implications of antenatal detection are discussed. 1999 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Fetus papyraceous; Obstructed labour
1. Introduction Fetus papyraceous only occurs in multiple pregnancy. The incidence is reported to be 1:184 twin births (¯1:12 000 live births) [1]. The fetus usually dies before 20 weeks’ gestation and becomes compressed and mummified between the amniotic sac of its co-twin and the uterine wall. The occurrence of fetus papyraceous is often associated with significant morbidity in the co-twin, namely antepartum stillbirth or intrauterine growth retardation. Maternal morbidity due to postpartum haemorrhage and infection resulting from the retention of the dead fetus has also been documented in the obstetric literature [2]. We report a case of fetus papyraceous as an unusual cause of obstructed labour at term and discuss the implications of antenatal detection.
2. Case report A 26-year-old primigravida was admitted at 39 weeks’ gestation because of spontaneous labour. Her antenatal *Corresponding author. Tel.: 1852-2632-2810; fax: 1852-2636-0008. E-mail address: [email protected] (W.C. Lau)
course had been uneventful. An ultrasound scan had been performed at 31 weeks’ gestation because the fetus was in breech presentation. No abnormalities were detected. On admission, the fetus was thought to be of average size and the head was 3 / 5 palpable above pelvic brim. The cervix was 3 cm dilated and effaced. However, on vaginal examination the fetal scalp was described by the midwife as nodular. There was neither decent of the presenting part nor dilatation of the cervix despite adequate augmentation of labour with oxytocin for 6 h. An emergency Caesarean section was performed due to failure of labour to progress. A normal female baby weighing 2.6 kg was delivered. A fetus papyraceous was found at the lower part of the uterus, covering the cervical os. The placenta and the mummified dead fetus were delivered. The mother was well after the operation. There was no evidence of infection or intravascular coagulopathy in the puerperium and the neonatal course was uneventful. The macerated fetus was a male and weighed 135 g. It was compressed with marked deformation and roughly corresponded to between 16 and 20 weeks’ gestation based on measurement of the crown–rump length. Pathological examination did not reveal any significant malformation. The placenta weighed 610 g and the membranes appeared to be diamniotic and dichorionic. Approximately one third
0301-2115 / 99 / $ – see front matter 1999 Elsevier Science Ireland Ltd. All rights reserved. PII: S0301-2115( 99 )00053-6
110
W.C. Lau, M.S. Rogers / European Journal of Obstetrics & Gynecology and Reproductive Biology 86 (1999) 109 – 111
Fig. 1. Fetus papyraceous and the twin placenta.
of the placenta was infarcted. The placental disc of the dead fetus showed severe degeneration with calcification and fibrin deposition while the placental disc of the surviving co-twin was unremarkable histologically. There was no evidence of abruptio placenta or velamentous insertion of the cord (Fig. 1).
3. Discussion Intrauterine death in a twin pregnancy can occur in any gestation. The death of one twin in the first half of pregnancy is relatively common. Fetus papyraceous usually results from missed abortion occurring at the end of the first trimester or early in the second trimester while the other fetus goes on to full development. The amniotic fluid is absorbed and the retained dead fetus is compressed between the sac of the surviving co-twin and the uterine wall. The retained fetus may undergo maceration. It eventually becomes desiccated and mummified so that it resembles parchment [3]. The problem of intrauterine death in one of a twin pair and its association with increased morbidity and mortality in the surviving co-twin has long been recognized. First
trimester single fetal demise in a twin has not been shown to have adverse consequences for the surviving twin [4]. The perinatal morbidity and mortality for the surviving co-twin is known to be considerably higher where second or third trimester loss occurs. Premature delivery may supervene or the second twin might succumb in utero from a variety of causes such as abruptio placenta, chorioamnionitis or intrauterine growth restriction. Rarely, it is associated with disseminated intravascular coagulation or congenital aplasia cutis of the surviving co-twin [5]. There is a higher risk of cerebral palsy among the survivors, particularly if the twins are monochorionic [6]. The diagnosis of fetus papyraceous is usually made during labour or after delivery. Bagga et al. recently reported a case of fetus papyraceous covering the site of a uterine perforation, found incidentally during Caesarean section for failure of labour to progress [7]. Ultrasound failed to detect the anomaly in our case because of its anatomical position: the papyraceous was presenting below the vertex of its co-twin and had descended into the bony pelvis. Transvaginal ultrasound would have been able to visualise the fetus if the diagnosis had been suspected following the midwife’s report. Early ultrasound had not been performed in this pregnancy and therefore the pres-
W.C. Lau, M.S. Rogers / European Journal of Obstetrics & Gynecology and Reproductive Biology 86 (1999) 109 – 111
ence of a second fetus was never suspected. Neither was there any history of vaginal bleeding or abdominal pain to suggest intrauterine death of a second fetus. However, failure to detect this anomaly in the antenatal period had minimal effect on this pregnancy. Early diagnosis would have resulted in elective Caesarean delivery rather than after 6 h of labour but Caesarean delivery itself was unavoidable as the fetus papyraceous obstructed descent of the co-twin. Routine scanning is useful for determination of the chorionicity of early twin gestations and for early diagnosis of a blighted twin. Serial scanning is useful for monitoring the progress of these pregnancies, especially in monochorionic twins: twin to twin transfusion syndrome is postulated as one of the causes of fetus papyraceous. Co-twin morbidity would not be expected to be a significant problem in dizygotic twins because of the absence of vascular anastomoses.
111
References [1] Livnat EJ, Burd L, Cadkin A et al. Fetus papyraceous in twin pregnancy. Obstet Gynecol 1978;51:41S–5S. [2] Landy HJ, Weingold AB. Management of a multiple gestation complicated by an antepartum fetal demise. Obstet Gynecol Survey 1989;44:171–6. [3] Cunningham FG, MacDonald PC, Gant NF et al. In: Williams obstetrics, 19th edition, Appleton and Lange, 1993, p. 666. [4] Landy HJ, Weiner S, Corson SL et al. The vanishing twin: ultrasonographic assessment of fetal disappearance in the first trimester. Am J Obstet Gynecol 1986;155:14–9. [5] Wagner DS, Klein RL, Robinson HB, Novak RW. Placental emboli from a fetus papyraceous. J Pediatr Surg 1990;25(2):538–42. [6] Pharoah PO, Cooke T. Cerebral palsy and multiple births. Arch Dis Child 1996;75(3):F174–7. [7] Bagga R, Goel P, Prasad GRV, Gupta I. Fetus papyraceous covering the site of uterine perforation found during caesarean section. Aust NZ J Obstet Gynecol 1997;3(37):360.
Case Report
Fetus papyraceous: an unusual cause of obstructed labour W.C. Lau*, M.S. Rogers Department of Obstetrics and Gynaecology, Prince of Wales Hospital, The Chinese University of Hong Kong, Shatin, NT, Hong Kong Received 2 November 1998; received in revised form 6 January 1999; accepted 22 February 1999
Abstract Fetus papyraceous is a relatively rare complication in twin pregnancy. The occurrence of fetus papyraceous is frequently associated with perinatal morbidity in the other twin, making antenatal diagnosis of this condition desirable. Ultrasound detection is not always possible due to anatomical position and technical difficulties. A case of fetus papyraceous, found during Caesarean section for obstructed labour is reported and the implications of antenatal detection are discussed. 1999 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Fetus papyraceous; Obstructed labour
1. Introduction Fetus papyraceous only occurs in multiple pregnancy. The incidence is reported to be 1:184 twin births (¯1:12 000 live births) [1]. The fetus usually dies before 20 weeks’ gestation and becomes compressed and mummified between the amniotic sac of its co-twin and the uterine wall. The occurrence of fetus papyraceous is often associated with significant morbidity in the co-twin, namely antepartum stillbirth or intrauterine growth retardation. Maternal morbidity due to postpartum haemorrhage and infection resulting from the retention of the dead fetus has also been documented in the obstetric literature [2]. We report a case of fetus papyraceous as an unusual cause of obstructed labour at term and discuss the implications of antenatal detection.
2. Case report A 26-year-old primigravida was admitted at 39 weeks’ gestation because of spontaneous labour. Her antenatal *Corresponding author. Tel.: 1852-2632-2810; fax: 1852-2636-0008. E-mail address: [email protected] (W.C. Lau)
course had been uneventful. An ultrasound scan had been performed at 31 weeks’ gestation because the fetus was in breech presentation. No abnormalities were detected. On admission, the fetus was thought to be of average size and the head was 3 / 5 palpable above pelvic brim. The cervix was 3 cm dilated and effaced. However, on vaginal examination the fetal scalp was described by the midwife as nodular. There was neither decent of the presenting part nor dilatation of the cervix despite adequate augmentation of labour with oxytocin for 6 h. An emergency Caesarean section was performed due to failure of labour to progress. A normal female baby weighing 2.6 kg was delivered. A fetus papyraceous was found at the lower part of the uterus, covering the cervical os. The placenta and the mummified dead fetus were delivered. The mother was well after the operation. There was no evidence of infection or intravascular coagulopathy in the puerperium and the neonatal course was uneventful. The macerated fetus was a male and weighed 135 g. It was compressed with marked deformation and roughly corresponded to between 16 and 20 weeks’ gestation based on measurement of the crown–rump length. Pathological examination did not reveal any significant malformation. The placenta weighed 610 g and the membranes appeared to be diamniotic and dichorionic. Approximately one third
0301-2115 / 99 / $ – see front matter 1999 Elsevier Science Ireland Ltd. All rights reserved. PII: S0301-2115( 99 )00053-6
110
W.C. Lau, M.S. Rogers / European Journal of Obstetrics & Gynecology and Reproductive Biology 86 (1999) 109 – 111
Fig. 1. Fetus papyraceous and the twin placenta.
of the placenta was infarcted. The placental disc of the dead fetus showed severe degeneration with calcification and fibrin deposition while the placental disc of the surviving co-twin was unremarkable histologically. There was no evidence of abruptio placenta or velamentous insertion of the cord (Fig. 1).
3. Discussion Intrauterine death in a twin pregnancy can occur in any gestation. The death of one twin in the first half of pregnancy is relatively common. Fetus papyraceous usually results from missed abortion occurring at the end of the first trimester or early in the second trimester while the other fetus goes on to full development. The amniotic fluid is absorbed and the retained dead fetus is compressed between the sac of the surviving co-twin and the uterine wall. The retained fetus may undergo maceration. It eventually becomes desiccated and mummified so that it resembles parchment [3]. The problem of intrauterine death in one of a twin pair and its association with increased morbidity and mortality in the surviving co-twin has long been recognized. First
trimester single fetal demise in a twin has not been shown to have adverse consequences for the surviving twin [4]. The perinatal morbidity and mortality for the surviving co-twin is known to be considerably higher where second or third trimester loss occurs. Premature delivery may supervene or the second twin might succumb in utero from a variety of causes such as abruptio placenta, chorioamnionitis or intrauterine growth restriction. Rarely, it is associated with disseminated intravascular coagulation or congenital aplasia cutis of the surviving co-twin [5]. There is a higher risk of cerebral palsy among the survivors, particularly if the twins are monochorionic [6]. The diagnosis of fetus papyraceous is usually made during labour or after delivery. Bagga et al. recently reported a case of fetus papyraceous covering the site of a uterine perforation, found incidentally during Caesarean section for failure of labour to progress [7]. Ultrasound failed to detect the anomaly in our case because of its anatomical position: the papyraceous was presenting below the vertex of its co-twin and had descended into the bony pelvis. Transvaginal ultrasound would have been able to visualise the fetus if the diagnosis had been suspected following the midwife’s report. Early ultrasound had not been performed in this pregnancy and therefore the pres-
W.C. Lau, M.S. Rogers / European Journal of Obstetrics & Gynecology and Reproductive Biology 86 (1999) 109 – 111
ence of a second fetus was never suspected. Neither was there any history of vaginal bleeding or abdominal pain to suggest intrauterine death of a second fetus. However, failure to detect this anomaly in the antenatal period had minimal effect on this pregnancy. Early diagnosis would have resulted in elective Caesarean delivery rather than after 6 h of labour but Caesarean delivery itself was unavoidable as the fetus papyraceous obstructed descent of the co-twin. Routine scanning is useful for determination of the chorionicity of early twin gestations and for early diagnosis of a blighted twin. Serial scanning is useful for monitoring the progress of these pregnancies, especially in monochorionic twins: twin to twin transfusion syndrome is postulated as one of the causes of fetus papyraceous. Co-twin morbidity would not be expected to be a significant problem in dizygotic twins because of the absence of vascular anastomoses.
111
References [1] Livnat EJ, Burd L, Cadkin A et al. Fetus papyraceous in twin pregnancy. Obstet Gynecol 1978;51:41S–5S. [2] Landy HJ, Weingold AB. Management of a multiple gestation complicated by an antepartum fetal demise. Obstet Gynecol Survey 1989;44:171–6. [3] Cunningham FG, MacDonald PC, Gant NF et al. In: Williams obstetrics, 19th edition, Appleton and Lange, 1993, p. 666. [4] Landy HJ, Weiner S, Corson SL et al. The vanishing twin: ultrasonographic assessment of fetal disappearance in the first trimester. Am J Obstet Gynecol 1986;155:14–9. [5] Wagner DS, Klein RL, Robinson HB, Novak RW. Placental emboli from a fetus papyraceous. J Pediatr Surg 1990;25(2):538–42. [6] Pharoah PO, Cooke T. Cerebral palsy and multiple births. Arch Dis Child 1996;75(3):F174–7. [7] Bagga R, Goel P, Prasad GRV, Gupta I. Fetus papyraceous covering the site of uterine perforation found during caesarean section. Aust NZ J Obstet Gynecol 1997;3(37):360.