914534

case-report2020
EJO0010.1177/1120672120914534European Journal of OphthalmologyRóżańska-Walędziak et al.

EJO European
Journal of
Ophthalmology
Case Report

European Journal of Ophthalmology

Spontaneous unilateral exophthalmos

1­–3
© The Author(s) 2020
Article reuse guidelines:
after a vaginal delivery sagepub.com/journals-permissions
https://doi.org/10.1177/1120672120914534
DOI: 10.1177/1120672120914534
journals.sagepub.com/home/ejo

Anna Różańska-Walędziak1, Oksana Szewczuk1,

Maciej Walędziak2 and Krzysztof Czajkowski1

Abstract
Introduction: Spontaneous orbital exophthalmos is an extremely rare incident during a vaginal delivery. In most cases,
it is associated with venous malformations and presents spontaneous resolution.
Case description: We report a case of orbital hematoma after vaginal delivery due to a superior ophthalmic vein
rupture. The patient presented proptosis of the right eye and diplopia immediately after the delivery and was diagnosed
with unilateral orbital hematoma. The patient was given conservative treatment with complete resolution of clinical
symptoms 4 weeks after the delivery.
Conclusion: Increased abdominal pressure during a vaginal delivery may lead to a spontaneous orbital hemorrhage.

Keywords
Exophthalmos, vaginal delivery, orbital hematoma
Date received: 14 September 2019; accepted: 3 March 2020

Introduction 10 days treatment with cefuroxime to prevent intrauterine

Spontaneous orbital hemorrhage is a very rare, vision-
threatening event. Non-traumatic orbital bleeding may be
due to vascular malformations, increased intracranial
venous pressure, systemic hypertension, coagulopathies as
well as Valsalva maneuver. As orbital veins do not contain
valves, the increased intraabdominal pressure transmitted
to the orbital veins may result in congestion and rupture.
The most common symptoms of retrobulbar hematoma are
proptosis, ophthalmoplegia, increased intraocular pres-
sure, loss of pupillary reflexes and optic disk, or retinal
pallor. Diagnosis requires computed tomography (CT) or
magnetic resonance imaging (MRI). The purpose of the
treatment is to reduce intraorbital pressure and protect the
optic nerve from possible damage, which can be obtained
by surgical means or medical conservative treatment.
Case description
A 33-year-old multigravida was admitted to a perinatology
clinic in the 32nd week of gestation after preterm prema-
ture rupture of membranes (pPROM). She presented no
contractions and the blood results did not show any signs
of inflammation. Because of pPROM, the patient received
infection and subcutaneous 0.04 mg enoxaparin due to bed
regime. Throughout the hospitalization, the cardiotocogra-
phy and ultrasonography results showed no abnormalities,
apart from decreased amniotic fluid levels as a result of
pPROM.
The patient had history of one vaginal birth in the 32nd
week of gestation, after pPROM 4 years earlier and cervi-
cal carcinoma in situ treated with cervical amputation
5 years earlier. She had myopia of −5 D in both eyes and
used correction glasses. She had no history of orbital
hemorrhage or exophthalmos. In the 19th week of gesta-
tion, the patient had a preventive cervical pessarium intro-
duced without complications. She received intravaginal
progesterone and in the 29th week of gestation two doses
1
2 nd Department of Obstetrics and Gynecology, Medical University of
Warsaw, Warsaw, Poland
2
Department of General, Oncological, Metabolic and Thoracic Surgery,
Military Institute of Medicine, Warsaw, Poland
Corresponding author:
Maciej Walędziak, Department of General, Oncological, Metabolic and
Thoracic Surgery, Military Institute of Medicine, Szaserów 128 Street,
00-141 Warsaw, Poland.
Email: maciej.waledziak@gmail.com
2 European Journal of Ophthalmology 00(0)
of 12 mg of intramuscular dexamethasone as a standard
procedure accelerating lung maturation and improving
neonatal results in case of preterm birth. The levels of
thyroid-stimulating hormone were normal throughout the
pregnancy and the patient had no history of hypertension.
The patient developed gestational diabetes mellitus, with
normal glucose levels when treated with diet.
After 2 weeks of hospitalization, at the 34th week of
gestation, the patient presented uterine contractions and
decelerations in the cardiotocography of the fetus as well as
the C-reactive protein levels increased to 59 mmol/L (with
the norm of 5 mmol/L). As cervical dilation of 4 cm was
stated in the vaginal examination, the pessarium was
removed. The patient received inhalations of nitrous oxide
as analgesic during the first stage of the labor. After 1 h, she
delivered a male infant weighing 2470 g, evaluated with 10
points Apgar. During the contractions, the patient did not
follow the advice of the medical staff and she kept pushing
with excessive strength. The blood pressure of the patient
was normal throughout the labor and after the delivery was
115/60 mm Hg, with heart rate of 80 beats per minute. Due
to incomplete placental detachment, the patient was quali-
fied to instrumental examination of the uterus.
Immediately after the delivery of the baby, the patient
complained of diplopia and pain in the right eyeball.
Unilateral exophthalmos was observed, the right eyeball
protruding from its socket. The pupils were symmetrical,
with normal direct reaction to light. The patient was
administered a neurological and ophthalmological consul-
tation. MRI results showed a hematoma of right orbit
probably due to a rupture of right upper ophthalmic vein.
The size of the hematoma was 24 × 8 × 34 mm, it was situ-
ated in the upper medial part of the orbit along the upper
ophthalmic vein and it caused displacement of the eyeball
and other orbital structures in the lower and parietal direc-
tion. No signs of arterial or venal thrombosis were found.
Magnetic resonance angiography (MRA) showed no
signs of pre-existing vascular malformations in the orbit
on both sides. There were neither lesions in the brain struc-
tures nor loci of contrast enhancement.
Fat reserve around optical nerves was retained, slightly
lesser in the right orbit. The motility of the right eyeball
was reduced in the upper direction. Bilateral diplopia aug-
mented when looking medially and upward. There were no
changes found in the eye fundus bilaterally. Visual acuity
was normal in both eyes. The patient was administered
250 mg of oral etamsylate and 50 mg rutoside trihydrate
three times daily and 500 mg calcium dobesilate twice a
day. Improvement of vision and resolution of clinical signs
(such as proptosis and mass effect) were observed after 2
weeks and an ophthalmologist consultation did not show
any persistent changes in the eye fundus. About 4 weeks
after the delivery the patient achieved complete recovery
with no reduction in visual acuity.
Conclusion
Spontaneous exophthalmos caused by orbital hematoma
is a condition that may lead to a vision loss. The majority
of orbital hemorrhages are associated with vascular mal-
formations. In the presented case, several possible predis-
posing risk factors, enoxaparin therapy, myopia, excessive
strength used by the patient during the contractions may
be considered, although the real background will remain
unsure. Patients with orbital hemorrhage most commonly
complain of pain, sense of pressure, diplopia, or loss of
vision.
The number of cases of spontaneous orbital hemor-
rhage during a vaginal delivery in the literature is scarce
and we found only several cases reported. Sullivan and
Wright1 analyzed 115 cases of patients with a diagnosis of
non-traumatic orbital hemorrhage and found that associ-
ated orbital vascular malformations were present in 90%
of cases. In 4 of 115 cases (3.5%) hemorrhage occurred
during childbirth, with venous anomalies present in three
cases (75%). They observed spontaneous resolution of
hemorrhage in 89% of cases and final reduction of visual
acuity in 20% of cases.
A case of spontaneous exophthalmos during a vaginal
delivery was published by Goldberg2 in the British Medical
Journal in 1948. The patient complained of dimness of
vision of the left eye and protrusion of the eye was noticed
during the second stage of labor. The patient did not
receive any treatment and completely lost vision in the
affected eye. Taking account of the date of the incident, no
imaging was available at the time that would allow to put
an adequate diagnosis and treatment. After 2 days of the
incident, the author observed retinal hemorrhages particu-
larly in the macular region.
Hamouda et al.3 presented a case of spontaneous sub­
periosteal hematoma of the orbit after a vaginal delivery
in 2006, with spontaneous resolution of the hematoma.
Bilateral proptosis and subperiosteal hemorrhages were
also reported after a cesarean section with a subsequent
hysterectomy in a patient with placenta previa by Atalla
et al.4
Some unusual causes may also be risk factors predis-
posing to orbital hemorrhage. A case of spontaneous ret-
robulbar hematoma caused by sneeze was reported by
Deveer et al.5 The patient was treated surgically and reso-
lution of clinical symptoms was obtained. Another case of
subperiosteal hematoma in the orbit due to self-induced
asphyxia was presented by Subiras et al.6 in a patient with
non-accomplished suicide attempt. The Valsalva maneu-
ver, known to cause hemorrhagic retinopathy, subconjunc-
tival hemorrhage, and periorbital petechiae may also lead
to subperiosteal orbital hemorrhage. In the case presented
by Katz and Carmody7 the increased abdominal pressure
was caused by excessive weight lifting. Chang et al.8
Różańska-Walędziak et al. 3
described two cases of pregnant women, who had under-
gone spontaneous orbital hemorrhage during subcutane-
ous heparin treatment respectively in the 12th and 26th
week of pregnancy. Vomiting, including hyperemesis
gravidarum may also lead to orbital hemorrhage, as
reported by Atalla et al.4 Graves thyroid disease may lead
to a potentially sight-threatening ophthalmopathy, rarely
progressive during pregnancy due to immunological
changes altering the course of the autoimmune disorders.9
Progression may appear in the postpartum period, rarely
during the last month of pregnancy, with exacerbation
immediately after the delivery.
In case of diminished vision, diplopia or signs of exoph-
thalmos observed during or after vaginal delivery, immedi-
ate ophthalmologic consultation and MRI should be
administered to speed up diagnosis and treatment. Orbital
imaging is helpful in the assessment of the lesions. Most
bleed are venous and self-limiting with rare necessity of sur-
gical intervention, which should be limited to patients with
optic nerve compromise or a localized persistent lesion.
Regardless of the form of treatment, any physical effort or
heavy lifting should be prohibited for at least 3 months. We
also assume that a patient after an intrapartum or postpartum
orbital hemorrhage should have a cesarean delivery in case
of any future pregnancies.
Consent for publication
We received consent from the patient to publish the case report.
Declaration of conflicting interests
The author(s) declared no potential conflicts of interest with respect
to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research,
authorship, and/or publication of this article.
ORCID iD
Maciej Walędziak https://orcid.org/0000-0003-4311-9995
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