.;S:^i'3
CASE REPORT
SPINA BIFIDA IN A CALF
C. E. DOIGE*
Introduction Congenital anomalies in domestic animals are of considerable economic importance; they deserve close examination and accurate description. Spina bifida, a congenital anomaly which refers to defective closure of the dorsal vertebral laminae, has been reported infrequently in cattle (1-3, 5, 7-10). It has been described in several different breeds of cattle (2, 3, 5, 9) and there is some evidence that it is a heritable defect (2, 9). A variety of chemical substances and environmental changes have been used to produce spina bifida in laboratory animals (4, 5). The purpose of this report is to describe the FIGURE 1. External appearance of sldn defect pathological findings observed in a case of spina located at the dorsal midline in the sacral region. bifida, arthrogryposis and fusion of the kidneys Note irregular surface and lack of hair. in a newborn calf. :
History and Gross Postmortem Findings A male, term, Hereford calf was presented for necropsy because of abnormalities involving the pelvic limbs and the sacral area of the skin. The calf was delivered by cesarean section and was stillborn. Superficial examination revealed that the hock and stifle joints of both limbs were fixed in extension. In addition, a circular defect in the skin (4 cm diameter) was present at the dorsal midline in the sacral region (Figure 1). This area was depressed, void of hair and did not appear to communicate with underlying structures. Removal of the skin over the lumbosacral area and deeper dissection revealed an oblong opening (1 x 2 cm in diameter) into the vertebral canal at the level of the second and third sacral vertebrae (Figure 2). This opening was surrounded by a ring of fibrous tissue which was adherent to the overlying skin and to the dura. The dura was thickened in the region of the defect and formed a flattened, oval plaque on the dorsal aspect of the spinal cord (Figure 3). Maceration of the vertebral column revealed incomplete closure of the dorsal laminae of the
:°
:\.
iY
':
:.
*i: .:
FIGURE 2. Sacral defect viewed after removal of the skin. TMe opening is surrounded by a ring of dense fibrous tissue and opens into the vertebral canal.
sixth lumbar and of all sacral vertebrae (Figure 4). Sacral vertebrae were malaligned (some degree of scoliosis was present) and ventral foramina were distorted (Figure 5). Muscles of the pelvic limbs were pale and reduced in mass. Hock and stifle joints could not *Department of Veterinary Pathology, Western be flexed even after removal of muscles and College of Veterinary Medicine, University of ligaments. Although articular surfaces were Saskatchewan, Saskatoon, Saskatchewan S7N OWO. judged to be normal, fixation of joints appeared 2
CAN. VET. JOUR., vol. 16, no. 1, January, 1975
SPINA BIFIDA
tensive replacement of muscle fibers by adipose tissue. In certain areas no recognizable muscle fibers were visible; only bands of collagen, blood vessels and adipose tissue were present (Figure 7). Muscle spindles were identified. The epithelial surface of the cutaneous defect was irregular and thrown into papillary projections. Surface epithelium was continuous with that of the surrounding skin but accessory structures such as hair follicles and sweat glands FIGURE 3. Dorsal view of sacral portion of the were absent. Pathological changes in the spinal spinal cord with dura removed in the region of the efect (arrowheads) shown in Figure 2.
FIGURE 5. Ventral view of sacral vertebrae. Note scoliosis and distortion of intervertebral foramina.
FIGuRE 4. Dorsal view of sacral vertebrae. The dorsal laminae of all sacral vertebrae have failed to unite. to be a consequence of tightness of the joint capsule and interlocking of articular surfaces. No abnormalities were recognized in the pectoral limbs. Both kidneys were fused to form a single, horse-shoe shaped structure (Figure 6). When the capsule was removed it was found that there was no fusion of renal parenchyma and that fusion involved capsular structures only.
Microscopic Findings Significant findings were confined to the skin, spinal cord and skeletal muscles. Vertebrae were not examined histologically. Sections of skeletal muscle (vastus lateralis, semitendinosus and peroneus tertius) had ex-
FiGuRE 6. Gross appearance of the single, horse-shoe shaped kidney that was present. Fusion (arrowhead) involved the capsule only. 23
CANADIAN VETERINARY JOURNAL
and suggested the defect was inherited as a recessive trait. Based on evidence desimple .7'~~~~~~~~A rived from nine calves with spina bifida, Nes (9) concluded that the condition was heritable and that a dominant gene with low penetrance and variable expressivity was involved. It is interesting to note that spina bifida can be produced in the offspring of laboratory animals by exposing pregnant females to a variety of chemical substances or environmental changes early in gestation (eight to 12 days in the rat and mouse). Treatments reported to induce spina bifida in laboratory animals include antagonists of folic acid, excessive oral doses of vitamin A, trypan blue, salicylates, thalidomide, FIGURE 7. Microscopic appearance of muscle hypothermia and hyperoxia (4, 5). In the case (semitendinosus). Note extensive replacement of described in this report there was no evidence muscle fibers by adipose tissue. H & E. x28. available to incriminate familial or environmental factors. However, the sporadic occurrence of the defect together with the incord were confined to the lumbar and sacral formation available from the experimental area. At the level of L6 the central canal was reproduction of the defect in laboratory animals found to be lined incompletely by ependyma. might lead the observer to favour the concept No recognizable spinal cord was evident at the of early in utero injury. level of S2-S3, only a thickened plaque of dura covering nerve bundles was present. At the level Summary of S4 the terminal portion of the spinal cord A case of spina bifida, arthrogryposis and was visible. At this level two central canals were fusion of the kidneys in a Hereford calf is seen but these canals did not communicate; presented. Gross and microscopic postmortem both were lined by ependymal cells. findings are described. Discuss-ion Resume The lesions associated with spina bifida vary L'auteur rapporte un cas de spina-bifida, in severity and are frequently associated with d'arthrogrypose et de fusion des reins, qu'il a. defects in the overlying skin. Meningocele and observe chez un veau "Hereford". I1 en decrit myelodysplasia may or may not be present. In les l6sions macroscopiques et histologiques. cattle, the level of the vertebral defect is commonly lumbar (1) or sacral (8-10) al- References though involvement of the thoracic vertebrae 1. ADELMANN, H. B. An extreme case of spina has been described (2). bifida with dorsal hernia in a calf. Anat. Rec. Arthrogryposis involving the pelvic limbs has 19: 29-34. 1920. been reported previously (8, 9) in calves with 2. Goss, L.J. and F. E. HULL. Spina bifida sacral spina bifida. In the case described herein (calf). Cornell Vet. 29: 239-240. 1939. it was thought that the vertebral defect led to 3. GREENE, H. J., H. W. LEipoLD, K. HUSTON and M. M. GUFFY. Bovine congenital defects: myelodysplasia and subsequently to defective Arthrogryposis and associated defects in innervation of some muscles of the pelvic calves. Am. J. vet. Res. 34: 887-891. 1973. limbs, neurogenic atrophy or hypoplasia of D. L. Spina bifida and the Arnoldmuscle and to arthrogryposis. It should be noted 4. GUNBERG, Chiari in the progeny of trypan malformation that a number of other causes of arthrogryposis blue injected rats. Anat. Rec. 126: 343-367. have been described (6). 1956. Several congenital defects have been de- 5. KALTER, H. Teratology of the Central Nerscribed in association with spina bifida in cattle. vous System. Induced and Spontaneous Malformations of Laboratory, Agricultural and These include the presence of a single kidney, Domestic Animals. Chicago and London: The aplasia of one uterine horn, atresia ani, kyphosUniversity of Chicago Press. 1968. coliosis and cleft diaphragm (2, 9). 6. H. W., K. HUSTON, L. C. HULBERT, LEIPOLD, Although the cause of spina bifida in cattle M. GUFFY and S. M. DENNIS. Congenital has not been firmly established, genetic factors syndrome in Hereford calves with kyphoshave been implicated. Goss and Hull (2) recoliosis, arthrogryposis and palatoschisis. ported four affected calves from the same sire Cornell Vet. 64: 123-135. 1974. 24
SPINA BIFIDA
9. NES, A. N. Spina bifida ledsaget av muskelkontraktur og andre defekter hos kalv. Nord. VetMed. 11: 33-54. 1959. 10. WHITTEM, J. H. Congenital abnormalities in calves: Arthrogryposis and hydranencephaly. J. Path. Bact. 73: 375-387. 1957.
7. MAMMERICKX, M. and J. LEUNEN. Anomalies foetales dans 1'espece bovine. Rev. Agr. (Brussels) 17: 1077-1094. 1964. 8. McFARLAND, L. Z. Spina bifida with myelomeningocele in a calf. J. Am. vet. med. Ass. 134: 32-34. 1959.
GAINES VETERINARY AWARD made to the CVMA Executive Committee by anyone, no later than March 31, 1975. Each nomination shall include a description of the work done by the one nominated, a statement of how the work has contributed to the advancement of small animal medicine and siirgery, a pertinent bibliography (if any), and suitable biographic information. The Award will consist of a gold medallion and a $500 cash award, and will be presented at an appropriate time during the Annual Convention. All communications may be addressed to the undersigned, J. R. Kinney Executive Secretary 360 Bronson Avenue Ottawa, Ontario K1R 6J3
To encourage progress in the field of smallanimal medicine and surgery, General Foods Limited, through Gaines Professional Service Centre, has made available the "Gaines Veterinary Award". The award will be made annually to that veterinarian whose work in either ciinical research or the basic sciences is judged to have contributed significantly to the advancement of small animal medicine and surgery. The work to be recognized shall have been made known either by publication in recognized veterinary periodicals or presentations at professional meetings. Primary consideration shall be given to work within the preceding five years with secondary consideration given to those individuals still active in the profession, but whose work was from a prior period. Nominations for the 1975 Award may be
PRIX ViTERINAIRE GAINES Dans le but d'encourager le progres en medecine et en chirurgie des petits animaux, la compagnie General Foods Limited, par l'entremise du Centre de Services professionnels Gaines, a institue le "prix veterinaire Gaines". Ce prix sera decerne annuellement a un veterinaire dont on aura jug6 que le travail a contribue eminemment a l'avancement de la medecine et de la chirurgie des petits animaux, soit en recherches cliniques ou soit en recherches fondamentales. La presentation de l'oeuvre a evaluer devra avoir fait l'objet d'une publication dans des revues veterinaires d'envergure ou d'une communication a des congres professionnels. On considerera en premier lieu les travaux executes au cours des cinq dernieres annees, bien que, en second lieu, on puisse aussi apprecier les oeuvres anterieures "a cette p6riode pour des membres qui demeu-
Toute personne peut, jusqu'au 31 mars 1975 au plus tard, presenter des candidats pour le prix de 1975 en s'adressant au Comite executif de I'ACV. Avec chaque recommandation, le proposeur devra soumettre une description des travaux de son candidat. II devra aussi demontrer comment ces travaux ont contribue a l'avancement de la medecine et de la chirurgie des petits animaux et soumettre une bibliographie pertinente (s'il en existe) en meme temps qu'une notice biographique. Le prix consistera en un medaillon d'or accompagnant une somme de $500.00. Le tout sera decerne a l'occasion du congres annuel. Toute correspondance 'a ce sujet peut etre adressee au soussigne. J. R. Kinney Secretaire executif 360 avenue Bronson Ottawa, Ontario K1R 6J3
rent toujours actifs. 25
CASE REPORT
SPINA BIFIDA IN A CALF
C. E. DOIGE*
Introduction Congenital anomalies in domestic animals are of considerable economic importance; they deserve close examination and accurate description. Spina bifida, a congenital anomaly which refers to defective closure of the dorsal vertebral laminae, has been reported infrequently in cattle (1-3, 5, 7-10). It has been described in several different breeds of cattle (2, 3, 5, 9) and there is some evidence that it is a heritable defect (2, 9). A variety of chemical substances and environmental changes have been used to produce spina bifida in laboratory animals (4, 5). The purpose of this report is to describe the FIGURE 1. External appearance of sldn defect pathological findings observed in a case of spina located at the dorsal midline in the sacral region. bifida, arthrogryposis and fusion of the kidneys Note irregular surface and lack of hair. in a newborn calf. :
History and Gross Postmortem Findings A male, term, Hereford calf was presented for necropsy because of abnormalities involving the pelvic limbs and the sacral area of the skin. The calf was delivered by cesarean section and was stillborn. Superficial examination revealed that the hock and stifle joints of both limbs were fixed in extension. In addition, a circular defect in the skin (4 cm diameter) was present at the dorsal midline in the sacral region (Figure 1). This area was depressed, void of hair and did not appear to communicate with underlying structures. Removal of the skin over the lumbosacral area and deeper dissection revealed an oblong opening (1 x 2 cm in diameter) into the vertebral canal at the level of the second and third sacral vertebrae (Figure 2). This opening was surrounded by a ring of fibrous tissue which was adherent to the overlying skin and to the dura. The dura was thickened in the region of the defect and formed a flattened, oval plaque on the dorsal aspect of the spinal cord (Figure 3). Maceration of the vertebral column revealed incomplete closure of the dorsal laminae of the
:°
:\.
iY
':
:.
*i: .:
FIGURE 2. Sacral defect viewed after removal of the skin. TMe opening is surrounded by a ring of dense fibrous tissue and opens into the vertebral canal.
sixth lumbar and of all sacral vertebrae (Figure 4). Sacral vertebrae were malaligned (some degree of scoliosis was present) and ventral foramina were distorted (Figure 5). Muscles of the pelvic limbs were pale and reduced in mass. Hock and stifle joints could not *Department of Veterinary Pathology, Western be flexed even after removal of muscles and College of Veterinary Medicine, University of ligaments. Although articular surfaces were Saskatchewan, Saskatoon, Saskatchewan S7N OWO. judged to be normal, fixation of joints appeared 2
CAN. VET. JOUR., vol. 16, no. 1, January, 1975
SPINA BIFIDA
tensive replacement of muscle fibers by adipose tissue. In certain areas no recognizable muscle fibers were visible; only bands of collagen, blood vessels and adipose tissue were present (Figure 7). Muscle spindles were identified. The epithelial surface of the cutaneous defect was irregular and thrown into papillary projections. Surface epithelium was continuous with that of the surrounding skin but accessory structures such as hair follicles and sweat glands FIGURE 3. Dorsal view of sacral portion of the were absent. Pathological changes in the spinal spinal cord with dura removed in the region of the efect (arrowheads) shown in Figure 2.
FIGURE 5. Ventral view of sacral vertebrae. Note scoliosis and distortion of intervertebral foramina.
FIGuRE 4. Dorsal view of sacral vertebrae. The dorsal laminae of all sacral vertebrae have failed to unite. to be a consequence of tightness of the joint capsule and interlocking of articular surfaces. No abnormalities were recognized in the pectoral limbs. Both kidneys were fused to form a single, horse-shoe shaped structure (Figure 6). When the capsule was removed it was found that there was no fusion of renal parenchyma and that fusion involved capsular structures only.
Microscopic Findings Significant findings were confined to the skin, spinal cord and skeletal muscles. Vertebrae were not examined histologically. Sections of skeletal muscle (vastus lateralis, semitendinosus and peroneus tertius) had ex-
FiGuRE 6. Gross appearance of the single, horse-shoe shaped kidney that was present. Fusion (arrowhead) involved the capsule only. 23
CANADIAN VETERINARY JOURNAL
and suggested the defect was inherited as a recessive trait. Based on evidence desimple .7'~~~~~~~~A rived from nine calves with spina bifida, Nes (9) concluded that the condition was heritable and that a dominant gene with low penetrance and variable expressivity was involved. It is interesting to note that spina bifida can be produced in the offspring of laboratory animals by exposing pregnant females to a variety of chemical substances or environmental changes early in gestation (eight to 12 days in the rat and mouse). Treatments reported to induce spina bifida in laboratory animals include antagonists of folic acid, excessive oral doses of vitamin A, trypan blue, salicylates, thalidomide, FIGURE 7. Microscopic appearance of muscle hypothermia and hyperoxia (4, 5). In the case (semitendinosus). Note extensive replacement of described in this report there was no evidence muscle fibers by adipose tissue. H & E. x28. available to incriminate familial or environmental factors. However, the sporadic occurrence of the defect together with the incord were confined to the lumbar and sacral formation available from the experimental area. At the level of L6 the central canal was reproduction of the defect in laboratory animals found to be lined incompletely by ependyma. might lead the observer to favour the concept No recognizable spinal cord was evident at the of early in utero injury. level of S2-S3, only a thickened plaque of dura covering nerve bundles was present. At the level Summary of S4 the terminal portion of the spinal cord A case of spina bifida, arthrogryposis and was visible. At this level two central canals were fusion of the kidneys in a Hereford calf is seen but these canals did not communicate; presented. Gross and microscopic postmortem both were lined by ependymal cells. findings are described. Discuss-ion Resume The lesions associated with spina bifida vary L'auteur rapporte un cas de spina-bifida, in severity and are frequently associated with d'arthrogrypose et de fusion des reins, qu'il a. defects in the overlying skin. Meningocele and observe chez un veau "Hereford". I1 en decrit myelodysplasia may or may not be present. In les l6sions macroscopiques et histologiques. cattle, the level of the vertebral defect is commonly lumbar (1) or sacral (8-10) al- References though involvement of the thoracic vertebrae 1. ADELMANN, H. B. An extreme case of spina has been described (2). bifida with dorsal hernia in a calf. Anat. Rec. Arthrogryposis involving the pelvic limbs has 19: 29-34. 1920. been reported previously (8, 9) in calves with 2. Goss, L.J. and F. E. HULL. Spina bifida sacral spina bifida. In the case described herein (calf). Cornell Vet. 29: 239-240. 1939. it was thought that the vertebral defect led to 3. GREENE, H. J., H. W. LEipoLD, K. HUSTON and M. M. GUFFY. Bovine congenital defects: myelodysplasia and subsequently to defective Arthrogryposis and associated defects in innervation of some muscles of the pelvic calves. Am. J. vet. Res. 34: 887-891. 1973. limbs, neurogenic atrophy or hypoplasia of D. L. Spina bifida and the Arnoldmuscle and to arthrogryposis. It should be noted 4. GUNBERG, Chiari in the progeny of trypan malformation that a number of other causes of arthrogryposis blue injected rats. Anat. Rec. 126: 343-367. have been described (6). 1956. Several congenital defects have been de- 5. KALTER, H. Teratology of the Central Nerscribed in association with spina bifida in cattle. vous System. Induced and Spontaneous Malformations of Laboratory, Agricultural and These include the presence of a single kidney, Domestic Animals. Chicago and London: The aplasia of one uterine horn, atresia ani, kyphosUniversity of Chicago Press. 1968. coliosis and cleft diaphragm (2, 9). 6. H. W., K. HUSTON, L. C. HULBERT, LEIPOLD, Although the cause of spina bifida in cattle M. GUFFY and S. M. DENNIS. Congenital has not been firmly established, genetic factors syndrome in Hereford calves with kyphoshave been implicated. Goss and Hull (2) recoliosis, arthrogryposis and palatoschisis. ported four affected calves from the same sire Cornell Vet. 64: 123-135. 1974. 24
SPINA BIFIDA
9. NES, A. N. Spina bifida ledsaget av muskelkontraktur og andre defekter hos kalv. Nord. VetMed. 11: 33-54. 1959. 10. WHITTEM, J. H. Congenital abnormalities in calves: Arthrogryposis and hydranencephaly. J. Path. Bact. 73: 375-387. 1957.
7. MAMMERICKX, M. and J. LEUNEN. Anomalies foetales dans 1'espece bovine. Rev. Agr. (Brussels) 17: 1077-1094. 1964. 8. McFARLAND, L. Z. Spina bifida with myelomeningocele in a calf. J. Am. vet. med. Ass. 134: 32-34. 1959.
GAINES VETERINARY AWARD made to the CVMA Executive Committee by anyone, no later than March 31, 1975. Each nomination shall include a description of the work done by the one nominated, a statement of how the work has contributed to the advancement of small animal medicine and siirgery, a pertinent bibliography (if any), and suitable biographic information. The Award will consist of a gold medallion and a $500 cash award, and will be presented at an appropriate time during the Annual Convention. All communications may be addressed to the undersigned, J. R. Kinney Executive Secretary 360 Bronson Avenue Ottawa, Ontario K1R 6J3
To encourage progress in the field of smallanimal medicine and surgery, General Foods Limited, through Gaines Professional Service Centre, has made available the "Gaines Veterinary Award". The award will be made annually to that veterinarian whose work in either ciinical research or the basic sciences is judged to have contributed significantly to the advancement of small animal medicine and surgery. The work to be recognized shall have been made known either by publication in recognized veterinary periodicals or presentations at professional meetings. Primary consideration shall be given to work within the preceding five years with secondary consideration given to those individuals still active in the profession, but whose work was from a prior period. Nominations for the 1975 Award may be
PRIX ViTERINAIRE GAINES Dans le but d'encourager le progres en medecine et en chirurgie des petits animaux, la compagnie General Foods Limited, par l'entremise du Centre de Services professionnels Gaines, a institue le "prix veterinaire Gaines". Ce prix sera decerne annuellement a un veterinaire dont on aura jug6 que le travail a contribue eminemment a l'avancement de la medecine et de la chirurgie des petits animaux, soit en recherches cliniques ou soit en recherches fondamentales. La presentation de l'oeuvre a evaluer devra avoir fait l'objet d'une publication dans des revues veterinaires d'envergure ou d'une communication a des congres professionnels. On considerera en premier lieu les travaux executes au cours des cinq dernieres annees, bien que, en second lieu, on puisse aussi apprecier les oeuvres anterieures "a cette p6riode pour des membres qui demeu-
Toute personne peut, jusqu'au 31 mars 1975 au plus tard, presenter des candidats pour le prix de 1975 en s'adressant au Comite executif de I'ACV. Avec chaque recommandation, le proposeur devra soumettre une description des travaux de son candidat. II devra aussi demontrer comment ces travaux ont contribue a l'avancement de la medecine et de la chirurgie des petits animaux et soumettre une bibliographie pertinente (s'il en existe) en meme temps qu'une notice biographique. Le prix consistera en un medaillon d'or accompagnant une somme de $500.00. Le tout sera decerne a l'occasion du congres annuel. Toute correspondance 'a ce sujet peut etre adressee au soussigne. J. R. Kinney Secretaire executif 360 avenue Bronson Ottawa, Ontario K1R 6J3
rent toujours actifs. 25
