Abstract • There is little information concerning long-term outcome of vertebral agenesis. In this series of eight patients with variable severity of vertebral agenesis, three died during infancy, two of associated congenital anomalies involving the cardiorespiratory system and one of unknown causes. The remaining five have had no life-threatening problems. They have normal intellect, and the developmental achievements are adequate for their age. The five patients with the highest lesions were those born to diabetic mothers. We also discuss current concepts of comprehensive team management of such infants. (Am J Dis Child 1982;136:533-537) References 1. Freedman B: Congenital absence of the sacrum and coccyx . Br J Surg 1950;37:299-303.Crossref 2. Pang D, Hoffman HJ: Sacral agenesis with progressive neurological deficit . Neurosurgery 1980;7:118-126.Crossref 3. Abraham E: Sacral agenesis with associated anomalies (caudal regression syndrome): Autopsy case report . Clin Orthop 1979;145:168-171. 4. Stanley JK, Owen R, Koff S: Congenital sacral anomalies . J Bone Joint Surg Br 1979;61B: 401-409. 5. Mariani AJ, Stern J, Khan AU, et al: Sacral agenesis: An analysis of 11 cases and review of the literature . J Urol 1979;122:684-686. 6. Andrish J, Kalamchi A, MacEwen GD: Sacral agenesis: A clinical evaluation of its management, heredity, and associated anomalies . Clin Orthop 1979;139:52-57. 7. Battaglia F, Lubchenco L: A practical classification of newborn infants by weight and gestational age . J Pediatr 1967;71:159-163.Crossref 8. White P: Pregnancy and diabetes, medical aspects . Med Clin North Am 1965;49:1015-1024. 9. Duhamel B: From the mermaid to anal imperforation: The syndrome of caudal regression . Arch Dis Child 1961;36:152-155.Crossref 10. Blumel J, Evans EB, Eggers GWW: Partial and complete agenesis or malformation of the sacrum with associated anomalies . J Bone Joint Surg Am 1959;41A:497-518. 11. Pouzet F: Les anomalies de développement du sacrum . Lyon Chir 1938;35:371-373. 12. Banta JV, Nichols O: Sacral agenesis . J Bone Joint Surg Am 1969;51A:693-703. 13. Cohn J, Bay-Nielsen E: Hereditary defect of the sacrum and coccyx with anterior sacral meningocele . Acta Paediatr Scand 1969;58:268-274.Crossref 14. Kenefick JS: Hereditary sacral agenesis associated with presacral tumor . Br J Surg 1973;60:271-274.Crossref 15. Finer NN, Bowen P, Dunbar LG: Caudal regression anomalad (sacral agenesis) in siblings . Genetics 1978;13:353-358. 16. Stewart JM, Stoll S: Familial caudal regression anomalad and maternal diabetes . J Med Genet 1979;16:17-20.Crossref 17. Renshaw TS: Sacral agenesis: A classification and review of 23 cases . J Bone Joint Surg Am 1978;60A:373-383. 18. Kûcera J: Exposure to fat solvents: A possible cause of sacral agenesis in mar . J Pediatr 1968;72:857-859.Crossref 19. Gardner WJ, Breuer AC: Anomalies of heart, spleen, kidneys, gut and limbs may result from an overdistended neural tube: A hypothesis . Pediatrics 1980;65:508-514. 20. Russell HE, Aitken GT: Congenital absence of the sacrum and lumbar vertebrae with prosthetic management: A survey of the literature and presentation of five cases . J Bone Joint Surg Am 1963;45A:501-508. 21. Elting JJ, Allen JC: Management of the young child with bilateral anomalous and functionless lower extremities . J Bone Joint Surg Am 1972;54A:1523-1530. 22. Perry J, Bonnet CA, Hoffer MM: Vertebral pelvic fusion in rehabilitation of patients with sacral agenesis . J Bone Joint Surg Am 1970;52A:288-294. 23. Current approaches to evaluation and management of children with myelomeningocele, Action Committee on Myelodysplasia, Section on Urology . Pediatrics 1979;63:663-667. 24. Rosenfelder BA: Vertebral agenesis. Review of the literature and presentation of 24 new cases . J Am Phys Ther Assoc 1968;48:203-217. 217 25. Gesell A, Amatruda C: Developmental Diagnosis: Normal and Abnormal Child Development, Clinical Methods and Practical Applications , ed 3. New York, Harper & Row Publishers Inc, 1962.
American Journal of Diseases of Children – American Medical Association
Published: Jun 1, 1982
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