Dermoid cyst (mature cystic teratoma) is a prevalent
ovarian neoplasia which might become complicated
by torsion, rupture, or dysplasia, whereas
super infection is a rare complication of these cysts
An infertile couple of afghan origin with low socio-economic and educational state came to our center to treat their 10 years primary infertility. The primary cause of infertility was determined to be azoospermia of the 28 years old male partner, and the couple was a good candidate for surgical sperm retrieval (testicular sperm extraction/TESE) and intracytoplasmic sperm injection (ICSI).
In preliminary studies before ICSI, the 26 years old female partner reported regular menstruation intervals, but she experienced secondary dysmenorrhea and deep dyspareunia for about a year.
She also gave a history of unspecified manipulation of uterine cavity by a midwife in Afghanistan to treat her infertility before the onset of these findings. She also complained of intermittent pelvic pain and nocturnal fever without chills starting after this manipulation. In examination of pelvic area, a mobile cystic formation of about 10 cm in diameter was palpable in right adnexal region. Transvaginal ultrasound examination showed a cystic formation with a size of 101×42 mm, including hyper-echoic solid components and a mural nodule, in the right ovary, while color Doppler examination revealed a pulsatility index (PI) of higher than one, so both findings confirmed a benign ovarian teratoma.
In hysterosalpingography, uterine cavity was normal and both tubes were patent, but the evidence of an adnexal mass with three complete teeth was observed. All tumor markers were within the normal limits [carbohydrate antigen19-9 (CA19-9)=17.1, carbohydrate antigen 125 (CA125)=18.7, carcino embryonic antigen (CEA)=2.4, and alpha-1-fetoprotein (α FP)=0.2] and other laboratory findings were all normal, as well [white blood cells (WBCs)=6200, erythrocyte sedimentation rate (ESR)=2, C- reactive protein (CRP+1), purified protein derivative (PPD)=negative, and chest X-ray (CXR)=normal].
Patient underwent laparoscopy with a diagnosis of dermoid cyst. During laparoscopic exam pelvic cavity, uterus and left ovary had normal view, and both tubes were patent. Right ovary contained a big cyst with a 5×10 cm diameter without any adhesion to surrounding tissues.
Grossly, the cyst contained abundant adipose tissue,
osteoid material and four well-formed teeth.
Laparoscopic cystectomy was performed, and the
remaining ovarian tissue was preserved. The mass
was sent for histopathologic evaluation. Microscopic
sections showed ovarian cyst with ciliated
stratified epithelium and foci of squamous lining.
Components such as mature osseous and chondroid
tissue were indentified. Some foci of acute
and chronic inflammatory cells infiltration and
actinomyces like organisms were also noted. The
histopathologic findings suggested the presence of
mature cystic teratoma with actinomycotic abscess
A Section of ovarian mature cystic teratoma showing fibro ossified tissue containing sulfur granule of actinomyces [using hematoxylin and eosin stain (H&E)].
Methinamine silver staining of actinomycosis sulfur granule.
Patient was treated for three months with amoxicillin 500 mg/TDS and doxycycline100 mg/BID after consultation with an infectious disease specialist. After the treatment period, patient’s fever and pelvic pain stopped. At this time, patient had serum follicle-stimulating hormone (FSH)=8.9 and serum anti-mullerian hormone (AMH)=1.4 and in ultrasonographic exam both ovaries had normal view.
ICSI was performed following a standard long protocol using gonadotropin-releasing hormone (GnRH) agonist test (Superfact, Buserelin, Aventis, Germany) and a combination of Fostimon and Merional (IBSA, Switzerland). Thirty six hours after injection of 10000 IU human chorionic gonadotropin (hCG), three oocytes were obtained from the left ovary. The right ovary remained inactive during the controlled ovarian hyperstimulation (COH) and no oocyte was retrieved from it. In total, two good quality embryos were formed and were transferred using transcervical technique. Serum βhCG level remained negative two weeks after embryo transfer.
Mature ovarian cystic teratoma or dermoid cyst
is the most prevalent ovarian germ cell tumor comprising about 20% of all ovarian neoplasms.
Dermoid cyst might become complicated by torsion,
rupture or develop to a malignant tumor, but
infection is a rare complication accruing in only
1% of mature ovarian cystic teratomas (
Luk et al. reported a case of dermoid cyst infection
following a dilation and curettage (D&C) procedure
with signs of tubo-ovarian abscess (
In a report of 11 cases of pelvic actinomycosis,
Marret et al. (
However, it should be considered that rare cases
of pelvic actinomycosis without a history of IUD
usage have been reported (
Our patient did not have any history of IUD usage or previous fertility. It is possible that manipulation of uterus in a non standard setting and using non sterile equipment in this patient had caused the transfer of germs into the uterus cavity and secondary infection of ovarian dermoid cyst.
Diagnosis of pelvic actinomycosis is hard, and
the infection is mostly recognized during the surgery
or in histopathological studies (
Our case shows that pelvic actinomycosis should be considered in women without a history of previous fertility or IUD usage even if the signs and symptoms are unspecific; especially the patient comes from a low level socio economic background. The presence of unusual findings like night fevers, chills, intermittent abdominal pain, and nonspecific digestive system signs can lead to consider a possible super infection of benign pelvic masses like dermoid cysts by the physician.
Our case and other cases reported in the literature indicate the importance of sending the samples of suspicious tissue during surgery for microbiological examination in parallel with cytopathologic examination, especially in the presence of these symptoms. Also, the long-term follow-up of patients after surgery has a high priority. There is a need for further studies on the outcome of COH/ ART in infertile women with pelvic actinomycosis.