Kamiel Vandenberghe

A comparison of methods for preoperative discrimination between malignant and benign adnexal masses: The development of a new logistic regression model☆☆☆★

OBJECTIVE The aim of this study was to assess the complementary use of ultrasonographic end points with the level of circulating CA 125 antigen by multivariate logistic regression analysis algorithms to distinguish malignant from benign adnexal masses before operation. STUDY DESIGN One hundred ninety-one patients aged 18 to 93 years with overt adnexal masses were examined by transvaginal ultrasonography with color Doppler imaging and 31 variables were recorded. The end points were the histologic classification of the tumor and the areas under the receiver-operator characteristic curves of alternative algorithms. RESULTS One hundred forty patients had benign tumors and 51 (26.7%) had malignant tumors: 31 primary invasive tumors (37% International Federation of Gynecology and Obstetrics stage I), 5 tumors of borderline malignancy (100% International Federation of Gynecology and Obstetrics stage I), and 15 tumors were metastatic and invasive. The most useful variables for the logistic regression analysis were the menopausal status, the serum CA 125 level, the presence of >/=1 papillary growth (>3 mm in length), and a color score indicative of tumor vascularity and blood flow. The optimized procedure had a sensitivity of 95.9% and a specificity of 87.1%. The area under the receiver-operator characteristic curve was significantly higher (P <.01) than the corresponding values from the independent use of serum CA 125 levels or indexes of tumor form or vascularity. CONCLUSION Regression analysis of a few complementary variables can be used to accurately discriminate between malignant and benign adnexal masses before operation.

Experience with fetoscopic cord ligation

OBJECTIVE In the case of a monochorionic multiple pregnancy with one non-viable fetus who compromises its co-twin, fetoscopic cord ligation may be performed. We describe our fetoscopic cord ligation technique and discuss the efficacy of cord ligation for salvaging the co-twin, based on available data. STUDY DESIGN Descriptive case series of four cases and review of the cases published up to 1996. RESULTS We performed four successful ligations. Of the 23 reported cases, which include the present series, two ligations failed. Four fetuses died in utero, and 17 were born alive at a mean of 8 weeks following the procedure. Two babies died in the perinatal period, a third after 60 days. Preterm uterine contractions do not seem to be a clinical problem. Preterm prelabour rupture of the membranes (PPROM) complicates about 40% of cases, the majority occurring prior to 32 weeks. CONCLUSION Fetoscopic cord ligation is a feasible procedure with a 71% survival rate and a high risk for PPROM.

PATHOGENESIS OF CONGENITAL CYSTIC ADENOMATOID MALFORMATION OF THE LUNG

Congenital cystic adenomatoid malformation is a rare developmental abnormality of the lung. In most earlier reported cases, the anatomy of the bronchial tree was poorly documented. We describe four cases studied following autopsy. Post‐mortem bronchography or serial microscopical examination showed segmental bronchial absence or atresia in each of them. Our observations provide further evidence pointing to bronchial atresia as being the primary defect leading to the development of congenital cystic adenomatoid malformation. The morphology of the lesion, i.e. the type of malformation, is determined by the extent of dysplastic lung growth beyond the atretic segment. The aetiology of the bronchial atresia is probably heterogeneous and may either represent a primary malformation, or be the result of vascular disruption.

Fetal cardiac tamponade due to an intrapericardial teratoma

A case of an intrapericardial tumor diagnosed in utero at 26 weeks of gestation is presented. The prenatal echocardiographic follow-up of an incipient hydrops fetalis determined the management and the emergency surgical treatment. Histologically, the tumor appeared to be a benign teratoma, grade I. In the postoperative period an unexpected mediastinal tumor was found and removed later. This tumor also appeared to be a benign teratoma, grade 0. Both teratomas were independent and therefore primary.

Carbon dioxide pneumoamnios causes acidosis in fetal lamb.

Recently developed techniques of video-endoscopic surgery may offer new hope for the future of fetal surgery. To allow this approach, the amniotic cavity has to be temporarily enlarged, either by carbon dioxide (CO2) insufflation or by amnioinfusion. In 6 anesthetized ewes, CO2 insufflation of the amniotic cavity produced severe fetal hypercapnia (from 57.6 +/- 1.6 to 87.0 +/- 7.0 torr) and acidosis (from 7.22 +/- 0.03 to 7.11 +/- 0.08) despite normal maternal CO2 pressure and pH. CO2 pneumoamnios does not therefore appear to be an ideal working medium. Fetal endoscopic surgery through amnioinfusion of physiologic fluid may be a safer alternative.

Tracheoscopic endoluminal plugging using an inflatable device in the fetal lamb model

OBJECTIVE Intra-uterine tracheal occlusion has been proposed to reverse pulmonary hypoplasia, an important prognostic factor in congenital diaphragmatic hernia. We aimed to evaluate the feasibility and pulmonary effects of tracheoscopic tracheal obstruction with a detachable balloon. STUDY DESIGN Fourteen mid-trimester fetuses out of 24 in 13 ewes underwent tracheoscopic balloon obstruction. Ten non-operated fetuses served as controls. Plugging was performed under fiber-tracheoscopy using a detachable balloon. Outcome measures consisted of: total operating time, tracheoscopy time, fetal survival, efficiency of plugging, and pulmonary effects. The Mann-Whitney test and linear regression were used for statistical analysis. RESULTS Mean operating time and tracheoscopy time were 65+/-12 and 6.6+/-3.9 min, respectively. One intra-operative death occurred in each group. The post-operative mortality was 2/13 for cases and 2/9 for controls. In all 14 fetuses, the trachea was successfully obstructed. In the 11 treated animals born alive, the lung-to-body-weight ratio was 0.060+/-0.01, while in controls it was 0.031+/-0.01 (P = 0.0001). In a subset of six fetuses obstructed for 14-18 days, mean-terminal-bronchial density was 0.95+/-0.59, compared to 2.06+/-0.80 for controls (P = 0.046). CONCLUSIONS Using fetal tracheoscopy, the trachea can successfully be obstructed with an inflatable balloon. Pulmonary hyperplasia is achieved when the obstruction lasts 2 weeks.

Tracheoscopic endotracheal occlusion in the ovine model: technique and pulmonary effects.

BACKGROUND/PURPOSE The purpose of this study was to assess the tracheoscopic placement of a detachable balloon to obstruct the trachea in the fetal lamb model and to study the pulmonary effects 2 weeks later. METHODS Thirteen pregnant ewes carrying 27 fetuses were used in this trial. Thirteen fetuses underwent endotracheal plugging at a mean gestational age of 95 (90 to 100) days (term, 145). Fourteen nonoperated fetuses served as controls. The intended duration of obstruction was 15 days. Study parameters consisted of operating time, fetal survival, performance of the balloon, and pulmonary changes. RESULTS Hysterotomy time was 41 +/- 25 minutes and tracheoscopy time was 7.2 +/- 3.4 minutes. There were no intraoperative deaths in this series. Postoperative fatalities occurred in 2 of 13 plugged fetuses and 1 of 14 controls. At completion of the experiment, the balloon was recovered intact in the trachea of 12 of 13 (92.3%) fetuses. The lung weight/body weight ratio was 76 +/- 16 mg/kg for plugged animals, while in controls it was 34 +/- 8 mg/kg (P = .0001). The lung volume/body weight ratio was 101 +/- 17 mL/kg in plugged fetuses, compared with 47 +/- 4 mL/kg for controls (P = .0002). Mean-terminal-bronchial-density was 0.937 +/- 0.303 for plugged animals, compared with 1.911 +/- 0.441 for controls (P = .0002). Mean linear intercept was 60.9 +/- 5.2 microm in treated fetuses, and 46.3 +/- 5.8 microm for controls (P = .0004). CONCLUSIONS Fetal endotracheal occlusion, using a tracheoscopically placed detachable balloon, is reliable, and results in pulmonary hyperplasia after 2 weeks of obstruction. This technique may be beneficial in treating the pulmonary hypoplasia seen in congenital diaphragmatic hernia. Further studies using this balloon device are now required to corroborate these findings.

Operative fetoscopy: new perspective in fetal therapy?

In a limited number of conditions in utero surgery may be life‐saving, such as some cases of congenital diaphragmatic hernia, cystic adenomatoid malformation of the lung, sacrococcygeal teratoma and lower urinary tract obstruction. Postoperative premature labour and its extreme invasiveness have been major drawbacks for open fetal surgery. More recently the merger of fetoscopy and advanced video‐endoscopic surgery has been the basis of the concept of endoscopic fetal surgery. In order to evaluate the opportunities of ‘fetendo’ surgery, animal models have been developed to test the safety of the endoscopic approach, and the feasibility of surgical manipulations on the fetus. In the non‐human primate, a lesser invasiveness of endoscopy over open surgical approach was demonstrated, by a significant decrease in uterine activity in comparison with hysterotomy.

A model for fetal surgery through intrauterine endoscopy

Video-endoscopic technology may offer new hope for the future of fetal surgery. Ten time-dated pregnant ewes (95 to 105 days; term, 145 days) were used to develop a model of fetal surgery through intrauterine endoscopy. Special 5-mm ports were designed with short, flexible, balloon-tipped shafts. The cannulas were inserted through purse-stringed hysterostomies. Amnioinfusion of warmed physiological fluid (Hartmann) was used as the work medium. Fetal heart rate and oxygen saturation (pulse oximetry), temperature, and amniotic pressure were monitored continuously through endoscopically placed sensors. Surgical procedures included subcutaneous dissection of the abdominal wall and ligation of the urethra in the male fetus (n = 7), ligation of the urachus within the umbilical cord (n = 8), and endoscopy of the fetal esophagus and trachea (n = 6). Bloodless surgery was achieved with a novel coagulation device that allows bipolar cutting and coagulation in an electrolyte-rich fluid medium. The mean fetal operative time was 147 minutes (range, 130 to 195). The intraamniotic temperature was maintained between 38.0 +/- 0.9 degrees C and 41.7 +/- 1.7 degrees C throughout the procedure. Oxygen saturation ranged from 51 +/- 20% to 72 +/- 14%, and fetal heart rate from 120 to 140 beats per minute. In conclusion, intrauterine endoscopy in a fluid medium allows fully monitored surgical procedures, lasting more than 2 hours, to be performed on the fetal lamb.

Trisomy of the short arm of chromosome 5: autopsy data in a malformed newborn with inv dup (5) (p13.1----p15.3).

In this paper we report detailed autopsy data of a malformed male newborn with Sp trisomy due to a de novo inverted 5p duplication, inv dup (5Xpl3.1→ pl5.3), and we compare these data with the findings in previous reports on 5p trisomy. Cerebral malformations, i.e. agenesis of corpus caliosum, and Dandy‐Walker cyst malformations, seem to be another characteristic finding in this partial autosomal duplication syndrome.