Maternal and fetal outcomes of intraplacental choriocarcinoma complicated by fetomaternal hemorrhage: a systematic review

Abstract

Introduction

Intraplacental choriocarcinoma is a gestational trophoblastic neoplasia located within the placenta. Due to the usual silent presentation, more than half of the cases are diagnosed incidentally. It has been demonstrated that this pathology is linked to feto-maternal hemorrhage (FMH), stillbirth, and intrauterine growth restriction. The aim of our review was to establish if there are recurrent signs that might lead to an early diagnosis and better management in cases complicated by FMH.

Materials and methods

We performed a systematic review of the literature from 2000 up to March 2023. The adopted research strategy included the following terms: (gestational choriocarcinoma obstetrics outcome) AND (intraplacental choriocarcinoma) AND (gestational choriocarcinoma). The MEDLINE (PubMed), Google Scholar, and Scopus databases were searched.

Results

The research strategy identified 19 cases of FMH coexisting with intraplacental choriocarcinoma (IC), as described in 17 studies. The perinatal mortality rate was 36.8%. In eight cases, histological diagnosis of IC was made post-delivery. Metastatic lesions were found in 75% (6/8) of described cases. One case of maternal death has been described. Chemotherapy was necessary in seven cases. Sporadical prenatal ultrasound signs were described.

Discussion

The diagnosis of IC is usually delayed, mostly due to aspecific symptoms and signs. Histological analysis of the placenta, when not routinely performed, should be performed when warning symptoms are encountered. The maternal prognosis was good, with a mortality rate of 5.5%. A fertility-sparing approach is always possible even in the presence of metastasis. Chemotherapy seems to be useful in cases of maternal and neonatal metastasis.

Keywords:

• Intraplacental choriocarcinoma
• fetomaternal hemorrhage
• gestational trophoblastic neoplasia
• maternal outcome
• neonatal outcome
• placenta

Author contributions

Conceptualization, G.S. and M.B.; methodology, G.S., R.G., and M.B.; software, R.G.; validation, G.S., M.B., M.G., and T.S; formal analysis, G.S., M.B., and S.C.; investigation, G.S., G.O., M.B., and R.G.; data curation, R.G., S.B., and G.S.; writing – original draft preparation, G.S., R.G., and S.C.; writing – review and editing, G.S. and R.G.; visualization, T.S., G.O., M.G., and S.B.; supervision, T.S. and M.G.; project administration, G.S. and M.B. All authors have read and agreed to the published version of the manuscript.

Ethical approval

The study was conducted in accordance with the guidelines of the Declaration of Helsinki and approved on 15/04/2020 by the Institutional Review Board of IRCCS Burlo Garofolo (RC 08/2020).

Disclosure statement

The authors declare no conflicts of interest.

Data availability statement

The authors confirm that data supporting the findings of this study are available within the article.

Additional information

Funding

This work was supported by the Ministry of Health, Rome, Italy, in collaboration with the Institute for Maternal and Child Health IRCCS Burlo Garofolo, Trieste – Italy.