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Abdelrhaman,, Ibrahim, and Elsadig: Sacrococcygeal teratoma

Case report

A 38-year-old woman was admitted to Khartoum Hospital maternity unit with pre-eclampsia at 38 weeks' gestation following poor antenatal care. Labour had to be induced and, following a delay due to the presence of an undiagnosed large sacrococcygeal tumour, a baby girl in the vertex presentation was delivered. The baby cried at birth, had an Apgar score of 7 at 1 minute and 10 at 5 minutes and her birthweight was 4.0 kg.

The tumour was larger than the baby's head and it covered both buttocks and extended forwards on either side of the anus, pushing the anus forwards so that it lay on its anterior wall. The two projections of the tumour met anterior to the anus. The mass was mainly cystic with a few palpable small nodules and was covered with healthy skin; minor sloughing was visible on the right side of the tumour, at the rupture site. Rectal examination showed that the mass extended upwards to the sacral promontory and was lying posterior to the rectum but extended laterally on either side. The mass weighed 400 g with a circumference of 38 cm; however, the baby was otherwise well and was not dysmorphic (Figure 1).

Investigations revealed her haemoglobin level was 16.0 g/dl, her white cell count was 10 × 109/l, her platelet count was 300 × 109/l and her electrolyte, urea and creatinine levels were normal.

The tumour was excised en bloc via the sacral approach. The pelvic component was carefully dissected, protecting the rectum and bladder, and a coccygectomy was performed. Postoperatively, the baby passed urine and meconium normally.

The histopathological report showed solid and cystic components with some serous fluid and a small amount of fatty tissue. These findings were consistent with a diagnosis of sacrococcygeal teratoma.

Discussion

Sacrococcygeal teratoma is one of the most common tumours in the neonatal period. It occurs in 1 in 20 000–40 000 live births.1,2 It is a germ-cell tumour and may be cystic, solid or mixed.3,4 The solid tumour is more vascular and is sometimes associated with other complications such as hydrops fetalis, urinary obstructions, stillbirth and dystocia.5,6 Advancement in diagnostic and therapeutic techniques has influenced clinical decisions in management and prognosis,7,8 and in most centres diagnosis is made during the antenatal period.5 Early surgical treatment is advocated to prevent malignant transformation and a minimum 3-year follow-up is required to detect recurrence or good overall prognosis.

The case presented here is typical of a patient with sacrococcygeal teratoma complicated by tumour rupture at delivery. Patients with ruptured tumours usually bleed heavily and can present with hypovolaemic shock with an increased risk of morbidity.3,4 Aggressive resuscitation with intravenous fluid, blood transfusion and intravenous antibiotics may be required to prevent shock or death; however, the patient presented in this case did not require resuscitation or preoperative transfusion. The baby survived the intrapartum period because the tumour was cystic, which is usually less vascular, and when it ruptured it prevented labour dystocia.

Conclusion

This case reports a complicated delivery and shows that an adverse outcome in sacrococcygeal teratoma can be prevented by early antenatal recognition of the problem, which is not always easy in the absence of routine ultrasonography. Delivery should be via caesarean section in cases of large or vascular tumours and vaginally if tumours are small.

Sacrococcygeal teratoma should always be suspected during labour when a delay occurs after delivery of the shoulders and it is worth emphasizing that early involvement of a paediatric surgeon could remarkably improve the outcome.

FIGURE 1

The baby after birth with the tumour visible at the anus.

6-3-7-fig1.jpg

References

1. 

Krushin B, Visnjic A, Gzmic A, et al. DNA ploidy analysis and cell proliferation in congenital sacrococcygeal teratoma. Cancer 2000; 89:932–7. http://dx.doi.org/10.1002/1097-0142(20000815)89:4〈932::AID-CNCR29〉3.0.CO;2-0

2. 

Hamilton C. Teratoma Cystic Exerpt. EMedicine, 2002. URL: http://emedicine.medscape.com/article/281850-overview (accessed 15 October 2013).

3. 

Schimdt B, Habrlik A, Uray E, Ratschek M, Lackner H, Höllwarth ME. Sacrococcygeal teratoma: clinical cause and prognosis with special view to long-term functional results. Pediatr Surg Int 1999; 15:573–6. http://dx.doi.org/10.1007/s003830050675

4. 

Kay S, Khalife S, Laberge JM, Shaw K, Morin L, Flageole H. Prenatal percutaneous needle drainage of cystic teratoma. J Pediatr Surg 1999; 34:1148–51. http://dx.doi.org/10.1016/S0022-3468(99)90587-0

5. 

Chuileannain FN, Woodrow N, de-Crespingy L. Prenatal diagnosis and management of sacrococcygeal teratoma. Aust N Z J Obstet Gynaecol 1999; 39:497–501. http://dx.doi.org/10.1111/j.1479-828X.1999.tb03143.x

6. 

Tongsong T, Wanapirac C, Piyamongkol W, Sudasana J. Prenatal sonographic features of sacrococcygeal teratoma. Int J Gynecol Obstet 1999; 67:95–101. http://dx.doi.org/10.1016/S0020-7292(99)00118-6

7. 

Ruangtrakool R, Nitipon A, Laohpensang M, et al. Sacrococcygeal teratoma: 25 years experience. J Med Assoc Thai 2001; 84:265–73.

8. 

Westerburg B, Feldstein VA, Sandberg PL, Lopoo JB, Harrison MR, Albanese CT. Sonographic prognostic factors in fetuses with sacrococcygeal teratoma. J Pediatr Surg 2000; 35:322–6. http://dx.doi.org/10.1016/S0022-3468(00)90032-0





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