A need for cautious interpretation of elevated serum germ cell tumor markers in children. Review and own experiences

2015 ◽  
Vol 9 (9) ◽  
pp. 923-932 ◽  
Author(s):  
Elzbieta Drozynska ◽  
Ewa Bien ◽  
Katarzyna Polczynska ◽  
Joanna Stefanowicz ◽  
Beata Zalewska-Szewczyk ◽  
...  
2020 ◽  
Vol 2020 ◽  
pp. 1-5 ◽  
Author(s):  
Minesh Nandi ◽  
Rahul Anil ◽  
Edward Hamaty ◽  
William Adams ◽  
David Stidd ◽  
...  

Extragonadal germ cell tumors are a rare entity that is more prevalent in infants and young children, with preference to midline structures. The category of intracranial germ cell tumors is divided into pure germ cell tumors (GCTs) versus nongerminomatous germ cell tumors (NGGCTs). They are usually present in the second decade of life with a male preponderance. We present here a rare case of intracranial NGGCT in a 21-year-old Romanian male, who presented with complaints of emesis, ataxic gait, and diplopia. A computed tomography scan of the head in the emergency department revealed a pineal/suprapineal mass along with obstructive hydrocephalus and dilated lateral and third ventricles without any bleeding. MRI of the cervical, thoracic, and lumbar spine showed no evidence of leptomeningeal metastasis. The patient had elevated serum markers of beta-hCG and AFP, which pointed towards a diagnosis of nongerm cell tumor, as in pure GCTs, these markers are normal. To relieve the obstruction from the mass effect, the patient had an endoscopic third ventriculostomy (EVT). However, after the procedure, he developed central diabetes insipidus as a complication with a triphasic response. Biopsy of the mass revealed atypical cells with granular architecture and atypical glands with positive immune histological markers for NGGCT. These findings supported the diagnosis of mixed germ cell tumor with yolk sac carcinoma and seminoma components. Patient’s transient central diabetes resolved with normalization in his urine output. He was eventually stabilized and returned to Romania for further management. In summary, intracranial germ cell tumors are rare brain tumors that should be distinguished based on histology and tumor markers as they will help in the guidance of therapy. An initial evaluation with neuroimaging, tumor markers, cytology from CSF, and biopsy is a must to distinguish further treatment and prognosis.


2013 ◽  
Vol 31 (15_suppl) ◽  
pp. 4557-4557
Author(s):  
Nabil Adra ◽  
Costantine Albany ◽  
Daniel Sonnenburg ◽  
Yan Tong ◽  
Nasser H. Hanna ◽  
...  

4557 Background: PRGCT represents 14% of germ cell tumors, with 2-year PFS of 50%. PRGCT is defined by primary mediastinal non-seminomatous germ cell tumor (PMNSGCT), non-pulmonary visceral metastasis (NPVM), AFP > 10,000 or hCG > 50,000. This analysis attempts to identify subsets of patients with more or less favorable outcomes among the poor risk groups. Methods: Retrospective analysis of all patients with testicular cancer seen at Indiana University (IU) from 2000-2010. 291 patients with PRGCT identified of whom 79 received initial therapy at IU. We analyzed the following variables: primary site testis/retroperitoneal (T/RP) vs. PMNSGCT, pulmonary vs. NPVM, and the amplitude of serum tumor markers. We identified groups of patients according to the level of tumor marker elevation with cutoff points of AFP 20,000 and hCG 200,000. Results: Mean age 29, mean AFP 8,283, mean hCG 185,667. 24% had PMNSGCT, 48% NPVM, 11% AFP>20,000, and 25% hCG>200,000. When hCG was analyzed as a continuous variable, every 10,000 unit increase in hCG caused the hazard of progression to increase by 1% (p value 0.01). Patients with NPVM had significantly worse PFS. NPVM with elevated hCG had worse outcome than NPVM with normal hCG. This did not correlate as well with AFP. PFS was worse with NPVM than elevated pre-chemotherapy tumor markers. Multiple different criteria for poor risk disease carried significantly worse impact on PFS and OS when compared to having a single criterion for poor risk disease. Conclusions: Our data indicate that patients with NPVM or more than one criteria for PRGCT have a worse outcome compared to other PRGCT subgroups. [Table: see text]


Urology ◽  
1994 ◽  
Vol 43 (1) ◽  
pp. 74-80 ◽  
Author(s):  
James A. Eastham ◽  
Timothy G. Wilson ◽  
Christy Russell ◽  
Thomas E. Ahlering ◽  
Donald G. Skinner

2006 ◽  
Vol 24 (18_suppl) ◽  
pp. 4550-4550 ◽  
Author(s):  
D. S. Sharp ◽  
B. S. Carver ◽  
S. E. Eggener ◽  
G. V. Kondagunta ◽  
R. J. Motzer ◽  
...  

4550 Background: The significance of late relapse (LR) of germ cell tumor (GCT) and its association with poor survival is becoming increasingly recognized. Methods: Between 1990 and 2004, 75 patients (pts) were managed for LR of GCT at MSKCC. Using our prospective databases and following IRB approval, clinical and pathologic parameters were reviewed. Estimates of cancer-specific survival were generated using the Kaplan-Meier method and Cox-proportional hazards model was used to assess for predictors of outcome. Results: Clinical stage (CS) at initial presentation was I in 15 (21%), II in 32 (44%), and III in 26 (36%) of pts. Management prior to LR included surveillance in 10 (13%) pts, RPLND only in 8 (11%) pts, chemotherapy only in 19 (25%) pts, and both chemotherapy (induction or adjuvant) and RPLND in 38 (51%) pts. The median time to LR was 6.8 (2.1–37.7) years. Overall, 56/75 (75%) of pts had LR in the retroperitoneum (RP), including 25 of 27 (93%) initially managed without RPLND prior to LR. At LR, 45/75 (60%) of pts were symptomatic and 46/75 (61%) pts had elevated serum tumor markers (STM) (78% AFP and 39% βHCG). Overall 5 year cancer-specific survival (CSS) was 59% (95% CI 47%–72%). Pts who underwent complete resection (with or without salvage chemotherapy) at time of LR (n = 44) had a 5 year CSS of 77% (95% CI 62%–92%). In contrast, 5 year CSS for patients treated without complete surgical resection (n = 31) was 39% (95% CI 21%–57%) (p = 0.0001). The pathology of 69 pts at LR revealed viable GCT in 39 (57%), only teratoma in 13 (19%), malignant transformation in 14 (20%), and fibrosis in 3 (4%). A multivariable analysis of pretreatment parameters showed that at time of LR, a solitary site of metastasis (HR 2.6, 95% CI 1.1–6.2, p = 0.03) and no prior chemotherapy (HR 4.2, 95% CI 1.2–14.2, p = 0.02) were predictive of improved cancer-specific survival. Elevated STM and presence of symptoms did not impact survival independently in this model. Conclusions: Although LR of GCT is associated with poor survival, pts who are chemotherapy naïve and have a solitary metastatic site at LR experience improved survival. The data suggest that meticulous control of the RP is critical to prevent LR in the RP. Survival is greatly improved if complete surgical excision of disease at LR is attained, highlighting the critical role for surgery in these patients. No significant financial relationships to disclose.


2016 ◽  
Vol 34 (2_suppl) ◽  
pp. 489-489
Author(s):  
Shingo Hatakeyama ◽  
Takahiro Yoneyama ◽  
Yasuhiro Hashimoto ◽  
Takuya Koie ◽  
Chikara Ohyama

489 Background: Specific tumor marker for seminoma is still lacking. Moreover, 10% to 15% of the patients with non-seminomatous germ cell tumor (NSGCT) can be expected to have normal marker levels. Glycan-based biomarkers for testicular germ cell tumor (TGCT) have not yet been established. We examined whether the serum N-glycan profiling can be applied to detection in patient with TGCT. Methods: We performed a N-glycan structural analysis of sera from 14 patients with GCT and age-matched 28 healthy volunteers using the glycoblotting methods and matrix-assisted laser desorption/ionization-time of flight mass spectrometry. The intensity of the N-glycans was compared between the TGCT patients and the volunteers to select TGCT associate N-glycans. Optimal cut-off values were determined by receiver operating characteristic (ROC) curves. Selected N-glycans were divided according to cut-off values, and positive numbers of TGCT associated N-glycan was added up to risk classification. Results: Six (43%) had seminoma, and eight (57%) patients had NSGCT in this study. The numbers of patients in stage I, II, III were 5, 2, and 4, respectively. Three patients had extragonadal tumor. The numbers of patients in IGCCC good, intermediate and poor risk were 10, 1, and 3, respectively. There were 3 patients (21%) with negative in any tumor markers. We identified 70 kinds of N-glycans in sera from healthy volunteers and GCT patients. A total 6 of N-glycans; m/z 2336.85, 2378.86, 2890.05, 3195.16, 3341.22, 3560.30 were selected as significantly high intensity in the patient with TGCT than in the healthy volunteers, with the area under the curve (AUC) of 0.81, 0.83, 0.86, 0.84, 0.81, and 0.78, respectively. Tumor associated N-glycans were classified as positive or negative, and scored from 0 to 6 points. Optimal cut-off score for detection was determined by ROC curve, and score > 3 were selected (AUC 0.90, P < 0.001). Based on this classification, 2 of 3 patients with negative tumor markers were categorized as a carrier for TCGT. Conclusions: Although the present study is small and preliminary, serum N-glycan analysis is a potential approaches to discover new biomarkers for TGCT. Further validation study is warranted.


Sarcoma ◽  
2000 ◽  
Vol 4 (4) ◽  
pp. 179-181 ◽  
Author(s):  
Michael Froehner ◽  
Hans-Juergen Gaertner ◽  
Andreas Manseck ◽  
Sven Oehlschlaeger ◽  
Manfred P. Wirth

Patient.A 65-year-old man was admitted with a large primary retroperitoneal tumor and an increased β-human chorionic gonadotropin (β-HCG) serum level. A germ cell tumor was suspected; however, a computed tomography-guided biopsy failed to enable tumor classification. After two courses of chemotherapy, the β-HCG serum level had returned to the normal level and a diagnostic laparotomy with incisional biopsy was performed. The immunohistochemical examination of the specimen identified the tumor as a retroperitoneal pleomorphic leiomyosarcoma.Discussion.Tumor markers play only a marginal role in the work-up of patients with soft tissue sarcomas. In men with suspected retroperitoneal sarcomas, however, the determination of germ cell tumor markers occasionally enables a preoperative distinguishing of primary retroperitoneal germ cell tumors with considerable consequences for management. In this setting, a retroperitoneal tumor associated with a moderately elevated β-HCG is a diagnostic dilemma, and surgeons should be aware of the pitfall of a β-HCG-producing leiomyosarcoma in the differential diagnosis.


2021 ◽  
Vol 15 (1) ◽  
Author(s):  
Motoi Tobiume ◽  
Shigeyuki Aoki ◽  
Genya Nishikawa ◽  
Hiroyuki Muramatsu ◽  
Kenzo Ono ◽  
...  

Abstract Background Extragonadal germ cell tumor (EGCT) is a relatively rare condition, reportedly representing 3–7% of all germ cell tumors. We report a patient who had metachronous testicular tumor with uncommon metastases 20 years after primary retroperitoneal EGCT treatment, along with a corresponding literature review. Case presentation A 49-year-old Japanese man visited our department in November 2017 with chief complaints of indolent right scrotum enlargement and a right inguinal mass. History showed that the patient visited our department of gastroenterology with chief complaints of blackish feces and ill complexion in February 1997. Computed tomography (CT) showed a right retroperitoneal tumor, which was removed in the same month. Histopathological examination showed a teratoma and yolk sac tumor. He was diagnosed with primary retroperitoneal EGCT and received three courses of chemotherapy (bleomycin/etoposide/cisplatin; BEP). Periodic imaging and the determination of tumor markers (alpha-fetoprotein [AFP], human chorionic gonadotropin [HCG], and lactate dehydrogenase [LDH]) showed no recurrence or metastasis during the 5 years postoperatively. Subsequently, he did not visit the outpatient ward. In August 1999, he underwent surgery of right hydrocele. Contrast-enhanced CT showed a 35-mm contrast effect with uneven content in the right testicle and enlarged nodes that raised suspicion for metastases in the right inguinal and right external iliac lymph nodes. All tumor markers were within normal ranges. He underwent right high orchiectomy and resection of the right inguinal lymph nodes in the same month. Histopathological findings revealed seminoma (pT1, pN2, M0, S0, and TNM stage IIB). He received postoperative chemotherapy, one course of BEP therapy, and three courses of etoposide and cisplatin therapy. Post-chemotherapy CT confirmed a complete clinical response at the right external iliac lymph nodes, and this response continued 12 months later. No recurrence or metastasis has been found so far. Conclusions We report a patient in whom a testicular tumor with uncommon metastases occurred 20 years after primary retroperitoneal EGCT treatment. After EGCT treatment, testicular relapses tend to occur after relatively long-term follow-up. After EGCT treatment, such patients must be closely monitored for testicular recurrences and onset of testicular tumor.


1993 ◽  
Vol 24 (2) ◽  
pp. 166-171 ◽  
Author(s):  
Jagdeesh N. Kulkarni ◽  
Murali R. Kamat

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