Umbilical Cord Hematoma: A Case Report and Review of the Literature.
1. IntroductionAlthough very rare, umbilical cord hematoma (UCH) is a real serious complication of pregnancy. It represents a rare cause of acute fetal distress that may be shown by the decrease of fetal movement or fetal death [1].
Lately, a case of UCH resulted in perinatal death at our department stimulating our interest in performing this review of the literature, emphasizing the research on pathogenesis, diagnosis, and management for UCH.
The study by Dipple et al., with 36 cases, is the largest series published on this topic so far. It estimates an incidence rate of 1 in 5505. Although umbilical cord complications may be the second most common cause of stillbirth [2], umbilical cord hematoma has been reported as a rare cause for stillbirth and fetal distress; the overall perinatal loss rate was approximately 50%, and the incidence of this disorder in live births would then be approximately one in 11,000 pregnancies.
Our review of the English literature resulted in 9 publications of 11 cases of UCH in the years 2008-2017 [3-11]. Of the 11 cases of spontaneous UCH reported in the 9 studies published in the last 10 years, 2 were stillbirths, 1 occurring antenatally and 1 on day 6 of life [6, 8]. Of the nine live born cases, 7 presented at term of gestational age, 2 preterm [3-5, 7, 9-11].
2. Materials and Methods
A review of the literature was conducted in order to identify the case reports reported in the English language. We searched PubMed MEDLINE electronic database published between 2008 and 2017 on https://www.ncbi.nlm.nih. gov/pubmed. The keywords used were as follows: "Umbilical," "Cord," and "Haematoma." Different combinations of the terms were used. Moreover, references in each article were searched to identify potentially missed studies. We chose 2008 as a starting year point for our literature search because this year was marked by a review of the literature by Gualandri et al. [12] for the years 1958 to 2008. From the authors' descriptions of individual case reports, we took available and reliable information about the possible predisposing factors, clinical presentation, diagnosis, and management (Table 1). Because of the lack of uniformity in the cases reviewed, we have not made any calculations with statistical significance.
3. Results
3.1. Pathogenesis. The exact etiology of UCH still remains unexplained. Many theories have been proposed but without final results. Probably a combination of different factors leads to UCH.
Risk factors for spontaneous umbilical cord hematoma are various. They include morphologic anomalies of the umbilical cord (both in length and in thickness), true knots, cord prolapse, traction or torsion, velamentous insertion of the cord, vessel wall abnormalities, umbilical cord cysts, abdominal trauma in pregnancy, postterm pregnancy, infections (chorioamnionitis and funisitis), deficiency of Wharton's jelly, congenital defects, and many more remain unexplained [13]. Fetal hypoxia and anemia may occur due to the compression of the umbilical vessels leading to perinatal asphyxia and stillbirth. Iatrogenic causes secondary to amniocentesis, in utero transfusions and diagnostic cordocentesis are also reported [14].
Spontaneous bleeding in the umbilical cord is due to a disruption of the vessel wall through which, in most cases, an extravasation of blood into Wharton's jelly occurs. [15]. A high intravascular pressure could be implicated in its formation. The hematoma can compromise the maternal-fetal circulation by compressing the vessels (umbilical arteries and vein) with subsequent fetal hypoxia or by the blood loss within the cord itself with anemia, leading to perinatal asphyxia and stillbirth [1].
In our analysis of 11 cases, 2 cases showed evidence of chorioamnionitis [7, 8], 1 case revealed single umbilical artery and marginal cord insertion with spontaneous avulsion of the umbilical artery [5], 1 case was secondary to composite heterozygous congenital factor VII deficiency, and in 7 cases no pathological condition was reported [3, 4, 9-11].
3.2. Diagnosis. The diagnosis is usually made postnatally, but in some cases it can be made by Doppler ultrasound scan prenatally, assessing the cord and the blood flow in the umbilical vessels [11, 12]. Cord hematomas can arise during pregnancy which can lead to fetal death [8], or may occur, more frequent, during labour giving rise to fetal distress and requiring immediate fetal delivery. Of the nine live born cases discussed in this review, 7 presented at term of gestational age, 2 preterm [3-5, 7, 9-11]; moreover, 7 cases presented with a complaint of decreased fetal movement [3-6, 8]. Abnormal fetal heart monitor tracing has been described in 6 cases [3-6]. In 3 cases, there were no changes in fetal movements or abnormalities in fetal heart rate. Stillbirth that occurs in the antenatal period is more difficult to explain than that occurring intrapartum since it is difficult to ascertain a cause of antenatal stillbirth [8].
Detailed physical examination of the placenta and cord confirmed the presence of the hematoma in all 11 cases described. During macroscopic examination, umbilical cord may have abnormal appearance with dark red discoloration and markedly increased thickness [10]. It may have a darkish bulge and a bluish discoloration [9]. Hystopathological examination of the placenta and cord confirmed the presence of hematoma and showed evidence of chorioamnionitis in two cases [7, 8]. The hystological examination of the tract of cord affected by the hematoma may show perivascular hemorrhagic infiltration, umbilical vessels compressed by the hemorrhagic effusion, fissures of the venous wall, alterations of the intima and middle tunica with the vessels wall markedly thinned by the reduction of the muscular component and also moderate inflammatory leukocytic infiltration of the umbilical vascular walls [12].
Autopsy plays an important role in investigating the cause of stillbirth that occurs in the antenatal period.
3.3. Case Report. A 29-year-old multipara woman, with an uncomplicated pregnancy, presented at 41 weeks and 3 days of gestation for elective labour induction. The patient showed Grade 1 obesity (BMI of 30kg/[m.sup.2]).
Labour was induced with a controlled-release hydrogel pessary containing 10 mg prostaglandin E2. The patient was placed on continuous fetal heart rate (FHR) monitoring. After 24 hours from the labour induction, the Bishop score was unchanged and the vaginal insert removed. 3 hours later, the induction continued with intravenous injection of oxytocin 10 UI. After approximately 1 hour, spontaneous rupture of membranes with amniotic clear fluid was observed.
8 hours after induction patient delivered a hypotonic, with no evidence of cardiac activity, male newborn. FHR corresponded to type 1 and 0 of Piquard criteria during the second stage of the labour.
Venous pH at birth was 7.11 (base excess, 16.2 mMol/L), and arterious pH was 6.96 (base excess, 14.2 mMol/L). Apgar score at 1 minute was 0. After 40 minutes of continuous resuscitation, the fetus was still asystolic, and it was therefore decided to stop the resuscitation efforts.
The gross examination of the placenta and of the umbilical cord revealed the presence of blackish-reddish material in the proximity of the placental insertion measuring approximately 3 cm. The umbilical cord presented vascular ectasia at 18 cm from the placental insertion. An hematoma of the cord was noted at 34 cm from the placental insertion; the hematoma was described as an infiltrate of 2 cm, in the tones of black and red, extended to the whole umbilical cord section.
The histological examination of the cord highlighted oedema of Wharton's jelly, circumscribed hematic infiltrates, marked venous ectasia with delamination and hematic infiltration of the venous walls, extensive hemorrhage of Wharton's jelly within the whole portion of the cord. The lumen of the vein was completely occluded by coagulated hematic material.
The histological examination of the placenta highlighted intense vascular congestion of villi and hematic infiltrates as for intervillous hematomas.
Measurements of crown heel, crown rump, head circumference, foot length and weight indicated a regular intrauterine development.
The internal gross examination and the hystopathological examination of lung tissue revealed elements indicating physiological respiration in presence of FHR. The above pattern confirmed that the fetus started the respiratory activity after being delivered before dying.
4. Discussion
The umbilical cord is called the fetal life line, and it is the vital link between the fetus and placenta. Various abnormalities are observed in the morphology and pathology of the umbilical cord but knowledge of them is quite poor.
A considerable number of stillbirths that are thought to be unexplained may be attributable to placental or cord pathologies. UCH can compromise the maternal-fetal circulation by compressing the vessels or because of the blood loss within the cord itself, leading to perinatal asphyxia and stillbirth [1].
Stillbirth can occur either antenatally or perinatally, but sometimes UCH is uneventful. In our case, the stillbirth was peripartum; the results from external inspection, hystopathological examination, and autopsy suggest the manifestation, before death, of a hyperacute asphyctic mechanism. Furthermore, macro- and microscopic analyses of the umbilical cord revealed pathological alterations indicating an acute trauma with compression, vascular laceration, and hemorrhagic infiltration.
This must be due to the occurrence of mechanical compression of the umbilical cord during labour, with acute interruption of the fetoplacental circulation. The cause of death is therefore attributable to an intrauterine asphyxia caused by acute mechanical compression of the umbilical cord, difficult to detect antenatally.
5. Conclusions
Cord accident (compromised umbilical blood flow) as a cause of stillbirth is underreported, mainly due to a lack of diagnostic criteria.
A complete fetopathological examination can state causality between hematoma and stillbirth, exclude another fetal or placental cause of death and consequently reassure the parents for the prognosis of another pregnancy. The issue of hematoma-related complications is also important because of its medicolegal aspect since litigation may occur. Timing of delivery should be based on gestational week as long as the fetus shows well-being signs. Preterm or urgent delivery should be performed in case of fetal distress or reduced movements.
Because of the rarity of this condition, every new case of UCH should be reported in order to improve the knowledge of predisposing factors, prenatal diagnosis, and clinical management.
https://doi.org/10.1155/2018/2610980
Conflicts of Interest
The authors declare that they have no conflicts of interest.
References
[1] M. Seoud, L. Aboul-Hosn, A. Nassar, A. Khalil, and I. Usta, "Spontaneous umbilical cord hematoma: a rare cause of acute fetal distress," American Journal of Perinatology, vol. 18, no. 2, pp. 99-102, 2001.
[2] L. Nappi, F. Trezza, P. Bufo et al., "Classification of stillbirths is an ongoing dilemma," Journal of Perinatal Medicine, vol. 44, no. 7, pp. 837-843, 2016.
[3] C. V. Towers, C. E. Juratsch, and T. J. Garite, "The fetal heart monitor tracing in pregnancies complicated by a spontaneous umbilical cord hematoma," Journal of Perinatology, vol. 29, no. 7, pp. 517-520, 2009.
[4] A. Barbati, M. G. Cacace, D. Fratini, T. Ceccarelli, F. Capanna, and G. C. Di Renzo, "Umbilical cord haematoma with altered fetal heart rate," Journal of Obstetrics and Gynaecology, vol. 29, no. 2, pp. 150-151, 2009.
[5] A. Kumar, C. Kaplan, S. Mokrian, and P. Ogburn, "Intact newborn survival after spontaneous umbilical cord vascular rupture before labor," Obstetrics & Gynecology, vol. 120, no. 2, pp. 489-490, 2012.
[6] C. Jouannelle, M. Giansily-Blaizot, F. Monpoux, F. Casagrande, M. Poiree, and E. Berard, "Spontaneous umbilical cord haematoma and congenital factor VII deficiency," Haemophilia, vol. 18, no. 1, pp. e24-e25, 2012.
[7] G. Tonni, M. P. Bonasoni, C. De Felice, A. Rossi, and S. Tonni, "Histopathological findings in spontaneous hematoma of the umbilical cord: severe hypoxic-ischemic encephalopathy in a term survived newborn," American Journal of Forensic Medicine and Pathology, vol. 36, no. 4, pp. 254-256, 2015.
[8] A. Abraham, S. Rathore, M. Gupta, and S. J. Benjamin, "Umbilical cord haematoma causing stillbirth-a case report," Journal of Clinical and Diagnostic Research, vol. 9, no. 12, pp. QD01-QD02, 2015.
[9] R. M. McAdams and S. Chabra, "Umbilical cord haematoma and adrenal haemorrhage in a macrosomic neonate with anaemia," BMJ Case Reports, vol. 2016, pp. bcr2015214140, 2016.
[10] K. E. Hooper and P. Sebire, "Spontaneous umbilical cord haematoma," Archives of Disease in Childhood-Fetal and Neonatal Edition, vol. 101, no. 4, p. F332, 2016.
[11] P. K. Arora, S. Mohandas, S. McAndrew, and V. Karody, "Spontaneous umbilical cord hematoma," Journal of Pediatrics, vol. 184, pp. 233-233.e1, 2017.
[12] G. Gualandri, F. Rivasi, A. L. Santunione, and E. Silingardi, "Spontaneous umbilical cord hematoma: an unusual cause of fetal mortality: a report of 3 cases and review of the literature," American Journal of Forensic Medicine and Pathology, vol. 29, no. 2, pp. 185-190, 2008.
[13] D. Feldberg, M. Ben-David, D. Dicker, N. Samuel, and J. Goldman, "Hematoma of the umbilical cord with acute antepartum fetal distress. A case report," Journal of Reproductive Medicine, vol. 31, no. 1, pp. 65-66, 1986.
[14] E. Chenard, A. Bastide, and W. D. Fraser, "Umbilical cord hematoma following diagnostic funipuncture," Obstetrics & Gynecology, vol. 76, no. 5, pp. 994-996, 1990.
[15] A. Dippel, "Hematomas of the umbilical cord," Surgery, Gynecology & Obstetrics, vol. 70, pp. 51-57, 1940.
Gennaro Scutiero, (1) Bernardi Giulia, (1) Piergiorgio Iannone, (1) Luigi Nappi, (2) Danila Morano, (1) and Pantaleo Greco (1)
(1) Department of Morphology, Surgery and Experimental Medicine, Section of Obstetrics and Gynecology, Azienda Ospedaliero-Universitaria S. Anna, University of Ferrara, Via Aldo Moro 8, 44121 Cona, Ferrara, Italy
(2) Department of Medical and Surgical Sciences, Institute of Obstetrics and Gynecology, University of Foggia, Viale L. Pinto, 71100 Foggia, Italy
Correspondence should be addressed to Piergiorgio Iannone; pg.iannone88@gmail.com
Received 17 December 2017; Accepted 21 February 2018; Published 26 March 2018
Academic Editor: John J. Moore
TABLE 1: Case reports described in literature.
Authors Age/parity Gestational Antenatal course
age (wk)
Towers 23/1 31 Decreased fetal
et al. [3] movements for 18
hours
22/0 40 Decreased fetal
movements for 30
hours
39/1 38 Absent fetal
movements for 14
hours
Barbati 44/1 40 Reduction of
et al. [4] fetal movements,
FHR with severe
reduced
variability of
<5 bpm and late
decelerations
Kumar 31/1 36 Two vessel-cord,
et al. [5] right pelvic
kidney, decreased
fetal movement
for 12 hours
Jouannelle Not given At term Decreased fetal
et al. [6] movements, fetal
heart
decelerations
Tonni 19/0 At term Loss of fetal
et al. [7] heart lasting
90 seconds, at
birth
Abraham 27/multipara 35 Decreased fetal
et al. [8] movements
(ultrasound scan
confirmed fetal
death)
McAdams 32/1 At term Uneventful
and
Chabra [9]
Hooper Not given At term Uneventful
and Sebire
[10]
Arora Not given 39 Uneventful
et al. [11]
Authors Mode of Macroscopical
delivery lesion
Towers Cesarean Umbilical cord
et al. [3] hematoma 3 x 2 cm
Cesarean Umbilical cord
hematoma 4 x 2 cm
Cesarean Umbilical cord
hematoma 3 x 2 cm
Barbati Cesarean Large cord
et al. [4] hematoma
(5 x 3.7 x 2.6)
at 3 cm from the
fetal insertion
Kumar Cesarean Marginal
et al. [5] umbilical cord
insertion,
avulsed umbilical
artery rupture:
single artery is
shown to be
ruptured at the
site of cord
insertion to the
placenta.
Jouannelle Cesarean Massive umbilical
et al. [6] cord hematoma at
the skin
junction, with
cord compression
Tonni Vaginal Fresh hematoma in
et al. [7] the cord
Abraham Vaginal Central cord
et al. [8] insertion.
Umbilical cord
had 4-5 sausage
shaped swellings
suggestive of
cord hematoma of
varying sizes all
along the length
of the cord with
the largest
measuring 6 x 3
cm
McAdams Vaginal Umbilical cord
and hematoma
Chabra [9]
Hooper Vaginal Umbilical cord
and Sebire proximal to the
[10] baby has dark red
discoloration and
increased
thickness,
measuring 4.5 in
diameter at the
widest part.
Arora Vaginal A 4 cm and 2 cm
et al. [11] wide reddish
purple, nontender
swelling in the
cord proximal to
the level of the
skin
Authors Hystopathological Fetal outcome
examination
Towers Hematoma None/AS 7 at
et al. [3] associated with 1 min and 9 at
umbilical vein. 5 min
Thrombotic
material was seen
within the vein, but
the vein was not
totally occluded; the
umbilical arteries
were compressed to
the side but patent.
Vein/arterial None/discharged
lumens were well at follow up
compressed but at 18 months/AS 2
both patent. at 1 min, 6 at 5 min
and 8 at 10 min
Vein/umbilical None/AS 8 at 1 min
arteries appeared and 9 at 5 min
patent.
Barbati Two arteries and Tachypnea,
et al. [4] one vein with no cyanosis, and
other abnormalities anemia without
in the form of knots any other physical
and loops. or neurological
Extravasation of damage/AS 6 at
blood into the 1 min and 9 at
surrounding 5 min
Wharton's jelly
caused by the
rupture of a dilated
umbilical artery
Kumar Fetal branch artery Tachycardia,
et al. [5] ruptured with tachypnea, and
a vessel wall polyuric acute
significant for mild kidney failure
acute inflammation secondary to
and necrotic muscle cortical-sparing
cells. acute tubular
necrosis;
discharged well at
14 day/AS 3 and 5 at
1 and 5 min
Jouannelle Not given Baby was flat/AS
et al. [6] 0 at 1 min, 3 at
5 min, 7 at 10 min.
Postnatal evolution:
coma, total
hypotonia, no
archaic reflex, and
hypoxic-ischemic
encephalopathy.
The newborn died
of multiorgan
failure on day 6 of
life.
Tonni A rupture in the Sever mixed acidosis,
et al. [7] wall of the fetal anemia, and
umbilical vein with severe HIE/AS 3 at 1,
discontinuity in the 5, and 10 min
layers of the Follow-up at the
subintimal and age of 4 years:
internal elastic spastic tetraplegia,
lamina. One seizures, central
umbilical artery deafness, and
presented blindness
peripheral
dissection,
subintimal myxoid
degeneration, and
widespread disruption
of the elastic
fibers; marked
reduction in
myofibroblasts in
Wharton's jelly.
Amniotic band at
umbilical cord
insertion into the
chorionic plate,
markedly congested
chorionic vessels
with dispersed
distribution and
convoluted
decourse.
The membranes
had diffuse
chorioamnionitis
(E. coli infection).
Abraham Cord had multiple Stillbirth
et al. [8] swellings suggestive
of umbilical cord
hematoma.
Chorioamnionitis
McAdams Not given None
and
Chabra [9]
Hooper Not given None
and Sebire
[10]
Arora Not given None AS 8 and 9 at 1
et al. [11] and 5 min
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