Immature Sacrococcygeal Teratoma Research Articles

A neonate presenting with large immature sacrococcygeal teratoma – a challenging case with recurrence and malignant component

A neonate presenting with large immature sacrococcygeal teratoma – a challenging case with recurrence and malignant component

Immature Sacrococcygeal Teratoma: A Case Report and Extensive Review of the Literature

Immature sacrococcygeal teratoma represents a histological form with rapid tumor growth, a risk of premature birth, an enhanced rate of complications, an increased risk of recurrence, and a higher mortality rate than the mature type. Thus, prenatal diagnosis of immature forms would significantly improve the prognosis of these cases. To this end, we performed an extensive literature review on the diagnosis, therapeutic management, and follow-up of immature teratomas. Regarding this medical conduct, we also presented our case. In conclusion, the early identification of immature sacrococcygeal teratomas with or without other associated structural abnormalities and their correct therapeutic approach are basic principles for a favorable evolution of these cases.

Pediatric presacral tumors with intraspinal extension: a rare entity with diagnostic challenges.

The presacral space is a potential space located between the rectum and the lumbosacral spine. It contains various primitive germ cell types that serve as the origin for a range of tumors. Imaging is crucial in characterizing, assessing the extent of and evaluating the treatment response to these tumors. We report a series of six cases of pediatric presacral tumors with intraspinal extension, including an immature sacrococcygeal teratoma (Altman type II), a malignant sacrococcygeal teratoma (Altman type IV), a neuroblastoma, a rhabdomyosarcoma, a clear cell sarcoma and an Ewing's sarcoma of the ilium. These tumors can be broadly categorized as tumors of germ cell, neuroblastic, mesenchymal and osteogenic origin. Despite overlapping imaging features, a review of the existing literature and careful retrospective observation revealed several distinctive features that aid in the optimal characterization of tumors. These include the tumor's epicenter, the pattern and degree of bone involvement, the status of sacral foramina and neural elements, and internal tumor characteristics such as the presence of fat, calcification, hemorrhage and necrosis.

Malignant recurrence after mature Sacrococcygeal teratoma: A meta-analysis and review of the literature.

Sacrococcygeal teratoma (SCT) is a rare extragonadal germ cell tumour mostly diagnosed during infancy and early childhood. Neonatal SCTs are mostly mature, but can also contain immature and/or malignant components. Recurrence of an SCT alters prognosis, especially when it is malignant, of which its mechanism is not yet fully understood. This study is a review and meta-analysis of the literature on malignant recurrences after an initially mature SCT. A literature search was performed to identify studies describing children with SCT and presenting specific information on histology of the initial tumour as well as the recurrence. Random effect models for mature recurrence and malignant recurrence after an initially mature SCT were employed to pool study-specific percentages in order to estimate an overall percentage and its associated 95 % confidence intervals (CI). Inverse variance method, which gives more weight to larger studies, was used to pool outcomes for the different studies. A total of 22 articles, comprising 1516 patients with SCT, were included in the meta-analysis. The pooled proportions of mature and malignant recurrences after mature SCT were 3 % (95 % CI 1-4 %) and 5% (95 % CI 3-6 %), respectively. Fifty-seven (56 %) of a total of 102 recurrences after resection of an initially mature SCT were malignant, mostly yolk sac tumour (YST). Many recurrences occurred within 1-6 years, however some occurred as long as 20 years after initial diagnosis. A substantial number of recurrences of mature SCT present as a malignant tumour. Overlooking malignant components on initial pathological evaluation and the progression of mature SCT cells to malignant cells may play a role. Treatment of mature SCTs with resection alone requires thorough follow-up of at least 6 years. Future research is needed to determine whether SCTs with malignant microfoci should be treated or followed-up differently from mature or immature SCTs. In addition, the value of serum biomarkers in follow-up after SCT needs to be further evaluated.

Half-life of alpha-fetoprotein in neonatal sacrococcygeal teratoma

BackgroundAlpha-fetoprotein (AFP) is useful as a tumor marker for sacrococcygeal teratoma (SCT). We investigated the half-life of AFP in SCT. MethodsNeonates who underwent surgical treatment for SCT between 1997 and 2016 were included in the study, whereas patients who died before or after surgery or had malignant germ cell tumors were excluded. ResultsFifty-five non-recurrent SCT patients (M:F = 18:37) were enrolled. They underwent surgery on average 7.4 ± 4.1 days after birth. Serum AFP was measured an average 4.25 ± 2.07 times per patient. We obtained 165 half-lives following the formula (M = Mo * (1/2) Δt/T). A positive correlation was observed between half-life and patient age using the formula T1/2 = 0.0597 × days +6.1643 (p < 0.001). It was different from recurrent SCT (T1/2 = 0.1196 × days −0.0633) (p < 0.05). Half-life was different between mature SCT (T1/2 = 0.0671 × days +4.3912) and immature SCT (T1/2 = 0.0433 × days +8.9339) (p < 0.05). ConclusionThe half-life of AFP in neonatal patients with SCT was prolonged in proportion to the age, and it was getting longer in recurrent tumor than non-recurrent tumor. The half-life of AFP was longer in immature teratoma than in mature teratoma. Level of EvidenceIV.

Immature Sacrococcygeal Teratoma with Microfoci of Yolk Sac Tumor in a Preterm Female Newborn- Is the Surgical Resection Enough?

Immature Sacrococcygeal Teratoma with Microfoci of Yolk Sac Tumor in a Preterm Female Newborn- Is the Surgical Resection Enough?

Ultrasound and magnetic resonance imaging in prenatal diagnosis of sacrococcygeal teratoma: Case report

Fetal tumors are relatively rare and the early prenatal diagnosis enables the prediction of possible complications and decision for appropriate treatment. This paper deals with the role of ultrasonography and magnetic resonance imaging in the early prenatal diagnosis of sacrococcygeal teratoma. A 21-year-old primigravida was found to have a large sacrococcygeal tumor measuring 25 x 30mm, presented mainly as a solid mass, when she underwent the initial ultrasound examination on the first day of 19th week of gestation. There were no other associated anomalies. The second ultrasound examination was obtained after 6 days and revealed that the tumor had doubled its size. The prominent enlargement raised suspicion of immature sacrococcygeal teratoma and thus, fetal magnetic resonance imaging was performed in the 20th gestation week. The examination showed a crococcygeal teratoma presented as a large solid mass with small microcystic areas, the largest diameter being 60mm, completely externally positioned with no intra-pelvic extension. Rapid tumor growth posed a threat to the fetal circulation and it was the main reason when opting for the termination of pregnancy at 20 weeks of gestation. Pathohystological examination confirmed the diagnosis of immature sacrococcygeal teratoma. In the prenatal period the magnetic resonance imaging of a fetal tumor initially diagnosed by ultrasound examination is an additional part of diagnostic procedure, and it is not contraindicated during pregnancy. Magnetic resonance imaging improves the delineation of the tumor. The prompt prenatal diagnosis of sacrococcygeal teratoma, as well as the detection of possible associated anomalies, is important for the pregnancy management, prediction of possible complications and choice of appropriate treatment. Ultrasonography and magnetic resonance imaging are noninvasive, compatible and complementary diagnostic modalities in evaluation of sacrococcygeal teratoma in prenatal period.

A case of immature sacrococcygeal teratoma diagnosed by prenatal ultrasonography

천미골기형종은 생식세포 종양 중 매우 드문 아형이지만, 신생아에서는 가장 흔한 선천성 기형으로 한센결절의 다능성 간세포 또는 미성숙 생식세포에서 기원하며, 35,000-40,000 출산 중 1-2명의 태아에서 발생한다. 대부분의 천미골기형종은 양성종양이지만 약 20%에서 악성이며, 신생아에서 악성천미골기형종은 매우 드물다. 천미골기형종의 산전진단은 임신 제1삼분기 또는 제2삼분기 초기에도 초음파를 이용하여 이루어진다. 천미골기형종은 태아 사망률과 유병률의 중요한 원인이 되며, 빠르게 자라는 종양 내부의 동정맥 단락에 의한 태아의 고박출심부전과 이후의 태아수종, 양수과다증, 조산과 강한 관련이 있다. 저자들은 33세 여성에서 첫아이 임신 21주 5일에 태아수종과 양수과다증을 동반한 드문 천미골미성숙기형종을 산전초음파로 진단하였기에 간단한 문헌 고찰과 함께 보고하는 바이다.

Study of the factors associated with recurrence in children with sacrococcygeal teratoma

Purpose The aim of this study was to explore effects of (1) histological involvement of resection margins with microscopic residue, (2) incomplete removal of coccyx, and (3) tumor spillage on recurrence and on survival in children operated upon for sacrococcygeal teratoma (SCT). Methods Retrospective review of 70 patients treated between 1960 and 2003. Results Fifty-four girls and 16 boys presented with SCT diagnosed prenatally (12), at birth (37), or later (21). Thirty-six percent of tumors were Altman type I, 27% type II, 18% type III, and 18% type IV. Histologically, mature teratoma was observed in 48 patients, immature teratoma in 11, yolk sac tumor (YST) in 9, embryonal carcinoma in one, and mixed tumor in one. Eighty-four percent of patients solely underwent surgical extirpation. Six (8.5%) patients died. However, mortality for the group of 42 patients treated during the past 15 years was as low as 2.5%. Tumor recurrence was observed in 5 patients, 2 of whom died. Of 3 patients with initially mature teratoma, 1 showed local immature recurrence and 2 malignant recurrences. One of the latter died. Of 2 patients with initially immature teratoma grade I, one relapsed with a benign lesion and one with YST leading to death. Possible eliciting factors had been demonstrated in 3 patients. Histological analysis of resection margins showed tumoral involvement in 11 patients (and also in one patient after resection of a recurrent tumor). Only one of those with YST focus in the resection margin showed recurrence. Intraoperative tumor spillage presented in 2 patients, who both died of metastatic disease. Spillage of tumoral cyst fluid occurred in 6, none developed recurrence. One of 5 patients whose coccyx had not been removed died of metastatic disease. One with immature teratoma developed a benign recurrent tumor. The other 3 showed no recurrence. Conclusions Microscopic involvement of the resection margins of mature or immature SCT is rarely associated with recurrence, provided there are no YST foci in the resection margins. A conservative attitude then appears to be justified. Spillage of cyst fluid was never associated with recurrence, unlike spillage of tumor and absence of removal of coccyx.

Sacrococcygeal Immature Teratoma in a Twin Pregnancy

Summary Objective Sacrococcygeal teratoma (SCT) is the most common congenital tumor of the newborn. About 20% of SCTs are malignant. We report the case of a dizygotic twin with a large immature SCT who was successfully delivered by cesarean section at 34 weeks' gestation and who underwent surgical removal of the tumor. Case Report A 21-year-old, gravida 2, para 0, abortus 1, woman was referred to Chung Shan Medical University (CSMU) Hospital at 33 weeks' gestation because of the diagnosis of a twin pregnancy with SCT in one twin. There were no remarkable findings by ultrasound examination during the second trimester. At 30 weeks of gestation, a sacral mass measuring 8.4 cm in diameter and with multi-cystic and solid components attached to the coccyx was identified sonographically. The patient was referred to CSMU Hospital and underwent planned cesarean section at 34 weeks of gestation. The sick twin was delivered smoothly and underwent surgical resection of the tumor at 1 week of age after magnetic resonance imaging studies. Immature SCT with free surgical margins was confirmed by pathologic examination. The infant did well postoperatively. Conclusion SCT diagnosed before birth can be managed by planned abdominal delivery and postnatal surgery. Patients with immature teratomas should be followed-up long-term for recurrence and distant metastases.

Paravesical suture granuloma simulating a local recurrence of the immature sacrococcygeal teratoma

It has been reported that paravesical suture granuloma after inguinal hernia repair may simulate a bladder, pelvic, or urachal malignancy in adults, and therefore, these lesions often pose a diagnostic and therapeutic dilemma. However, such a case has not been reported in the pediatric age group to date. The authors report a paravesical suture granuloma after left inguinal hernia repair in a child that mimics a local recurrence of the totally excised immature sacrococcygeal teratoma.

CT Findings of Sternal Masses

Purpose : To describe and characterize the CT findings of the sternal masses. Materials and Methods : We retrospectively reviewed the medical records, pathologic reports, and CT findings of 16 patients whose chest CT revealed sternal masses. Two primary tumors were found, namely chondrosar-comas. Twelve metastatic tumors had arisen were from lung cancer (n=4), breast cancer (n=3), hepatoma (n=2), osteosarcoma (n=1), carotid body paraganglioma (n=1), and immature sacrococcygeal teratoma (n=1). Others were Castleman ’s disease (n=1) and inflammatory pseudotumor (n=1). Results : Chondrosarcomas were large expansile osteolytic masses showing a variable degree of cortical break-through and containing punctate chondroid calcifications. Most sternal metastases (83 %) were located in the manubrium and were accompanied by metastasis in other bones (83 %). Metastatic tumors were nonspecific osteolytic soft tissue masses showing homogeneous or inhomogeneous enhancement, except for those which arose from an osteosarcoma and a lung cancer, and showed osteoblastic lesions. Castleman ’s disease was seen as an ill-defined enhanced soft tissue mass involving the sternum and adjacent soft tissue. Inflammatory pseudotumor appeared as an infiltrating lesion around the sternoclavicular joint and was accompanied by scle-rosis and the erosion of opposing sternal and clavicular ends. Conclusion : Most of the sternal masses are due to malignant neoplasms, among which metastatic tumors are more common than primary ones. Metastatic tumors affect the manubrium more commonly than the body portion, and most also affect other bones in the thorax. CT findings of metastatic tumors are non-specific and thus do not suggest their origin. Non-neoplastic masses are not readily differentiated from malignant tumors on the basis of CT findings alone and require pathological confirmation.

Ultrasonic and magnetic resonance imaging of fetal sacrococcygeal teratoma.

To evaluate ultrasonic and magnetic resonance imaging (MRI) and Doppler examination in fetal sacrococcygeal teratoma (SCT), in respect to the postnatal findings and histological type of the tumor. Nine pregnancies complicated by histologically mature fetal sacrococcygeal teratoma in four cases and by immature/malignant teratoma in five cases. Transabdominal ultrasonic imaging and Doppler velocity waveforms were recorded during the second or last trimester in all cases, first trimester ultrasound examination was carried out in six cases and last trimester MRI in five cases. These findings were compared with postnatal and operative findings of the children. Ultrasound examination did not reveal intrapelvic parts of SCT, but this was possible by MRI. Velocity waveforms of the tumor arteries were similar in all histological types and the resistance index varied from 60 to 70. The mean gestational age at antepartal diagnosis was 25.2 weeks. Large tumor size with relatively large proportion of solid components was often recognized in cases with malignant/immature histology. Antepartal MRI is useful for examination of fetal SCT, but reliable differentiation of mature and immature SCT is not possible antepartally.

Yolk sac tumor identified at autopsy after surgical excision of immature sacrococcygeal teratoma

Although the predictive value of immature elements in sacrococcygeal teratomas is unclear, there are reports of malignant recurrence after surgical resection of immature sacrococcygeal teratomas. The recurrent tumors are presumed to arise from small residual malignant foci not identified at the time of surgical resection. In this report a premature female infant was delivered at 29 weeks' gestation with a large sacrococcygeal teratoma. The tumor weighed 1,350 g. It was largely cystic with a focal nodular and variegated appearance. Histologically, the tumor was a grade 1 immature teratoma with a predominance of neuroglial elements. No malignant elements were identified in any of 26 sections examined. The infant died intraoperatively of cardiovascular complications related to the large vascular supply of the tumor but had a grossly complete resection of tumor. At autopsy, a small microscopic focus of yolk sac tumor was identified adjacent to the sacrum anteriorly. Had the infant survived, this focus might well have been a source for malignant recurrence.