Life-threatening rupture of interstitial ectopic pregnancy: a rare presentation of metastatic invasive mole.

An invasive mole is an uncommon type of gestational trophoblastic disease, and if considering its implantation in an interstitial extrauterine location, we are facing a rarer condition.There are 14 cases described of interstitial ectopic gestational trophoblastic disease. As far as we know, we present the third case of invasive mole within interstitial location, in this case with pulmonary metastases.The diagnosis of an interstitial implantation is challenging. Our patient was initially diagnosed with an intrauterine hydatidiform molar pregnancy, and a uterine aspiration was performed. Two weeks later, she presented with haemodynamical instability due to a severe haemoperitoneum. A laparotomy was immediately performed and revealed a ruptured interstitial pregnancy with molar vesicle extrusion. Besides its rarity, we highlight the clinical presentation with hypovolaemic shock due to rupture of ectopic pregnancy in a young nulliparous woman, which required an emergent surgical approach with lifesaving purpose while preserving future fertility.

Manejo clínico de la neoplasia trofoblástica gestacional: presentación de un caso / Clinical management of gestational trophoblastic neoplasia: presentation of a case

La enfermedad trofoblástica gestacional es una entidad poco frecuente que se produce por una proliferación anormal de la placenta. Engloba un diverso espectro de entidades histológicas, que conllevan a su vez diversas implicaciones clínicas. Unas son de carácter benigno (mola parcial y mola completa, placentomegalia, nódulo del sitio placentario) y otras de carácter maligno, estas últimas reciben en común la denominación de Neoplasia Tofoblástica Gestacional (NTG) y tienen un alto potencial de metastatización. Forman parte de las NTG: la mola invasiva, el tumor trofoblástico del sitio placentario, el tumor trofoblástico epitelioide y el coriocarcinoma gestacional. Lo más común es que la NTG debute tras la aparición de una gestación molar, pero también es posible que ocurra tras otro tipo de evento obstétrico como una gestación a término, o una gestación ectópica. Es pues de vital importancia realizar un correcto seguimiento tras evacuar una gestación molar, realizando una monitorización de los valores séricos de la BhcG y sospechando una enfermedad trofoblástica persistente ante los supuestos que posteriormente describiremos.La principal herramienta terapéutica para la NTG es el uso de la quimioterapia, aunque también se puede optar por la cirugía endeterminados casos. Habrá que valorar de modo individualizado en función de la histología, score pronóstico y deseos genésicos futuros de la paciente. Afortunadamente, la tasa de supervivencia y de curación de la NTG con un tratamiento y seguimiento adecuado es muy elevada. (AU)

Gestational trophoblastic disease is a rare entity that is caused by an abnormal proliferation of the placenta. It encompasses adiverse spectrum of histological entities, which carry various clinical implications. Some of them are benign (partial mole and complete mole, placentomegaly, placental site nodule) and others of a malignant nature, which are known as Gestational TrophoblasticNeoplasia (GTN) and have a high potential for metastasization. Are part of the GTN: invasive mole, trophoblastic tumor of theplacental site, trophoblastic tumor epithelioid and gestational choriocarcinoma. The most common is that NTG debuts after theappearance of a molar gestation, but it also may occur after another type of obstetric event such as a term gestation, or an ectopicgestation. It is therefore of vital importance to carry out a correct follow-up after evacuating a molar gestation, monitoring the serumvalues of BhcG and suspecting a persistent trophoblastic disease in the event that we will later describe. The main therapeutic toolfor NTG is the use of chemotherapy, although surgery can also be chosen in certain cases. It will be necessary to assess individuallyaccording to histology, prognostic score and future genetic desires of the patient. Fortunately, the survival and cure rate of NTG with proper treatment and follow-up is very high. (AU)

The diagnostic use of magnetic resonance imaging for acute abdominal and pelvic pain in pregnancy.

OBJECTIVES: Acute abdomino-pelvic pain in pregnancy represents a diagnostic challenge. In many cases, radiological and laparoscopic diagnostic modalities are hazardous or contraindicated. Magnetic Resonance Imaging (MRI) is not commonly used for this indication and the results are not widely published. DESIGN AND SETTING: A single-center retrospective observational study. POPULATION: 34 cases of pregnant women with abdomino-pelvic pain who underwent MRI as an additional modality when clinical, laboratory and ultrasound (USS) findings were indeterminate. METHODS: Case notes were reviewed where pregnant women underwent a MRI investigation for abdominal-pelvic pain. Primary Obstetric indications for an MRI eg placenta accreta were excluded. MAIN OUTCOME MEASURES: The differential diagnosis after; 1) history and physical examination and 2) with the addition of USS and 3) with the further addition of an MRI were all individually compared to the eventual diagnosis. RESULTS: The diagnoses reached by MRI corresponded with the final diagnosis in 22 out of 23 cases. In the remaining 11 cases MRI accurately ruled out presence of pathology. MRI was inaccurate in 1 case. CONCLUSION: The additional use of MRI was more accurate than clinical assessment and USS combined. The accurate exclusion of pathology in 11 cases is particularly significant. MRI should be considered in cases of abdomino-pelvic pain in pregnant women.

Molar pregnancy with normal viable fetus presenting with severe pre-eclampsia: a case report.

BACKGROUND: While gestational trophoblastic disease is not rare, hydatidiform mole with a coexistent live fetus is a very rare condition occurring in 0.005 to 0.01% of all pregnancies. As a result of the rarity of this condition, diagnosis, management, and monitoring will remain challenging especially in places with limited resources and expertise. The case we report is an interesting rare case which presented with well-described complications; only a few similar cases have been described to date. CASE PRESENTATION: We report a case of a 21-year-old local Sarawakian woman with partial molar pregnancy who presented with severe pre-eclampsia in which the baby was morphologically normal, delivered prematurely, and there was a single large placenta showing molar changes. CONCLUSION: Even though the incidence of this condition is very rare, recognizing and diagnosing it is very important for patient care and it should be considered and looked for in patients presenting with pre-eclampsia.

Predictive value of some hematological parameters for non-invasive and invasive mole pregnancies.

AIM: The aim of this study was to discriminate mole pregnancies and invasive forms among cases with first trimester vaginal bleeding by the utilization of some complete blood count parameters conjunct to sonographic findings and beta human chorionic gonadotropin concentration. MATERIALS AND METHODS: Consecutive 257 cases with histopathologically confirmed mole pregnancies and 199 women without mole pregnancy presented with first trimester vaginal bleeding who admitted to Zeynep Kamil Women and Children's Health Training Hospital between January 2012 and January 2016 were included in this cross-sectional study. The serum beta HCG level at presentation, and beta hCG levels at 1st, 2nd and 3rd weeks of postevacuation with some parameters of complete blood count were utilized to discriminate cases with molar pregnancy and cases with invasive mole among first trimester pregnants presented with vaginal bleeding and abnormal sonographic findings. RESULTS: Levels of beta hCG at baseline (AUC = 0.700, p < 0.05) and 1st (AUC = 0.704, p < 0.05), 2nd (AUC = 0.870, p < 0.001) and 3rd (AUC = 0.916, p < 0.001) weeks of postevacuation period were significant predictors for the cases with persistent disease. While area under curve for mean platelet volume is 0.715, it means that mean platelet volume has 21.5% additional diagnostic value for predicting persistency in molar patients. For 8.55 cut-off point for mean platelet volume, sensitivity is 84.6% and specificity is 51.6%. Area under curve for platelet/lymphocyte ratio is 0.683 means that platelet/lymphocyte ratio has additional 18.3% diagnostic value. For 102.25 cut-off point sensitivity is 86.6% and specificity is 46.2. CONCLUSIONS: Simple, widely available complete blood count parameters may be used as an adjunct to other risk factors to diagnose molar pregnancies and predict postevacuation trophoblastic disease.

Laparoscopic Management of an Invasive Mole Perforating the Uterus.

STUDY OBJECTIVE: To show the possibility of conservative laparoscopic management in a case of invasive mole perforating the uterus. DESIGN: Video with explanations. SETTING: An invasive mole is a potentially life-threatening complication of gestational trophoblastic disease [1]. This is a case of a 24-year-old female presenting with abdominal pain and vaginal bleeding. There have been several previous reports of cases of uterine perforation by an invasive mole, all of which were managed with abdominal hysterectomy [2-7]. To our knowledge, this is the first report of an invasive mole perforation with active bleeding managed by laparoscopy without hysterectomy. INTERVENTIONS: Sonography revealed a large amount of fluid and a 3 × 3-cm heterogeneous lesion next to the posterior uterine wall. Her hemoglobin level dropped from 10.6 mg/dL to 8.6 mg/dL, and her ß-human chorionic gonadotropin level was 19,004 mIU/mL. On laparoscopy, ∼2500 mL of hemoperitoneum was found, along with an actively bleeding bulging mass in the posterior uterine wall. This mass was dissected, and hemostasis was secured with sutures and electrocoagulation. Pathology confirmed the diagnosis of a complete mole. After surgery, the patient was treated with 5 courses of a methotrexate-folinic acid regimen. Her recovery was uneventful. CONCLUSION: Uterine perforation by an invasive mole can be managed conservatively with laparoscopic surgery and postoperative chemotherapy. The transmural lesion will increase the risk of future uterine rupture during pregnancy in this patient.

Mola invasiva / Invasive mole

La mola invasiva es poco frecuente y se caracteriza por la excesiva proliferación trofoblástica y penetración del trofoblasto dentro del miometrio, pero sin tendencia hacia la diseminación metastásica. El objetivo del trabajo es presentar un caso diagnosticado de neoplasia trofoblástica gestacional, tipo mola invasiva. Se realizaron estudios complementarios como: ecografía transabdominal, determinaciones de la fracción beta de la gonadotropina coriónica humana y una biopsia endometrial por legrado uterino. El resultado fue embarazo molar. Al ingreso le fue administrado metotrexato sistémico 25 mg intramuscular y ácido folínico un ámpula intramuscular de 15 mg diariamente, ambos durante cinco días. Posteriormente, a los 20 días se realizó histerectomía total abdominal sin anexectomía. La evolución clínica fue por consulta externa. Además, se realizó: radiografía de tórax, ultrasonografía de pelvis, dosificación de gonadotropina coriónica humana en su fracción beta, hemograma completo, tiempo de coagulación, sangramiento y transaminasa glutámico pirúvica evolutivas. En el manejo de la mola invasiva el tratamiento médico combinado con el quirúrgico fue efectivo y bien tolerado por la paciente(AU)

Invasive mole is an infrequent condition characterized by excessive trophoblastic proliferation and trophoblast penetration into the myometrium, without a tendency to metastatic dissemination. The purpose of the study is to present a case diagnosed with gestational trophoblastic neoplasia of the invasive mole type. The following complementary tests were performed: transabdominal echography, human chorionic gonadotropin beta subunit determinations, and endometrial biopsy by uterine curettage. The result was molar pregnancy. Upon admission the patient was administered systemic methotrexate 25 mg intramuscularly, as well as an intramuscular ampoule of folinic acid 15 mg daily, both during five days. Total abdominal hysterectomy without anexectomy was performed 20 days later. Clinical evolution was followed up on an outpatient basis. The following tests were also performed: chest radiography, pelvic ultrasonography, human chorionic gonadotropin beta dosage, complete blood count, clotting time, bleeding and evolutive glutamic-pyruvic transaminase. During management of the invasive mole, the combination of clinical and surgical treatments proved effective and was well assimilated by the patient(AU)

Spontaneous renal hemorrhage caused by invasive mole: a case report.

CASE: The authors report a case with spontaneous renal hemorrhage caused by invasive mole. The diagnosis was gestational trophoblastic disease (GTD), with metastasis to brain, kidneys, and lungs at Stage IV. The patient was given etoposide-methotrexate-actinomycin D plus cyclophosphamide-vincristine (EMACO) treatment regimen for 11 times including three times with consolidation chemotherapies. Laparoscopically-assisted vaginal hysterectomy (LAVH) + laparoscopic-assisted left renal excision + evacuation of the left perirenal hematoma were performed during the eighth chemotherapy. CONCLUSION: Post-operational pathological examination revealed trophoblasts within the lesions present in uterine fundus and the residue images of a few trophoblasts present in the left renal mass.

[Spontaneous renal hemorrhage caused by invasive mole: a case report].

Gestational trophoblastic tumors (GTTs) are malignant lesions that often cause abnormal genital bleeding and may present with hemoptysis, intraperitoneal bleeding or acute neurologic deficits. GTTs are generally highly chemosensitive with more favorable outcomes than other comparable malignancies. Here we report a rare case of invasive mole (FIGO stage IV, WHO score16) presenting with renal subcapsular hematoma due to bleeding renal metastasis. The patient had a pretreatment ß-human chorionic gonadotrophin (ß-HCG) level of 462 047 mIU/ml and received combined chemotherapy with etoposide, methotrexate, actinomycin-D, cyclophosphamide and vincristine with also adjuvant surgeries including hysterectomy and nephrectomy. The patient recovered well and the tumor has remained in complete remission for one year and a half.

Ovarian hyperstimulation syndrome in a spontaneous pregnancy with invasive mole.

Spontaneous ovarian hyperstimulation syndrome (sOHSS) is an extremely rare event. Herein, we report a case of severe sOHSS with invasive mole in a 29-year-old woman. In this case the full-blown OHSS developed after evacuation when the serum ß-hCG value was declining. Also noticeable was a very high level of cancer antigen-125. Molecular biology study of the follicle-stimulating hormone (FSHR) gene did not detect exonic mutations, but revealed the presence of c.-29G>A (rs1394205) in the 5'-non-coding region of exon 1. The A307T and S680N polymorphisms of exon 10 of FSHR was Thr307 Asn680. Although sOHSS is a rare entity, clinicians must bear the differential diagnosis of sOHSS in mind if a patient presents with gross ascites and other symptoms of ovarian cancer, which also may be signs of OHSS. Whether the single nucleotide polymorphism rs1394205 affects the level of transcriptional activity of the FSHR gene needs to be studied in the future.

Ovarian hyperstimulation syndrome in a spontaneous pregnancy with invasive mole: report of a case.

It is known that most cases of Ovarian Hyperstimulation Syndrome (OHSS) are associated with the therapies for ovulation induction. However, OHSS may rarely be associated with a spontaneous ovulatory cycle, usually in the case of multiple gestations, hypothyroidism or polycystic ovary syndrome. We report a case of severe OHSS in spontaneous pregnancy with invasive mole in a 34 years old woman. The clinical picture showed abdominal pain, massive ascites, nausea, dyspnea and amenorrhea. After imaging examinations and laboratory tests, the diagnosis was established. The patient was managed expectantly with no complications. Although spontaneous ovarian hyperstimulation is a rare entity, it is important that the physician recognizes this condition. Prompt diagnosis and successful management is likely to avoid serious complications, which may develop rapidly.

Nephrotic syndrome associated with invasive mole: a case report.

Gestational trophoblastic disease describes a number of gynaecological tumours that originate in the trophoblast layer, including hydatidiform mole (complete or partial), placental site trophoblastic tumour, choriocarcinoma and gestational trophoblastic neoplasia (GTN). Invasive moles are responsible for most cases of localized GTN. Two cases of GTN previously reported in the literature exhibited membranous glomerulonephritis (MGN). However, histologic examinations in our case did not reveal evidence of MGN. Clinical features and pathologic findings were consistent with minimal change disease associated with an invasive mole. In the present case, we observed complete remission of nephrotic syndrome following removal of the invasive mole.

Invasive mole: Indonesian perspective.

OBJECTIVE: To share our experiences concerning the characteristics of invasive mole (IM), including transformation and progression, its propensity to perforate, diagnostic and therapeutic procedures and prognosis. STUDY DESIGN: A cross-sectional, descriptive, analytic study, conducted at Hasan Sadikin Hospital, Bandung, Indonesia. RESULTS: There were 27 cases of IM, histologically confirmed. Seventy and three tenths percent were preceded by complete mole, 18.5% by partial mole and 11.2% by uncertain pathology. The average patient age, gravidity, transformation period and uterine size were 34.4 years, 4.4 pregnancies, 2.6 months and 14.3 weeks, respectively. Ultrasound findings in 20 cases were: 9 (45%) diagnosed as IM, 8 (40%) as choriocarcinoma, 2 (10%) as hydatidiform mole and 1 (5%) doubtful. Perforation occurred in 18 (66.7%) cases, 61.1% to the abdominal cavity, 5.5% to the uterine cavity and parametrium, 11.1% to the abdominal and uterine cavity and 16.7% to the abdomen and parametrium. There were 3 deaths, 2 due to perforation to the parametrium and 1 to the abdomen. Two cases developed metastasis, 1 to the vagina and the other to the lungs. Both survived. The surgical intervention rate was 96.3%, with all patients having hysterectomy except for 1 exploratory laparotomy. CONCLUSION: IM should be suspected in multiparous patients with a history of mole and short transformation period. Ultrasound as a diagnostic tool is promising, but still needs further study. The incidence of perforation is high with possibility of death, yet the prognosis remains good provided that the acute period is managed appropriately. The method of follow-up is similar to mole.

A perplexing case of gastrointestinal haemorrhage.

Choriocarcinoma is a gestational trophoblastic tumour with a high metastatic potential but presentation with gastrointestinal haemorrhage due to jejunal mucosal metastasis is very rare. A 25-year-old Nepali woman presented with severe anaemia and massive gastrointestinal haemorrhage after normal pregnancy following evacuation of a hydatiform mole. During laparotomy, the patient was found to have extensive jejunal mucosal metastases.

Mola invasora y abdomen agudo: a propósito de un caso / Invasive mole and the acute abdomen: a case study

La mola invasora es una eventual complicación de la mola hidatiforme parcial o total. Se manifiesta por una penetración profunda de las vellosidades coriales con degeneración hidrópica y estructuras trofoblásticas dentro del miometrio o invasión de los vasos uterinos. La mayoría deriva de una mola completa, aunque las parciales también se han descrito con una frecuencia 10 veces menor. El diagnóstico es por sospecha analítica (aumento progresivo de la ß-hCG) y de imagen (ecografía con o sin Doppler y/o RM uterina). Las posibles complicaciones son: hemorragia uterina grave, perforación uterina que produce hemoperitoneo y abdomen agudo, y progresión a coriocarci noma. Con el tratamiento citostático adecuado (metotrexato o dactinomicina) el pronóstico es excelente, produciéndose curaciones en prácticamente el 100% de los casos. Hemos presentado el caso clínico de una paciente de 27 años con una mola invasora que ocasionó una perforación uterina y un cuadro de hemoperitoneo y abdomen agudo que precisó de intervención quirúrgica. Se consiguió realizar cirugía conservadora resecando el fundus uterino, lugar de implantación de la mola, y suturándolo. Posteriormente se administraron 73 mg de metotrexato intramuscular a la semana, durante 8 semanas. La paciente lleva 16 meses asintomática, con ß-hCG negativa y ecografía pélvica normal, en busca de una nueva gestación (AU)

Radiologically identified molar invasion into pelvic arteriovenous shunts.

A case of radiologically identified molar invasion into extensive arteriovenous shunts (AVSs) is described. CT and MRI revealed a large uterine mass, accompanied by multiple AVSs. Dynamic MRI and pelvic angiography demonstrated multiple trophoblastic cysts invading into the AVSs. Resected specimen confirmed the diagnosis of invasive mole. Dynamic MRI was very useful in determining the etiology of AVS.

Mola y preeclampsia: una presentación clínica atípica / Mola and preeclampsia: an atypical clinical presentation

Se presenta un caso clínico de mola invasora y preeclampsia, la cual tuvo una presentación atípica, involucrando, por lo difícil del diagnóstico inicial, a los servicios de medicina interna y ginecología-obstetricia. Se analiza el caso, su fisiopatología y su adecuado tratamiento