Sacral agenesis associated with teratoma: report of a case

Borges, Antonio Cesar G.; Festugato, Rodinei Roberto; Cerqueira, José Carlos S. de; Souza, Miguel Ramos de

doi:10.1590/S0004-282X1987000100011

Abstracts

A case of a 29-year-old male with a huge sacrococcygeal teratoma completely removed by surgery, and associated with a sacral agenesis is reported. Roentengenograms revealed fusion of the T12, LI, L2 and L3 vertebral bodies, absence of the left 12th rib, partial agenesis of sacrococcygeal elements and a lumbar scoliosis convex to the left. Cystometry and myelograms were normal. No such association has been found in the literature reviewed. Some topics on the embryology of these lesions are discussed.

Relata-se o caso de paciente masculino de 29 anos de idade que apresentava enorme teratoma sacrococcígeo, que foi completamente removido cirurgicamente, associado com agenesia sacral. As radiografias mostraram fusão de Tl2, LI, L2, L3, ausência da 12* costela esquerda e agenesia parcial de elementos sacrococcígeos. Não foram encontradas alterações na cistometria, nem na mielografia. Na literatura consultada este tipo de associação não foi encontrado. Alguns tópicos sobre a embriologiada desta lesão são discutidos.

Agenesia sacral associada a teratoma

Sacral agenesis associated with teratoma: report of a case

Antonio Cesar G. Borges^I; Rodinei Roberto Festugato^II; José Carlos S. de Cerqueira^III; Miguel Ramos de Souza^IV

^IProfessor Adjunto - Trabalho da Disciplina de Neurologia e Neurocirurgia da Faculdade de Medicina da Universidade Federal de Pelotas

^IIProfessor Assistente - Trabalho da Disciplina de Neurologia e Neurocirurgia da Faculdade de Medicina da Universidade Federal de Pelotas

^IIIMédico Estagiário - Trabalho da Disciplina de Neurologia e Neurocirurgia da Faculdade de Medicina da Universidade Federal de Pelotas

^IVAcadêmico de Medicina - Trabalho da Disciplina de Neurologia e Neurocirurgia da Faculdade de Medicina da Universidade Federal de Pelotas

RESUMO

Relata-se o caso de paciente masculino de 29 anos de idade que apresentava enorme teratoma sacrococcígeo, que foi completamente removido cirurgicamente, associado com agenesia sacral. As radiografias mostraram fusão de Tl2, LI, L2, L3, ausência da 12* costela esquerda e agenesia parcial de elementos sacrococcígeos. Não foram encontradas alterações na cistometria, nem na mielografia. Na literatura consultada este tipo de associação não foi encontrado. Alguns tópicos sobre a embriologiada desta lesão são discutidos.

SUMMARY

A case of a 29-year-old male with a huge sacrococcygeal teratoma completely removed by surgery, and associated with a sacral agenesis is reported. Roentengenograms revealed fusion of the T12, LI, L2 and L3 vertebral bodies, absence of the left 12th rib, partial agenesis of sacrococcygeal elements and a lumbar scoliosis convex to the left. Cystometry and myelograms were normal. No such association has been found in the literature reviewed. Some topics on the embryology of these lesions are discussed.

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1. ADAMS, R.D. & WEGNER, W. - Congenital cyst of the spinal meninges as a cause of intermittent compression of the spinal cord. Arch. Neurol. Psychiat 58:57, 1947.
2. ALTMAN, R.P.; RANDOLPH, J.G. & LILLY, U.R. - Sacrococcygeal teratoma. Amer. Acad. Pediatrics Surgical Section Survey, 1973. J. pediatr. Surg. 9:389, 1974.
3. BUCY, P.C. & HAYMOND, H.E. - Lumbosacral teratoma associated with spina bifida occulta: report of a case with review of the literature. Amer. J. Pathol. 8:339, 1932.
4. DONNELLAN, W.A. & SWENSON, 0. - Benign and malignant sacrococcygeal teratomas. Surgery 64:834, 1968.
5. GARRISON, J.E. & KASDON, D.L. - Intramedullary spinal teratoma: a case report and review of the literature. Neurosurgery 7:509, 1980.
6. GELLIS, S.S. & FEINGOLD, M. - Caudal dysplasia sindrome. Amer. J. Dis. Child. 116:407, 1968.
7. GROSFELD, J.L.; BALLANTINE, T.V.N.; LOWE, D. - Benign and malignant teratomas in children: analysis of 85 patients. Surgery 80:297, 1976.
8. HANSEBOUT, P.R. & BERTRAND, G. - Intraspinal teratoma simulating protruded intervertebral disc. J. Neurosurg. 22:374, 1965.
9. IZANT, R.J. & FILSTON, H.C. - Sacrococcygeal teratomas: analysis of 43 cases. Amer. J. Surg. 130:617, 1975.
10. KALLEN, B. - Early embryogenesis of central nervous system with special reference to closure defects. Dev. Med. Child. Neurol. (Suppl.) 16:44, 1968.
11. KOONTZ, W.W. & PROUT, G.R. - Agnesis of the sacrum and the neurogenic bladder. J. amer. med. Assoc. 203:481, 1968.
12. KUBIE, L.S. & FULTON, J.F. - A clinical and pathological study of two teratomatous cysts of the spinal cord containing mucus and ciliated cells. Surg. Gynecol. Obstet. 47:297, 1928.
13. LAKE, P.A. & PITTS, F.W. - Atypical myelomeningocele with associated anomalies. J. Neurosurg. 31:234, 1969.
14. LAWSON, F.L. - Paravertebral teratoid tumor with scoliosis: report of cases. J. amer. med. Assoc. 151:271, 1953.
15. LOMBARDI, V. - Giant coccygeal teratoma in the newborn. J. Neurosurg. 30:747, 1969.
16. LOURIE, H. - Sacral agenesis: case report. J. Neurosurg. 38:92, 1973.
17. MITGANG, R.N. - Teratoma occurring within a myelomeningocele: case report. J. Neurosurg 37:448, 1972.
18. PANG, D. & HOFFMAN, H.J. - Sacral agenesis with progressive neurological defitic. Neurosurgery 7:118, 1980.
19. PASSARGE, E. - Congenital malformations and maternal diabetes. Lancet 1:324, 1965.
20. PASSARGE, E. & LENZ, W. - Syndrome of caudal regression in infants of diabetic mothers; observations of further cases. Pediatrics 37:672, 1966,
21. PRICE, D.L..; DOOLING, E.C. & RICHARDSON, E.P. - Caudal dysplasia (caudal regression syndrome). Arch. Neurol. 23:212, 1970.
22. PROP, N.; FRENSDORF, E.L.. & van der STADT, F.R. - A postvertebral endoder-mal cyst associated with axial deformities: a case showing the «endodermal-ectodermal adhesion syndrome». Pediatrics 39:555, 1967.
23. REWCASTL.E, N.B. & FRANCOEUR, J. - Teratomatous cysts of the spinal canal: with sex chromatin studies. Arch. Neurol. 11:91, 1964.
24. RHANEY, K. & BARCLAY, G.P.T. - Enterogenous cysts and congenital diverticula of the alimentary canal with abnormalities of the vertebral column and spinal cord. J. Pathol. Bacteriol. 77:457, 1959.
25. ROSENBAUM, T.J.; SOULE, E.H. & ONOFRIO, B.M. - Teratomatous cysts of the spinal canal: case report. J. Neurosurg. 49:292, 1978.
26. RUSNAK, S.L. & DRISCOL.L, S.G. - Congenital spinal anomalies in infants of diabetic mothers. Pediatrics 35:989, 1965.
27. SARNAT, H.B.; CASE, M.E. & GRAVISS, R. - Sacral agenesis: neurologic and neuropathologic features. Neurology 26:1124, 1976.
28. SMITH, E.D. - Congenital sacral anomalies in children. Aust. N.Z.J. Surg. 29: 165, 1959.
29. UGARTE, N.; GONZALEZ-CRUSSI. F. & SOTELO-AVILA C. - Diatematomyelia associated with teratomas: report of two cases. J. Neurosurg 53:720, 1980.

Publication Dates

Publication in this collection
22 June 2011
Date of issue
Mar 1987

This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

[1] 1. ADAMS, R.D. & WEGNER, W. - Congenital cyst of the spinal meninges as a cause of intermittent compression of the spinal cord. Arch. Neurol. Psychiat 58:57, 1947.

[2] 2. ALTMAN, R.P.; RANDOLPH, J.G. & LILLY, U.R. - Sacrococcygeal teratoma. Amer. Acad. Pediatrics Surgical Section Survey, 1973. J. pediatr. Surg. 9:389, 1974.

[3] 3. BUCY, P.C. & HAYMOND, H.E. - Lumbosacral teratoma associated with spina bifida occulta: report of a case with review of the literature. Amer. J. Pathol. 8:339, 1932.

[4] 4. DONNELLAN, W.A. & SWENSON, 0. - Benign and malignant sacrococcygeal teratomas. Surgery 64:834, 1968.

[5] 5. GARRISON, J.E. & KASDON, D.L. - Intramedullary spinal teratoma: a case report and review of the literature. Neurosurgery 7:509, 1980.

[6] 6. GELLIS, S.S. & FEINGOLD, M. - Caudal dysplasia sindrome. Amer. J. Dis. Child. 116:407, 1968.

[7] 7. GROSFELD, J.L.; BALLANTINE, T.V.N.; LOWE, D. - Benign and malignant teratomas in children: analysis of 85 patients. Surgery 80:297, 1976.

[8] 8. HANSEBOUT, P.R. & BERTRAND, G. - Intraspinal teratoma simulating protruded intervertebral disc. J. Neurosurg. 22:374, 1965.

[9] 9. IZANT, R.J. & FILSTON, H.C. - Sacrococcygeal teratomas: analysis of 43 cases. Amer. J. Surg. 130:617, 1975.

[10] 10. KALLEN, B. - Early embryogenesis of central nervous system with special reference to closure defects. Dev. Med. Child. Neurol. (Suppl.) 16:44, 1968.

[11] 11. KOONTZ, W.W. & PROUT, G.R. - Agnesis of the sacrum and the neurogenic bladder. J. amer. med. Assoc. 203:481, 1968.

[12] 12. KUBIE, L.S. & FULTON, J.F. - A clinical and pathological study of two teratomatous cysts of the spinal cord containing mucus and ciliated cells. Surg. Gynecol. Obstet. 47:297, 1928.

[13] 13. LAKE, P.A. & PITTS, F.W. - Atypical myelomeningocele with associated anomalies. J. Neurosurg. 31:234, 1969.

[14] 14. LAWSON, F.L. - Paravertebral teratoid tumor with scoliosis: report of cases. J. amer. med. Assoc. 151:271, 1953.

[15] 15. LOMBARDI, V. - Giant coccygeal teratoma in the newborn. J. Neurosurg. 30:747, 1969.

[16] 16. LOURIE, H. - Sacral agenesis: case report. J. Neurosurg. 38:92, 1973.

[17] 17. MITGANG, R.N. - Teratoma occurring within a myelomeningocele: case report. J. Neurosurg 37:448, 1972.

[18] 18. PANG, D. & HOFFMAN, H.J. - Sacral agenesis with progressive neurological defitic. Neurosurgery 7:118, 1980.

[19] 19. PASSARGE, E. - Congenital malformations and maternal diabetes. Lancet 1:324, 1965.

[20] 20. PASSARGE, E. & LENZ, W. - Syndrome of caudal regression in infants of diabetic mothers; observations of further cases. Pediatrics 37:672, 1966,

[21] 21. PRICE, D.L..; DOOLING, E.C. & RICHARDSON, E.P. - Caudal dysplasia (caudal regression syndrome). Arch. Neurol. 23:212, 1970.

[22] 22. PROP, N.; FRENSDORF, E.L.. & van der STADT, F.R. - A postvertebral endoder-mal cyst associated with axial deformities: a case showing the «endodermal-ectodermal adhesion syndrome». Pediatrics 39:555, 1967.

[23] 23. REWCASTL.E, N.B. & FRANCOEUR, J. - Teratomatous cysts of the spinal canal: with sex chromatin studies. Arch. Neurol. 11:91, 1964.

[24] 24. RHANEY, K. & BARCLAY, G.P.T. - Enterogenous cysts and congenital diverticula of the alimentary canal with abnormalities of the vertebral column and spinal cord. J. Pathol. Bacteriol. 77:457, 1959.

[25] 25. ROSENBAUM, T.J.; SOULE, E.H. & ONOFRIO, B.M. - Teratomatous cysts of the spinal canal: case report. J. Neurosurg. 49:292, 1978.

[26] 26. RUSNAK, S.L. & DRISCOL.L, S.G. - Congenital spinal anomalies in infants of diabetic mothers. Pediatrics 35:989, 1965.

[27] 27. SARNAT, H.B.; CASE, M.E. & GRAVISS, R. - Sacral agenesis: neurologic and neuropathologic features. Neurology 26:1124, 1976.

[28] 28. SMITH, E.D. - Congenital sacral anomalies in children. Aust. N.Z.J. Surg. 29: 165, 1959.

[29] 29. UGARTE, N.; GONZALEZ-CRUSSI. F. & SOTELO-AVILA C. - Diatematomyelia associated with teratomas: report of two cases. J. Neurosurg 53:720, 1980.