(Acta Paediatr Jpn 1992; 34: 547
- 550)
Spinal Arachnoid Cyst in a Newborn Infant Kazuhiro Hashimoto, M.D.,’ Motoya Hashimoto, M.D.,’ Satoshi Homma, M.D.,’ Yoshiteru Ohsone, M.D., Hisaya Hasegawa, M.D.,’ Jiro Takei, M.D.,l Yoshikazu Kida, M.D..l Yutaka Takeuchi, M.D.,l and Minako Ohba, M.D.’ Division of Neonatology, Matudo City Hospital, Matudo City, Chiba, and ’Department of Pediatrics, Tohoku University School of Medicine, Sendai City, Miyagi, Japan
A male infant was born with asphyxia following a prolonged breech delivery at 39 weeks of gestation. He had a chylohemothorax from birth. Soon after birth he exhibited flaccid paraplegia with an absence of deep tendon reflexes in the lower extremities. At 17 days of age, magnetic resonance imaging (MRI) disclosed a hypodense area in the thoracic cord between C7 and Th4 in TI-weighted images, and spinal sonography revealed an echo-free space compressing the spinal cord in the same region observed with MRI. A spinal cyst was removed surgically at 24 days of age. The cyst was confirmed by pathology to be a true arachnoid cyst between C7and Th4. The cause of this cyst may have been a spinal cord injury during the perinatal period. Key Words Spinal arachnoid cyst, Spinal tumor, Newborn infants, Birth trauma
Introduction
Case Report
Spinal cord tumors are rarely observed in newborn infants. Congenital spinal tumors such as lipomas, teratomas and neuroblastomas have been reported in the literature [ 1-41 but, to our knowledge, spinal arachnoid cysts have never been reported. In cases of intracranial arachnoid cysts the origin has been considered to have resulted from traumatic damage to the brain [5]. We report a newborn infant with a spinal arachnoid cyst in the thoracic cord between C7 and Th,.
A male infant was born after a prolonged breech delivery at 39 weeks of gestation, weighing 2,540 g. His parents were non-consanguineous. His family history was unremarkable. The Apgar score was 1 and 6 at 1 and 5 min, respectively. He was intubated and transferred to our neonatal intensive care unit (NICU). On admission he exhibited flaccid paraplegia with the absence of deep tendon reflexes. Plantar grasp reflexes in the lower extremities were noted. Abdominal reflexes were absent, but cremaster and anocutaneous reflexes were weakly present. He did not respond to painful stimuli to the skin area innervated from the Th, to Th,, cord level. Movements of his upper extremities were normal. His head circumference was within normal limits. Physical examination disclosed no abnormal findings in the chest or abdomen.
Received March 13, 1992 Revised April 14, 1992 Accepted April 24, 1992 Correspondence address: Dr Kazuhiro Hashimoto, Division of Child Neurology, Tottori University School of Medicine, 86 Nishimachi, Yonago 683, Tottori, Japan.
548 ( 5 8 ) Hashimoto et al.
v
Echo-free space
Spinal cord
Fig. 1 : Longitudinal spinal ultrasonography. Note the echo-free space and compressed spinal cord.
Neither nevi nor localized hirsutism on the back were observed. The activities of liver transaminase, lactic dehydrogenaseand creatine phosphokinasewere increased. Metabolic acidosis was noted. Little cerebrospinal fluid (CSF) was obtained from the lumbar puncture. There were no bacteria in the CSF culture. Tumor markers, such as a-fetoprotein (AFP) and vanillylmandelic acid (VMA), were negative but the activity of
neuron-specific enolase in the CSF was mildly elevated. In plain roentgenographs of the chest and spine, a compressed deformity of the third thoracic vertebra was observed. Cranial computerized tomography (CT) and sonography of the brain were within normal limits. At 2 days of age, respiratory failure gradually worsened because of a chylohemothorax. Drainage from the thorax was performed on both sides for 10 days. He was fed on medium chain triglyceride
Fig. 2: MRI (T,-weighted image) at 24 days. Decreased signal intensity (arrowed) in the anterior subdural space extending from C, to Th,, a backward depressed cord and forward distorted vertebral bodies above the third vertebral body can be noted.
Acta Paediatr Jpn
Spinal arachnoid cyst in a newborn (59) 549 (MCT) milk for 30 days. At 17 ,days of age, sonograms (U-sonic RT 2800, Tosiba Medical Co, Ltd, Tokyo, Japan) with a 3.75 MHz linear probe demonstrated an echo-free space that compressed the spinal cord backward (Fig. 1). Magnetic resonance imaging (MRI), with a Magnetom operating at 0.5 T (Siemens Co, Ltd, Tokyo, Japan) produced TI-weighted images (spin-echo: pulse interval, 200 msec; time interval, 15 msec) that demonstrated decreased signal intensity between the vertebral bodies and the spinal cord extending from C7to Th4. The spinal cord appeared to be thin and was distorted backward. The vertebral bodies were distorted forward above the third vertebral body. On the administration of gadoliniumDTPA, no enhanced area was observed in Tiweighted images (Fig. 2). At 24 days of age, an operation for the spinal tumor was performed. The spinal cord was pressed backward but the diameter seemed to be normal, and a membrane-bound cyst in front of the thoracic cord between C7 and Th4 was confirmed. The membrane of the cyst was totally removed. After the operation, the patient’s left ankle began to move but his right ankle did not move at all, and
the sensory disturbance remained. At 80 days of age, MRI was performed again and TI-weighted images demonstrated decreased signal intensity, with irregular margins in the subdural space behind the spinal cord extending from C7 and Th, (Fig. 3). The spinal cord between C7 and Th, was atrophic and the signal intensity within the anterior space, in which the removed cyst had been located, was the same as in the CSF. The nature of the area exhibiting irregularly decreased signal intensity was unclear. An operation to examine this area was undertaken at 3 months of age, when it was found that the posterior arachnoid space was full of fatty tissue. At present (1 year of age), his paraplegia is gradually improving but the sensory disturbance remains.
Discussion A spinal cord lesion was suspected in our patient from the following neurological findings: flaccid paraplegia, absence of deep tendon reflexes restricted to the lower extremities, and sensory disturbance at levels between Th4 and Th12. Spinal sonograms and MRI clearly demon-
Fig. 3: MRI (T,-weighted image) at 80 days. Irregular decreased signal intensity in the posterior subdural space extending from C , to Th,, spinal cord atrophy at the levels of C,-Th, and distorted vertebrae can be noted.
Vol. 34 No. 5 October 1992
550 (60) Hashimoto et al. strated an arachnoid cystic lesion extending between C, and Th,. In spinal sonograms, the lesion was observed in real time as an echo-free space that compressed the spinal cord. In TIweighted images on MRI, the lesion was observed to be as hypodense as CSF and was not enhanced, and thus an arachnoid cyst was suspected. There have been a few reports of spinal tumors in infancy [l-41. Most of these spinal tumors were located in the sacral or thoracolumbar portions, and most of the tumors were lipomas, teratomas or neuroblastomas. To our knowledge, there has been no report of a spinal arachnoid cyst in infancy. Generally, the origin of an arachnoid cyst is thought to be congenital or traumatic. In the present case, a compressed deformity of the third thoracic vertebral body diagnosed at birth, distortion of the vertebral bodies at the level of the third vertebral body, and severe atrophy of the spinal cord at the same level strongly suggested traumatic damage to the spine during the perinatal period. However, there was no direct evidence of the cause of the trauma, except for the asphyxiated breech delivery and the chylohemothorax. Also, the spinal cord atrophy may be secondary to the compression by the cyst or may be the result of spinal damages due to birth trauma.
In conclusion, MRI and spinal sonography were non-invasive and helpful for delineating the lesion in the spinal cord [6-81, which is a difficult area for diagnosis in small infants.
References 1.
2.
3.
4.
5. 6.
7. 8.
Tibbs PA, James HE, Rorke LB, Schut L, Bruce DA. Midline hamartomas masquerading as meningoceles or teratomas in the newborn infant. J Pediatr 1976; 89: 928-933. Packer RJ, Zimmerman RA, Sutton LN, Bilaniuk LT, Bruce DA, Schut L. Magnetic resonance imaging of spinal cord disease of childhood. Pediatrics 1986; 78: 251-256. Gregorios JB, Green B, Page L, Thomsen S , Monforte H. Spinal cord tumors presenting with neural tube defects. Neurosurgery 1986; 19: 962966. Mawk JR, Leibrock LG, McComb RD, Trembath EJ. Metameric capillary hemangioma producing complete myelographicblock in an infant. J Neurosurg 1987; 67: 456-459. Balsubramaniam C, Laurent J, Rouah E et al. Congenital arachnoid cysts in children. Pediatr Neurosci 1989; 15: 223-228. Babyn SP, Chuang HS, Daneman A, Davidson SG. Sonographic evaluation of spinal cord birth trauma with pathologic correlation. Am J Roentgen 1988; 15 1: 763-766. Brant-Zawadzki M et at. Magnetic resonance imaging of the central nervous system: Ch 18, The Spine; p 289-328. Packer JR, Zimmerman AR et al. Magnetic resonance imaging of spinal cord disease of childhood. Pediatrics 1986; 78: 251-256.
Acta Paediatr Jpn
- 550)
Spinal Arachnoid Cyst in a Newborn Infant Kazuhiro Hashimoto, M.D.,’ Motoya Hashimoto, M.D.,’ Satoshi Homma, M.D.,’ Yoshiteru Ohsone, M.D., Hisaya Hasegawa, M.D.,’ Jiro Takei, M.D.,l Yoshikazu Kida, M.D..l Yutaka Takeuchi, M.D.,l and Minako Ohba, M.D.’ Division of Neonatology, Matudo City Hospital, Matudo City, Chiba, and ’Department of Pediatrics, Tohoku University School of Medicine, Sendai City, Miyagi, Japan
A male infant was born with asphyxia following a prolonged breech delivery at 39 weeks of gestation. He had a chylohemothorax from birth. Soon after birth he exhibited flaccid paraplegia with an absence of deep tendon reflexes in the lower extremities. At 17 days of age, magnetic resonance imaging (MRI) disclosed a hypodense area in the thoracic cord between C7 and Th4 in TI-weighted images, and spinal sonography revealed an echo-free space compressing the spinal cord in the same region observed with MRI. A spinal cyst was removed surgically at 24 days of age. The cyst was confirmed by pathology to be a true arachnoid cyst between C7and Th4. The cause of this cyst may have been a spinal cord injury during the perinatal period. Key Words Spinal arachnoid cyst, Spinal tumor, Newborn infants, Birth trauma
Introduction
Case Report
Spinal cord tumors are rarely observed in newborn infants. Congenital spinal tumors such as lipomas, teratomas and neuroblastomas have been reported in the literature [ 1-41 but, to our knowledge, spinal arachnoid cysts have never been reported. In cases of intracranial arachnoid cysts the origin has been considered to have resulted from traumatic damage to the brain [5]. We report a newborn infant with a spinal arachnoid cyst in the thoracic cord between C7 and Th,.
A male infant was born after a prolonged breech delivery at 39 weeks of gestation, weighing 2,540 g. His parents were non-consanguineous. His family history was unremarkable. The Apgar score was 1 and 6 at 1 and 5 min, respectively. He was intubated and transferred to our neonatal intensive care unit (NICU). On admission he exhibited flaccid paraplegia with the absence of deep tendon reflexes. Plantar grasp reflexes in the lower extremities were noted. Abdominal reflexes were absent, but cremaster and anocutaneous reflexes were weakly present. He did not respond to painful stimuli to the skin area innervated from the Th, to Th,, cord level. Movements of his upper extremities were normal. His head circumference was within normal limits. Physical examination disclosed no abnormal findings in the chest or abdomen.
Received March 13, 1992 Revised April 14, 1992 Accepted April 24, 1992 Correspondence address: Dr Kazuhiro Hashimoto, Division of Child Neurology, Tottori University School of Medicine, 86 Nishimachi, Yonago 683, Tottori, Japan.
548 ( 5 8 ) Hashimoto et al.
v
Echo-free space
Spinal cord
Fig. 1 : Longitudinal spinal ultrasonography. Note the echo-free space and compressed spinal cord.
Neither nevi nor localized hirsutism on the back were observed. The activities of liver transaminase, lactic dehydrogenaseand creatine phosphokinasewere increased. Metabolic acidosis was noted. Little cerebrospinal fluid (CSF) was obtained from the lumbar puncture. There were no bacteria in the CSF culture. Tumor markers, such as a-fetoprotein (AFP) and vanillylmandelic acid (VMA), were negative but the activity of
neuron-specific enolase in the CSF was mildly elevated. In plain roentgenographs of the chest and spine, a compressed deformity of the third thoracic vertebra was observed. Cranial computerized tomography (CT) and sonography of the brain were within normal limits. At 2 days of age, respiratory failure gradually worsened because of a chylohemothorax. Drainage from the thorax was performed on both sides for 10 days. He was fed on medium chain triglyceride
Fig. 2: MRI (T,-weighted image) at 24 days. Decreased signal intensity (arrowed) in the anterior subdural space extending from C, to Th,, a backward depressed cord and forward distorted vertebral bodies above the third vertebral body can be noted.
Acta Paediatr Jpn
Spinal arachnoid cyst in a newborn (59) 549 (MCT) milk for 30 days. At 17 ,days of age, sonograms (U-sonic RT 2800, Tosiba Medical Co, Ltd, Tokyo, Japan) with a 3.75 MHz linear probe demonstrated an echo-free space that compressed the spinal cord backward (Fig. 1). Magnetic resonance imaging (MRI), with a Magnetom operating at 0.5 T (Siemens Co, Ltd, Tokyo, Japan) produced TI-weighted images (spin-echo: pulse interval, 200 msec; time interval, 15 msec) that demonstrated decreased signal intensity between the vertebral bodies and the spinal cord extending from C7to Th4. The spinal cord appeared to be thin and was distorted backward. The vertebral bodies were distorted forward above the third vertebral body. On the administration of gadoliniumDTPA, no enhanced area was observed in Tiweighted images (Fig. 2). At 24 days of age, an operation for the spinal tumor was performed. The spinal cord was pressed backward but the diameter seemed to be normal, and a membrane-bound cyst in front of the thoracic cord between C7 and Th4 was confirmed. The membrane of the cyst was totally removed. After the operation, the patient’s left ankle began to move but his right ankle did not move at all, and
the sensory disturbance remained. At 80 days of age, MRI was performed again and TI-weighted images demonstrated decreased signal intensity, with irregular margins in the subdural space behind the spinal cord extending from C7 and Th, (Fig. 3). The spinal cord between C7 and Th, was atrophic and the signal intensity within the anterior space, in which the removed cyst had been located, was the same as in the CSF. The nature of the area exhibiting irregularly decreased signal intensity was unclear. An operation to examine this area was undertaken at 3 months of age, when it was found that the posterior arachnoid space was full of fatty tissue. At present (1 year of age), his paraplegia is gradually improving but the sensory disturbance remains.
Discussion A spinal cord lesion was suspected in our patient from the following neurological findings: flaccid paraplegia, absence of deep tendon reflexes restricted to the lower extremities, and sensory disturbance at levels between Th4 and Th12. Spinal sonograms and MRI clearly demon-
Fig. 3: MRI (T,-weighted image) at 80 days. Irregular decreased signal intensity in the posterior subdural space extending from C , to Th,, spinal cord atrophy at the levels of C,-Th, and distorted vertebrae can be noted.
Vol. 34 No. 5 October 1992
550 (60) Hashimoto et al. strated an arachnoid cystic lesion extending between C, and Th,. In spinal sonograms, the lesion was observed in real time as an echo-free space that compressed the spinal cord. In TIweighted images on MRI, the lesion was observed to be as hypodense as CSF and was not enhanced, and thus an arachnoid cyst was suspected. There have been a few reports of spinal tumors in infancy [l-41. Most of these spinal tumors were located in the sacral or thoracolumbar portions, and most of the tumors were lipomas, teratomas or neuroblastomas. To our knowledge, there has been no report of a spinal arachnoid cyst in infancy. Generally, the origin of an arachnoid cyst is thought to be congenital or traumatic. In the present case, a compressed deformity of the third thoracic vertebral body diagnosed at birth, distortion of the vertebral bodies at the level of the third vertebral body, and severe atrophy of the spinal cord at the same level strongly suggested traumatic damage to the spine during the perinatal period. However, there was no direct evidence of the cause of the trauma, except for the asphyxiated breech delivery and the chylohemothorax. Also, the spinal cord atrophy may be secondary to the compression by the cyst or may be the result of spinal damages due to birth trauma.
In conclusion, MRI and spinal sonography were non-invasive and helpful for delineating the lesion in the spinal cord [6-81, which is a difficult area for diagnosis in small infants.
References 1.
2.
3.
4.
5. 6.
7. 8.
Tibbs PA, James HE, Rorke LB, Schut L, Bruce DA. Midline hamartomas masquerading as meningoceles or teratomas in the newborn infant. J Pediatr 1976; 89: 928-933. Packer RJ, Zimmerman RA, Sutton LN, Bilaniuk LT, Bruce DA, Schut L. Magnetic resonance imaging of spinal cord disease of childhood. Pediatrics 1986; 78: 251-256. Gregorios JB, Green B, Page L, Thomsen S , Monforte H. Spinal cord tumors presenting with neural tube defects. Neurosurgery 1986; 19: 962966. Mawk JR, Leibrock LG, McComb RD, Trembath EJ. Metameric capillary hemangioma producing complete myelographicblock in an infant. J Neurosurg 1987; 67: 456-459. Balsubramaniam C, Laurent J, Rouah E et al. Congenital arachnoid cysts in children. Pediatr Neurosci 1989; 15: 223-228. Babyn SP, Chuang HS, Daneman A, Davidson SG. Sonographic evaluation of spinal cord birth trauma with pathologic correlation. Am J Roentgen 1988; 15 1: 763-766. Brant-Zawadzki M et at. Magnetic resonance imaging of the central nervous system: Ch 18, The Spine; p 289-328. Packer JR, Zimmerman AR et al. Magnetic resonance imaging of spinal cord disease of childhood. Pediatrics 1986; 78: 251-256.
Acta Paediatr Jpn
