Case-presentation
A 32-year-old woman presented in 2019 with secondary subfertility two
years after a full-term pregnancy. Her first pregnancy was complicated
by a preeclampsia. In October 2016, at 36 weeks and 6 days, she
delivered a daughter vaginally after induction of labor. The placenta
was retained and removed manually, which was complicated by post-partum
hemorrhage. In January 2017 a hysteroscopy was performed due to
complaints of irregular vaginal bleeding and suspicion of retained
products of pregnancy. A suspected remnant of the placenta was removed
hysteroscopically and an intra uterine device (IUD Mirena) was inserted
as a contraceptive. The pathology report described normal placental
tissue. The IUD was removed in the beginning of 2018 because of a
renewed child wish. In March 2019, a hysteroscopy was performed because
of secondary subfertility and the suspicion of Asherman’s syndrome.
Before the procedure, no abnormalities were seen on transvaginal
ultrasound in the myometrium. The endometrial thickness was relatively
thin (2.7mm) postovulatory. During the hysteroscopy a small lesion of
placenta-like tissue (1cm) was resected. The histologic examination
showed linear, superficial linear lesional tissue of 1 cm, with a
thickness of 1.2 mm, consisting of hyalinized eosinophilic material with
interspersed hyperchromatic, pleomorphic and intermediate type
trophoblast. The cells had mainly clear cytoplasm and focally contained
eosinophilic cytoplasm. The trophoblastic cells were arranged in cords
and small nests. Mitoses and necrosis were not present in the
hysterectomy specimen but were seen in the previous histology sample.
Some chorionic villi were seen to suggest trophoblast accompanying
products of conception. Immunohistochemistry showed that these cells
were positive for p63 and showed a Ki-67 or MIB-1 proliferation index of
8-10%. Immunophenotype was that of proliferating intermediate
trophoblast. These characteristics were consistent with the diagnosis of
APSN.
Following the pathology result, hCG-levels were determined and found to
be below the cut-off limits. A CT-scan chest/abdomen and MRI pelvis were
performed to exclude an infiltrating, malignant mass, no abnormalities
or lesions suspected of metastases were seen. An expectant approach was
discussed as the woman wished to conceive again. She was counseled about
the association of APSN with PSTT and ETT. Her case was discussed in a
national and international multi-disciplinary team. Consensus was to
perform three-montly monitoring using serum hCG-levels, transvaginal
ultrasound and alternating MRI/hysteroscopy if not pregnant. None of the
tests used showed abnormalities during follow-up. The woman achieved two
full-term pregnancies two years apart from each other, the pregnancies
were uneventful except for a retained placenta in both deliveries
resulting in a manual removal of the placenta. Histological examination
of both placenta’s did not show any signs of abnormality. The MRI scans
performed during the pregnancies were discussed with a specialist in
placental MRI scans.
In May 2022, six months after the last pregnancy, a hysterectomy was
performed. An atypical, linear and superficial lesion of 1.1 cm was
found in the endometrium and pathology results remained consistent for
APSN with a Ki-67 proliferation rate of 8-10%. Macroscopically the
lesion could not be detected. The woman was discharged from follow-up.