Cavernous hemangioma of the cervix and pregnancy: a case report

doi:10.4236/ojog.2011.14043

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Open Journal of Obstetrics and Gynecology, 2011, 1, 221-224

doi:10.4236/ojog.2011.14043 Published Online December 2011 (http://www.SciRP.org/journal/ojog/

OJOG

Published Online December 2011 in SciRes. http://www.scirp.org/journal/OJOG

Cavernous hemangioma of the cervix and pregnancy:

a case report

Samar Elkhateb1,2*, Mostafa Azzouzi Idrissi2, Kamilia Laabadi1,2, Leila Chbani2,3, Hikmat Chaara1,2,

AbdIlah Melhouf1,2

1Department of Gynaecology and Obstetrics II, University Hospital Hassan II, Sidi Mohamed Ben Abdellah University, Fez, Morocco;

2Sidi Mohamed Ben Abdullah University, Fez, Morocco;

3Department of Pathology, University Hospital Hassan II, Sidi Mohamed Ben Abdullah University, Fez, Morocco.

Email: *[email protected]

Received 5 June 2011; revised 24 July 2011; accepted 10 August 2011.

ABSTRACT

Purpose: Cavernous haemangiomas of the uterine cer-

vix are very rare and usually harmless. It is a benign

tumor that may cause gynecological obstetrical com-

plications. Although it is a benign condition but it can

have serious consequences for the mother as well as

for the baby. The treatment is often surgical and dia-

gnosis is histological. Methods and Result: we repor-

ted a case of cavernous haemangioma of the cervix in

a 25-year old multiparous patient, referred to our

Centre in the 33 - 34 gestational weeks for premature

labor with membranes rupture. A 3 cm × 6 cm soft

regular purplish red mass is found in the uterine cer-

vix. Tocolosys, prophylactic antibiotic therapy and

corticotherapy for fetus pulmonary maturation were

then decided after elimination of chorioamniotite si-

gns. Caesarean has been made during labor, a week

later, for tumor previa. The tumor excision has been

made through the vagina after fetus extraction. The

histological examination confirmed diagnosis of the

cavernous haemangioma. Discussion: Further to this

observation, the clinical, histological and therapeutic

characteristics of the cavernous haemangioma unu-

sual presentation were drawn up and literature enri-

ched since this pathology is rare. Most previous repor-

ts in the literature consist of single-case histories, and

the experience of individual institutions is limited. The

patients dramatically present dilemmas in the disease

management. This can result in uncontrolled bleeding

especially during operative delivery and may require

hysterectomy. To avoid unexpected bleeding from hae-

mangioma, patient s should be repeatedly examined for

haemangioma of the birth canal, and special care shou-

ld be taken in choosing the delivery mode.

Keywords: Cavernous Haemangioma; Uterine Cervix;

Pregnancy; Tocolytic Therapy

1. INTRODUCTION

Cavernous haemangioma of uterine cervix is a rare vas-

cular malformation. It is a benign pathology. Its seat is

primarily visceral and generally hepatic [1,2-4]. This

cervical lesion may cause vaginal metrorrhagia or pains

[1,2-9]. It seems that the tumor can vary. The different-

tial diagnosis includes a cervical malignant tumor. The

anatomopathologic study proves the benign and vascular

nature of the tumor [10]. The treatment is generally sur-

gical [1,2]. Many gynecological and/or obstetrical com-

plications are often associated. The gynecological com-

plications are inter-menstrual spotting, menometrorrha-

gia, post-menopause metrorrhagia, post coitus metrorr-

hagia, infertility and dyspareunia [1-2,7-9]. The obstet-

rical complications are the premature rupture of mem-

branes, the fetal death in the uterus, the postpartum hae-

morrhage and disseminated intravascular coagulation

[3-5,11-13]. The dilemma that may arise is the delivery

way in a pregnant mother having a cervical cavernous

haemangioma [3-17]. In this report, we describe a hae-

mangioma of the uterine cervix in a 25-year old multi-

parous patient, who presented premature labor with me-

mbranes rupture in the 33 - 34 gestational weeks. Caesa-

rean has been made during labor, a week later, for tumor

previa. The tumor excision has been made through the

vagina after fetus extraction. The histological study con-

firmed diagnosis. Apparently, the reported case empha-

sizes the importance of special care in the choice of the

delivery mode. This case is presented in order to increa-

se awareness on the existence of Cavernous haeman-

gioma of the uterine cervix during pregnancy, which

may cause life-threatening bleeding at the delivery time

and obstetrical complications.

S. Elkhateb et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 221-224

222

2. CASE REPORT

Mrs. S.Z, 25 year-old, multiparous, G6 P2 1EV with two

late abortions, referred to our Centre for premature rup-

ture of the membranes at eight-month pregnancy. The

clinical examination objectivized a 3 cm × 6 cm soft re-

gular purplish red mass, overhanging through the vulva.

At the vaginal touch, this formation expands to the cer-

vix anterior lip. At the obstetrical examination, several

parameters are found: The fundal height at 26 cm, Fetal

Heart Beat, beginning uterine contractions associated to

modifications in the uterine cervix and Bishop scores

less than or equal to 4. An obstetrical ultrasonography e-

valuated the pregnancy at the 34th week of amenorrhea

with the fetus weight estimated at 2508 grams. The ma-

turity signs are absent and placenta is localized on the

anterior fundic wall. Nifedipine tocolysis, prophylactic

antibiotic therapy and antenatal corticosteroids for fetal

lung maturation were then decided after elimination of

Chorioamniotic signs. One week later, the patient ente-

red in the active phase of labor. A caesarean was decided

because of the previa obstacle. The new born child is a

female premature, at 33 weeks - 34 weeks of amenorrhea,

Apgar score at 8/10, with a birth weight of 2350 g. After

fetus extraction, the tumor excision, at the edge of the

cervix, through the vagina was carried out (Figur e 1).

The anatomopathological study of the piece revealed

the cavernous haemangioma. The Histological analysis

detects a benign tumor growth, without normal cervical

stroma, made up from vascular grooves, of various sizes,

full of blood. The walls of these grooves are very fine,

composed of regular and flattened endothelium, and re-

sting directly on neighboring fibrous tissue (Figure 2).

Figure 1. Soft regular purplish-red mass overhanging through

the vulva.

Figure 2. Fibrous tissue containing large cavities filled with

blood and lined by regular endothelial cells.

3. DISCUSSION

The uterine cervix cavernous haemangioma is a vascular

benign tumor growth primarily made up of superficial

and/or deep dilated large blood-vessels. It can touch

many internal organs such as the liver, the spleen, the

pancreas, the gastrointestinal tract, the skin, the uterus

and sometimes the brain. The cervical localization is rare

[1-12]. In Table 1, we present 15 cases found in the lite-

rature. 5 cases are associated with pregnancy besides our

case (Table 1).

Most cases in these series presented post-coitus met-

rorrhagias that were regarded as endocervical polyp in

the clinical examination. These lesions were suspected to

be malignant tumors at vaginal touch. Although the ute-

rine cervix haemangioma is generally asymptomatic [7],

35% of the reported cases were associated to vaginal

bleedings (menometrorrhagia, post-coitus metrorrhagia)

[8,18-20]. Changes during pregnancy under hormonal

influence can present a risk of obstetrical complications

[8-13]. A quick growth of the lesion, during pregnancy,

may generate premature rupture of membranes, prema-

ture Labor, fetal death in-utero, postpartum haemorrhage

and disseminated intravascular coagulation [20]. These

complications require delivery by Caesarean [13-17].

Surgical excision is curative in most of cases. Because

the haemangioma is a benign vascular lesion with a high

hemorrhagic risk, conservative therapies have been sug-

gested such as the use of sclerosing agents, cryotherapy,

and CO2 laser excision (carbon dioxide laser) [8]. The

cervical haemangioma that especially presents serious

complications, can be treated by local excision, conisa-

tion, or through ablation by laser [9-10]. These therapeu-

tic methods permit to preserve fertility in the young

S. Elkhateb et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 221-224 223

Table 1. Review of various cases of the uterine cervix cavernous haemangioma in the literature.

Ahern [18] 1978 Patient found to have cervical haemangioma on ultrasound, managed in a conservative way.

Jackson [12] 1993 Cervical haemangioma is diagnosed in an asymptomatic multiparous woman or during pregnancy,

expectant management is warranted.

Petry [11] 1994 31-year old multiparous woman in the 34 + 1 gestational week, who presented herself with a fast growing

cervical haemangioma, indicates that this benign tumor may cause obstetrical complications.

Padmanabhan [19] 2001 Oral Contraceptive Pills related cervical haemangioma and focal nodular hyperplasia of the liver.

Riggs [20] 2003 Patient presented intractable bleeding after Medical termination of pregnancy. Required hysterectomy

on Histopathology-cavernous haemangioma.

Kondi-Pafiti [10] 2003 9 unusual vascular tumors of the female genital tract and to investigate the problems in the differential diagnosis from

other genital lesions. A capillary haemangioma of the endometrium, one capillary and one cavernous haemangioma

of the cervix, 6 vulva lesions (one capillary, and one cavernous haemangioma and 4 angiokeratomas) and one

haemangiosarcoma of the mons pubis in a 76-year-old patient were diagnosed. The differential diagnosis of the

vascular tumors must be made mainly from endometriotic lesions and melanomas. Immunohistochemistry

(S-100, CD31, CD34, CKS, EMA) aid in the Diagnosis. Local excision is appropriate for the benign vascular lesions.

Baxi [7] 2005 Haemangioma of the cervix was found in a 60-year-old female patient with prolapse of uterus. The haemangioma

was incidentally detected during routine histopathological examination of the organ removed by surgery.

Wang [9] 2005 Examination of a 39-year-old patient presenting pelvic pain revealed a noticeable pelvic mass consisting of a 10 c

tumor extending from the cervix to the retro peritoneum. The pathology report demonstrated spindle cell

haemangioma. This is a rare case of spindle cell haemangioma of the cervix after transvaginal resection.

Ozyer [8] 2006 Reported a case of cavernous haemangioma of the cervix in a 53-year-old patient complaining about

postcoital spotting.

Tanaka [17] 2007 The Patient has BRBNS, haemangioma of the uterine cervix appeared during pregnancy.

Caesarean section was performed without complications, One month after delivery,

cervical haemangiomas were found to have decreased in size.

El Khateb 2008 25-year old multiparous patient in the 33 - 34 gestational week for premature labor with membranes rupture. A 3 cm

× 6 cm soft regular purplish red mass is found in the uterine cervix. Caesarean has been made for

tumor previa interference. The tumor excision has been made through the vagina after caesarean.

Reggiani Bonetti [6] 2009 3 cases of haemangioma of the cervix in asymptomatic women, diagnosed as cavernous haemangioma. All

tumors were immunoreactive for CD31, CD34, and factor-VIII-related antigen. Focal expression of

estrogen receptors was detected. No positivity was obtained with progesterone receptor antibodies.

The presence of estrogen receptor in the endothelial cells of the haemangioma of the cervix suggests

a direct role of this hormone in the haemangioma development. A possible target therapy is discussed.

female patients [1-5]. The surgical excision has always

been essential to eliminate a subjacent malignant tumor,

including malignant vascular lesions [10]. In gyneco-

logical practice, the embolization treatment for arterio-

venous malformations was crowned with success in pa-

tients presenting serious menorrhagia with relief of sym-

ptoms and preservation of their capacity of reproduc-

tion [14]. However, the cervical haemangioma can be

treated, if necessary, by hysterectomy [15,18,20]. Spon-

taneous regression of the lesion has been reported by

Tanaka et al. [17].

The uterine cervix cavernous haemangioma is a rare

lesion given its localization. The diagnosis of this benign

tumor is histological. It is purely morphological and

does not require any complementary technique. As in the

case of other localizations, lesions are large-sized, less

marked-out and often invading the underlying structures.

They are inclined not to regress and require thus a sur-

gical treatment to avoid bleedings, which are often fre-

quent and sometimes important. In Histology, they are

composed of dilated vessels, full of blood and lined with

regular endothelial cells, without atypical mitoses. The

vascular walls are sometimes thickened with adventitial

fibrosis. Calcifications may also be present. The differ-

rential histological diagnosis can be made if there is a

benign lesion having a vascular component of reactional

nature like the pyogenic or botryomycosis granuloma.

However, the lesion polyploidy aspect, its lobule well

marked-out character and the presence of abundant in-

flammatory infiltrate plead in favor of the reactional

nature of the lesion [10-16].

The cervical cavernous haemangioma can also be

found in particular cases of generalized vascular mal-

formations such as Blue rubber bleb nevus syndrome

(BRBNS), which is a rare disease showing venous mal-

formations on the skin, on the gastro-intestinal tract and

S. Elkhateb et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 221-224

224

on other internal organs. Norifumi Tanaka reported the

case of a patient who presented the BRBNS disease with

the outbreak of several haemangiomas on the uterine

cervix during pregnancy. A Caesarean was carried out.

One month postpartum reduction in the size of the le-

sions was observed. The lesion, induced by pregnancy,

increased during pregnancy and regressed after delivery

[17].

Padmanabhan et al. reported the rare association of ca-

vernous haemangioma of the cervix with the focal no-

dular hyperplasia of the liver in a patient using oral con-

traceptives [19].

Reggiani L et al. present three cases of cervix hae-

mangioma in asymptomatic women, diagnosed as ca-

vernous haemangioma in two cases and capillary hae-

mangioma in one. All tumors were immune-reactive to

CD31, CD34, and factor-VIII-related antigen. Focal ex-

pression of estrogen receptors was detected. No positivi-

ty was obtained with progesterone receptor antibodies.

The presence of estrogen receptor in the cervix haeman-

gioma endothelial cells suggests a direct role of this hor-

mone in haemangioma development. A possible target

therapy is discussed [6].

4. CONCLUSIONS

The uterine cervix cavernous haemangioma is a very

rare pathology in pregnant women. This benign vascular

lesion may cause gynecological-obstetrical complica-

tions. It is recommended to perform a Caesarean. Some-

times, the consequences could also be serious. Although,

delivery by natural ways is possible but Caesarean is

recommended. The differential diagnosis is very difficult

because it is a rare lesion. Uterine atony or disseminated

intravascular coagulation can be observed, likely to lead

to hysterectomy. The final diagnosis can only be made

via the histological study.

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