Saeed Al-Hindi,MD,FRCST,CAB*
Jaffar Al-Khuzaie,MD**
Mohammed Al-Awadhi,MDCM,FRCS(C)***
Ashok Kumar Malik,MD,FRCpath****
An infant of in-vitro fertilization (IVF) product presented with right testicular swelling since birth. The patient underwent right groin exploration and orchidectomy. Histology confirmed malignant teratoma. Literature has been reviewed for the incidence of malignancy in IVF babies.
Bahrain Med Bull 2001;23(4):185-86
The testicle is an uncommon site of teratomas in infancy and childhood. Teratomas characteristically contains elements derived from all the three embryonic germ layers1. Testicular teratomas in prepubertal children are distinct from adult in their pathological feature and biologic behaviour. We document a case of infant produced by IVF who had a teratoma of right testis.
The Case
Six month old infant, a product of IVF by normal delivery, presented with right scrotal swelling since birth. He was referred to the hospital suspected of a right hydrocele. The family history was negative for any cancers. On examination, there was right scrotal swelling, firm in consistency, non tender and negative for transillumination test. Groin lymph nodes were not palpable bilaterally. Laboratory investigations for complete blood count, urea, electrolytes, serum creatinine and liver function tests were within normal limits. Alpha fetoprotein was 271 ?g/l (N: 0-15 ?g/l) and the other tumor markers (Ca 25, Ca 15-3, Ca 19-9, CEA and ?-HCG) were within normal levels. Scrotal and pelvic ultrasound showed homogeneous right testicular mass with no evidence of lymph node enlargement.
MRI showed gross right testicular enlargement which was four times as compared to the left, both spermatic cords appeared normal and no obvious lymphadenopathy in the
retroperitoneum. Patient underwent right testicular frozen section followed by orchidectomy as section proved malignancy. Right testis weighed 16 grams and measured 3.5 x 2.5 cms. Slicing did not reveal any cystic areas. Cut surface appeared greyish smooth. Microscopy showed aggregates and clusters of immature neuroepithelial tubular structures interspersed with primitive embryonal stroma (Fig.1). Rosettes were noted and frequent mitotic figures, admixtures of mature and immature tissues representing mainly endo and mesoderms were also seen. Based on morphologic appearances, a diagnosis of Grade III immature teratoma involving the testis was given. Two weeks after surgery, alpha fetoprotein dropped to 52 ?g/l. Right groin swelling was noted 4 months after surgery, which was excised suspecting recurrence or involvement of lymph node. The mass measured 2.8 cms in it’s maximum diameter. Cut surface revealed cystic gelatinous appearance. Microscopically, it showed tell-tale features of mature cystic/ solid tetatoma. Few lymph nodes adjacent to the mass showed reactive changes only. One week later, alpha fetoprotein level was within normal limits.
DISCUSSION
Testicular tumors in infants and children are uncommon and account only 1-2% of all pediatric solid tumors2.
Testicular tumours classification includes germ cell tumors (yolk sac tumor, teratoma, teratocarcinoma and seminoma), gonadal stromal tumors (Leydig and Sertoli cell tumors, and intermediate forms), gonadoblastomas, tumors of supporting tissue, lymphomas and leukemias, tumor-like lesions, secondary tumors and tumors of adnexa3.
Teratomas are heterogeneous group of tumors which may be benign, potentially malignant, or malignant depending on the evolutionary characteristics and site or organ of origin4,5. Testicular teratomas in prepubertal children are distinct from adult testicular teratomas as well as from teratomas located elsewhere in the body, both with respect to their pathological features and biological behavior5,6.
The sacrococcygeal region and ovary appear to be the most common sites of origin. Other rare sites of origin are retroperitoneal, mediastinal, abdominal, parotid and testicular4. Testicular teratoma rarely presents before one year of age1. Alpha-fetoprotein has been detected in the blood of two-thirds of children with malignant teratoma but not in benign teratoma7. The overall mortality for children with mature teratomas was significantly less than for those with immature tumors1.
In the era of in-vitro fertilization
(IVF), the only suggested fetal complications have been spina bifida and
transposition of the great vessels8. The association between IVF and pediatric
cancer has been described in sporadic case reports. However, tumors arising
in the first months of life are likely to be related to factors associated
with conception or pregnancy. The procedure of IVF is prone to carcinogenic
effect due to hormonal enhancement of ovulation, preservation of pregnancy
or micromanipulation on germ cells and the fertilized ovum9.
CONCLUSION
Malignant testicular teratoma is very rare in infants. In a review of literature, few reports have been found describing the association between product of IVF and malignancy. However, no report has been found about malignant testicular teratoma in this group.
REFERENCES
1. Tapper D, Lack EE. Teratomas
in Infancy and Childhood: A 54 year Experience at the Children’s Hospital
Medical Center. Ann Surg. 1983;198:398-410.
2. Kay R. Prepubertal
testicular tumor registry. Urol Clin North Am 1993:20:1-5.
3. Kaplan GW, Cromie WJ, Kelalis
PP, et al. Gonadal Stromal Tumors: a report of the
Prepubertal
Testicular Tumor Registry J Urol. 1986;136(1 Pt 2):300-2.
4. Sinnah D, Prathap K,
Somasundram K. Teratoma in Infancy and Childhood. Cancer
1980;
46:630-32.
5. Jones P. Campbell PE. Tumours
in Infancy in Childhood. Oxford: Blackwell
Scientific
Publications 1976;911:45-6.
6. Gupta DK, Kataria R, Sharma
MC. Prepubertal testicular teratomas. Eur J Pediatr
Surg 1999;9:173-6.
7. Smith JB, O’Neill RT. Alpha-fetoprotein.
Occurance in germinal cells and liver
malignancies. Am J Med 197;51:767-71.
8. Lancaster PA. Congenital malformations
after in-vitro fertilization. Lancet
1987;2:1392-3.
9. Toren A, Sharon N, Mandel M,
et al. Two embryonal cancers after in vitro
fertilization.
Cancer 1995;76:2372-4.
----------------------------------------------
* Chief Resident
** Resident
*** Consultant Surgeon
Department of Surgery
**** Consultant Pathologist
Department of Pathology
Salmaniya Medical Complex
State of Bahrain
