Prognostic accuracy of factors associated with poor outcome in prenatally diagnosed sacrococcygeal teratoma: A systematic review and meta-analysis.

Several factors associated with poor outcome in patients with prenatally diagnosed sacrococcygeal teratoma (SCT) have been found. However, the prognostic accuracy of these factors has not been well established. Therefore, we aimed to systematically review the prognostic accuracy of factors associated with poor outcome in these patients. We queried Search Premier, COCHRANE Library, EMCARE, EMBASE, PubMed, ScienceDirect, and Web of Science databases to identify studies regarding patients with prenatally diagnosed SCT. Poor outcome was defined as termination of pregnancy (TOP), intrauterine fetal death (IUFD), or perinatal death. We estimated the odds ratio of factors associated with poor outcome. Eleven studies (447 patients) were included. Overall mortality, including TOP, was 34.9%. Factors associated with poor outcome in fetuses with prenatally diagnosed SCT were cardiomegaly, hypervascular tumor, solid tumor morphology, fetal hydrops, and placentomegaly. A tumor volume to fetal weight ratio (TFR) of >0.12 before a gestational age of 24 weeks is predictive of poor outcome. The prognostic accuracy of factors associated with poor outcome in fetuses prenatally diagnosed with SCT seems promising. Factors associated with cardiac failure such as cardiomegaly, hypervascular tumor, solid tumor morphology, fetal hydrops, placentomegaly, and TFR >0.12 were found to be predictive of poor outcome.

Angiolipoma sacro. Reporte de un caso / Sacral angiolipoma. A case report

Fundamento: Los angiolipomas son tumores benignos que se presentan en adultos con una localización, preferentemente, en el espacio epidural posterior torácico. Objetivo: Presentar un caso que debutó con un traumatismo axial lumbosacro donde se evidenció una localización poco común de la lesión y sin relación con las estructuras del canal raquídeo. Presentación del caso: Hombre de 25 años que se cayó y debido a ello se le hizo un traumatismo directo en la región sacrococcígea con dolor y aumento de volumen regional, asociado a parestesias glúteas. Los estudios radiológicos evidenciaron una fractura del cóccix y la presencia de una lesión ubicada en las partes blandas, de aspecto redondeado, homogéneo, sólido, de poco más de 50 mm de diámetro. Se le realizó tratamiento quirúrgico que consistió en coccigectomía subperióstica y exéresis macroscópica de la masa. El estudio histológico concluyó el diagnóstico de un angiolipoma. Conclusiones: Los angiolipomas son tumores raros que tienen características radiológicas peculiares, requieren de alta sospecha clínico-imagenológica para indicar los estudios y el tratamiento. La exéresis total es recomendada para evitar la recurrencia y mejorar el pronóstico.

Background: Angiolipomas are benign tumors that appear in adults with special location in the posterior thoracic epidural position. Objective: To present a case that appeared with a lumbosacral axial trauma where a non-common lesion location was evidenced with no relation among the structures of the spinal canal. Case presentation: 25-year-old man who fell down, consequently suffered a painful direct trauma to the sacrococcygeal region and increased regional volume, associated with gluteal paresthesias. Radiological studies showed a fracture of the coccyx and presence of a lesion located in the soft tissues, with a rounded, homogeneous, solid aspect, a little more than 50 mm in diameter. Surgical treatment consisted of subperiosteal coccygectomy and macroscopic excision of the mass. Histological study concluded the diagnosis of an angiolipoma. Conclusions: Angiolipomas are rare tumors with peculiar radiological features, they require high clinical-imaging suspicion for studies and treatment. Total excision is recommended to avoid recurrence and improve prognosis.

A single-center experience of magnetic resonance imaging findings of fetal sacrococcygeal teratomas.

BACKGROUND: Sacrococcygeal teratomas (SCT) are known as rare tumors, but they are the most common tumor in fetuses and newborns. This study aims to present fetal magnetic resonance imaging (MRI) findings of SCT diagnosed prenatally and compare them with that of the prenatal Ultrasound (US) findings. METHODS: Eleven patients diagnosed as SCT prenatally by US and further assessed by MRI are included. MRI was performed via a 1.5-T magnet with a body coil. The presence, size, content extension, and compressive effects of each mass were determined and correlated with US findings and with postnatal examinations, including surgery and pathology. As complications, the presence of ascites and skin edema or pleural or pericardial effusion was diagnosed as hydrops. The amniotic fluid index was calculated for the assessment of oligo- or polyhydramnios. RESULTS: US findings are found strongly correlated with MRI findings. An agreement on the extent of each mass was observed in eight patients, disagreement in one fetus was an extension of the tumor within the spinal canal recognized only at MR and assessment of intrapelvic-abdominal extension was superior in MRI. There were n = 6 type I, n = 2 type II, n = 1 type III and, n = 2 type IV tumors. MRI was superior to US for detecting displacement of the colon (n = 3), intrapelvic-intraabdominal extension (n = 1), urinary tract complication (n = 2), and intraspinal extension (n = 1). DISCUSSION: MRI is found to be superior to US especially in the assessment of intrapelvic and intraspinal extension of the tumor, colonic displacement, and complications.

[Prenatal diagnosis and prognostic factors analysis of fetal sacrococcygeal teratoma].

Objective: To investigate the prenatal diagnosis and prognostic factors of fetal sacrococcygeal teratoma (SCT). Methods: A retrospective analysis was performed on 41 pregnant women who were diagnosed with fetal SCT by prenatal ultrasound at the Women's Hospital, Zhejiang University School of Medicine from January 2014 to September 2021. The prenatal imaging features and pregnancy outcomes, including tumor volume to fetal weight ratio (TFR), proportion of solid tumor, tumor growth rate (TGR), fetal hydrops, placentomegaly and polyhydramnios were analyzed. Receiver operating characteristic (ROC) curve was used to determine the critical values of TFR and TGR for predicting adverse fetal outcomes. Results: (1) Among the 41 pregnant women with fetal SCT, the diagnostic gestational week of ultrasound was (24.2±2.9) weeks (range: 18-28 weeks). Among them, 1 case progressed to fetal hydrops and induced labor at 22 weeks of gestation, 1 case developed intrauterine death and induced labor at 29 weeks of gestation, and 39 pregnancies continued until delivery. Among the 39 cases of continued pregnancy, 1 case underwent cesarean section at 31 weeks of gestation due to malignant polyhydramnios and increased fetal cardiothoracic ratio in the third trimester, 1 case underwent cesarean section at 32 weeks of gestation due to fetal heart failure, and 1 case underwent cesarean section at 32 weeks of gestation due to fetal heart failure and hydrops. The other 36 cases underwent surgical resection of tumor within 3 weeks after birth with good prognosis. (2) TFR>0.12 before 28 weeks of gestation could predict poor fetal prognosis, with a sensitivity of 100.0%, a specificity of 86.1% and an area under curve (AUC) of 0.922 (P<0.01). Among the fetuses with TFR>0.12, 5/10 had poor prognosis, while the fetuses with TFR≤0.12 all had good prognosis (100%,31/31), and the difference between the two groups was statistically significant (P<0.001). (3) TGR>48 cm3/week could predict poor fetal prognosis with a sensitivity of 100.0%, a specificity of 78.3% and an AUC of 0.880 (P<0.05). (4) Among the 28 SCT fetuses delivered in our hospital, the incidence rate of poor fetal prognosis was 0 (0/20) in those with solid tumor component<50%, and 5/8 in those with solid tumor component ≥50%, and the difference between the two groups was statistically significant (P<0.01). The incidence rate of poor fetal prognosis was 2/2 in those with placentomegaly (all with fetal hydrops), and 12% (3/26) in those without placentomegaly. The risk of poor fetal prognosis was 8.67 times higher in those with placentomegaly than those without placentomegaly, and the difference between the two groups was statistically significant (P<0.05). The incidence rate of poor fetal prognosis in those with polyhydramnios was 3/7, and 10% (2/21) in those without polyhydramnios, but there was no statistically significant difference between the two groups (P>0.05). Conclusion: TFR combined with solid tumor morphology, TGR, and presence of placentomegaly could predict the adverse pregnancy outcomes of fetal SCT.

Ultrasound determination of the dural sac to sacrococcygeal membrane distance in premature neonates.

BACKGROUND: Caudal epidural analgesia is the most common regional anesthetic performed in infants. Dural puncture, the most common serious complication, is inversely proportional to age. Measuring the distance from the sacrococcygeal membrane to the dural sac may prevent dural puncture. This study measures the sacrococcygeal membrane to dural sac distance using ultrasound imaging to determine feasibility of imaging and obtaining measurements. METHODS: Sacral ultrasound imaging of 40 preterm neonates was obtained in left lateral decubitus, a typical position for caudal blockade. No punctures were made. The sacrococcygeal membrane and termination of the dural sac were visualized, and the distance measured. The spinal levels of the conus medullaris and dural sac termination were recorded. RESULTS: 20 males and 20 females former preterm neonates with an average weight (SD; range) of 1740 (290; 860-2350) g and average age (SD; range) of 35.0 (1.35; 32.2-39) weeks gestational age at the time of imaging. The average sacrococcygeal membrane to distal dural sac distance (SD; range) was 17.4 (3.1; 10.6-26.3) mm. Overall, the weights correlated positively with the distance but the coefficient of variation was large at 23%. The conus medularis terminated below the L3 level and dural sac below the S3 level in 20% and 10% of subjects respectively with hip flexion. CONCLUSION: Ultrasound can be used to measure the sacrococcygeal membrane to dura distance in preterm neonates prior to needle insertion when performing caudal block and demonstrates large variability. Ultrasound imaging may identify patients at risk for dural puncture. When ultrasound is not available, needle insertion less than 3 mm/kg beyond the puncture of the sacrococcygeal membrane should prevent dural contact in 99.9% of neonates.

Continuous ultrasound-guided sacral intervertebral block for postoperative analgesia in neonates.

A sacrococcygeal teratoma and a sacral agenesis represent a contraindication to a caudal block. We report two cases where the optimal use of ultrasound helped place a sacral intervertebral catheter in two neonates. Radiological and ultrasound studies of the sacrum and spine should be available before performing the procedure.

Angiolipoma sacro. Reporte de un caso / Sacral angiolipoma. A case report

Fundamento: Los angiolipomas son tumores benignos que se presentan en adultos con una localización,preferentemente, en el espacio epidural posterior torácico.Objetivo: Presentar un caso que debutó con un traumatismo axial lumbosacro donde se evidenció unalocalización poco común de la lesión y sin relación con las estructuras del canal raquídeo.Presentación del caso: Hombre de 25 años que se cayó y debido a ello se le hizo un traumatismo directo en laregión sacrococcígea con dolor y aumento de volumen regional, asociado a parestesias glúteas. Los estudiosradiológicos evidenciaron una fractura del cóccix y la presencia de una lesión ubicada en las partes blandas, deaspecto redondeado, homogéneo, sólido, de poco más de 50 mm de diámetro. Se le realizó tratamiento quirúrgicoque consistió en coccigectomía subperióstica y exéresis macroscópica de la masa. El estudio histológico concluyóel diagnóstico de un angiolipoma.Conclusiones: Los angiolipomas son tumores raros que tienen características radiológicas peculiares, requierende alta sospecha clínico-imagenológica para indicar los estudios y el tratamiento. La exéresis total esrecomendada para evitar la recurrencia y mejorar el pronóstico. [AU]

A rare case of fetus in fetu in the sacrococcygeal region: CT and MRI findings.

BACKGROUND: Fetus in fetu is a rare condition in which a malformed fetus is found in the body of a living twin. The retroperitoneum is the most common location of this condition. However, the sacrococcygeal region is a rare site of the disease. The presence of vertebral bodies and limbs differentiates FIF from teratoma. Imaging modalities are important for diagnosing FIF. CASE PRESENTATION: A 12-months old boy was hospitalized because of a mass in the sacrococcygeal region. CT showed a large, complex mass with bony structure resembling sacrococcygeal bone, hip bone and the femur in the sacrococcygeal region of the boy. The blood supply of the mass was from the aorta of the host. MRI revealed the mass was connected with the dilated sacral canal of the host, which resulted in tethered cord. A preoperative diagnosis of FIF was made and surgery was performed to remove the mass. Surgical removal and subsequent pathological examination revealed the anencephalic fetus had limb buds and a sacrum but no axial skeleton, which supported the diagnosis of FIF. Conclusions CT and MRI played important roles in diagnosing FIF based on the location of the lesion.

Solitary vaginal paraganglioma with mature sacrococcygeal teratoma: a rare case report.

BACKGROUND: Vaginal paraganglioma are rare, atypical, solitary tumors which originate from the female genital tract. Sacrococcygeal teratoma are also rare neoplasms which derive from one (or more) primordial germ cell layers. Here we report a unique case of vaginal paraganglioma with sacrococcygeal teratoma. CASE PRESENTATION: A 44-year-old female experienced paroxysmal hypertension, palpitations and dizziness for almost six years. Enhanced CT and MRI highlighted two abnormal soft tissue lesions located in the left vaginal wall and coccyx anteriorly, and Iodine-131 metaiodobenzylguanidine (131I-MIBG) demonstrated abnormal radioactive uptake in perineum area. Endocrine tests showed elevated plasma normetanephrine (NMN) and 24 h urine norepinephrine. There was a well-circumscribed soft tissue lesion of approximately 3.5 cm in the left lateral vaginal wall which could be palpated during bimanual examination, together with a 1.5 cm tumor in the posterior wall of the rectum. We completely resected the two lesions in stages with the support of a senior gynecologist and general surgeons. Postoperative histopathological examinations suggested the vaginal paraganglioma and mature sacrococcygeal teratoma. Targeted sanger sequencing for the 36 mostly common paraganglioma-related genes, with a depth of 1000x, revealed no mutations. Post-operatively, plasma NMN and 24 h urine norepinephrine returned to the normal range and her symptoms completely disappeared. CONCLUSIONS: We reported an extremely rare case and the successful treatment of functional vaginal paraganglioma coexisting with adult sacrococcygeal teratoma.

Factors associated with poor outcome in fetuses prenatally diagnosed with sacrococcygeal teratoma.

AIM OF THE STUDY: Outcome of fetuses, prenatally diagnosed with sacrococcygeal teratoma (SCT), is still poorly documented. This study assesses the incidence and prenatal predictors of outcome in all fetuses prenatally diagnosed with SCT. METHODS: This is a retrospective study on all fetuses prenatally diagnosed with SCT from 1998 to 2018 in the Netherlands. Poor outcome was defined as terminations of pregnancy (TOP) because of expected unfavorable outcome, intrauterine fetal death, or early neonatal death. Potential risk factors for poor outcome were analyzed. MAIN RESULTS: Eighty-four fetuses were included. Sixteen (19.0%) TOPs were excluded from statistical analysis. Eleven of the remaining 68 fetuses had poor outcome. Overall mortality was 32.1%, with a mortality excluding TOPs of 13.1%. Thirteen fetal interventions were performed in 11 (13.1%) fetuses. Potential risk factors for poor outcome were the presence of fetal hydrops (OR: 21.0, CI: 2.6-275.1, p = 0.012) and cardiomegaly (OR: 10.3, CI: 1.9-55.8, p = 0.011). CONCLUSIONS: The overall mortality of fetuses prenatally diagnosed with SCTs including tTOP was 32.1%. This high mortality rate was mainly due to termination of pregnancy. Mortality excluding TOP was 13.1%. Potential risk factors for poor outcome were fetal hydrops and cardiomegaly.

A modified MRI protocol for the increased detection of sacrococcygeal fractures in patients with thoracolumbar junction fractures.

This study aimed to identify concurrent thoracolumbar junction (TLJ) and sacrococcygeal (SC) fractures using a modified MRI protocol and analyze the risk factors associated with tandem fractures. We retrospectively investigated patients with MRI-confirmed TLJ fractures from January 2017 to March 2020. Patients were divided into two study groups: study 1 with a modified MRI protocol and study 2 with a routine protocol. The modified protocol included an extended field of view of sagittal scans in L-spine MRI covering the full SC spine. In study 1, frequency of concurrent TLJ and SC fractures was investigated. And we analyzed risk factors and compared CT and MRI for detecting SC fractures. In study 2, co-occurrence of both fractures was investigated. A total of 176 and 399 patients with TLJ fractures were enrolled in study 1 and 2, then SC fractures were identified in 53 (30.14%) and 36 patients (9.02%), respectively. An axial loading trauma mechanism was a significant risk factor (Odds ratio 7.0, p < 0.001), and MRI was more sensitive than CT in detecting SC fractures (p < 0.002). Thus, a modified MRI protocol was useful to detect the high occurrence of SC fractures in TLJ fractures, which concurrent fractures increased by an axial loading mechanism.

Description of a giant hypothalamic hamartoma associated with an immature ruptured giant sacrococcygeal teratoma: a case report.

Giant hypothalamic hamartomas (GHH) are rare neonatal intracerebral congenital malformations responsible for gelastic epilepsy and/or endocrine disturbances. Sacrococcygeal teratomas (SCT) are fetal neoplasms associated with perinatal morbidity and mortality, especially hemorrhagic complications in giant examples (GSCT). Here, we describe an immature ruptured GSCT complicated by hemorrhagic shock at 32-week gestation boy requiring an emergency delivery, followed immediately by urgent surgical removal. A brain lesion resembling a GHH was also present on the antenatal MRI. In order to exclude metastatic immature teratoma or glioma, a biopsy was performed by a retro-sigmoidal approach, which confirmed the nature of the hamartoma. Here, we describe for the first time the association of a ruptured immature GSCT associated with a GHH.

Imaging anatomy of the vertebral canal for trans-sacral hiatus puncture of the lumbar cistern.

A standard lumbar puncture may be impossible for many anatomic or technical reasons. Previous accounts of caudal epidural anesthesia and other procedures via the sacral hiatus prompted us to test if image-guided percutaneous trans-sacral hiatus access to the lumbosacral subarachnoid cistern would be anatomically feasible. To study vertebral canal morphometry and curvature, we analyzed midsagittal computed tomography-myelogram images of 40 normal subjects and digitally measured sacral curvatures between S1 to S5 and S2 to S4 using two methods whereby a lower angle signifies a straighter sacrum. We measured midsagittal vertebral canal area, hiatus width, dural sac termination levels, and distance from sacral hiatus to the dural sac tip (needle distance). Subjects were F:M = 25:15, with a mean age of 44.9 years. The two S1-S5 full sacral curvature mean angles were 57.3° and 60.4°. Almost all sacral hiatuses were at S4, and dural sac terminations were at S1-S2. The mean S2-S4 sacral curvature was 25.1°, and the mean needle distance was 57.7 mm. Using two-way analysis of variance, there were significant sex differences for needle distances (p = .001), and full and limited sacral curvatures (p = .02, and p = .046, respectively). There were no significant linear regression correlations between age and sacral curvature, needle distance, canal area, or hiatus width. Therefore, despite a frequently prominent full sacral curvature, the combination of S1-S2 dural sac termination plus a relatively straight trajectory of the lower vertebral canal between S2 and S4 support the theoretical feasibility of percutaneous trans-sacral hiatus and vertebral canal access to the lumbosacral cistern using a standard spinal needle.

Sacrococcygeal teratoma in one twin: a case report and literature review.

BACKGROUND: Sacrococcygeal teratoma is one of the most common congenital tumors in newborns and infancy. The incidence is 1 per 20,000-40,000 live births. Ultrasonography is an optimal method for prenatal screening and diagnosis of fetal sacrococcygeal teratoma. MRI can be used to assist in the diagnosis. However, sacrococcygeal teratoma in the twin pregnancy is rare. CASE PRESENTATION: We reported a case of one twin with sacrococcygeal teratoma in dichorionic-diamniotic twin pregnancy.One twin with sacrococcygeal teratoma was diagnosed at the second trimester by ultrasonic examination and another twin was normal. A regular and careful antenatal care was conducted by the multidisciplinary team. The parents refused to perform the fetal MRI and examine the chromosome of both twin.At 37 + 1 of gestation, planned cesarean section was performed. The healthy male co-twin (twin A) weighed 2880 g.The male twin with SCT (twin B) weighed 2900 g, complying with 6 × 3 × 3 cm cystic and solid mass in sacrococcygeal region. At four days of age twin B underwent excisional surgery of the sacrococcygeal teratoma and coccyx and discharged 7 days after surgery. The mother and both babies were followed up and are all in good health until now. CONCLUSION(S): Sacrococcygeal teratoma in twin pregnancy is rare. Early antenatal diagnosis is important. Once the sacrococcygeal teratoma is diagnosed, clinicians should be aware of the associated maternal and fetal complications. Expecting parents should be counseled by the multidisciplinary team about the management and prognosis of the STC twin and co-twin. Prompt surgical excision of the sacrococcygeal teratoma after birth should be suggested.

Imaging Coccygeal Trauma and Coccydynia.

The coccygeal region has complex anatomy, much of which may contribute to or be the cause of coccyx region pain (coccydynia). This anatomy is well depicted at imaging, and management is often dictated by what structures are involved. Coccydynia is a common condition that is known to be difficult to evaluate and treat. However, imaging can aid in determining potential causes of pain to help guide management. Commonly, coccydynia (coccygodynia) occurs after trauma and appears with normal imaging features at static neutral radiography, but dynamic imaging with standing and seated lateral radiography may reveal pathologic coccygeal motion that is predictive of pain. In addition, several findings seen at cross-sectional imaging in patients with coccydynia can point to a source of pain that may be subtle and easily overlooked. Radiology can also offer a role in management of coccygeal region pain with image-guided pain management procedures such as ganglion impar block. In addition to mechanical coccyx pain, a host of other conditions involving the sacrococcygeal region may cause coccydynia, which are well depicted at imaging. These include neoplasm, infection, crystal deposition, and cystic formations such as pilonidal cyst. The authors review a variety of coccydynia causes, their respective imaging features, and common management strategies.©RSNA, 2020.

Sacrococcygeal teratoma with preterm delivery: a case report.

BACKGROUND: Sacrococcygeal teratoma is one of the most frequently prenatally diagnosed neoplasias. Obstetric ultrasound has a role in the diagnosis and management of these tumors during pregnancy. In this report, we describe a multidisciplinary approach in a case of a patient with sacrococcygeal teratomas and preterm delivery, as well as postnatal outcomes. CASE PRESENTATION: A 26-year-old Caucasian woman at 20.3 weeks of gestation with a normal gestational course and no relevant medical or surgical history was referred to our institution with a sacrococcygeal mass diagnosis. Magnetic resonance imaging confirmed the diagnosis of sacrococcygeal teratoma type I according to the Altman classification. Follow-up with ultrasound showed an increase in the size of the mass up to 190 × 150 mm, high Doppler flow, and severe polyhydramnios. At 35.1 weeks of gestation, the patient had premature rupture of membranes, and an emergency cesarean section was performed due to recurrent late decelerations detected by fetal heart rate monitoring. Afterward, surgery was performed successfully at 36 hours of life. Posterior controls revealed normal and healthy child growth. CONCLUSIONS: This case report demonstrates the importance of a multidisciplinary approach to offer the best neonatal outcomes by performing early surgery, as well as the need for follow-up by ultrasound in order to minimize complications by assessing mass growth, Doppler flow, and amniotic fluid.

Analysis of magnetic resonance signal intensity changes in the sacrococcygeal region of patients with uterine fibroids treated with high intensity focused ultrasound ablation.

Objective: To evaluate the magnetic resonance (MR) signal intensity changes in the sacrococcygeal region of patients with uterine fibroids treated with high intensity focused ultrasound (HIFU).Materials and Methods: Two hundred and sixty-seven patients with uterine fibroids treated with HIFU between January and December 2016 were retrospectively reviewed. All patients underwent enhanced pre- and post-HIFU MRI. Multivariate analysis was used to assess the relationship between the factors and the signal intensity changes in the sacrum and the soft tissue adjacent to the sacrum.Results: Among the 267 patients, 122 (46%) had MR signal intensity changes in the sacrum and/or the soft tissue adjacent to the sacrum after HIFU. Multivariate analysis showed that the position of the uterus, the distance from the dorsal side of the fibroid to the sacrum, and the ablation efficiency were significantly correlated with MR signal intensity changes in the sacrum and the soft tissue adjacent to the sacrum. Further analysis showed a significant relationship between the location of the MR signal intensity changes and uterine size, the enhancement degree of the uterus. Leg pain was only seen in patients with MR signal intensity changes both in the sacrum and the soft tissue adjacent to the sacrum.Conclusions: The location of the uterus, the distance between the dorsal side of the fibroids to the sacrum, and ablation efficiency have a significant relationship with the MR signal intensity changes. The size of the uterus and the degree of enhancement are related to the locations of MR signal changes.