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Germ Cell Tumors, Pediatric


Basics


Description


Germ cell tumors (GCTs) are a heterogeneous group with a suspected common cell of origin, the primordial germ cell.  
  • Their location can be gonadal or extragonadal.
  • The numerous histologic subtypes are broadly classified as mature or immature teratomas and malignant GCTs.
  • See "Brain Tumor" chapter for primary CNS GCT.

Epidemiology


The incidence has a bimodal distribution with a smaller peak in early infancy and a larger peak in adolescence.  
  • In children younger than 15 years of age, GCTs occur at a rate of 2.4 cases per million children, and they account for 2-3% of all malignancies.
  • Between 15 and 19 years of age, extracranial GCTs account for approximately 14% of cancer diagnoses.
  • Teratomas and germinomas are the predominant histologic subtypes of early infancy and adolescence respectively.
  • Sacrococcygeal teratomas account for 50% of childhood teratomas. They are most prevalent in infants (1:40,000 live births); with a female predominance (4:1)

Risk Factors


  • Sex chromosome abnormalities are associated with an increased risk for GCTs.
  • Klinefelter syndrome is associated with an increased risk of mediastinal GCTs.
    • Approximately 50% of adolescent mediastinal GCTs are associated with a cytogenetic diagnosis of Klinefelter syndrome.
  • Turner syndrome with any portion of Y chromosome material, Swyer syndrome, nonscrotal partial androgen insensitivity, Frasier syndrome, and males with Denys-Drash syndrome are all associated with streak gonads at increased risk for developing GCTs.
  • History of cryptorchidism is associated with an increased risk of testicular GCT.

Genetics
Familial GCT has been described in 1.5-2% of adult GCTs and a similar contribution to adolescent GCTs is presumed.  

General Prevention


  • Prophylactic gonadectomy is recommended for streak gonads in the specific syndromes mentioned above because of the increased risk of developing GCTs.
  • Guidelines on the management of cryptorchidism recommend the following to decrease the future risk of testicular GCT:
    • Orchidopexy by 18 months of age
    • Consideration of orchiectomy of an undescended testis in all boys with a normal contralateral testis when orchidopexy is not feasible
    • Consider orchiectomy or biopsy in a post pubertal boy with cryptorchidism.
    • Counsel all men with a history of cryptorchidism and/or their parents regarding the long-term risk of testicular cancer.

Etiology


  • GCTs are hypothesized to originate from primordial germ cells containing neoplastic genetic aberrations.
  • Arrested migration of primordial germ cells is presumed to explain the midline location of extragonadal GCTs.
  • The postpubertal peak in the incidence of GCTs suggests hormonal factors are involved in their growth.
  • Isochromosome 12p or i(12)p is present in more than 80% of postpubertal GCTs. GCTs without the i(12)p typically have gain of 12p chromosomal material.
    • Mature and immature ovarian tumors are biologically distinct in their lack of association with the i(12)p.
    • A number of other genetic mutations have been observed in testicular GCTs.
  • Malignant GCTs in children younger than age 4 years typically contain cytogenetic abnormalities of chromosomes 1, 3, 6, and others.
    • The i(12)p is rarely seen in this group.
    • Deletion of 1q36 is present in 80-100% of infantile malignant GCTs in testicular and extragonadal sites.
    • Recurrent genetic changes of infantile yolk sac tumors include loss of 6q24-qter, gain of 20q and 1q, loss of 1p, and c-myc or n-myc amplification.

Diagnosis


History


  • Acute or chronic abdominal pain is the presenting symptom in up to 80% of ovarian GCTs.
    • Vaginal bleeding, amenorrhea, and constipation may also be present.
  • Pediatric testicular tumors typically present as nontender scrotal masses.
  • GCTs can present with severe, acute abdominal or testicular pain secondary to gonadal torsion.
  • A history of abnormal sexual development may be an indication of an underlying sex chromosome abnormality associated with an increased risk of a GCT.
  • Sacrococcygeal teratomas typically present with swelling.
    • Pregnancies may be associated with polyhydramnios or high-output cardiac failure.
    • Other congenital anomalies are seen in up to 18% of patients.
  • Mediastinal tumors can present with respiratory compromise in younger children, whereas adolescents may have a more insidious presentation.

Physical Exam


  • A palpable abdominal mass may be present with an ovarian GCT.
  • A palpable nontender testicular mass is typical of testicular GCT.
  • Sacrococcygeal GCTs can present with a palpable mass or signs of spinal cord compression.
  • Especially in younger children, a mediastinal GCT may present with potentially critical respiratory compromise or superior vena cava syndrome.
  • Inappropriate absence or presence of sexual development may be seen with hormonally active GCTs.

Diagnostic Tests & Interpretation


Lab
  • Serum tumor markers include AFP and beta-hCG help to diagnose, monitor, and stage GCTs.
    • An acute rise can be seen after initiating chemotherapy.
  • Workup should include a CBC and differential, chemistry panel, liver function tests, uric acid, and LDH to evaluate for other malignancies or organ dysfunction.

Imaging
  • Plain radiograph: may reveal mature calcified tissues, such as bone or teeth, within tumor
  • Chest radiograph: shows mediastinal mass
  • CT scan: necessary to evaluate the primary site and regional disease
  • Transscrotal ultrasound: initial imaging of testicular mass, can reveal calcifications or heterogenous features.
  • Prenatal MRI: helpful for prenatal counseling and preoperative planning of fetal sacrococcygeal teratomas
  • Chest CT and bone scan: indicated to evaluate for metastatic disease when malignancy is suspected

Diagnostic Procedures
Tissue is mandatory, since histology is essential to the classification of GCTs.  
Pathologic Findings
  • GCTs are broadly classified as teratomas or malignant GCTs.
  • Teratomas contain elements of all three germ cell layers (ectoderm, mesoderm, and endoderm).
    • They can be mature, immature, or immature with malignant elements.
  • Major malignant histologic subtypes include yolk sac tumor, embryonal carcinoma, gonadoblastoma, choriocarcinoma, and mixed malignant GCT.
    • Histologic subtype does not necessarily correlate with tumor biology, patient age, site of origin, or patient prognosis.

Differential Diagnosis


  • Sacrococcygeal: pilonidal cyst, meningocele, lipomeningocele, hemangioma, abscess, bone tumor, epidermal cyst, chondroma, lymphoma, ependymoma, neuroblastoma, glioma
  • Abdominal: Wilms tumor, neuroblastoma, lymphoma, rhabdomyosarcoma, hepatoblastoma, retained twin fetus
  • Vaginal: rhabdomyosarcoma (sarcoma botryoides), clear cell carcinoma
  • Ovarian: cyst, appendicitis, pregnancy, pelvic infection, hematocolpos, sarcoma, lymphoma, other ovarian tumors
  • Testicular: epididymitis, testicular torsion, infarct, orchitis, hernia, hydrocele, hematocele, rhabdomyosarcoma, lymphoma, leukemia, other testicular tumors
  • Mediastinal: Hodgkin and non-Hodgkin lymphoma, leukemia, thymoma

Treatment


Medication


  • The administration of chemotherapy is based on malignant potential, local tumor extent, surgical outcomes, and the patient's medical history.
  • The typical 1st-line chemotherapy regimen for malignant GCTs includes cisplatin, etoposide, and bleomycin or PEB.
    • Cyclophosphamide in addition to PEB is being investigated in the treatment of high-risk GCTs.
    • Cisplatin is associated with a risks of ototoxicity and nephrotoxicity.
    • PEB therapy is associated with a risk of secondary myelodysplasia or acute myeloid leukemia.
    • Bleomycin is associated with a risk of pulmonary toxicity.

Surgery/Other Procedures


  • Every effort should be made to preserve fertility in gonadal teratomas. An experienced pediatric-gynecology oncologic surgeon is critical.
  • Sacrococcygeal teratoma
    • A complete surgical resection including the coccyx is curative.
    • Close postoperative follow-up should include monitoring of tumor markers.
    • Fetal surgery should be considered if signs of hydrops are seen.
  • Mature teratoma
    • Full surgical excision, irrespective of site, is curative in prepubescent patients.
    • Postpubescent patient with testicular teratoma (mature or immature) is at risk for retroperitoneal metastatic recurrence, so adjuvant chemotherapy and retroperitoneal lymph node resection are additional treatment considerations.
  • Immature teratoma
    • Complete surgical resection is therapy of choice; close observation and tumor marker evaluation for normalization
    • In cases of elevated AFP and incomplete surgical resection, chemotherapy should be offered given risk of microscopic foci of endodermal sinus tumor.
  • Teratoma with malignant components
    • Surgery plus chemotherapy with etoposide, cisplatin or carboplatin, and bleomycin
    • Patients with residual disease should have additional surgery and additional chemotherapy if total resection is not possible.
    • High-dose chemotherapy with autologous stem cell support and radiation are reserved for salvage therapy in recurrent disease.

Ongoing Care


  • Serial physical exams and imaging studies of primary site
  • Tumor markers (AFP or β-hCG) if elevated at diagnosis
  • If chemotherapy or radiation therapy used, need to monitor for secondary malignancies, long term (See "Cancer Therapy Late Effects" chapter); short term, need to monitor blood counts, chemistries, renal function, and audiology

Additional Reading


  • Barksdale  EM, Obokhare  I. Teratomas in infants and children. Curr Opin Pediatr.  2009;21(3):344-349.  [View Abstract]
  • Hedrick  HL, Flake  AW, Crombleholme  TW, et al. Sacrococcygeal teratoma: prenatal assessment, fetal intervention, and outcome. J Pediar Surg.  2004;39(3):430-438.  [View Abstract]
  • Koulouris  CR, Penson  RT. Ovarian stromal and germ cell tumors. Semin Oncol.  2009;36(2):126-136.  [View Abstract]
  • Lakhoo  K. Neonatal teratomas. Early Hum Dev.  2010;86(10):643-647.  [View Abstract]
  • Mannuel  HD, Hussain  A. Update on testicular germ cell tumors. Curr Opin Oncol.  2010;22(3):236-241.  [View Abstract]

Codes


ICD09


  • 199.1 Other malignant neoplasm without specification of site
  • 238.0 Neoplasm of uncertain behavior of bone and articular cartilage
  • 164.9 Malignant neoplasm of mediastinum, part unspecified
  • 183.0 Malignant neoplasm of ovary

ICD10


  • C80.1 Malignant (primary) neoplasm, unspecified
  • D48.0 Neoplasm of uncertain behavior of bone/artic cartl
  • C38.3 Malignant neoplasm of mediastinum, part unspecified
  • C56.9 Malignant neoplasm of unspecified ovary

SNOMED


  • 402878003 Germ cell tumor (disorder)
  • 281561000 Sacrococcygeal teratoma (disorder)
  • 278042005 Malignant teratoma of mediastinum (disorder)
  • 254869000 malignant germ cell tumor of ovary (disorder)

FAQ


  • Q: What is the chance of cure for immature/malignant teratomas?
  • A: With current chemotherapy as outlined earlier, overall survival is 85-97% (dependent on disease stage).
  • Q: Can a benign tumor recur? If so, can it then be malignant?
  • A: Yes. If there is residual tissue left behind, the tumor can recur. If there were unrecognized areas of malignancy, the recurrence can be a malignant teratoma. The greatest risk for the latter is with the immature teratomas.
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