Identification and treatment of primary cervical gestational trophoblastic neoplasia: a retrospective study of 13 patients and literature review

Xiaoyu Wang,Junjun Yang,Tong Ren,Fengzhi Feng,Yang Xiang,Jun Zhao,Xirun Wan

doi:10.1186/s13023-021-02111-w

Abstract

BackgroundPrimary cervical gestational trophoblastic neoplasias (GTNs) are extremely rare ectopic GTNs. Such lesions are difficult to diagnose clinically because of their rarity, with abnormal vaginal bleeding of a non-specific cause being the most common symptom. To that end, this retrospective study aimed to identify the clinical characteristics of cervical GTN and to explore diagnostic and therapeutic strategies.ResultsThirteen patients diagnosed with primary cervical GTN at the Department of Gynecology, Peking Union Medical College Hospital, Beijing, China, between June 1, 1988 and May 31, 2020 were included in the study. All patients had irregular vaginal bleeding, including six who presented with massive bleeding. Seven patients (53.8%) were initially misdiagnosed with a cervical pregnancy. All patients received chemotherapy; 11 (84.6%) also underwent hysterectomy because of chemoresistant lesions or uncontrolled bleeding. All patients achieved complete remission; however, two women (15.4%) experienced a relapse during the median follow-up period of 35 months. A comprehensive review of English-language literature published between 1980 and 2020 identified 22 case reports encompassing 27 patients. The definitive diagnosis was achieved via pathology in 26 of them (96.3%), and hysterectomy was performed in 21 (77.8%).ConclusionsOwing to its rarity and nonspecific symptoms, the diagnosis of primary cervical GTN is challenging and often relies on pathology. The combination of chemotherapy and hysterectomy is the main therapeutic strategy for this disease.

Highlights

Primary cervical gestational trophoblastic neoplasias (GTNs) are extremely rare ectopic Gestational trophoblastic neoplasia (GTN)
Unlike most patients with GTN that can be diagnosed clinically and achieved complete remission (CR) by chemotherapy alone, the definitive diagnoses of primary cervical GTN were primarily made based on pathological examinations, while most patients underwent hysterectomy for uncontrolled bleeding or chemoresistant lesions
Hysterectomy was the initial treatment in 15.4% and 29.6% of patients in our retrospective study and among those we reviewed in the literature, respectively, and as many as 81.8% and 65%, respectively, underwent this surgical procedure during chemotherapy owing to the presence of chemoresistant lesions in the remaining patient

Summary

Introduction

Primary cervical gestational trophoblastic neoplasias (GTNs) are extremely rare ectopic GTNs. Such lesions are difficult to diagnose clinically because of their rarity, with abnormal vaginal bleeding of a non-specific cause being the most common symptom To that end, this retrospective study aimed to identify the clinical characteristics of cervical GTN and to explore diagnostic and therapeutic strategies. Primary cervical GTN is an extremely rare type of ectopic GTN that is difficult to clinically diagnose because of its rarity; non-specific abnormal vaginal bleeding is the most common symptom [2]. This disease can mimic other, more common, cervical lesions such as cervical pregnancy, threatened abortion, cervical polyp, or cervical neoplasia. We performed a retrospective analysis and literature review of the clinical characteristics and therapeutic processes of primary cervical GTN with the aim of contributing to improvements in the diagnostic accuracy of this disease and establishing better therapeutic strategies

Methods

Results

Discussion

Conclusion