Intracranial growing teratoma syndrome mimicking tumor relapse: a diagnostic dilemma

2009 ◽  
Vol 3 (5) ◽  
pp. 392-396 ◽  
Author(s):  
Doo-Sik Kong ◽  
Do-Hyun Nam ◽  
Jung-Il Lee ◽  
Kwan Park ◽  
Jong Hyun Kim ◽  
...  
Keyword(s):  
Tumor Recurrence ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Clinical Manifestations ◽  
Serum Marker ◽  
Study Cohort ◽  
Diagnostic Dilemma ◽  
Primary Tumors ◽  
Growing Teratoma Syndrome ◽  
Second Look

Object It is important to differentiate growing teratoma syndrome (GTS) from tumor recurrence in the setting of an enlarging residual mass present after treatment of intracranial germ cell tumors (GCTs). The aim of this study was to determine the incidence of intracranial GTS and present its clinical manifestations in detail. Methods The authors performed a retrospective cohort study of 52 consecutive patients with newly diagnosed intracranial GCTs who presented between January 2000 and December 2006. The records were screened to identify a study cohort in which all patients had regrowing tumor mass despite normalization of tumor markers during or after treatment of GCTs. Results In 6 (11.5%) of 52 patients the pathological diagnosis was GTS. The median patient age at diagnosis was 14.5 years (range 2 months–17 years), and the primary tumors included 4 mixed GCTs and 2 immature teratomas. After second-look surgery, histological testing revealed the lesions to be mature teratoma in all patients. Three of 6 patients subsequently underwent radiation therapy and 1 patient received additional chemotherapy for spinal seeding. Conclusions In enlarging residual masses after treatment of intracranial GCTs, GTS should be kept in mind in the differential diagnosis of tumor recurrence especially if there is a radiographic mismatch with serum marker test results. If technically feasible, second-look surgery may be necessary for an accurate diagnosis.

scholarly journals Impact of Teratoma on the Cumulative Incidence of Disease-Related Death in Patients With Advanced Germ Cell Tumors

2019 ◽  
Vol 37 (26) ◽  
pp. 2329-2337 ◽  
Author(s):  
Samuel A. Funt ◽  
Sujata Patil ◽  
Darren R. Feldman ◽  
Robert J. Motzer ◽  
Dean F. Bajorin ◽  
...  
Keyword(s):  
Germ Cell ◽  
Cumulative Incidence ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Adverse Outcomes ◽  
Term Survival ◽  
Risk Status ◽  
Primary Tumors ◽  
Platinum Based Chemotherapy

PURPOSE In men with metastatic germ cell tumors (GCTs), risk-directed treatment is determined, in part, by a distinction between seminoma and nonseminomatous GCT (NSGCT). The importance of NSGCT cell type is uncertain. We evaluated the long-term impact of teratoma on survival in patients with NSGCT. METHODS Prechemotherapy, primary tumors from patients who received platinum-based chemotherapy were studied, and the histology was confirmed by a genitourinary pathologist. The cumulative incidence of disease-related death (CIDD) was the primary end point, and a competing-risk analysis was performed. RESULTS Tumors were available from 232 patients, including 193 with NSGCT. An element of teratoma was present in 82 NSGCT primary tumors (42%). With a median follow-up of 17 years (range, 0.3 to 35 years), 58 patients with NSGCT died, 47 as a result of GCT and 11 as a result of other causes. Most GCT deaths occurred within the first 5 years and were associated with pretreatment risk status ( P < .001). Death as a result of other causes rose steadily after 15 years and was not associated with risk status ( P = .66). A higher CIDD was observed in patients who had NSGCT with teratoma than those with NSGCT without teratoma and seminoma (5-year CIDD rate, 27.4%, 17.4%, and 10.3%, respectively; P = .03). A higher CIDD was observed in patients who had NSGCT with mature teratoma compared with those with either NSGCT with immature teratoma or NSGCT without teratoma (5-year CIDD rate, 38.1%, 19.9%, and 17.4%, respectively; P = .01). CONCLUSION The presence of teratoma, particularly mature teratoma, in an NSGCT primary tumor is associated with a higher CIDD, consistent with the hypothesis that differentiation is associated with adverse outcomes. Death as a result of non-GCT causes is not associated with risk status and must be separated from GCT death when evaluating long-term survival.

Novel Diagnostic Methods and Posttreatment Clinical Phenotypes Among Intracranial Germ Cell Tumors

Neurosurgery ◽  
2020 ◽  
Vol 87 (3) ◽  
pp. 563-572 ◽  
Author(s):  
Hirokazu Takami ◽  
Avital Perry ◽  
Christopher S Graffeo ◽  
Caterina Giannini ◽  
David J Daniels
Keyword(s):  
Cohort Study ◽  
Germ Cell ◽  
Statistical Significance ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Extent Of Resection ◽  
Diagnostic Methods ◽  
Cell Fraction ◽  
Clinical Phenotypes ◽  
Second Look

Abstract BACKGROUND Central nervous system (CNS) germ cell tumors (GCT) are rare and complex pediatric neoplasms, the optimal management of which remains an area of active investigation. OBJECTIVE To present an updated cohort study, with particular attention to novel diagnostic methods and posttreatment clinical phenotypes. METHODS A single-institution cohort study of 80 primary, neurosurgically managed, CNS GCTs was conducted at Mayo Clinic, 1988-2017. RESULTS Postchemotherapy resection (eg, second-look surgery) was frequently required (27.0%), especially after adjuvant therapies for nongerminomatous GCTs (NGGCTs; 14 of 28 cases, excluding mature teratoma) and significantly associated with pineal lesions, as compared to neurohypophyseal or bifocal lesions (43.6% vs 5.9% vs 6.7%, P = .004), a finding that retained statistical significance after adjusting for index extent of resection and histology (P = .04). Essentially every NGGCT case underwent at least 1 craniotomy, either on presentation, as second-look surgery, or following local recurrence. Mature teratomatous tissue was highly incident in second-look specimens (84.2%), even among lesions initially diagnosed as germinomas. Pretreatment cerebrospinal fluid (CSF) cell fraction analysis demonstrated an association between single lesions and neutrophil predominance, whereas nongerminomatous GCTs were associated with increased monocyte fractions. CONCLUSION CNS GCTs are clinically heterogeneous lesions, resulting in numerous opportunities for improved understanding and clinical management via novel diagnostic and therapeutic protocols. Samples from second-look surgeries for recurrent germinomas frequently demonstrate teratomatous tissue, suggesting possible underdiagnosis of mixed GCTs—particularly among pineal lesions. GCT subtypes demonstrate differential cell fraction distributions on CSF analysis, a novel and perhaps diagnostically helpful finding that requires validation in external cohorts.

Second-Look Surgery for Intracranial Germ Cell Tumors

Neurosurgery ◽  
2015 ◽  
Vol 76 (6) ◽  
pp. 658-662 ◽  
Author(s):  
Hideki Ogiwara ◽  
Chikako Kiyotani ◽  
Keita Terashima ◽  
Nobuhito Morota
Keyword(s):  
Germ Cell ◽  
Tumor Markers ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Residual Tumor ◽  
Complete Response ◽  
Mixed Germ Cell Tumor ◽  
Second Look ◽  
Intracranial Germ Cell Tumors ◽  
Atypical Cells

Abstract BACKGROUND: The role of second-look surgery in intracranial germ cell tumors (GCTs) needs to be reviewed. OBJECTIVE: To present our experience of second-look surgery in patients with intracranial GCTs who showed less than complete response despite normalizing or decreasing tumor markers after chemotherapy. METHODS: Retrospective review of 7 patients who underwent second-look surgery for an intracranial GCT was performed. RESULTS: Of 23 consecutive patients with newly diagnosed intracranial GCTs treated between August 2003 and August 2013, 7 patients (30%) underwent second-look surgery. The mean age was 9.4 years. The initial diagnoses were mixed germ cell tumor in 5 and immature teratoma in 2. Second-look surgery was performed after 1 to 3 courses of chemotherapy. Magnetic resonance imaging at the surgery demonstrated increasing residual tumor in 4 and stable residual tumor in 3. Tumor markers were normalized in 5 and nearly normalized in 2. Gross total resection was achieved in all patients. Histopathology at second-look surgery revealed mature teratoma in 5, fibrosis with atypical cells in 1, and fibrosis in 1. All patients subsequently underwent additional chemoradiation therapy according to the initial diagnosis. All patients are alive with no evidence of recurrence, with a mean follow-up of 48 months. CONCLUSION: Second-look surgery plays an important role in the treatment of intracranial GCTs. Surgery may be encouraged at a relatively early phase after chemotherapy when the residual tumor increases or does not change size despite normalized or nearly normalized tumor markers in order to achieve complete resection and improve outcome.

Management of growing teratoma syndrome: Aachen University experience.

2014 ◽  
Vol 32 (4_suppl) ◽  
pp. 385-385
Author(s):  
David J. K. P. Pfister ◽  
Daniel Porres ◽  
Andrea K. Thissen ◽  
Charlotte Piper ◽  
Axel Heidenreich
Keyword(s):  
Tumor Markers ◽  
Surgical Resection ◽  
Systemic Chemotherapy ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Vena Cava ◽  
Retroperitoneal Lymph Node Dissection ◽  
Tumor Diameter ◽  
Growing Teratoma Syndrome ◽  
Complete Surgical Resection

385 Background: Growing teratoma syndrome (GTS) is an infrequent clinical phenomenon. GTS is defined as an enlarging metastatic mass during systemic chemotherapy for advanced nonseminomatous germ cell tumors (NSGCT) despite decreasing serum tumor markers. Complete surgical resection of the mass is mandatory to achieve a favourable outcome. We report on our single center experience in the management of GTS. Methods: Between January 2000 and August 2009 postchemotherapeutic retroperitoneal lymph node dissection (PCRPLND) was performed in 162 patients (pts) with advanced NSGCT. Fourteen pts (4.9%) fulfilled the criteria of a GTS: enlarging metastatic mass in the retroperitoneum or visceral organs during systemic chemotherapy with normalized or regredient tumor markers. In all cases of GTS a complete radical bilateral PCRPLND including the resection of adjacent visceral and vascular structures was performed. Results: Median patient age was 24.5 (18 to 52) years. All patients exhibited NSGCT with a good or intermediate prognosis according to IGCCCG; in all cases the primary tumor contained predominantly (greater than 50%) mature teratoma; 10 and 4 patients presented with clinical stage IIC and III, resp. Median tumor diameter at time of surgery was 6,5 (3,0-35)cm. Tumor markers were normalized in 12 out of 14 patients and markers plateauted in 2 out of 14 patients. Tumor masses were localized in the retroperitoneum in 12 pts.; two patients had additional pulmonary metastases which were resected in a second approach. Median time from start of chemotherapy to surgery was 4.8 (1.5 to 26.5) months Median surgical time was 265 (165 to 585) minutes, and median blood loss 650 (450 to 2,000) ml. Four pts required resection of the inferior vena cava or abdominal aorta with implantation of a prosthetic graft; adjunctive nephrectomy was performed in three pts. After a median follow-up of 4.2 years two pts developed recurrent disease; the remainder are alive without evidence of disease. Conclusions: GTS is a rare phenomenom among pts with advanced NSGCT and necessitates complete surgical resection of all masses with curative intention. Surgery should be considered at time of progression to facilitate complete removal of the mass. Due to the complex surgery, treatment should be performed at specialized centers.

scholarly journals GCT-70. INTRACRANIAL GROWING TERATOMA SYNDROME IN CHILDREN

2020 ◽  
Vol 22 (Supplement_3) ◽  
pp. iii342-iii342
Author(s):  
Maria Carter Febres ◽  
Carol S Bruggers ◽  
Holly Zhou ◽  
Arie Perry ◽  
John Kestle ◽  
...  
Keyword(s):  
Germ Cell ◽  
Obstructive Hydrocephalus ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Residual Tumor ◽  
Complete Resection ◽  
Pineal Tumor ◽  
Tumor Biopsy ◽  
Growing Teratoma Syndrome ◽  
Multiagent Chemotherapy

Abstract Germ cell tumors account for less than 5% of all intracranial malignancies in children. Intracranial growing teratoma syndrome (GTS) is a rare pathophysiologic process characterized by growth of mature teratoma elements of a non-germinomatous germ cell tumor (NGGCT) during or following treatment with chemotherapy, in addition to normalization of or declining AFP/βHCG of the cerebral spinal fluid (CSF)/serum. A 13-year-old male presented with headache, emesis, and diplopia. MRI of the brain/spine revealed a localized 3.1 x 3.1 x 3.2 cm pineal tumor. Biopsy confirmed NGGCT (germinoma, immature and mature teratoma). Serum AFP (227ng/ul) and βHCG (12 IU/L) and CSF AFP (21ng/ul) and βHCG (31 IU/L) were elevated. Prior to cycle two of chemotherapy, he developed unstable gait and moderate hearing loss. Repeat MRI brain demonstrated tumor enlargement (4.4 x 5.2 x 5.1 cm) and obstructive hydrocephalus, although serum AFP/βHCG had normalized. Gross total resection of tumor confirmed GTS, without residual immature/malignant elements. Following six cycles of multiagent chemotherapy (carboplatin, etoposide, ifosfamide) and proton beam craniospinal irradiation (36 Gy with 18 Gy boost), he remains free of disease at eleven months since diagnosis. The pathogenesis of GTS remains unclear. Care must be taken to avoid misdiagnosing GTS as progressive NGGCT, as treatment and prognosis differ significantly. Second-look surgery, with a goal of complete resection, should be considered in cases of NGGCT when residual tumor grows during or following therapy, as this may represent GTS. Although histologically benign, GTS can be fatal. In patients with GTS, complete resection is usually curative.

scholarly journals Metastatic Mature Teratoma and Growing Teratoma Syndrome in Patients with Testicular Non-Seminomatous Germ Cell Tumors

2021 ◽  
Vol 22 ◽  
Author(s):  
Daniel B. Green ◽  
Francisco G. La Rosa ◽  
Paul G. Craig ◽  
Francesca Khani ◽  
Elaine T. Lam
Keyword(s):  
Germ Cell ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Growing Teratoma Syndrome

scholarly journals GCT-14. SECOND-LOOK SURGERY FOR INTRACRANIAL GERM CELL TUMORS

2020 ◽  
Vol 22 (Supplement_3) ◽  
pp. iii330-iii330
Author(s):  
Hideki Ogiwara
Keyword(s):  
Germ Cell ◽  
Tumor Markers ◽  
Mature Teratoma ◽  
Germ Cell Tumors ◽  
Residual Tumor ◽  
Complete Response ◽  
Immature Teratoma ◽  
Total Resection ◽  
Mixed Germ Cell Tumor ◽  
Second Look

Abstract OBJECTIVE The authors present their experiences of second-look surgery in patients with intracranial GCTs who showed less than complete response despite normalizing or decreasing tumor markers after chemotherapy. METHODS Retrospective review of 14 patients who underwent second-look surgery for an intracranial GCT was performed. RESULTS Of 40 consecutive patients with newly diagnosed intracranial GCTs treated between August 2003 and 2019, 14 patients (35%) underwent second-look surgery. The mean age was 9.2 years. The initial diagnoses were mixed germ cell tumor in 6, immature teratoma in 4, yolk sac tumor in 2, and germinoma 2. Second-look surgery was performed after 1–3 courses of chemotherapy. Magnetic resonance imaging (MRI) at the surgery demonstrated increasing residual tumor in 8 and stable residual tumor in 6. Tumor markers were normalized in 10 and nearly-normalized in 4. Gross total resection was achieved in 12 patients and near-total resection in 2. Histopatholgy at second-look surgery revealed mature teratoma in 6, immature teratoma in 3, fibrosis with atypical cells in 2, and fibrosis in 3. Eleven patients subsequently underwent additional chemo-radiation therapy according to the initial diagnosis. All patients are alive with no evidence of recurrence with a mean follow-up of 69 months. CONCLUSIONS Second-look surgery plays an important role in the treatment of intracranial GCTs. Surgery may be encouraged at a relatively early phase after chemotherapy when the residual tumor increases or does not change the size despite normalized or nearly-normalized tumor markers in order to achieve complete resection and improve the outcome.

scholarly journals GCT-53. CASE OF INTRACRANIAL GROWING TERATOMA SYNDROME WITH DIFFICULTY IN TIMING OF RESECTION

2020 ◽  
Vol 22 (Supplement_3) ◽  
pp. iii339-iii339
Author(s):  
Hidenobu Yoshitake ◽  
Hideo Nakamura ◽  
Yuta Hamamoto ◽  
Yusuke Otsu ◽  
Jin Kikuchi ◽  
...  
Keyword(s):  
Radiation Therapy ◽  
Endoscopic Third Ventriculostomy ◽  
Obstructive Hydrocephalus ◽  
Mature Teratoma ◽  
Timing Of Surgery ◽  
Optimal Timing ◽  
Third Ventriculostomy ◽  
Mixed Tumor ◽  
Gland Tumor ◽  
Growing Teratoma Syndrome

Abstract BACKGROUND Intracranial Growing teratoma syndrome(iGTS) is a phenomenon in which a tumor with a teratoma component grows during treatment, and its pathological tissue is often a mature teratoma. Here we report a case of iGTS in which the timing of surgery was determined by tumor markers and changes in tumor size on MRI images. CASE-REPORT: 11-year-old boy with a short stature. He developed a headache and we found a pineal gland tumor on MRI. Due to obstructive hydrocephalus, an endoscopic third ventriculostomy and biopsy were performed. The pathological diagnosis was mature teratoma, but AFP was elevated at 104.2 ng/mL. Considering NGGCT, we started chemoradiation immediately. Despite the declining AFP, it gradually increased, at which point we suspected iGTS. Resection was considered, but at some point tumor growth had stopped, so radiation therapy and a second course of ICE therapy preceded the resection. Thereafter, the tumor was completely removed, and a third course of ICE therapy was performed. DISCUSSION The onset mechanism of iGTS has not been elucidated, and its prediction is difficult. Early resection of the tumor is required, but discontinuation of radiation therapy and side effects of chemotherapy also need to be considered. In our case, resection was performed after normalization of AFP and recovery of myelosuppression. The patient followed an uneventful course, but the timing of resection was controversial. CONCLUSION We experienced a case of iGTS in NGGCT, a mixed tumor with mature teratoma. The optimal timing of the resection was discussed and literature was reviewed.

Intraperitoneal Radioactive Phosphorus (32P) Versus Observation After Negative Second-Look Laparotomy for Stage III Ovarian Carcinoma: A Randomized Trial of the Gynecologic Oncology Group

2003 ◽  
Vol 21 (15) ◽  
pp. 2849-2855 ◽  
Author(s):  
Mahesh A. Varia ◽  
Frederick B. Stehman ◽  
Brian N. Bundy ◽  
Jo Ann Benda ◽  
Daniel L. Clarke-Pearson ◽  
...  
Keyword(s):  
Ovarian Cancer ◽  
Relative Risk ◽  
Ovarian Carcinoma ◽  
Tumor Recurrence ◽  
Stage Iii ◽  
Gynecologic Oncology Group ◽  
Consolidation Therapy ◽  
Risk Of Death ◽  
Second Look ◽  
Significant Difference

Purpose: The objectives of this prospective randomized study of consolidation therapy were to evaluate recurrence-free survival (RFS), overall survival (OS), and the morbidity of intraperitoneal (IP) chromic phosphate suspension (32P) therapy in patients with stage III epithelial ovarian carcinoma who have no detectable evidence of disease at the second-look laparotomy (SLL) procedure after primary chemotherapy. Patients and Methods: In a multi-institution clinical cooperative trial, 202 eligible patients with a negative SLL were randomly selected to receive either 15 mCi IP 32P (n = 104) or no further therapy (NFT; n = 98). Results: With a median follow-up of 63 months in living patients, 68 patients in the IP 32P group (65%) and 63 patients in the NFT group (64%) have developed tumor recurrence. The relative risk of recurrence is 0.90 (IP 32P to NFT) (90% confidence interval [CI], 0.68 to 1.19). The 5-year RFS rate is 42% and 36% for the IP 32P and NFT groups, respectively; the difference is not statistically significant (log-rank test, P = .27). There was no statistically significant difference in OS (P = .19). The relative risk of death is 0.85 (IP 32P to NFT) (90% CI, 0.62 to 1.16). Sixteen patients (8%) experienced grade 3 or 4 adverse effects, with eight in each respective group. Conclusion: Intraperitoneal chromic phosphate did not decrease the risk of relapse or improve survival for patients with stage III epithelial ovarian cancer after a negative SLL. Despite complete pathologic remission at SLL after initial surgery and platinum-based chemotherapy, 61% of stage III ovarian cancer patients had tumor recurrence within 5 years of negative SLL. This indicates a need for more effective initial therapy and further studies of consolidation therapy.

scholarly journals Oligometastatic Growing Teratoma Syndrome: A Case for an Aggressive Surgical Approach

2015 ◽  
Vol 9 (3) ◽  
pp. 163-165 ◽  
Author(s):  
William S. Gange ◽  
Robert H. Blackwell ◽  
John Biemer ◽  
Güliz A. Barkan ◽  
Maria M. Picken ◽  
...  
Keyword(s):  
Surgical Approach ◽  
Clinical Stage ◽  
Mature Teratoma ◽  
Disease Free Survival ◽  
Testicular Germ Cell Tumor ◽  
Testicular Germ Cell ◽  
Growing Teratoma Syndrome ◽  
Aggressive Surgical Approach ◽  
Metastatic Sites

Growing teratoma syndrome is an infrequent presentation of testicular cancer. We present a case of growing teratoma syndrome in a patient who initially presented with clinical stage I nonseminomatous testicular germ cell tumor, who subsequently developed large volume oligometastases to the retroperitoneum, thorax, and thigh. Despite two regimens of chemotherapy, his disease progressed. Complete surgical extirpation of all gross tumors confirmed mature teratoma. An aggressive surgical approach, including postchemotherapy resection of all known metastatic sites, can provide long-term disease-free survival.

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