Spontaneous uterine perforation revealing an invasive mole: a case report

¹Obstetrics and Gynecology Department, Mohammed VI University Hospital Center, Marrakesh, Morocco, ²Morphosciences Research Laboratory, Faculty of Medicine and Pharmacy of Marrakesh, Cadi Ayyad University, Marrakech, Morocco, ³Department of Radiology, Mother and Child Hospital, University Hospital of Mohamed VI, Cadi Ay-yad University, Marrakesh, Morocco

^&Corresponding author
Kawtar Iraqi Houssaini, Obstetrics and Gynecology Department, Mohammed VI University Hospital Center, Marrakesh, Morocco

Introduction    

Gestational trophoblastic disease comprises a spectrum of disorders arising from abnormal trophoblastic proliferation, ranging from hydatidiform mole to malignant trophoblastic tumors. Among these entities, invasive mole is characterized by my-ometrial infiltration of molar villi following a complete or partial molar pregnancy. Although most cases are detected during post-molar surveillance through persistent-ly elevated serum β-hCG levels, rare presentations include uterine perforation and life-threatening intra-abdominal haemorrhage. Such complications are now rarely encountered because of earlier diagnosis and improved post-molar surveillance [1,2].

Its occurrence beyond the usual reproductive age is exceptionally rare and may pose significant diagnostic challenges, particularly in atypical clinical settings. Inad-equate post-molar surveillance can delay recognition of the disease, allowing pro-gression and increasing the risk of severe complications. Spontaneous uterine perfo-ration in postmenopausal women has only rarely been reported in the literature [2,3]. We report a case of spontaneous uter-ine perforation revealing an invasive mole in a 55-year-old post-menopausal woman with a history of documented molar pregnancy and incomplete follow-up, providing valuable insights into the diagnosis and management of this rare life-threatening complication.

Patient and observation     

Patient information: a 55-year-old postmenopausal woman, mother of one child, presented to the gynecological emergency department with acute ab-dominal pain. Her medical and family histories were unremarkable. Six months be-fore admission, she had undergone uterine evacuation for a complete hydatidiform mole confirmed by histopathological examination. She was subsequently lost to post-molar follow-up.

Timeline of current episode: the patient was admitted with acute ab-dominal pain and hypovolemic shock. Emergency investigations revealed massive hemoperitoneum and elevated serum β-hCG levels. Surgical exploration was per-formed, and histopathological examination confirmed an invasive mole. Postopera-tive staging identified pulmonary metastases, and etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine (EMA-CO) chemo-therapy was subsequently initiated.

Clinical findings: the patient was hemodynamically unstable and com-plained of severe acute abdominal pain. Physical examination revealed generalized abdominal tenderness, while speculum examination showed no active gynecological bleeding and an intact lower genital tract.

Diagnostic assessment: initial management included placement of two peripheral venous lines, blood sampling, and preparation of blood products. Pelvic ultrasonography demonstrated a heterogeneous pelvic mass associated with abundant intraperitoneal fluid. Computed tomography revealed a 7 cm heterogeneous intrauterine mass with massive hemoperitoneum (Figure 1, Figure 2). Serum β-hCG level was 139.000 IU/L.

Diagnosis: uterine perforation secondary to gestational trophoblastic neoplasia was suspected based on the clinical presentation, elevated serum β-hCG level, and imaging findings. Histopathological examination of the hysterectomy specimen confirmed the diagnosis of invasive mole.

Therapeutic interventions: initial resuscitation was followed by an emergency exploratory laparotomy. Intraoperative findings revealed massive hemoperitoneum, a uterus enlarged to approximately 8 weeks of gestation, and two uterine perforations at the fundus and posterior wall. Multiple hydatidiform ves-icles were present in the abdominal cavity, while both adnexa were normal (Figure 3). A total hysterectomy with bilateral salpingo-oophorectomy was performed, followed by extensive peritoneal lavage (Figure 4, Figure 5).

Follow-up and outcome of interventions: the patient received trans-fusion support with two units of packed red blood cells and four units of fresh frozen plasma. The postoperative recovery was uneventful. Thoracoabdom-inopelvic computed tomography revealed four bilateral pulmonary metastases with-out extra-pulmonary spread (Figure 6). The Fédération internationale de gynécologie et d'obstétrique (FIGO) score was 13, indicating high-risk gestational trophoblastic neoplasia. Multi-agent EMA-CO chemotherapy was initiated with weekly serum β-hCG monitoring until normalization, which was achieved after four cycles. Treatment was well tolerated, and two consolidation cycles were subse-quently completed. No recurrence was observed after one year of follow-up.

Patient perspective: the patient understood the severity of her condi-tion, the importance of treatment and follow-up. She adhered to the management plan and expressed gratitude for the favorable, lifesaving outcome.

Informed consent: written informed consent for publication of this case report and accompanying images was obtained from the patient.

Discussion     

Invasive mole is a form of gestational trophoblastic neoplasia characterized by myometrial invasion of hydropic chorionic villi. It complicates approximately 15-20% of complete hydatidiform moles and 1-5% of partial hydatidiform moles [1]. Most cases occur during the reproductive years and are diagnosed during post-molar surveillance. To our knowledge, invasive mole in peri- or postmenopausal women remains exceptionally rare, particularly when presenting with spontaneous uterine perforation [2,3]. Our case was further complicated by haem-orrhagic shock and pulmonary metastases, emphasizing the potential consequences of delayed diagnosis.

The transition from hydatidiform mole to invasive disease is driven by persistent trophoblastic proliferation and progressive myometrial infiltration. Reported risk fac-tors include extremes of maternal age, vitamin A deficiency, genetic or geographic susceptibility, and a history of molar pregnancy [1]. Deep invasion may lead to uter-ine destruction, perforation, and metastatic spread [2,4]. In our patient, advanced maternal age was the most prominent contributing factor, while the absence of post-molar surveillance likely delayed diagnosis and allowed disease progression.

Diagnosis is primarily based on elevated serum β-hCG levels and imaging findings following molar evacuation [5]. Although abnormal uterine bleeding is the usual presentation, invasive mole may rarely manifest as an acute abdomen due to uter-ine perforation and hemoperitoneum [2,4]. A hypervascularized myometrial mass with a low uterine artery resistance index on doppler examination is highly sugges-tive of gestational trophoblastic neoplasia [4]. In our patient, the diag-nosis was established by integrating clinical, biological, radiological, and histopatho-logical findings.

Uterine perforation with hemodynamic instability is a surgical emergency requiring immediate intervention [6,7]. Extensive uterine destruction may require hysterec-tomy, although fertility-preserving approaches, including partial uterine resection or minimally invasive surgery followed by chemotherapy, have been reported in se-lected younger patients [6,8]. Mitani et al. reported term pregnancies in four of five patients managed conservatively after ruptured invasive mole [8]. Following post-operative staging and classification as high-risk gestational trophoblastic neoplasia (FIGO score 13), our patient received EMA-CO chemotherapy in accordance with current recommendations [5,9].

Despite high-risk disease, serum β-hCG levels normalized after four cycles of chemotherapy, with complete remission maintained at one year. This outcome high-lights the high chemosensitivity of gestational trophoblastic neoplasia, even in advanced cases [5]. Invasive mole with spontaneous uterine perforation in a postmenopausal woman remains exceptionally rare [2,3]. This case emphasizes the importance of strict post-molar surveillance and serial β-hCG monitoring for ear-ly detection and prevention of potentially life-threatening complications.

Conclusion     

Spontaneous uterine perforation secondary to invasive mole is an exceptionally rare complication of gestational trophoblastic neoplasia. This case highlights the diagnostic difficulties associated with atypical presentations in postmenopausal women and the importance of considering this diagnosis in the presence of hemoperitoneum and hemodynamic instability. Early recognition and appropriate multidisciplinary management can result in favorable outcomes, even in high-risk disease. Post-molar surveillance, including serial β-hCG monitoring, remains essential for early detection and prevention of severe complications.

Competing interests     

The authors declare no competing interests.

Authors' contributions     

Patient management and Manuscript revision: Iraqi Houssaini Kawtar and Ahlam Bassir. Data collection and interpretation: Iraqi Houssaini Kawtar and Btissam Zouita. Manuscript drafting: Ilyass Essaoudy and Iraqi Houssaini Kawtar. Study supervision: Ahlam Bassir and Abderraouf Soummani. All the authors read and approved the final version of this manuscript.

Acknowledgements     

The authors would like to thank the medical and nursing staff involved in the patient's care.

Figures     

Figure 1: massive hemoperitoneum on abdominopelvic computed tomography: abdominopelvic computed tomography demonstrating massive hemoperitoneum (yellow arrow)
Figure 2: invasive uterine mass extending to the serosa: pelvic computed tomography demonstrating a heterogeneous intrauterine mass (white arrow) extending to the uterine serosa (blue arrow)
Figure 3: intraoperative uterine perforations: surgical view showing two uterine perforations associated with invasive trophoblastic disease
Figure 4: hysterectomy specimen with molar vesicles: macroscopic appearance of the hysterectomy specimen demonstrating multiple molar vesicles
Figure 5: yometrial invasion by invasive mole: sagittal section of the hysterectomy specimen showing deep myometrial invasion reaching the uterine serosa
Figure 6: pulmonary metastases on chest computed tomography: chest computed tomography showing multiple bilateral intraparenchymal and subpleural pulmonary micronodules suggestive of metastatic disease

References