In-utero aspiration vs expectant management of anechoic fetal ovarian cysts: open randomized controlled trial.

OBJECTIVE: To assess the efficacy and safety of in-utero aspiration (IUA) of anechoic fetal ovarian cysts. METHODS: This multicenter, prospective, randomized open trial in two parallel groups included women from nine outpatient fetal medicine departments with singleton pregnancy ≥ 28 weeks of gestation and a female fetus with an ultrasound-diagnosed simple ovarian cyst, defined as a single fully anechoic cystic structure measuring ≥ 30 mm. They were allocated randomly to IUA under ultrasound guidance or expectant management. All procedures were performed by trained senior obstetricians. Primary outcome was need for neonatal intervention, by laparoscopy, laparotomy or transabdominal aspiration. Secondary outcomes were in-utero involution of the cyst and oophorectomy at birth. Analyses were conducted according to the intention-to-treat principle. RESULTS: Of 61 participants, 34 were allocated to IUA and 27 to expectant management. Three IUA procedures (9%) could not be performed (one due to fetal position and two due to aspirations being dry). The remaining 31 IUA procedures were uneventful. The incidence of neonatal intervention did not differ significantly between the IUA and the expectant management groups (20.6% vs 37.0%; relative risk (RR), 0.55; 95% CI, 0.24-1.27). Nonetheless, IUA was associated with increased incidence of in-utero involution of the cyst (47.1% vs 18.5%; RR, 2.54; 95% CI, 1.07-6.05) and reduced rate of oophorectomy (3.0% vs 22.0%; RR, 0.13; 95% CI, 0.02-1.03) compared with expectant management. CONCLUSION: IUA of anechoic fetal ovarian cysts, compared with expectant management, was not associated with a reduction in overall neonatal interventions but was associated with a reduced oophorectomy rate. Copyright © 2017 ISUOG. Published by John Wiley & Sons Ltd.

Pregnancy and primary Sjögren's syndrome: management and outcomes in a multicentre retrospective study of 54 pregnancies.

OBJECTIVES: Primary Sjögren's syndrome (pSS) is one of the most common autoimmune diseases, mainly affecting women during the fourth decade of life. During pregnancy, the presence of anti-Ro/SSa and anti-La/SSb antibodies increases the risk of congenital heart block (CHB). Foetal and pregnancy outcomes in pregnant women with pSS compared with the general population are difficult to evaluate because of confounding factors including age and body mass index (BMI). METHOD: The aim of this case-control study was to analyse the impact of pSS in pregnant women on foetal and pregnancy outcomes. RESULTS: We enrolled 19 women with pSS (54 pregnancies) matched by age and BMI to 216 controls. Patients with pSS delivered significantly earlier (38 weeks + 3 days vs. 39 weeks + 2 days) and experienced more spontaneous abortions [< 22 weeks of gestation (WG)] than the controls [n = 16/54 (30.0%) vs. n = 1/216 (0.4%); p < 0.00001]. Preterm delivery (≤ 37+6 WG) was significantly higher in the pSS group than in the control group (29% vs. 12%, p = 0.04). pSS activity significantly affected the birthweight percentile, which was lower in pregnancies occurring after the diagnosis of pSS than in those occurring before (32.43 ± 21.57 vs. 60.46 ± 27.37; p = 0.008). No case of CHB was observed. CONCLUSIONS: pSS is responsible for an increased risk of spontaneous abortion. The duration of pregnancy is lower in patients with than without pSS, with more premature deliveries. Pregnancies that occur after the onset of the disease result in lower birthweight percentile children than when pSS is not clinically overt.

[Congenital mucocele of the ventral face of the tongue]. / Mucocèle congénitale de la face ventrale de la langue.

BACKGROUND: Congenital cystic lesions of the oral cavity are an extremely rare occurrence. Their prenatal diagnosis is essential since they can impede respiratory and swallowing functions. We describe a case that was detected prenatally and discuss its management. CASE REPORT: A 21-year-old primigravida patient who was 23 weeks pregnant was referred to our obstetrics and gynecology center after fetal ultrasonography showed a cystic lesion of the oral cavity. She had no family history of any congenital anomalies. Ultrasonography showed a male fetus with an anechoic mass measuring 21×11 mm encompassing the entire oral cavity, evoking either a mucocele or a cystic hygroma. Magnetic resonance imaging (MRI) showed a fetus with a wide-open mouth, due to a well-demarcated protruding cystic mass with no solid component, suggestive of a mucocele. A prenatal sonographically guided percutaneous needle aspiration of mucous fluid was performed at 33 gestational weeks. Although the mucocele decreased significantly in size, it nevertheless continued to expand progressively. After an uncomplicated pregnancy, the patient had spontaneous onset of labor at 39 weeks of gestation. An iterative aspiration was performed in the same manner in utero, resulting in a complete collapse of the mucocele. If needed, intubation could be considered. A 3030-g male was born by vaginal delivery, without respiratory distress. Clinical examination showed the extremely opened mouth and confirmed the presence of a large cystic mass approximately 4 cm in diameter, of sublingual origin and encompassing the entire oral cavity. The continuous protrusion of the tongue was responsible for the infant's inability to close the mouth and be breastfed. After insertion of a feeding tube, the newborn had maxillofacial surgery consisting in marsupialization of the cyst at 2 days of age. The mucocele decreased in size and the postoperative course was uneventful. No recurrence was observed at 6 months' follow-up. DISCUSSION AND CONCLUSION: Congenital mucoceles of the tongue are very rare benign lesions of the oral cavity, resulting from extravasation or retention of mucus from minor salivary glands. Their prevalence is unknown and, to our knowledge, less than ten cases of prenatal diagnosis have been previously reported. Such cystic lesions can cause respiratory distress and swallowing disorders in newborns. They are usually suspected on ultrasonography. MRI highlights the nature of the lesion and its locoregional connections with muscles and blood vessels. It provides a good analysis of the soft tissues and can distinguish between the muscles of the tongue and the pathologic mass. However, the use of CT has been reported when the diagnosis was made after childbirth or in adulthood. Given the risks of interference of the lesion with respiratory and swallowing functions, intrauterine decompression of the mucocele can be an option to prevent respiratory distress at birth and the need for neonatal intubation. Mucoceles provide somewhat confusing and disturbing ultrasonographic appearances, which can be stressful for the medical team and parents. Prenatal diagnosis and early surgical intervention (marsupialization, complete excision of the cyst or the salivary gland) can prevent risks of breathing distress and breastfeeding problems. Therefore, this strategy is essential to offer fast and satisfactory management of this rare but anxiety-producing congenital situation.

[Epidemiology of loss pregnancy]. / Épidémiologie des pertes de grossesse.

OBJECTIVES: Study of epidemiology of pregnancy loss. MATERIALS AND METHOD: A systematic review of the literature was performed using Pubmed and the Cochrane library databases and the guidelines from main international societies. RESULTS: The occurrence of first trimester miscarriage is 12% of pregnancies and 25% of women. Miscarriage risk factors are ages of woman and man, body mass index greater than or equal to 25kg/m(2), excessive coffee drinking, smoking and alcohol consumption, exposure to magnetic fields and ionizing radiation, history of abortion, some fertility disorders and impaired ovarian reserve. Late miscarriage (LM) complicates less than 1% of pregnancies. Identified risk factors are maternal age, low level of education, living alone, history of previous miscarriage, of premature delivery and of previous termination of pregnancy, any uterine malformation, trachelectomy, existing bacterial vaginosis, amniocentesis, a shortened cervix and a dilated cervical os with prolapsed membranes. Fetal death in utero has a prevalence of 2% in the world and 5/1000 in France. Its main risk factors are detailed in the chapter.

[Prenatal diagnosis of cerebral tumors and differential diagnosis]. / Diagnostic anténatal des tumeurs cérébrales fœtales et diagnostics différentiels.

Fetal brain tumors are rare and have different histologies. Although the definitive diagnosis relies on the histopathology of the tumor, it is useful to distinguish the tumors potentially curable from the tumors rapidly fatal after birth. Nevertheless, some intracranial masses are not tumors. We report four cases of intracerebral masses diagnosed prenatally corresponding to different histological lesions: teratoma, fetus-in-fetu, chraniopharyngioma, hemangioma. We discuss the elements of the differential diagnosis, which can be identified prenatally.

Fetal obstructive uropathy complicated by urinary ascites: outcome and prognostic value of fetal serum ß-2-microglobulin.

OBJECTIVES: To determine whether the prognostic value of fetal serum ß-2-microglobulin is altered and whether the occurrence of fetal urinary ascites prevents kidney damage in cases of fetal obstructive uropathy with urinary ascites. METHODS: This was a retrospective study of cases of fetal bilateral obstructive uropathy that occurred between 2006 and 2010, for which both fetal serum and ascites samples were sent to our laboratory for analysis. ß-2-microglobulin was assayed in both fetal serum and the corresponding ascites. Renal outcome was analyzed. Histological features of the kidney in cases of termination of pregnancy and renal function of liveborn infants were recorded. RESULTS: Fourteen cases with analysis of fetal serum and fetal ascites in a context of urinary obstruction were included. Renal outcome was unfavorable in eight cases (57%) and favorable in six (43%). When fetal serum ß-2-microglobulin was < 5 mg/L, renal outcome was favorable in all cases (4/4). When fetal serum ß-2-microglobulin was ≥ 5 mg/L, 8/10 cases (80%) had an unfavorable renal outcome (sensitivity, 100%; specificity, 66%). CONCLUSION: Fetal serum ß-2-microglobulin reliably predicts postnatal renal outcome in obstructive uropathy complicated by urinary ascites. Moreover, urine extravasation does not seem to protect fetal renal function.

[Emergency cervical cerclage during mid-trimester of pregnancy: Experience of Clermont-Ferrand]. / Cerclage cervical en urgence au deuxième trimestre de la grossesse : expérience clermontoise.

OBJECTIVES: The objective of this study was to identify factors associated with favourable perinatal outcome after emergency cervical cerclage during mid-trimester of pregnancy. PATIENTS AND METHODS: This is a retrospective study of all cases who underwent emergency cervical cerclage between 16 to 28 weeks of gestation (WG) over a period of 16 years in a University Hospital. RESULTS: Among the 32 cases, the postnatal survival rate (day 28) was 80%. Delivery occurred at a mean gestational age of 33.1 WG [18-41.3 WG] and after 37 WG in 39% of cases. The perinatal outcome was improved by absence of bleeding (P=0.01), unripened cervix (P=0.02), cervical dilatation below 2 cm (P=0.002), no protruding membranes (P=0.02) and more advanced gestational age at the procedure (P=0.005). When no uterine contraction and no maternal blood inflammation were observed at admission, an expectancy of 48 hours before the procedure did not improve significantly perinatal outcome (gestational age at birth and survival rate [P=0.1 and P=0.3 respectively]). DISCUSSION AND CONCLUSION: Perinatal outcome after emergency cerclage depends on cervical status and gestational age at procedure. It is not influenced by an expectancy of 48 hours before intervention for patients with no uterine contraction and no maternal blood inflammation at admission.

[Favourable outcome after fetoscopic laser surgery for twin-twin transfusion syndrome: experience of an emerging centre]. / Résultats favorables d'une équipe émergente dans le traitement du syndrome transfuseur-transfusé par photocoagulation-laser.

OBJECTIVES: Our objective was to report perinatal outcome during the first three years of an emerging centre for laser photocoagulation in twin-twin transfusion syndrome (TTTS) and to compare with outcome observed earlier in the same centre when management consisted in recurrent amniodrainage. PATIENTS AND METHODS: We conducted a single centre retrospective study. We compared perinatal outcome of 19 consecutive cases of mid trimester TTTS managed by amniodrainage over a 10-year period with 49 cases of TTTS managed by laser photocoagulation over a 3-year period. RESULTS: Laser photocoagulation increased survival rate at birth (P=0.02) and at postnatal day 28 (P=0.01). Neurologic and cardiologic complications did not differ significantly (P=0.5 and P=0.3 respectively). We observed a significant increase in survival of the donor after laser coagulation at birth (P=0.04). DISCUSSION AND CONCLUSION: Our study demonstrated better outcome after laser photocoagulation. Early results of an emerging centre appeared comparable to those of more experienced centres.

[Ballantyne syndrome caused by materno-fetal Parvovirus B19 infection: about two cases]. / Syndrome de Ballantyne secondaire à une infection maternofœtale à Parvovirus B19: à propos de deux cas.

Ballantyne's syndrome also known as Mirror syndrome is the association of fetal hydrops and maternal hydric retention. The maternal condition is often misdiagnosed as preeclampsia. We report two cases of Ballantyne syndrome associated with materno-fetal Parvovirus B19 infection. In the first case, the syndrome occurred at 26GW in a context of premature rupture of membranes. Parents and medical staff opted for termination of pregnancy because of the poor fetal prognosis. Maternal symptoms regressed after delivery. In the second case, the patient presented a Ballantyne's syndrome at 25GW. Intrauterine transfusions reversed symptomatology. Fetal hydrops of any etiology can be associated with this syndrome. Specific treatment of the fetus can avoid maternal complication allowing continuation of the pregnancy.

Prenatal diagnosis of lobar bronchial atresia.

We report three cases of fetal lobar bronchial atresia referred to our Fetal Medicine Center during the mid-trimester of pregnancy over the last 15 years. Lobar bronchial atresia can mimic a main stem bronchial atresia on mid-trimester ultrasound examination as it induces extensive lobar enlargement, major mediastinal shift and eversion of the diaphragm. It was associated with severe pulmonary hypoplasia in all three cases, even though polyhydramnios and ascites were absent in two. Termination of pregnancy was performed at parental request after extensive counseling in each of the cases and necropsy confirmed one or two enlarged lung lobes leading to major compression of the remaining lobe(s) of the ipsilateral lung, the contralateral lung and the heart. No other anomalies were observed and the karyotype was normal in all cases.

[What type of delivery for twins?]. / Quelle voie d'accouchement pour les jumeaux?

OBJECTIVES: To determine if perinatal and neonatal morbidity and mortality is improved by a planned caesarean section for twins before and at term. METHODS: A systematic search was conducted in Medline between May 2001 and December 2008. Randomised controlled studies and meta-analysis were researched at first. RESULTS: There is no evidence to support a policy of planned caesarean section or vaginal delivery for twins before term or at term whatever the presentation of the first twin. There is also no evidence to support a policy of caesarean section or vaginal delivery for a patient with a history of prior caesarean section. Vaginal delivery must be made in the presence of an obstetrician, an anaesthesiologist, and a paediatrician in a level maternity adapted to the risks of the future newborn. CONCLUSION: Otherwise, there is no evidence to support a policy of planned caesarean delivery for twins but the type of delivery has to be decided with the informed patient.

[Which invasive diagnostic procedure should we use for twin pregnancies: chorionic villous sampling or amniocentesis?]. / Quel prélèvement choisir pour les grossesses gémellaires: choriocentèse ou amniocentèse?

OBJECTIVE: To describe invasive diagnostic procedures (amniocentesis/choriocentesis) and subsequent risks in twin pregnancies. MATERIAL AND METHODS: PubMed and Cochrane database investigations were conducted using following key words: twin gestation, amniocentesis, chorionic villous sampling, karyotype. Guidelines for twin management edicted by different societies were reviewed. RESULTS: Risk of pregnancy loss after invasive diagnostic procedure in twin pregnancies seems to be slightly higher to singletons, i.e about 1.5-2% after mid-trimester amniocentesis and about 2% after first trimester choriocentesis. Dual sampling is not always mandatory but can be performed on parent's request. Specific risks are associated with twins: redundant sampling, permutation or misidentification of affected twin in case of discordant status. Procedures should be performed by highly-skilled operators under permanent ultrasound-guidance. A scheme describing placental locations and funicular insertions appears to be useful for correct identification. If foeticide can be anticipated, diagnostic procedure and foeticide should be performed by the same operators. For amniocentesis, one or two needles can be used except for cases with infectious disease (two needles and two separate insertions required). For choriocentesis, sampling should be performed close to funicular insertions. CONCLUSION: First-trimester choriocentesis makes earlier diagnosis and earlier foeticide possible compared with mid-trimester amniocentesis. Both techniques require highly-skilled operators to reduce subsequent risks in the context of twin pregnancies.

Prenatal diagnosis of thrombosis of the dural sinuses: report of six cases, review of the literature and suggested management.

OBJECTIVES: To describe and assess the sonographic findings, evolution and clinical implications of thrombosis of the fetal dural sinuses. METHODS: We compiled a multicenter report of the outcomes of five cases with a prenatal diagnosis of thrombosis of the dural sinuses, and one case in which thrombosis of the dural sinus was diagnosed at necroscopy after termination of pregnancy. Prognostic factors are discussed, and suggestions made for prenatal and postnatal management. RESULTS: The mean (range) gestational age at diagnosis of thrombosis of the dural sinuses in the five cases in which it was made prenatally was 25.2 (22-31) weeks. In these five cases, diagnosis was made by sonography and confirmed by magnetic resonance imaging (MRI), which showed a blood clot in the region of the torcular herophili. Three of the six cases delivered vaginally with favorable sonographic findings, and normal clinical neurological development. Two pregnancies were terminated at the request of the parents. In one of these cases the prognosis was poor, with signs of fetal decompensation or cardiac failure; the pregnancy was terminated and necropsy revealed thrombosis of the occipital dural sinuses associated with a hemangioma. One infant, in whom the thrombosis developed in conjunction with a dural sinus malformation, died at 4 months of age. CONCLUSIONS: Thrombosis of the cerebral venous circulation can occur antenatally and is detectable by fetal real-time and color Doppler ultrasound examination. A review of the literature supports targeted evaluation of the fetus by serial ultrasound imaging and MRI to help guide the diagnosis, and to improve the counseling and management of such cases. Partial or total regression, isolated abnormality, absence of fetal decompensation or signs of cardiac failure and favorable clinical evolution are suggestive of favorable prognosis. In such cases, non-interventional neonatal management is recommended.