Results
Of the initial 476 identified articles, 23 full text articles were
assessed for eligibility. Eventually, 15 case reports were included in
this review (figure S1) . Baseline patient characteristics,
clinical presentation, mode of diagnosis, pregnancy course and the
maternal and neonatal outcomes in the 15 pregnancies described in these
articles are given in table S1 and S2.
Of the 15 pregnancies, two (2/15, 13.3%) resulted in a stillbirth
possibly due to the uterine hemangioma. The first one occurred at 28
weeks after massive thrombosis of the uterine and placental vessels, the
latter following acute rupture of the uterine hemangioma at 36 weeks.
Another two pregnancies were delivered preterm (2/15, 13.3%). One
patient delivered at 35 weeks after preterm prelabour rupture of the
membranes (PPROM) at 26 weeks, and a second patient had a cesarean
section at 30 weeks due to progressive abdominal discomfort and the
assumed bleeding risk of the hemangioma. Forty percent of women (6/15,
40.0%) went into labour spontaneously, which resulted into a vaginal
delivery in four cases. Most patients were delivered by cesarean section
(10/15, 66.7%), of which seven (7/10, 70%) were unplanned/in an
emergency setting. The postpartum period was complicated by a hemorrhage
in eight women (8/15, 53.3%), which necessitated a hysterectomy in four
cases (4/8, 50%). Two women developed progressive hypovolemic shock
(2/8, 25%). Of those for whom data was provided (5/8, 62.5%), all had
an estimated blood loss of ≥ 1000 mL. Furthermore, another two patients
developed a pulmonary embolism in the postpartum period, ultimately
fatal in one of them. Perinatal outcomes were mentioned in only six
reports (6/15, 40%). Only half of these cases (3/6, 50%) had an
uneventful outcome. One case of respiratory problems due to preterm
birth at 30 weeks was described beyond the two stillbirths mentioned
above.
The hemangioma was diagnosed before delivery in the majority of cases
(10/15, 66.7%), generally during the second trimester of pregnancy.
Most often, symptoms such as abdominal discomfort, dyspnea, or vaginal
bleeding and/or the finding of an enlarged uterus led to a work-up. Four
patients were referred for abnormal ultrasound findings, which in all
but one case were suspicious for a partial mole. The antenatal diagnosis
was generally based on ultrasound findings, MRI was used to confirm the
hemangioma in only one patient.
Half of the patients (5/10, 50%) with an established diagnosis of
hemangioma before delivery had a non-planned cesarean section. In two of
these patients (2/5, 40%), this was for reasons related to the
hemangioma: one patient had a cesarean section for fetal death at 28
weeks and another patient was delivered at 30 weeks due to progressive
abdominal discomfort as mentioned earlier. Five of the 10 antenatal
diagnosed patients (5/10, 50%) developed a postpartum hemorrhage, which
necessitated a hysterectomy in only one of them. Among the patients with
an antenatal diagnosis of a uterine hemangioma, one more patient
underwent a hysterectomy at 17 weeks because of recurrent syncopes and
the presumed risk of uterine rupture.
These numbers contrast with the undiagnosed group in which three out of
the five patients (3/5, 60%) had a postpartum hemorrhage and a
hysterectomy was necessary in all of them (3/3, 100%). There were no
cases of maternal mortality in the group of antenatal diagnosed
patients.