Asymptomatic congenital cystic adenomatoid malformation of the lung: Is it time to operate?

ObjectiveThe optimal management of congenital adenomatoid malformation of the lung remains controversial. Prenatal ultrasonographic analysis has increasingly discovered asymptomatic lesions, raising questions about the need for and timing of surgical treatment for asymptomatic congenital adenomatoid malformation. The aim of our study was to analyze the short-term postoperative outcome of symptomatic congenital adenomatoid malformations compared with asymptomatic malformations.MethodsAll the data of patients presenting with congenital adenomatoid malformations histologically diagnosed and operated on between 1998 and 2005 at our institution were retrospectively reviewed. Patients were divided into 2 groups: group A comprised asymptomatic infants, and group B comprised symptomatic infants. Major outcomes considered were the length of ventilation, pleural drainage, and hospital stay. Postoperative morbidity and mortality were also evaluated. Asymptomatic patients were further stratified for age at the time of the operation to evaluate whether age at surgical intervention affects the outcome. The Fisher's exact and Mann–Whitney tests were used as appropriate.ResultsFifty-seven patients were consecutively treated. Thirty-five patients were given diagnoses of asymptomatic lesions and were enrolled into group A, whereas 22 patients presenting with symptoms were entered into group B. The lengths of ventilation, pleural drainage, and hospital stay were significantly longer in patients with symptomatic congenital adenomatoid malformations. Moreover, symptomatic patients presented with a higher postoperative complication rate. The age-based stratification of asymptomatic children did not show any difference on either postoperative mortality or major outcome considered.ConclusionChildren with congenital adenomatoid malformations operated on when asymptomatic present a better short-term outcome than symptomatic children. In addition, age at the time of the operation does not negatively affect the outcome. Our findings support early surgical treatment for asymptomatic congenital adenomatoid malformation

Is counselling for CCAM that difficult? Learning from parental experience

Objective: Cystic adenomatoid malformations (CCAM) are relatively rare developmental abnormalities of the lung. Despite outcome is usually benign, parents often exhibit high anxiety level. The purpose of the present study was to collect parents’subjective experience of communication of diagnosis when expecting a fetus with CCAM

When uncertainty generates more anxiety than severity: the prenatal experience with cystic adenomatoid malformation of the lung

Aim: To assess reasons for higher levels of anxiety generated by prenatal counselling of a condition with good outcome such as cystic adenomatoid malformation (CCAM) of the lung compared to a life-threatening malformation such as congenital diaphragmatic hernia (CDH). Materials and methods: The Spielberger State-Trait Anxiety Inventory (STAI-S) was used to measure anxiety in two groups of mothers carrying a fetus with the respective malformation. Results: Forty-four mothers completed the questionnaire (CCAM, n=21 and CDH, n=23). Before consultation, the mean STAI-S scores in the CCAM group (44.80±5.92) and in CDH group (44.05±4.96) were not significantly different but was significantly reduced in both groups after consultation (CCAM 44.80 vs. 41.60, P=0.014 and CDH 44.05 vs. 34.35, P=0.0001). The groups were not significantly different regarding gestational age at diagnosis. Conclusions: After initial prenatal counselling, uncertainty about prenatal outcome and lack of defined management plans in CCAM seems to be more important than higher mortality rate occurring in CDH.Peer Reviewe

The management of fetal ovarian cysts

Ovarian torsion causing the loss of an ovary represents the most common complication of fetal ovarian cysts and occurs more frequently before than after birth. Thus, treatment of fetal simple ovarian cysts should be performed antenatally; however, criteria for prenatal decompression still need to be evaluated. Previous experience of the authors showed that large simple cysts have a poor outcome, whereas preliminary attempts of their "in utero" aspiration were all successful and uneventful. The authors evaluated the outcome of fetal simple ovarian cysts after prenatal aspiration and considered criteria for this procedure. The outcome of cysts showing a prenatal ultrasound pattern of torsion also was studied. This prospective study includes 73 ovarian cysts (48 simple, 25 showing torsion) diagnosed in 72 fetuses from June 1992 to June 1999, and followed up until spontaneous resolution or surgery. Prenatal aspiration was performed in the case of simple cysts >/=5 cm in diameter. The outcome of these cysts was compared with that of similar cysts not aspirated in the authors previous study (X(2). Cysts with an US pattern of torsion persisting at birth were operated on. The outcome of simple cysts less than 5 cm and cysts with a prenatal ultrasound appearance of torsion also was evaluated. Prenatal decompression was performed without any complications in 14 cases: 12 (86%; 95% CI: 0.68 to 1.00) regressed subsequently; 2 (14%; 95% CI: 0.00 to 0.32) showed torsion postnatally. This outcome is significantly better than that of similar cysts not aspirated in the authors' previous study(10) (P =.0002). Among the 34 simple cysts less than 5 cm, 26 (76%; 95% CI: 0.62 to 0.90) resolved spontaneously; 8 (24%; 95% CI: 0.10 to 0.38) had complications, 7 of which showing torsion (diameter at evidence of torsion, 4.4 cm [median]; range, 3.3 to 5.2 cm). Among the 34 cysts showing torsion (25 with initial US pattern of torsion + 9 subsequently complicated simple cysts), 24 (71%; 95% CI: 0.56 to 0.86) required oophorectomy; 9 (26%; 95% CI: 0.11 to 0.41) spontaneously disappeared at ultrasound, one of which required surgery for intestinal obstruction secondary to adhesion of a necrotic ovary; one patient (3%; 95% CI: 0.00 to 0.09) was lost to follow-up. Prenatal aspiration of ovarian cysts appears effective and safe: a "cutoff" of 4 cm should be investigated. Cysts with ultrasound pattern of torsion persisting postnatally require surgery; options for their management, when sonographically disappearing and asymptomatic, need to be investigated. Copyright 2002 by W.B. Saunders Company

Prenatal diagnosis of left isomerism with normal heart: a case report

Objective: Left isomerism, also called polysplenia, is a laterality disturbance associated with with paired leftsidedness viscera and multiple small spleens. Left isomerism, heart congenital abnormalities and gastrointestinal malformation are strongly associated. Methods: We present a case of prenatal diagnosis of left isomerism in a fetus with a structurally normal heart. Conclusion: Left isomerism syndrone may coesist with a structurally normal heart. If prenatal left isomerism is suspected, even in presence of a normal heart, is mandatory to esclude sign of gastrointestinal abnormalities, as late poly hy dramnios, and cardiac rhytm disturbance during the pregnancy and neonatal age

Prenatal diagnosis of a large abdominal cyst – Recommendations and management

This case report describes a twin fetus diagnosed in the third trimester with an enlarging abdominal mass that was confirmed on fetal magnetic resonance imaging (MRI) to be a hepatic mesenchymal hamartoma (HMH) without involvement of the placenta. Serial ultrasonography demonstrated progressive enlargement of the mass and infant was delivered at 33 + 6 weeks gestational age following maternal complications. On the fifth day, at laparotomy, a huge mass connected to the lower portion of the liver was completely resected. Microscopic evaluation confirmed a hepatic mesenchymal hamartoma. Postoperative recovery was uneventful and the infant was discharged at 4 weeks. The antenatal diagnosis of the hepatic mesenchymal hamartoma by fetal MRI and subsequent follow-up by serial ultrasonography emphasizes the importance of combining these 2 modalities for optimal management of the pregnancy to allow a favorable outcome