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Umbilical Cord Hematoma: A Case Report and Review of the Literature.

1. Introduction

Although 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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Author:Scutiero, Gennaro; Giulia, Bernardi; Iannone, Piergiorgio; Nappi, Luigi; Morano, Danila; Greco, Pant
Publication:Obstetrics and Gynecology International
Article Type:Report
Date:Jan 1, 2018
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