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Prenatal Diagnosis Nov 2015To describe the epidemiology and outcomes of sacrococcygeal teratoma (SCT) and identify the factors affecting prognosis in a population-based cohort. (Review)
Review
OBJECTIVES
To describe the epidemiology and outcomes of sacrococcygeal teratoma (SCT) and identify the factors affecting prognosis in a population-based cohort.
METHODS
Analyses of fetal SCTs from a population-based congenital anomaly register between 1995 and 2012, linked to regional datasets. A systematic literature review was performed for published studies on perinatal SCT (1995 to 2012).
RESULTS
Thirty-six confirmed SCT cases were identified, giving a total prevalence of 0.30 per 10 000 births (95%CI 0.20-0.39). Twenty-three cases (63.9%) were diagnosed prenatally. There were six false positive prenatal diagnoses, and the positive predictive value of ultrasound for SCT was 79.3%. Secondary complications in prenatally diagnosed cases were polyhydramnios (27.2%), fetal hydrops (9.1%) and rapidly growing tumour (54.0%). The perinatal (PNMR) and infant mortality rates were 333.3 per 1000 births and 285.7 per 1000 live births, respectively. All stillbirths and infant deaths occurred in cases diagnosed prenatally. Factors associated with higher PNMR in registerable births were solid, vascular tumour composition (1000), polyhydramnios (667), premature delivery (667) and rapidly growing tumour (454). In the systematic review, prenatal hydrops fetalis and prematurity were the most morbid association in SCT.
CONCLUSION
Prenatal ultrasound was relatively sensitive and specific in diagnosing SCT with good survival rates in live-born cases.
Topics: Adult; Cohort Studies; England; Female; Humans; Hydrops Fetalis; Infant, Newborn; Infant, Premature; Perinatal Mortality; Polyhydramnios; Predictive Value of Tests; Pregnancy; Prevalence; Retrospective Studies; Sacrococcygeal Region; Sensitivity and Specificity; Stillbirth; Teratoma; Ultrasonography, Prenatal
PubMed: 26114890
DOI: 10.1002/pd.4641 -
Pediatric Radiology Oct 2014Ablation techniques are widely used for solid malignant tumors in adults. There is no large series assessing the effectiveness of local ablative therapies in the... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Ablation techniques are widely used for solid malignant tumors in adults. There is no large series assessing the effectiveness of local ablative therapies in the treatment of malignant or aggressive benign lesions in children.
OBJECTIVE
To review the existing evidence on the techniques and results of ablation for pediatric solid malignant or aggressive benign tumors.
MATERIALS AND METHODS
We searched MEDLINE for papers published between 1995 and 2012 that reported outcomes of radiofrequency, microwave and cryoablation, interstitial laser therapy, irreversible electroporation and percutaneous ethanol injection for patients younger than 18 years old. Data collection included factors related to the patient, tumor biology, ablation technique and cancer-specific endpoints. Additional series of predominantly adults including data on patients younger than 18 years old were also identified.
RESULTS
We identified 28 patients treated by ablation in 29 regions: 5 patients undergoing ablation for liver lesions, 9 patients for lung metastases, 11 patients for bone and/or soft tissue and 4 patients for kidney or pancreas. The ablation was performed to treat primary tumors, local recurrences and metastases. The histology of the tumors was osteosarcoma in 6 patients, Wilms tumor in 3, rhabdomyosarcoma in 3, hepatoblastoma in 3, desmoid tumor in 3, adrenocortical carcinoma in 2 and a single case each of leiomyosarcoma, Ewing sarcoma, paraganglioma, solid-pseudopapillary neoplasm, sacrococcygeal teratoma, hepatic adenoma, juxtaglomerular cell tumor and plantar fibromatosis. Eighteen of the patients (64%) experienced a complication, but only 6 (21%) of these needed treatment other than supportive care.
CONCLUSIONS
Although ablative techniques are feasible and promising treatments for certain pediatric tumors, large multicenter prospective trials will be needed to establish efficacy.
Topics: Ablation Techniques; Child; Child, Preschool; Female; Humans; Infant; Infant, Newborn; Liver Neoplasms; Male; Practice Patterns, Physicians'; Prevalence
PubMed: 24821394
DOI: 10.1007/s00247-014-3001-5 -
Ultrasound in Obstetrics & Gynecology :... Jun 2014Large solid sacrococcygeal teratomas (SCT) can cause high-output cardiac failure and fetal or neonatal death. The aim of this study was to describe the outcomes of... (Review)
Review
OBJECTIVE
Large solid sacrococcygeal teratomas (SCT) can cause high-output cardiac failure and fetal or neonatal death. The aim of this study was to describe the outcomes of minimally invasive antenatal procedures for the treatment of fetal SCT.
METHODS
A case review was performed of five fetuses with a large SCT treated antenatally using minimally invasive techniques, and a systematic literature review on fetal therapy for solid SCTs was carried out.
RESULTS
Five women were referred between 17 + 5 and 26 + 4 weeks' gestation for a large fetal SCT with evidence of fetal cardiac failure. Vascular flow to the tumors was interrupted by fetoscopic laser ablation (n = 1), radiofrequency ablation (RFA; n = 2) or interstitial laser ablation ± vascular coiling (n = 2). There were two intrauterine fetal deaths. The other three cases resulted in preterm labor within 10 days of surgery. One neonate died. Two survived without procedure-related complications but had long-term morbidity related to prematurity. The systematic literature review revealed 16 SCTs treated minimally invasively for (early) hydrops. Including our cases, six of 20 hydropic fetuses survived after minimally invasive therapy (30%). Survival after RFA or interstitial laser ablation was 45% (5/11). Of 12 fetuses treated for SCT without obvious hydrops and for which perinatal survival data were available, eight (67%) survived. Mean gestational age at delivery after minimally invasive therapy was 29.7 ± 4.0 weeks. Survival after open fetal surgery in hydropic fetuses was 6/11 (55%), with a mean gestational age at delivery of 29.8 ± 2.9 weeks.
CONCLUSIONS
Fetal therapy can potentially improve perinatal outcomes for hydropic fetuses with a solid SCT, but is often complicated by intrauterine death and preterm birth.
Topics: Adult; Child, Preschool; Embolization, Therapeutic; Female; Fetal Death; Fetal Diseases; Fetoscopy; Heart Failure; Humans; Infant; Infant, Newborn; Laser Therapy; Perinatal Death; Pregnancy; Pregnancy Outcome; Prenatal Care; Sacrococcygeal Region; Spinal Neoplasms; Teratoma
PubMed: 24488859
DOI: 10.1002/uog.13315