|Year : 2021 | Volume
| Issue : 3 | Page : 172-175
Fetal mummification complicating a dichorionic twin gestation: Clinical pearls for medical education
Olusoji Edward Jagun1, Oluwaseyitan Adesegun2
1 Twin Specialist Clinic; Department of Obstetrics and Gynecology, Olabisi Onabanjo University Teaching Hospital, Sagamu, Ogun State, Nigeria
2 Twin Specialist Clinic; Department of Community Medicine, Babcock University Teaching Hospital, Ilishan-Remo, Ogun State, Nigeria
|Date of Submission||02-Dec-2020|
|Date of Acceptance||12-Aug-2021|
|Date of Web Publication||28-Sep-2021|
Dr. Oluwaseyitan Adesegun
Department of Community Medicine, Babcock University Teaching Hospital, Ilishan-Remo, Ogun State
Source of Support: None, Conflict of Interest: None
Fetus papyraceous is a complication of multiple gestation characterized by single intrauterine fetal death of a twin, co-existing with a normal twin. It can present without problems to the mother and surviving twin but it increases the risk of death to the surviving twin, preterm delivery, dystocia, and other fetomaternal complications. This case aims to draw attention to the rare finding of a mummified twin coexisting with a normal twin, as well as the clinical management of such presentation. We present a case of a 32-year-old multigravida with no living children, who spontaneously conceived a twin pregnancy that was complicated by fetus papyraceous and eventual (inevitable) preterm delivery. The pregnancy was managed conservatively by frequent fetomaternal monitoring to allow for adequate fetal lung maturity. The surviving twin and mother remained stable postpartum. Frequent ultrasonographic monitoring along with conservative management to prolong pregnancy until fetal lung maturity is ascertained, are useful strategies in managing such a case, and the management of each case should be individualized.
The following core competencies are addressed in this article: Medical knowledge, Patient care.
Keywords: Dichorionic twin gestation, fetus papyraceous, mummified twin, Nigeria
|How to cite this article:|
Jagun OE, Adesegun O. Fetal mummification complicating a dichorionic twin gestation: Clinical pearls for medical education. Int J Acad Med 2021;7:172-5
|How to cite this URL:|
Jagun OE, Adesegun O. Fetal mummification complicating a dichorionic twin gestation: Clinical pearls for medical education. Int J Acad Med [serial online] 2021 [cited 2021 Nov 30];7:172-5. Available from: https://www.ijam-web.org/text.asp?2021/7/3/172/326814
| Introduction|| |
Multiple gestation is a common presentation in Nigeria, reported to be one of the highest in the world. Intrauterine death of a single twin, with subsequent flattening or “mummification” of the dead fetus, is termed “fetus papyraceous,” and is one of the rare complications in twin and higher-order pregnancies. It occurs in 1 in 184 twin pregnancies. We present a case of fetus papyraceous that co-existed with a normal co-twin, in a multiparous client being managed for recurrent stillbirths. This case is unique because it highlights the morphology of a rare presentation of single intrauterine fetal death co-existing with a normal fetus, and demonstrates the challenges in clinical management as well as the risks to the surviving fetus and mother. It furthermore presents an opportunity for medical education. Informed consent was obtained from the patient. The authors adhered to the CARE guidelines in reporting this case.
| Case Report|| |
The case is that of a 32-year-old woman of the Yoruba tribe in South-West Nigeria, a gravida 6 para 5 (none alive), now para 6 (1 alive) who was booked at our specialist Obstetrics and Gynecology practice in South-West Nigeria. She had a history of recurrent unexplained stillbirths on five occasions, spanning a duration of 5 years (2014–2019). Each incident was preceded by an ultrasound diagnosis of intrauterine fetal demise, requiring induction of labor and eventual vaginal delivery. She had no history of diabetes mellitus, autoimmune diseases, Rhesus incompatibility, or TORCHES infections. She did not consume alcohol, cigarettes, recreational drugs, or herbal concoctions, and had no history of congenital anomalies in any of the previous stillbirths. She however spontaneously conceived in November 2019, which was confirmed to be a dichorionic diamniotic twin gestation at 23 weeks + 4 days, when she presented to the facility for antenatal booking. The ultrasound scan revealed that one twin (Twin I) was alive and well, with fetal biometric dating of about 24 weeks gestational age (GA), normal placental location and morphology, and adequate amniotic fluid volume. The second twin (Twin II) however showed no signs of life, with fetal biometric dating of about 19 weeks + 2 days GA. The patient had a positive family history of twinning in her first-degree relative. During the index pregnancy, the patient had recurrent episodes of preterm contractions from 25 to 33 weeks of gestation managed with tocolytics and anxiolytics. Her packed cell volume, fasting blood glucose, and glycosylated hemoglobin were normal throughout pregnancy. Doppler ultrasound of the live twin showed a normal blood flow pattern and fetal weight above the 50th percentile.
She however presented to the facility at 34 weeks + 2 days with persistent uterine contraction and passage of show. Vagina examination revealed that she was in the active phase of labor. She was eventually delivered of a live male fetus via spontaneous vertex delivery, birth weight 2.75 kg (APGAR scores 8 at 1 min, 10 at 5 min) [Figure 1]. She subsequently expelled a dead, mummified second twin along with the products of conception [Figure 2], [Figure 3]. Labor was managed actively. Mother and baby were stable postpartum.
|Figure 2: Dorsal view of dead (mummified) twin with products of conception|
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|Figure 3: Ventral view of dead (mummified) twin with products of conception|
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| Discussion|| |
While the rate of monozygotic (identical) twinning is constant globally across populations at about 4/1000 live births, Nigeria along with other countries in West and Central Africa are known to have the highest incidence rates of twinning in the world at more than 18/1000 live births, contributed mostly by dizygotic (fraternal) twinning. The Southwest part of Nigeria (where our patient is from) is particularly noted to have the highest incidence rates of twinning in the country, with up to 60/1000 live births documented in some states. Higher-order gestation occurs less frequently, with previously documented incidence rates of 3.37 per thousand live births. Scholars have postulated that this high incidence of twinning in Southwest Nigeria may be attributed to the dietary factors such as the consumption of yam which is a common staple in the region.
Twinning and higher-order gestations are associated with a higher risk of obstetric complications, such as spontaneous abortion, hypertensive disorders, placenta previa, fetal malformations, and preterm labor to mention a few. Single intrauterine fetal demise of one of the twins is one of the possible outcomes of a twin pregnancy and could complicate up to 6% of twin pregnancies. The surviving twin is often at risk of mortality, preterm delivery, and neurodevelopmental abnormalities, more so in monochorionic twins than in dichorionic twins. However, maternal coagulopathy from the retention of the dead twin is seldom reported. Fetus papyraceous occurs as a result of resorption of the amniotic fluid and body fluids of the dead fetus, and compression of the fetus between the membranes of the living twin and the uterine wall, giving it a characteristic “parchment paper” appearance. In the index case the dead fetus was wrapped round in its own membranes, with the whole body fluid resorbed, giving the resemblance of an “Egyptian mummy”. This was as a result of the timing of fetal death, as the fetus was dated at about 19 weeks' GA at initial evaluation. The degree of compression is a function of the time between fetal death and delivery. This is unlike vanishing twin syndrome where the death of a twin occurs much earlier in pregnancy with complete resorption of the dead twin. Researchers have documented fetus papyraceous complicating labor, by delaying or halting fetal descent. The index case was managed conservatively with tocolysis and anxiolysis until the inevitable delivery of the surviving twin. In the absence of contra-indications, vaginal delivery is permissible; however, the decision on mode of delivery should be made on a case-by-case basis.
| Conclusion|| |
This case describes fetus papyraceous, a rare complication of multiple gestation. It also illustrates the precipitous rise in the risk of preterm birth or intrauterine fetal demise in the surviving twin. Students and medical professionals need to be aware of these risks which come with twin or higher-order pregnancies, and the management options when presented with such clinical problems – these include frequent ultrasonographic monitoring and conservative management to prolong pregnancy until fetal lung maturity is ascertained. These would help improve feto-maternal outcomes. This case also contributes clinical images to the journal's database on a rare complication of twin pregnancy.
Declaration of patient consent
The authors certify that they have obtained appropriate patient consent documentation. Accordingly, the mother of the twin pregnancy gave consent for the report, the associated images, and other clinical information to be published in the journal. The consenting parent understands that any other identifying information will not be published and due efforts will be made to protect privacy, but anonymity cannot be guaranteed.
Financial support and sponsorship
No financial support was received for this report.
Conflicts of interest
There are no conflicts of interest.
Ethical conduct of research
The authors of this manuscript declare that this scientific work complies with reporting quality, formatting, and reproducibility guidelines set forth by the EQUATOR Network (in this case, CARE guideline). The authors also attest that this clinical investigation was determined to not require Institutional Review Board/Ethics Committee review, and the corresponding protocol/approval number is not applicable.
| References|| |
Smits J, Monden C. Twinning across the developing world. PLoS One 2011;6:e25239.
Livnat EJ, Burd L, Cadkin A, Keh P, Ward AB. Fetus papyraceus in twin pregnancy. Obstet Gynecol 1978;51:41s-5s.
Akinboro A, Azeez MA, Bakare AA. Frequency of twinning in southwest Nigeria. Indian J Hum Genet 2008;14:41-7.
] [Full text]
Fajolu IB, Ezeaka VC, Adeniyi OF, Iroha EO, Egri-Okwaji MT. Prevalence and outcome of higher order multiple pregnancies in Lagos, Nigeria. J Matern Fetal Neonatal Med 2013;26:1342-5.
Nylander PP. The twinning incidence of Nigeria. Acta Genet Med Gemellol (Roma) 1979;28:261-3.
Hillman SC, Morris RK, Kilby MD. Co-twin prognosis after single fetal death: A systematic review and meta-analysis. Obstet Gynecol 2011;118:928-40.
Mackie FL, Rigby A, Morris RK, Kilby MD. Prognosis of the co-twin following spontaneous single intrauterine fetal death in twin pregnancies: A systematic review and meta-analysis. BJOG 2019;126:569-78.
Yaman Tunç S, Ağaçayak E, Yaman Görük N, İçen MS, Fındık FM, Evsen MS, et al.
Single intrauterine demise in twin pregnancies: Analysis of 29 cases. Turk J Obstet Gynecol 2015;12:226-9.
Benirschke K. Intrauterine death of a twin: Mechanisms, implications for surviving twin, and placental pathology. Semin Diagn Pathol 1993;10:222-31.
Matovelo D, Ndaboine E. Fetus papyraceus causing dystocia in a rural setting: A case report. J Med Case Rep 2015;9:178.
[Figure 1], [Figure 2], [Figure 3]