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Introduction We present a rare case of long‐term survival following metastasectomy for lumbar metastasis with growing teratoma syndrome. Case presentation An 18‐year‐old man presented with left scrotal mass and lumbago. Alpha‐fetoprotein was elevated to 648.8 ng/mL, while human chorionic gonadotropin and lactate hydrogenase were normal. Pathology of left inguinal orchiectomy revealed immature teratoma, and computed tomography confirmed a single metastasis in the second lumbar vertebra. After two courses of bleomycin, etoposide, cisplatin chemotherapy, alpha‐fetoprotein decreased, but computed tomography confirmed an enlarged lumbar metastasis. A vertebral biopsy demonstrated teratoma with a dominant mature component, and growing teratoma syndrome was suspected. Following additional etoposide, cisplatin chemotherapy, and normalization of alfa‐fetoprotein, total spondylectomy was performed. Vertebral pathology proved mature teratoma. After adjuvant chemotherapy, he has been recurrence‐free for 17 years. Conclusion Spondylectomy of a single metastatic vertebra contributed to long‐term survival in a testicular teratoma case.

Growing teratoma syndrome (GTS) is characterized by a reduction in serum tumor markers despite the growth of a benign mature teratomatous mass following chemotherapy for germ cell tumors. Gliomatosis peritonei (GP) typically accompanies ovarian teratomas, marked by the dissemination of mature glial tissue across the peritoneum. The concurrent presence of GTS and GP after treatment for ovarian immature teratoma (IMT) is notably rare, with approximately 20 reported cases. This case involves a 25-year-old patient who underwent surgical removal of an adnexal mass, which was later diagnosed as stage IIIA grade 3 ovarian IMT with parametrial involvement. Following two cycles of bleomycin, etoposide, and cisplatin chemotherapy, imaging identified new lesions adjacent to the liver and on the pelvic peritoneum. A second fertility-sparing surgery was performed, and paraffin pathology confirmed a mature teratoma within the excised specimen. Additionally, the resected pelvic peritoneum revealed nodules of mature glial tissue consistent with GP. The coexistence of GP with GTS post-IMT surgery presents a diagnostic challenge in distinguishing between malignant and benign components, which is critical to avoid unnecessarily aggressive surgical and chemotherapeutic treatments. Recognizing such cases may enable fertility-sparing surgery for these patients. [ABSTRACT FROM AUTHOR]

The main objective is to evaluate the feasibility of laparoscopic fertility-sparing surgery in women with growing teratoma syndrome. Retrospective cohort study. Chinese tertiary university hospital. Patients with growing teratoma syndrome who underwent fertility-sparing surgery between January 2015 and August 2023. Baseline characteristics and surgical outcomes were evaluated, including clinical information, surgical procedures, operative time, intraoperative blood loss, complications, length of hospital stay, and follow-up information. Twenty-six patients with ovarian growing teratoma syndrome underwent fertility-sparing surgery: 12 had laparoscopic surgery and 14 underwent laparotomic surgery. In the laparoscopic group, the median age of the patients during initial management of immature teratoma or mixed malignant ovarian germ cell tumor was 14.0 years (interquartile range, 13.0–24.5 years). Eleven patients were nulliparous. The primary ovarian tumor was pure immature teratoma in 10 patients and mixed ovarian germ cell tumor in 2 patients. Complete laparoscopic tumor resection was achieved in 11 patients. Patients in the laparoscopic group had shorter median operative time (76.5 vs 180.0 minutes, p =.001), lower estimated blood loss (20.0 vs 400.0 mL, p <.001), and decreased postoperative hospital stay (2.0 vs 7.0 days, p <.001) compared with laparotomic surgery. There was no conversion to laparotomy and no perioperative complications. Histologic examination confirmed mature teratoma in all cases. During a median follow-up of 21.9 months (interquartile range, 7.6–44.9 months), 11 patients were alive without disease and 1 was alive with disease. One pregnancy was achieved postoperatively. Laparoscopic fertility-sparing surgery may represent a feasible option in well-selected patients with ovarian growing teratoma syndrome. Surgery should be performed in gynecologic oncology centers by experienced staff trained in endoscopic procedures. More research and long-time follow-up are needed to determine the oncologic outcomes and safety of laparoscopic surgery in this population. [Display omitted] [ABSTRACT FROM AUTHOR]

Purpose: The aim of this case report is to emphasize the significance of the growing teratoma syndrome. Growing teratoma syndrome is frequently misdiagnosed due to its low prevalence, with an estimated incidence of 19% among all immature ovarian teratomas and a lack of experience among healthcare professionals. It is characterized by the growth of benign tumoral tissue during or after chemotherapy for malignant germ cell tumors. Case report: Our case is about a 46-year-old patient diagnosed with an immature teratoma who was treated unsuccessfully with surgery and chemotherapy. The patient was then referred to our hospital for a second opinion, where this unknown entity was diagnosed and underwent complete surgical debulking, including abdominal wall resection and subsequent repair. Conclusion: Physicians need to be aware of rapidly growing masses during or after chemotherapy because early recognition of this syndrome is essential for the adequate treatment of our patients. [ABSTRACT FROM AUTHOR]

Objective: Gliomatosis peritonei (GP) is a rare entity characterized by multiple mature glial tissue implants in association with ovarian teratomas in the peritoneum and omentum. To date, only 100 cases have been published. Not much is known about the origin, clinicopathological profile or prognosis of GP. SOX2 and OCT4 are recently recognized markers of embryonic stem cell differentiation. Here, the role of SOX2 and OCT4 in the pathogenesis of 11 cases of GP are reported and clinicopathological factors are described. Material and Methods: This was a retrospective study of six years duration (2017-2022). All the cases of GP were retrieved from archives, the diagnosis was confirmed and clinicopathological factors were noted. Immunohistochemical (IHC) investigation for glial fibrillary acid protein (GFAP) and S100 was noted wherever available. IHC for SOX2 and OCT4 was performed using an avidin-biotin technique. Results: There were 11 cases of GP identified. The median age was 29 years and 1/11 cases had nodal gliomatosis as well. There were eight cases of immature teratoma and three cases of mature cystic teratoma. SOX2 was positive in all foci of GP, while OCT4 was negative. These foci were also positive for GFAP and S100. Conclusion: A possibility of GP should be considered as a differential, clinically and radiologically, in cases of omental nodularity. Adequate sampling at the time of surgery is essential to rule out metastasis or growing teratoma syndrome. SOX2, a stem cell marker inducing neural differentiation, may play a crucial role in the development of GP in association with other transcription factors. [ABSTRACT FROM AUTHOR]

Molecular analysis of the growing teratoma syndrome has not been extensively studied. Here, we report a 14-year-old boy with a growing mass during treatment for a mixed germ cell tumor of the pineal region. Tumor markers were negative; thus, growing teratoma syndrome was suspected. A radical resection via the occipital transtentorial approach was performed, and histopathological examination revealed a teratoma with malignant features. Methylation classifier analysis confirmed the diagnosis of teratoma, and DMRT1 loss and 12p gain were identified by copy number variation analysis, potentially elucidating the cause of growth and malignant transformation of the teratoma. The patient remains in remission after intense chemoradiation treatment as a high-risk germ cell tumor. [ABSTRACT FROM AUTHOR]

Introduction: The aim of the study was to evaluate the perioperative risks and outcomes of ultra-radical surgery in patients with extensive metastatic ovarian growing teratoma syndrome (GTS). Methods: We conducted a retrospective study of patients with extensive metastatic ovarian GTS treated in our hospital between 2000 and 2022. Patients' clinical characteristics, surgical treatment, and outcomes were evaluated. Results: Overall, 13 patients were identified, and the median age at diagnosis of ovarian immature teratoma (IT) was 24 years (range: 5–37). The median interval between IT diagnosis and presenting GTS was 8 months (range: 2–60), with a median surgery delay of 5 months (range: 3–300). Peritoneum and liver were the most commonly affected sites (100%), followed by bowel (12 patients, 92.3%), diaphragm (12 patients, 92.3%), adnexa (9 patients, 69.2%), omentum (8 patients, 61.5%), uterus (7 patients, 53.8%), in the descending order. The mean operation time was 316 min (range: 180–625), and the mean blood loss volume was 992 mL (range: 200–5,000). Peritoneal metastasectomy (13 patients, 100%), diaphragmatic metastasectomy (12 patients, 92.3%), metastasis removal from the bowel (8 patients, 61.5%), partial hepatectomy (4 patients, 30.8%), bowel excision and anastomosis (1 patient, 7.7%) were also applied to achieve optimal debulking. R0 was achieved in 9 (69.2%) patients. A high rate of intraoperative blood transfusion (8 patients, 61.5%) and admission to the intensive care unit (9 patients, 69.2%) were observed, and the median postoperative hospitalization time was 8 days (range: 4–22). After a median follow-up of 3.3 years, 9 patients were free of disease, and 4 were alive with stable residual diseases. Conclusion: The survival outcomes in extensive metastatic ovarian GTS were satisfactory after ultra-radical surgery, while a proper therapeutic plan should be established due to the high perioperative risks. [ABSTRACT FROM AUTHOR]

Growing teratoma syndrome (GTS) is a rare disease that occurs in patients with immature teratoma (IMT) or mixed malignant germ cell tumors. Its characteristics are that during adjuvant chemotherapy or after chemotherapy after surgery treatment, the tumor markers are normal, the residual lesions of surgery are enlarged or other masses appear, and the postoperative pathological examination only shows mature teratoma tissue, without malignant germ cells. Complete surgical resection is the preferred option for the treatment for GTS, while immunoregulation, chemotherapy, and radiofrequency can stop the disease progression. The diagnosis of this disease is clear, but the incidence is low, and it is easy to be misdiagnosed as malignant tumor recurrence. A case of ovarian GTS after IMT is reported. Relevant literature is reviewed to improve the understanding of the disease, enrich clinical experience, and reduce misdiagnosis and underdiagnosis. [ABSTRACT FROM AUTHOR]

Ovarian immature teratoma is a common ovarian germ cell malignant tumor that occurs in adolescent females. Surgical resection is the main treatment, and postoperative adjuvant chemotherapy is required if necessary. This case is an immature differentiated teratoma in the left ovary found during cesarean section. After 3 courses of chemotherapy with BEP regimen, the liver teratoma metastasis occurred. The pathological results after the second tumor resection suggest that mature teratoma is considered as growing teratoma syndrome (GTS). During the course of follow-up, the liver lesions were suspicious and reappeared, but gradually disappeared. Follow -up for 1 year showed no recurrence. During the treatment of ovarian immature teratoma, GTS is rare, has a special pathogenesis, and is easily misdiagnosed as recurrence or progression of ovarian immature teratoma. Therefore, it is necessary to understand GTS and pay attention to it in clinical practice. [ABSTRACT FROM AUTHOR]

Abstract Background Primary liver immature teratoma is extremely rare and only 4 cases have been reported, let alone with growing teratoma syndrome (GTS) and/or gliomatosis peritonei (GP). Case presentation Here, we report a case of a 44-year-old female presenting with progressive abdominal distension and elevated serum alpha fetal protein (AFP) level. CT/MRI scans revealed a large cystic-solid mass in the right lobe of the liver, accompanied with implant or metastasis in the abdominal cavity. Pathologic examination at biopsy suggested immature teratoma. After 4 cycles of chemotherapy, an MRI showed a slight increase in tumor size. Therefore, surgical resection of the right lobe of the liver was performed. The final histological diagnosis was a mature teratoma (tumor size 28 cm × 14 cm × 13 cm), with no residual immature component, and the diagnosis of GTS was considered. The patient continued to receive 2 courses of postoperative chemotherapy. An abdominal CT scan revealed innumerable miliary nodules in bilateral adnexal areas 2 months after surgery. Histologically, large numbers of mature glia were observed, supporting the diagnosis of GP. Conclusions We report for the first time a case of primary liver immature teratoma with GTS and GP in an adult. Longer follow-up is needed to assess definitive efficacy.

Context Growing teratoma syndrome (GTS) is a rare entity seen following chemotherapy for metastatic nonseminomatous germ cell tumors, characterized by increase in size of the metastatic deposits, with normal serum tumor markers. Aims In this article, we aim to describe the various clinicoradiological presentations of GTS treated at our center. Design All patients who satisfied the GTS criteria from 2001 to 2019 were included. Characteristic imaging appearances along with sites of primary lesion and metastatic disease, stage and risk stratification at diagnosis, details of chemotherapy, details of surgical treatment and histopathology, levels of tumor markers, serum β-human chorionic gonadotropin, lactate dehydrogenase, and alpha fetoprotein levels at baseline and at the end of all chemotherapy were analyzed. Results The significant radiological findings observed were an increase in the fat and cystic components and appearance of coarse calcifications within the lesions. Majority of the cases were male patients (87.5%) with testicular primaries and GTS transformation in nodal metastases being the most common occurrence (75%). All eight cases (100%) showed an increase in size and cystic component, whereas four out of eight cases (50%) had presence of internal septations and internal calcification. Conclusion Early recognition of this entity and clinical decision making through serial radiological imaging are of utmost importance as these growing deposits are resistant to chemotherapy and radiotherapy, with complete surgical excision being the only curative and definitive treatment option.

Coexistent growing teratoma syndrome (GTS) and gliomatosis peritonei (GP) arising during chemotherapy of ovarian immature teratoma (IMT) is extremely rare and can be misdiagnosed as recurrent or progressive disease. We present a 33-year-old woman diagnosed with GTS with synchronous GP during chemotherapy of IMT. She underwent ovarian cystectomy due to ovarian immature teratoma and chemotherapy were administered. The α-fetoprotein (AFP) concentration decreased from 28.7 ng/mL to normal after the second cycle. Four days after the third cycle of chemotherapy, ultrasound and CT revealed an 8-cm mass with negative tumor markers in the pouch of Douglas. An exploratory laparotomy was conducted, and a smooth round cystic-solid 8-cm mass was noted in the pouch of Douglas. Extensive peritoneal seeding glial nodules were also observed on the surface of the uterus, peritoneum, and omentum. The patient underwent a partial omentectomy, intact resection of the tumor, and resection of most of the glial nodules. Postoperative pathology demonstrated a pure mature cystic teratoma component in the mass, as well as diffuse GP involving the uterine serosa, peritoneum, and omentum; this diagnosis of GTS with synchorous GP should be considered in IMT patients with mass newly identified during chemotherapy while tumor markers are normal after treatment.

Growing teratoma syndrome (GTS) is rare and can mimic disease recurrence in patients with a history of immature teratoma. Benign hypermetabolic lymphadenopathy found on staging and surveillance computed tomography (CT) and positron emission tomography (PET) may lead to the presumption of metastatic malignancy. We report a case of a 38 year old with mixed mature and immature teratomas who developed new peritoneal masses after adjuvant chemotherapy despite a normalization of tumor markers. In addition to low FDG uptake observed in these peritoneal masses, a PET scan showed hypermetabolic lymphadenopathy and pulmonary and spleen lesions suggesting widespread metastases. Subsequent surgical resection confirmed a mixed pathology with GTS and sarcoidosis. We reviewed the current literature evidence of GTS and sarcoidosis as a benign cause of lymphadenopathy in cancer patients. We emphasize the importance of a tissue diagnosis before instituting therapy for presumed cancer recurrence to avoid potentially fatal diagnostic traps and management errors. A multiple disciplinary team approach is imperative in managing patients with suspected recurrent immature teratomas.

While cancer cure is the primary goal, fertility preservation is also a cornerstone of the underlying principle of treatment for ovarian germ cell tumors. Growing teratoma syndrome (GTS) presents with growth of mature teratomas during or after chemotherapy. We report a case of successful treatment of GTS in the anterior abdominal wall involving reconstruction. A 23-year-old woman with a suspected right ovarian mature teratoma with torsion underwent emergency laparoscopically assisted extracorporeal ovarian cystectomy. Histopathological findings revealed a grade 1 immature teratoma. After two months, postoperative α-fetoprotein (AFP) levels increased, and disseminated lesions developed not only in the pelvic cavity but also in the abdominal wound where the tumor had been extracted using an extracorporeal technique at the time of primary surgery. The patient underwent laparoscopic right salpingo-oophorectomy, excision of multiple peritoneal nodules, and biopsy of abdominal wall mass. The left rectus abdominis muscle tumor could not be removed. All of these nodules were diagnosed as metastatic immature teratomas. Although the patient received three cycles of chemotherapy, the residual tumor in the abdominal wall grew remarkably despite post-chemotherapy normalization of AFP levels. Both rectus abdominis muscles involving the residual tumors were removed and reconstructed using a left tensor fascia lata muscle flap. Histopathologically, the residual tumors were identified as mature teratomas with no immature elements, resulting in GTS. The patient got pregnant without the need of fertility treatment and gave birth uneventfully by cesarean section. Thus, reconstruction with a tensor fascia lata muscle flap facilitated complete removal of GTS while preserving fertility.

This case report describes the management of a 30-year-old male patient with a history of an advanced nonseminomatous germ cell tumor, hip fracture complicated by extensive deep vein thrombosis and pulmonary embolism, and on apixaban presenting with asymptomatic intracardiac teratoma and abdominopelvic metastases. Multidisciplinary intervention, including successful surgical excision of the intracardiac mass, highlights the importance of coordinated care and vigilant follow-up in optimizing patient outcomes and preventing life-threatening complications., Competing Interests: The authors have reported that they have no relationships relevant to the contents of this paper to disclose.

Background: Primary liver immature teratoma is extremely rare and only 4 cases have been reported, let alone with growing teratoma syndrome (GTS) and/or gliomatosis peritonei (GP). Case presentation: Here, we report a case of a 44-year-old female presenting with progressive abdominal distension and elevated serum alpha fetal protein (AFP) level. CT/MRI scans revealed a large cystic-solid mass in the right lobe of the liver, accompanied with implant or metastasis in the abdominal cavity. Pathologic examination at biopsy suggested immature teratoma. After 4 cycles of chemotherapy, an MRI showed a slight increase in tumor size. Therefore, surgical resection of the right lobe of the liver was performed. The final histological diagnosis was a mature teratoma (tumor size 28 cm × 14 cm × 13 cm), with no residual immature component, and the diagnosis of GTS was considered. The patient continued to receive 2 courses of postoperative chemotherapy. An abdominal CT scan revealed innumerable miliary nodules in bilateral adnexal areas 2 months after surgery. Histologically, large numbers of mature glia were observed, supporting the diagnosis of GP. Conclusions: We report for the first time a case of primary liver immature teratoma with GTS and GP in an adult. Longer follow-up is needed to assess definitive efficacy. [ABSTRACT FROM AUTHOR]

Growing teratoma syndrome (GTS) is rare. It is fatal if the teratoma is unresectable. A standard systemic therapy is not established. The efficacy of interferon-alpha (IFN-α) for GTS was described but the treatment periods were relatively short. A 23-year-old Japanese male with bulky retroperitoneal lymph node and multiple lung metastases that progressed to GTS was administered 6 × 106 units of natural IFN-α 2 × /week. Since the IFN-α treatment suppressed both lesions' growth, it was continued for >10 years. The patient is well with controlled metastases (135 months since the IFN-α′s initiation). This is apparently the longest follow-up of INF-α treatment for GTS.

Germ cell tumors of the testis comprosing of seminomas, or non-seminomatous germ cell tumors are the most common solid neoplasms in young men. There are rare cases where seminoma and non-seminomatous cells are seen together. Non-seminomatous germ cell tumors which have generally good prognosis may feature with growing masses, despite chemotherapy treatment and consequent diminishing or normal tumor markers. These cases are called Growing Teratoma Syndrome (GTS). Growing Teratoma Syndrome is diagnosed with: (i) normalization of tumor markers (AFP and / or hCG) that have been elevated before chemotherapy, (ii) increased tumor volume during or after chemotherapy, and (iii) biopsy result consistent with mature teratoma. In the management of GTS, the early and complete surgical removal of the tumor constitutes the main approach. Early diagnosis and correct intervention GTS cases have a very good prognosis. Herein, we report a patient diagnosed with GTS after two courses of chemotherapy.

Context Growing teratoma syndrome (GTS) is a rare entity seen following chemotherapy for metastatic nonseminomatous germ cell tumors, characterized by increase in size of the metastatic deposits, with normal serum tumor markers. Aims In this article, we aim to describe the various clinicoradiological presentations of GTS treated at our center. Design All patients who satisfied the GTS criteria from 2001 to 2019 were included. Characteristic imaging appearances along with sites of primary lesion and metastatic disease, stage and risk stratification at diagnosis, details of chemotherapy, details of surgical treatment and histopathology, levels of tumor markers, serum β-human chorionic gonadotropin, lactate dehydrogenase, and alpha fetoprotein levels at baseline and at the end of all chemotherapy were analyzed. Results The significant radiological findings observed were an increase in the fat and cystic components and appearance of coarse calcifications within the lesions. Majority of the cases were male patients (87.5%) with testicular primaries and GTS transformation in nodal metastases being the most common occurrence (75%). All eight cases (100%) showed an increase in size and cystic component, whereas four out of eight cases (50%) had presence of internal septations and internal calcification. Conclusion Early recognition of this entity and clinical decision making through serial radiological imaging are of utmost importance as these growing deposits are resistant to chemotherapy and radiotherapy, with complete surgical excision being the only curative and definitive treatment option. [ABSTRACT FROM AUTHOR]

Coexistent growing teratoma syndrome (GTS) and gliomatosis peritonei (GP) arising during chemotherapy of ovarian immature teratoma (IMT) is extremely rare and can be misdiagnosed as recurrent or progressive disease. We present a 33-year-old woman diagnosed with GTS with synchronous GP during chemotherapy of IMT. She underwent ovarian cystectomy due to ovarian immature teratoma and chemotherapy were administered. The α-fetoprotein (AFP) concentration decreased from 28.7 ng/mL to normal after the second cycle. Four days after the third cycle of chemotherapy, ultrasound and CT revealed an 8-cm mass with negative tumor markers in the pouch of Douglas. An exploratory laparotomy was conducted, and a smooth round cystic-solid 8-cm mass was noted in the pouch of Douglas. Extensive peritoneal seeding glial nodules were also observed on the surface of the uterus, peritoneum, and omentum. The patient underwent a partial omentectomy, intact resection of the tumor, and resection of most of the glial nodules. Postoperative pathology demonstrated a pure mature cystic teratoma component in the mass, as well as diffuse GP involving the uterine serosa, peritoneum, and omentum; this diagnosis of GTS with synchorous GP should be considered in IMT patients with mass newly identified during chemotherapy while tumor markers are normal after treatment. [ABSTRACT FROM AUTHOR]

Abstract Background This study was performed to clarify the treatment outcome of patients with primary mediastinal germ cell tumors (PMGCTs), focusing on the clinical manifestations and management during definitive therapy and long‐term follow‐up. Methods In this study, we retrospectively reviewed the medical records of patients with PMGCTs treated at Shinshu University School of Medicine, and examined the clinical profiles and treatment outcomes of 22 patients (mean age of 29 years) with primary mediastinal GCTs treated at our hospital between 1983 and 2019. Results Five patients were diagnosed with pure seminoma and 17 had nonseminomatous GCT. A total of 21 patients were treated with cisplatin‐based chemotherapy and 15 patients (68.2%) underwent thoracic surgery after chemotherapy. Although all cases of nonseminomatous GCT were negative for tumor markers after cisplatin‐based chemotherapy, two cases showed variable GCT cells and two had somatic components (angiosarcoma and rhabdomyosarcoma) in resected specimens. Three relapsed soon after surgery. Growing teratoma syndrome developed during chemotherapy in four cases. Urgent thoracic surgery was performed in three patients, but one case was inoperable. The calculated 10‐year overall survival rates were 100% in mediastinal seminoma and 64.7% in NSGCT. During follow‐up, second non‐GCT malignancies developed in three patients (colon cancer, 190 months; thyroid cancer, 260 months; non‐small cell lung cancer, 250 months after the initial chemotherapy) and one patient with primary mediastinal seminoma was associated with multiple type I endocrine tumors. Conclusions Our experiences demonstrated that long‐term survival and/or cure can be achieved with adequate chemotherapy followed by local surgical treatment even in patients with mediastinal GCTs. However, the clinical manifestations and biological behaviors during and/or after chemotherapy were complex and varied. In addition, the development of secondary malignancies should be taken into consideration for long‐term follow‐up. Clinicians should be aware of the various clinical features and secondary malignancies in primary mediastinal GCTs.

Growing teratoma syndrome (GTS) is rare and can mimic disease recurrence in patients with a history of immature teratoma. Benign hypermetabolic lymphadenopathy found on staging and surveillance computed tomography (CT) and positron emission tomography (PET) may lead to the presumption of metastatic malignancy. We report a case of a 38 year old with mixed mature and immature teratomas who developed new peritoneal masses after adjuvant chemotherapy despite a normalization of tumor markers. In addition to low FDG uptake observed in these peritoneal masses, a PET scan showed hypermetabolic lymphadenopathy and pulmonary and spleen lesions suggesting widespread metastases. Subsequent surgical resection confirmed a mixed pathology with GTS and sarcoidosis. We reviewed the current literature evidence of GTS and sarcoidosis as a benign cause of lymphadenopathy in cancer patients. We emphasize the importance of a tissue diagnosis before instituting therapy for presumed cancer recurrence to avoid potentially fatal diagnostic traps and management errors. A multiple disciplinary team approach is imperative in managing patients with suspected recurrent immature teratomas. [ABSTRACT FROM AUTHOR]

We present two cases of 10 and 27 years old girls with recurrence of immature teratoma after an incomplete surgical staging. In both cases, there were huge abdominopelvic masses despite decrease in tumor markers with chemotherapy. Complete surgical resection of these masses was done, and histopathology showed only mature teratoma. In growing teratoma syndrome, the metastatic tumors do not decrease in size despite the normalization of serum tumor markers with chemotherapy. Because of its high recurrence, timely complete surgical resection is preferred treatment. [ABSTRACT FROM AUTHOR]

Growing teratoma syndrome (GTS) is a condition characterized by tumor growth during or after chemotherapy for a germ cell tumor, albeit with normal tumor marker levels in the absence of histopathological evidence of immature teratoma components. We encountered a 10-cm large GTS lesion in the para-aorti nodes after fertility-preserving surgery for a grade 3 ovarian immature teratoma. The patient was a 20 year old woman who presented to the hospital with complaints of abdominal pain and swelling. Imaging examination revealed an ovarian tumor mass measuring 24 cm in the abdominal cavity, suspected to be composed of a mixture of fat and other components. The α-fetoprotein (AFP) levels were elevated at 853 ng/mL. We elected to perform fertility-preserving surgery. The surgical findings included a tumor in the right ovary, which was excised without rupture with adnexectomy. The histopathological diagnosis was grade 3 immature teratoma. Palpation of the pelvic and para-aortic lymph node areas did not reveal significant lymphadenopathy. Subsequently, a para-aortic node metastasis (major axis: 8 cm) was discovered before chemotherapy (19 days after surgery). We confirmed that there was no swelling before surgery and assumed that the immature teratoma had recurred. Chemotherapy was initiated, and the serum AFP levels normalized after 4 courses of bleomycin, etoposide, and cisplatin (BEP) therapy. However, the para-aortic node metastasis had grown further (major axis: 10 cm). Another open surgery was performed. The nodal mass was completely excised and pathology revealed only mature teratoma. Growing teratoma syndrome should be considered in the setting of a recurrent mass with negative tumor markers. [ABSTRACT FROM AUTHOR]

Introduction: A mixed germ cell tumor with a teratoma component can become enlarged following chemotherapy, and such an event is diagnosed as growing teratoma syndrome. Removing large, hypervascular tumors including a tumor encased by developed vasculatures from the pineal region is challenging during a single operation. Case report: A 15-year-old male underwent chemotherapy for mixed germ cell tumors according to the KSPNO G082 protocol. This case of a mixed germ cell tumor with growing teratoma syndrome was recognized very early during chemotherapy. The tumor was completely removed during the staged operations. First, the anteriorly located tumor on the third ventricle was removed via the transcallosal interforniceal approach, and 1 month later, the occipital transtentorial approach was used for the pineal tumor with decreased vascularity. Conclusion: Performing staged operations could be recommended for large hypervascular pineal tumors, which can be safely removed during the second operation once vascularity has decreased. [ABSTRACT FROM AUTHOR]

Introduction Surgery for postchemotherapy residual nonseminomatous germ cell tumors may be difficult due to exceptional lesion size and location. Case presentation A 47‐year‐old man presented with swelling and pain in the left scrotum. Computed tomography revealed a solid occupied lesion in the left scrotum with huge metastases in the left lung and pleura. Results of a left high inguinal orchiectomy indicated a pathological diagnosis of germ cell tumors of several histological types. The patient declined postoperative chemotherapy but returned to our department 10 months later with dyspnea. Serum tumor marker levels were restored to normal range by adjuvant chemotherapy. Thereafter, an extrapleural pneumonectomy was performed for the remaining tumors. He has since been asymptomatic without recurrence or dyspnea for over 5 years. Conclusion Extrapleural pneumonectomy is a valid treatment option for the management of huge pleural and pulmonary metastases of nonseminomatous germ cell tumors.

Growing teratoma syndrome is defined as conversion of a metastatic immature tumor to a mature tumor after adjuvant chemotherapy and remains an area of investigation because of its unclear pathogenesis. Because of its risk of malignant transformation, the primary treatment strategy for pediatric patients is surgical resection. In this report we present a case of a pediatric patient with recurrent growing teratoma syndrome who was treated with chemotherapy, debulking procedures, and cryoablation for the growing nodules throughout her abdominal cavity. The patient has had a good clinical outcome without recurrent malignant tumor. These masses do not always regress with chemotherapy and complete surgical excision or ablation should be attempted when possible. [ABSTRACT FROM AUTHOR]

Gliomatosis peritonei (GP) is a rare clinical condition characterized by presence of mature glial cells in the peritoneum. Growing teratoma syndrome (GTS) is described as an uncommon phenomenon that could be related to the incidence of non-seminomatous germ cell tumors. We report a case of a patient treated for immature ovarian teratoma, in whom both GP and GTS were observed, an association to date scarcely described in literature. A 15-year-old girl presented to the emergency department with severe pain in her lower abdomen and right lumbar region. Upon admission, concentration of α-fetoprotein (AFP) was 1500 ng/mL and β−human chorionic gonadotropin (β-hCG) less than 2 ng/mL. A computed tomography (CT) scan of the abdominal cavity and pelvis confirmed the presence of an anomalous mass in the abdominal cavity and pelvis. Initial surgery was performed. Histopathology revealed the presence of immature teratoma with epithelial elements. Normalization of AFP was achieved within 8 weeks. Five months after surgery, a progressive increase in AFP was noted. Magnetic resonance imaging (MRI) and CT scans of the pelvis minor showed local relapse. Evaluation of the remission after 2 blocks of preoperative chemotherapy revealed the presence of a large tumor mass in the pelvis minor, despite normalization of the AFP concentration. After opening the abdominal walls, numerous abnormal white nodules were observed in the peritoneum. Histopathology revealed the presence of mature glial tissue (gliomatosis peritonei). The remaining tumor mass was removed, and histopathology confirmed existence of mature teratoma (growing teratoma syndrome). Postoperative chemotherapy was continued. To date, the patient remains under clinical and laboratory remission. Concomitant incidence of GP and GTS, although rare, should always be taken into consideration in pediatric patients with diagnosis of either condition. [ABSTRACT FROM AUTHOR]

We report a rare case with the late occurrence of growing teratoma syndrome (GTS). A 24-year-old woman with Grade 3 immature teratoma of ovary underwent complete surgery and chemotherapy. Nineteen years later, she developed hematuria and pelvic mass that was completely resected and pathology revealed mature cystic teratoma. She has regularly followed up with tumor marker and computed tomography every three months. No evidence of disease has been detected throughout 14 years. In addition, we present a brief review of literature of ovarian GTS in the last decade. We have found that advanced stage, high grade, or early recurrence of germ cell tumor (GCT) could be the risk factors of GTS. It tends to appear within 1 year if the patients had the incomplete resection of primary disease. We stress the importance of long-term follow-up after treatment GCT to early recognition and treatment.

Growing teratoma syndrome (GTS) is a condition in which poorly differentiated cells in a mixed-germ cell tumor (GCT) regress after chemotherapy, and the number of well-differentiated components increases. A 60-year-old man had an 8.0 cm mediastinal tumor with strong 18F-fluorodeoxyglucose (FDG) uptake [maximum standardized uptake value (SUVmax): 9.2], which was diagnosed as a GCT. After chemotherapy, serum alpha fetoprotein, beta-human chorionic gonadotropin, and tumor 18F-FDG uptake decreased (SUVmax: 3.9), but the tumor volume increased. The tumor was completely resected, and pathology confirmed the diagnosis of GTS. 18F-FDG positron emission tomography after chemotherapy reflects the proliferation of highly differentiated tumor components with poor 18F-FDG uptake. [ABSTRACT FROM AUTHOR]

A 25-year-old man was initially diagnosed with a giant mediastinal nonseminomatous germ cell tumor. Chemotherapy was administered and the tumor markers were normalized; however, the tumor grew in size (20 cm), invading the left brachiocephalic vein (BCV) and the superior vena cava (SVC). Bilateral transmanubrial approach with median sternotomy and bilateral clamshell thoracotomy was required for the complete resection of the giant tumor with the SVC reconstruction. Bilateral TMA provided the surgical field of view around the bilateral phrenic nerves and bilateral BCVs. Given the final pathological diagnosis of the mature teratoma, this was considered growing teratoma syndrome where surgery is the only treatment option. [ABSTRACT FROM AUTHOR]

Introduction: Patients affected by metastatic germ cell tumors may occasionally experience enlargement of masses with concurrent normalization of tumor markers during or after chemotherapy. This phenomenon is described as growing teratoma syndrome (GTS). The aim of the pre sent study is to assess the prevalence of GTS in the pediatric population and its implications in terms of surgical outcome., Patients and Methods: The clinical notes of patients diagnosed with stage III and IV malignant germ cell tumors from January 2010 until December 2020 at our Institution were retrospectively reviewed. The prevalence of GTS, treatment strategies, survival, and outcome were analyzed., Results: Thirty-three patients with high-stage malignant germ cell tumors were diagnosed in our institution in the analyzed period. Nine patients (28%) had radiologic evidence of enlargement of persistent masses with normal markers after chemotherapy; these patients were classified as GTS patients. All nine patients underwent resection of metastatic lymph nodes, and six had surgery on visceral metastases. In six patients, radical excision of all metastatic sites was achieved; five patients are alive and in complete remission, while one died because of peri-operative complications. Out of the three patients who could not achieve radical excision of the metastases, two died of progressive disease, and one is alive with progressive disease., Conclusions: Patients affected by GTS have a risk of progression of chemotherapy-resistant disease and death. Radical surgical excision is essential to achieve disease control and long-term survival., (© 2024 The Author(s). Pediatric Blood & Cancer published by Wiley Periodicals LLC.)

Ekhlas Ahmed Abdulraheem,1 Maram K Sait,2 Khalid H Sait,2 Nisreen M Anfinan,1 Osama R Elkadi3 1Department of Obstetrics and Gynecology, Gynecology Oncology Unit, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia; 2Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia; 3Department of Pathology, Drexel University College of Medicine, PA, USA Abstract: Growing teratoma syndrome is a rare entity of tumors, it arises seldomly from ovarian and testicular carcinoma. It presents with disseminating masses of mature teratoma during or following chemotherapy of malignant germ cell tumors. We are reporting a 19-year old presented with recurrent left ovarian mass and supra renal large mass close to the porta hepatis was seen on magnetic resonance imaging. This patient was treated 3 years ago for stage I immature teratoma with left ovarian cystectomy and chemotherapy. Surgical excision of the left ovary and the abdominal mass required meticulous dissection, and the mass was shaved off the porta hepatis with no intraoperative or postoperative complications. Pathology showed mature teratoma. She has no recurrent 5 years after treatment. To the best of our knowledge, this is the first case report describing close relation of growing teratoma syndrome to the porta hepatis, no such case report like this has been reported in our region. Keywords: malignant germ cell tumor, porta hepatis, growing teratoma syndrome

Mediastinal nonseminomatous germ cell tumor (NSGCT) is rare. NSGCT shows excellent response to cisplatin-based chemotherapy. However, some tumors continue to enlarge despite normal tumor markers after chemotherapy, a rare condition called growing teratoma syndrome (GTS). Recognition of this condition is imperative for prompt surgical resection to prevent further cardiopulmonary compression and to improve survival. Multidisciplinary team meeting is important for perioperative preparation and care to improve the outcome of this high-risk surgery. Here, we report two cases of mediastinal GTS and underwent surgical resection, of which one died of pericardial decompression syndrome and the other is well.

Background: This study was performed to clarify the treatment outcome of patients with primary mediastinal germ cell tumors (PMGCTs), focusing on the clinical manifestations and management during definitive therapy and long‐term follow‐up. Methods: In this study, we retrospectively reviewed the medical records of patients with PMGCTs treated at Shinshu University School of Medicine, and examined the clinical profiles and treatment outcomes of 22 patients (mean age of 29 years) with primary mediastinal GCTs treated at our hospital between 1983 and 2019. Results: Five patients were diagnosed with pure seminoma and 17 had nonseminomatous GCT. A total of 21 patients were treated with cisplatin‐based chemotherapy and 15 patients (68.2%) underwent thoracic surgery after chemotherapy. Although all cases of nonseminomatous GCT were negative for tumor markers after cisplatin‐based chemotherapy, two cases showed variable GCT cells and two had somatic components (angiosarcoma and rhabdomyosarcoma) in resected specimens. Three relapsed soon after surgery. Growing teratoma syndrome developed during chemotherapy in four cases. Urgent thoracic surgery was performed in three patients, but one case was inoperable. The calculated 10‐year overall survival rates were 100% in mediastinal seminoma and 64.7% in NSGCT. During follow‐up, second non‐GCT malignancies developed in three patients (colon cancer, 190 months; thyroid cancer, 260 months; non‐small cell lung cancer, 250 months after the initial chemotherapy) and one patient with primary mediastinal seminoma was associated with multiple type I endocrine tumors. Conclusions: Our experiences demonstrated that long‐term survival and/or cure can be achieved with adequate chemotherapy followed by local surgical treatment even in patients with mediastinal GCTs. However, the clinical manifestations and biological behaviors during and/or after chemotherapy were complex and varied. In addition, the development of secondary malignancies should be taken into consideration for long‐term follow‐up. Clinicians should be aware of the various clinical features and secondary malignancies in primary mediastinal GCTs. [ABSTRACT FROM AUTHOR]

To study the clinical, biochemical, radiological, pathological characteristics, surgical treatment details, and follow-up of growing teratoma syndrome (GTS) patients. This is a retrospective study of GTS treated in the Department of Gynaecological Oncology at a regional cancer institute from March 2000 to March 2020. A total of 303 cases of germ cell ovarian cancers were treated, and 8 (2.6%) of 303 cases recurred as GTS during this period. The patients presenting with recurrent GTS were studied for clinical, radiological, tumor markers, surgical management, histopathology, and post-operative follow-up details that were analyzed retrospectively. The Kaplan-Meier curve was used for the survival analysis. The 8 out of 303 cases of germ cell ovarian cancers recurred as GTS and the incidence rate is 2.6% during this period. In the six (75%) of eight cases, the histopathology report was immature teratoma ovaries. The five cases (62.5%) were in advanced stage. All the eight recurrent GTS cases received optimal surgical cytoreduction. The overall disease-free survival is 85.7% and one patient has recurrence after the surgery for GTS at 23rd month of follow-up visit. All the patients are alive till date. The GTS represents a rare clinical and pathological phenomenon. Nevertheless, GTS should be considered as one of the differential diagnosis in young patients having normal tumor markers with recurrent carcinomatosis following the primary treatment germ cell tumors of ovaries. The optimal cytoreduction of recurrent GTS leads to prolonged survival and possible cure in young patients. [ABSTRACT FROM AUTHOR]

Background: Growing teratoma syndrome (GTS) is a very uncommon phenomena. Given its lower prevalence, there is little data about clinichopathological features and management of GTS. Literature about disease mostly composed of case reports. In this study, we aimed to report patients characteristics and treatment modalities in our center within a relatively large cohort.Patients and Methods: We retrospectively reviewed the clinical records 21 patients who fulfilled criteria of GTS. Survival analysis was performed by using the Kaplan-Meier method with the Long-rank test. p<0.05 was considered statistically significant.Results: The median age at diagnosis was 25 (range 17-51). A total of 12 patients could have undergone surgery. Of patients who underwent surgery, 5 patients remained fully disease free, and 7 patients had experienced disease recurrences. Nine patients had unresectable disease, and treated with either platin-based chemotherapy or interferone α2b. Of those, 5 patients eventually had undergone autologous stem cell transplantation (ASCT) with surprisingly promising response rates. One patient had complete response and three patients had partial response. One patient died soon after ASCT due to infectious complication.Conclusion: GTS is an unique entity with regard to its clinicopathological features and available treatment options as we mentioned in the text. Despite various agents reported to have efficacy in case reports, surgery remains as the mainstay of treatment. According to result of our study, ASCT and platin-based chemotherapy regimens may be feasible options for patients with unresectable disease. [ABSTRACT FROM AUTHOR]

We report a rare case with the late occurrence of growing teratoma syndrome (GTS). A 24-year-old woman with Grade 3 immature teratoma of ovary underwent complete surgery and chemotherapy. Nineteen years later, she developed hematuria and pelvic mass that was completely resected and pathology revealed mature cystic teratoma. She has regularly followed up with tumor marker and computed tomography every three months. No evidence of disease has been detected throughout 14 years. In addition, we present a brief review of literature of ovarian GTS in the last decade. We have found that advanced stage, high grade, or early recurrence of germ cell tumor (GCT) could be the risk factors of GTS. It tends to appear within 1 year if the patients had the incomplete resection of primary disease. We stress the importance of long-term follow-up after treatment GCT to early recognition and treatment. [ABSTRACT FROM AUTHOR]

Purpose: Intracranial growing teratoma syndrome (iGTS) is a rare phenomenon of paradoxical growth of a germ cell tumor (GCT) during treatment despite normalization of tumor markers. We sought to evaluate the frequency, clinical characteristics and outcome of iGTS in Western countries. Methods: Pediatric patients from 22 North American and Australian institutions diagnosed with iGTS between 2000 and 2017 were retrospectively evaluated. Results: From a total of 777 cases of central nervous system (CNS) GCT, 39 cases of iGTS were identified for an overall frequency of 5%. Pineal region was a more frequent location for iGTS as compared to cases of GCT without iGTS (p < 0.00001). In patients with an initial tissue diagnosis of GCT, immature teratoma was present in 50%. Serum AFP or ßhCG was detectable in 87% of patients (median values 66 ng/mL and 44 IU/L, respectively). iGTS occurred at a median of 2 months (range 0.5–32) from diagnosis, in the majority of patients. All patients underwent surgical resection, leading to gross total resection in 79%. Following surgery, all patients resumed adjuvant therapy or post treatment follow-up for GCT. At a median follow-up of 5.3 years (range 0.2–11.8), 37 (95%) of patients are alive, including 5 with stable residual mass. Conclusion: iGTS occurs in 5% of patients with GCT in Western countries. Tumors of the pineal region and GCT containing immature teratoma appear to be associated with a higher risk of developing iGTS. Complete surgical resection is the mainstay of treatment. Overall survival of patients developing iGTS remains favorable. [ABSTRACT FROM AUTHOR]

Intracranial growing teratoma syndrome (iGTS) is rare phenomenon which is observed in non-germinomatous germ cell tumor (NGGCT) after chemotherapy. The clinical features of iGTS are rapidly increasing in size compared with relapse, no elevation of tumor marker in spite of tumor regrowth, multiple cystic lesions in cranial imaging, and histopathologically diagnosed as mature teratoma. Here we present a 14-year-old man with iGTS which was revealed at 44 months after initial chemotherapy. He was diagnosed as pineal immature teratoma by histopathological specimen, and we performed chemotherapy and radiation therapy. After this treatment, we found growing cystic lesion in tumor removal cavity at 26 months after surgery. The histopathological findings showed dermoid cyst in first salvage surgery. Following this result, we observed him without adjuvant chemotherapy. However he had continuous headache, abnormal eye movement at 44 months after initial treatment. Cranial MRI showed regrowing cyst. In second salvage surgery, mature teratoma was demonstrated on histopathological specimen, and we diagnosed as iGTS. Although most reported iGTSs show rapid increasing after initial chemotherapy, few reported cases show regrowth at chronic phase as our case. In long-term follow-up of germ cell tumor, iGTS is important as differential diagnosis. [ABSTRACT FROM AUTHOR]

To evaluate the diagnostic, surgical, and oncological outcomes of patients with growing teratoma syndrome (GTS). Patients diagnosed with ovarian immature teratoma (IMT) between 1980 and 2018 at Peking Union Medical College Hospital (PUMCH) were evaluated for the development of GTS. Their clinical characteristics, surgical and pathological data, and oncological outcomes were collected. Between 1980 and 2018, 175 cases of IMT were referred to PUMCH. Thirty-five patients subsequently developed GTS with a crude rate of approximately 20%. The median interval between the initial diagnosis of IMT and the first occurrence of GTS was 18.5 months (range, 6–78 months). Residual disease (P < 0.001) and gliomatosis peritonei (GP) at initial surgery (P = 0.023) were independent risk factors for GTS development. Fertility-sparing surgery for GTS was performed in 27 patients and four patients achieved five singleton pregnancies. The median follow-up time was 73 months (range, 11–401 months). Eleven patients developed at least one recurrence. Residual disease after GTS surgery was associated with GTS recurrence (P = 0.001). By the end of follow-up, 27 patients were alive without disease and the other eight patients were alive with disease. The presence of residual disease and GP at initial surgery are risk factors for GTS. Complete surgical resection is the cornerstone for treatment of GTS. The presence of residual disease after surgery for GTS is a risk factor for GTS recurrence. Fertility-sparing surgery should be performed because spontaneous pregnancy is possible. The overall prognosis of GTS is excellent. • The incidence of GTS in ovarian immature teratoma is 20%. • Residual disease and gliomatosis peritonei are independent risk factors for development of GTS. • Residual disease after surgery for GTS is a risk factor for GTS recurrence. • Fertility-sparing surgery should be performed in GTS as spontaneous pregnancy is possible. • Overall prognosis of GTS is excellent. [ABSTRACT FROM AUTHOR]

Background: Growing teratoma syndrome is a rare syndrome that affects patients with nonseminomatous germ-cell tumors (NSGCTs). It is characterized by recurrent growing masses that appear during or after chemotherapy in the presence of normal levels of tumor markers. Histological examination is the only way to confirm the diagnosis. Case Presentation: We present the case of a 36-year-old man who developed recurrent masses after curative treatment for NSGCT of the testicle. His tumor markers were normal. The patient was cured after multiple surgical procedures. Conclusions: Close follow-up after treatment for NSGCT is very important for early detection of this syndrome, which can occur even many years after tumor onset. Normal blood makers can be misleading, and surgery remains the only curative treatment.

Introduction and Relevance: Paraneoplastic hyponatremia is often secondary to syndrome of inappropriate antidiuretic hormone secretion (SIADH) by tumour cells. Immature ovarian teratomas (IOT) are uncommon and may present with SIADH., Case Report: A 26-year-old female presented with a 3-month history of abdominal pain and constipation. Imaging identified a mixed solid-cystic right ovarian mass containing fat and peritoneal deposits. Biochemistry showed severe, refractory hyponatremia (117 mmol/l). She underwent diagnostic fertility-preserving right salpingo-oophorectomy and resection of peritoneal nodules with the aim to achieve symptom control and hyponatraemia resolution. Pathology revealed a FIGO Stage 2 Grade 2 IOT with extensive benign peritoneal gliomatosis. Initial management was conservative. After 6 months of active follow-up, a rise in AFP, and recurrent hyponatremia supported the decision to administer three cycles of Bleomycin-Etoposide-Cisplatin chemotherapy. One month later, given radiological disease progression despite satisfactory biomarker response, cytoreductive surgery with complete macroscopic resection was performed. Pathology consisted solely of peritoneal mature glial elements: a growing teratoma syndrome (GTS). The patient remains disease-free after 2 years of surveillance., Clinical Discussion: Specimen histological assessment from the patient's initial surgery showed immature neuroectodermal tubules, which are thought to be the source of vasopressin secretion. The authors hypothesise that recurrent hyponatremia and rising AFP levels represented postoperative disease relapse. Biochemical response despite radiological disease progression was pathognomonic of a GTS., Conclusion: Paraneoplastic SIADH secondary to an IOT must be considered in female patients presenting with abdominal symptoms and hyponatremia. Management requires a multidisciplinary approach. Serum electrolytes are useful surveillance biomarkers supplementary to tumour markers., Competing Interests: The authors declare no conflicts of interest., (Copyright © 2024 The Author(s). Published by Wolters Kluwer Health, Inc.)

Growing teratoma syndrome (GTS) represents a rare yet significant complication following treatment for non-seminomatous germ cell tumors (NSGCT), characterized by the growth of mature teratoma elements despite prior chemotherapy. We present the case of a 30-year-old male who, following orchidectomy for NSGCT and subsequent chemotherapy, developed acute abdominal pain and pulmonary metastasis. Despite normal serum tumor markers, imaging revealed a large retroperitoneal mass encasing significant vessels. Surgical excision led to symptom resolution. This case underscores the diagnostic challenges GTS poses, the importance of imaging in diagnosis, and the efficacy of prompt surgical intervention in achieving favorable outcomes., Competing Interests: The authors have declared that no competing interests exist., (Copyright © 2024, Nimodia et al.)

The term "growing teratoma syndrome (GTS)" has been used as follows: patients with germ cell tumor (GCT) who present with enlarging original/metastatic masses during or after appropriate systemic chemotherapy despite normalized serum markers. In other words, the definition of the term GTS is not fully established. We analyzed and reviewed our case series regarding GTS that developed after the treatment of central nervous system (CNS) nongerminoatous germ cell tumors (NGGCTs). Our institutional review board approved this retrospective study. Between 2003 and 2018, we treated 16 patients (16 males; age ranging from 5.4 to 51.9 years, median 13.8) with CNS-NGGCT at our institution. We reviewed those patients and also reviewed the literature about GTS of CNS. We defined primary GTS (p-GTS) as the enlargement of cyst size and/or solid tumor occurred during treatment in the absence of marker elevation, and recurrent GTS (r-GTS) as the enlargement of teratoma after complete response of initial tumors. Among 16 patients with CNS-NGGCT, we surgically confirmed mature/immature teratoma components in 15 patients. Two patients underwent surgical removal of tumor before neoadjuvant therapy, and among the rest 14 patients, 6 developed p-GTS, and 2 patients underwent salvage surgery during chemo-/chemoradiotherapy. Those with histologic diagnosis of immature teratoma during salvage surgery had a shorter interval from the initiation of chemoradiotherapy compared with mature teratoma (P < 0.05). One patient developed r-GTS. In the literature review, most of the p-GTS consisted of enlargement with the multicystic component. Histologic diagnosis of immature teratoma during salvage surgery was observed in earlier stages of chemoradiotherapy (P < 0.05, log-rank test). Previous history of p-GTS might be a risk factor of r-GTS. The incidence of p-GTS, enlargement of the cystic component during treatment, is not rare. Physicians need to be aware of this important phenomenon. [ABSTRACT FROM AUTHOR]

Following a presentation with abdominal pain, a 22-year-old female was diagnosed with a massive primary liver immature teratoma with evidence of omental and pelvic metastases. Despite chemotherapy, the teratoma continued to rapidly increase in size. Significant treatment-associated myelosuppression was challenging as the patient did not want to receive any blood products (religious objections). The only feasible approach was surgical resection. Surgical resection of the primary tumor and abdominal metastases was undertaken despite unappealing perioperative risk with histological specimens demonstrating only mature teratoma. We report the first case of a liver teratoma suggestive of growing teratoma syndrome treated with myelosuppressive chemotherapy and major hepatectomy without the use of any blood products. [ABSTRACT FROM AUTHOR]