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Spinal injury and neonatal death
Spinal injury and neonatal death Report
of six cases
CARL
E.
WALTER,
LUKE
G.
TEDESCHI,
Framingham,
M.D. M.D.
Massachusetts
Six out of 8 consecutive infants at this hospital displayed at postmortem examination intrarachidian hemorrhage and concurrent spinal cord and brain stem injury. Since not until recently has examination of the rachidian space been made part of the routine autopsy in stillborn and newborn infants, this series, although impressive, may reflect a chance occurrence and does not necessarily express the actual incidence with which the lesion takes place. In the absence of any other suitable explanation, this occurrence focuses attention on the need of lasting consideration of the problem of trauma as a cause of neonatal death and crippling of neurological functions. Assuming that in these 6 cases the rachidian injury was traumatic in origin, in every instance the trauma had apparently been so mild as to escape recognition. Predisposing factors, including the role of the physiologic hypoxia associated with the neonatal period, are given consideration.
u
N DER
THE
TERM
Of
“SpiIlal
sudden death of the infant after labor. Another possibility is that the infant survives for a short period, after showing manifestations of respiratory depression, spinal shock, and hyaline membrane disease. Third, the infant may survive with transient or permanent signs and symptoms of paralysis and spasticity. Respiratory depression and hypoxia may concurrently lead to devastation of the cerebral cortex with ultimate blighting of mentation and motor deficit, as seen in cerebral palsy. Damage to the brain stem or to the nuclei of cranial nerves and spinal nerve root lesions may in turn be responsible for paralysis of the diaphragm and random deficit of cranial nerves.? Patterns of damage at the postmortem table lack specificity and bear the unpredictable imprint of any injury related to trauma. Vertebral fracture and dislocation with transectional injury of the cord have been described by early investigators but are rarely seen nowadays. Epidural, subdural, and intradural hemorrhage, of varying severity and distribution, occasionally dural tears, arachnoid congestion, and subarach-
a&IO-
plexy,” Kennedy,l in 1836, called attention to spinal trauma at birth as a cause of lethal injury to the cord and brain stem. In the course of time, several other investigators have substantiated the observation.g-6 Renewed interest on the subject has been elicited by the work of Towbin. Among the causes of spinal cord injury at birth, he stresses the effects of excessive traction, flexion, torsion of the vertebral column, causing stretch injury, and compression of the spinal cord and brain stem structures; contributing factors include intrauterine fetal malposition, such as brow, face, breech presentation, dystocia, prematurity, primiparity, precipitate delivery, intraspinal vascular occlusive phenomena, and vertebral and foramen magnum malformations in the fetus. The outcome of the trauma cannot be predicted. One possibility is stillbirth, or
From the Department of Pathology, Framingham Union Hospital, and the Boston University Medical Center. 272
Volume Number
106 2
noid hemorrhage are the most startling findings. The cervical and upper thoracic regions are most commonly involved, and not infrequently the entire length of the spinal canal shows heavy accumulation of blood. The hemorrhage often extends outward into the intervertebral structures and infiltrates the soft tissues which envelop the spinal column. Damage to the vertebral bones and tears of joint capsules and ligaments may be easily overlooked, but they may occur as actually demonstrated in sevrral cases of Stoltzenberg’s* series. The longitudinal continuity of the cord is usually preserved (nontransectional segmental injury), but intrinsic lesions almost invariably occur, primarily vascular congestion and thrombosis, with focal hemorrhage, malacia, and acute neuronal damage which are responsible for crippling of neurological functions. Fibrous thickening of the meninges, meningeal adhesions, hemosiderin deposits, and reactive gliosis are the expected sequalae, and they have been recorded in infants who survived, for a length of time, the injury sustained at birth.G In combination with spinal trauma, stretch injury of the brain stem is not uncommon, with laceration of the cerebellar peduncles, cerebellar hemispheres, and other structures about the fourth ventricle. Former obstetric techniques, with wide indications for version and extraction of the fetus in face and breech presentations, were held responsible to a great extent for trauma to the spinal cord. However, not infrequently, as in some of the cases of Crothers and Putnam,G no evidence could be found to the effect that the infant had sustained undue trauma at birth. The concept that abnormal intrauterine position from longitudinal to transverse (flying fetus), may result in hyperextension of the fetal head and eventually in spinal trauma is also well documented.1° If one of the major goals of pathologic observation is to establish preventive measures: the message by Hellstrom and Sallmanderll that hyperestension of the head can be demon-
Spinal
injury
and
neonatal
death
273
strated roentgenologically after labor has started, and that permanent injuries resulting from this position can probably be avoided by timely cesarean section, represents a step in the right direction. The clinical diagnosis of neonatal spinal cord and brain stem injury is often missed. Furthermore, since the spinal cord is not routinely examined at postmortem examination and thus the injury may escape detection, no reliable data are available on the actual incidence of neonatal spinal trauma. A fair estimate is that from 10 to 33 per cent of the deaths occurring in the neonatal period are due to damage of spinal structures. The finding of spinal cord injury in 6 out of 8 consecutive postmortem examinations in newborn infants at this hospital establishes a situation impressive enough to warrant this communication. Not until recently, approximately one year ago, when we came across the first case here reported, was the spinal cord routinely included in the postmortem examination of stillborn and newborn infants. Thus in this series, as in those of others, it remains undetermined how many other deaths previously ascribed to hyaline membrane disease or to other conditions might have actually turned out to be due to rachidian trauma, had the spinal cord been examined. With increasing awareness of the problem and the inclusion of spinal cord examination as part of the routine autopsy, more realistic figures can soon be expected. Report of cases Case No. 1 (A-67-192). The patient was a well-developed, apparently healthy, 3,730 gram, term male infant. Gestation and delivery were uneventful. Three days after birth, he was found dead in the crib. Review of the medical record and questioning of the attending physician and nursery personnel failed to provide any clue as to the unexpected death. A postmortem examination was performed, and death was attributed to cardiorespiratory collapse secondary to spinal cord injury. The heart chambers were dilated, the lungs edematous, and the liver, spleen, and kidneys were congested. The brain showed marked edema with
274
Walter
and Tedeschi
.Irne~-.
Fig. 1. Epidural hemorrhage in the cervical and upper thoracic segment. Case No. old mature male infant; normal gestation and delivery. Sudden death.
flattening of convolutions but no apparent herniations of brain parts. Opening of the spinal canal by the anterior route showed epidural hemorrhage in the cervical and upper thoracic segment (Fig. 1). Smaller amounts of blood were present also in the subdural spinal space, and the arachnoid membranes were congested with focal hemorrhages. The microscopic study was limited to small histologic sections from the grossly altered portions of cord. Prominent features included acute neuronal damage, focal areas of malacia and hemorrhage, and diffuse tissular congestion and edema. These alterations were most severe in the upper cervical segment but to a lesser extent they were present also in the brain stem. No fractured vertebrae were detected, and capsules and ligaments were apparently intact. Case No. 2 (A-67-200). The patient was a premature male infant of 33 weeks’ gestational age and 760 grams in weight. He was delivered vaginally in cephalic presentation. The mothcl was febrile and the membranes had ruptured 2 weeks previously. After birth the infant had periods of respiratory depression with apnea and cyanosis. A generalized systolic murmur was present on the precordial region. Evidence of respiratory depression gradually increased and death occurred 9 hours post partum. Autopsy showed cardiac dilatation, disseminated pulmonary atelectasis, congestion of liver and spleen, and prominent cerebral edema. Anterior opening of the spinal canal showed epi-
Janual-y J. Obstet.
15. 19ill Gyncc.
3-day-
dural and, to a lesser extent, subdural hemorrhage extending from the upper cervical down to the lumbar segments. The blood was freshly clotted. The arachnoid was congested and the hemorrhagic process extended into the soft tissues surrounding the spinal column. Dural tears and damage to vertebral bones, joint caps&c, and ligaments could not be shown. The longitudinal continuity of the cord was preserved hut intrinsic lesions could be demonstrated by the microscopic sections, primarily focal hemorrhage and malacia, most prominent in the brain stem and upper cervical region. Case No. 3 (A-68-3). The patient was a premature female infant, 6 pounds 4 ounces in weight, of a gestational age of 35% weeks. Delivery was uncomplicated, with cephalic presentation. Respiratory distress was present at birth, with shallow breathing, periods of apnea, and tachycardia. Respiratory depression became increasingly severe, and death occurred the following day. Postmortem examination showed cardiac dilatation, pulmonary edema with focal areas of atelectasis, and congestion of liver and spleen. Anterior opening of the spinal canal revealed large amounts of freshly clotted blood filling the epidural space from the cervical down to thr lumbar segments. Subdural blood clots were preqent in the cervical region and at this level the arachnoid was congested and hemorrhagic. The hemorrhage extended into the soft tissues surrounding the vertebrae and microscopically into
Volume Number
106 2
Spinal
injury
and
neonatal
death
275
Fig. 2. Large amounts of freshly clotted blood on the epidural surfaces of the cervical, thoracic and lumbar spinal cord. Case No. 4, premature male infant delivered by cesarean section at the eighth month of gestation because of placenta previa. Death 40 hours after delivery.
the spinal nerve roots. The cord was edematous and congested with acute neuronal damage and focal areas of hemorrhage and malacia, most prominent in the upper cervical segment. Case No. 4 (A-68-51). A male infant, 4 pounds 2 ounces, was delivered by cesarean section at the eighth month of gestation because of placenta previa. Incisions, of both the abdominal and uterine walls, were adequate. From birth he showed shallow respiration with periods of apnea and cyanosis. Respiratory depression became progressively severe, and death occurred 40 hours post partum. Postmortem examination showed cardiac dilatation and visceral congestion. The lungs were firm, poorly aerated, and microscopically showed hyaline membrane disease. The spinal canal contained large amounts of freshly clotted blood on the epidural surfaces of the cervical, thoracic, and lumbar spinal cord (Fig. 2). The soft tissues surrounding the cervical spine were infiltrated with blood, but vertebrae and ligaments were apparently intact. The arachnoid was congested, with focal hemorrhages in the cervical tract, the subjacent cord was also malacic, and microscopically showed acute neuronal damage, tissue edema and hemorrhage, and thrombosis of small branches of spinal artery. A spinal nerve at the point of exit from the dura showed perineural hemorrhage. Case No. 5 (A-68-112). This 6-week-old male infant was the third child of a mother who had had two previously uncomplicated pregnancies.
In the third pregnancy, a rising titer against anti-D was noted, and the mother underwent cesarean section. After birth the baby required several exchange transfusions which were well tolerated. While he was being followed at home after discharge from the hospital, an anemia was noted, with the hemoglobin falling in the range of 8.2 to 8.8 Cm. The baby otherwise appeared to be doing quite well, and no other significant abnormalities were noted. He was found dead in the crib. Postmortem examination revealed moderate to large amounts of soft, gelatinous, reddish brown clots on the epidural surfaces of the cervical and upper thoracic cord. Microscopically, diffuse epidural hemorrhage was noted (Fig. 3 ) . The arachnoid at several points of thr cervical segment was lifted by blood clots (Fig. 4) and the cord itself was edematous with acute neuronal damage, particularly pronounced in the dorsal and lateral gray columns. Viscera were congested and the lungs showed scattered areas of atelectasis. Case No. 6 (A-66-121). This 2-day-old white male, fulI-term infant, was delivered by elective repeat cesarean section. The pregnancy was normal in all respects. The baby was vigorous initially with an Apgar rating of 9 out of a possible 10. The birth weight was 6 pounds 11 ounces, and the initial physical examination was completely within normal limits. Approximately one-half hour after delivery, the infant developed respiratory distress and was
276
Walter
and
Tedeschi
Amel-.
Fig. 3. Microscopic view of epidural delivered close to term by cesarean crib 6 weeks after delivery.
Fig.
4. Massive
collection
of blood
hemorrhage (cervical segment). section because of rising anti-D
in
the subarachnoid
noted to have a dusky color. The cyanosis decreased after the administration of oxygen. Rapid, shallow respirations with substernal retractions and fine, crepitant rgles were heard bilaterally. A portable chest film taken 8 hours after delivery showed normal-appearing heart and lungs. However, 24 hours after delivery the infant was noted to have increased respiratory distress and a fever of 100.4O F. He was given Kanamycin and Ampicillin and administration
space.
Same
January 15: 19io J. Obstet. Gynec.
Case No. 5, male infant titer. Found dead in the
case as Fig.
3.
of oxygen was continued. The baby’s condition remained essentially unchanged and he died 48 hours after birth. Autopsy showed severe bilateral atelectasis of the lungs with hyaline membranes and focal superimposed acute pneumonia. From the cervical down to the lumbar level, the spinal dura was coated by an irregular layer of clotted blood (Fig. 5). Lesser amounts of blood were present also in the subdural space. The spinal cord was
Volume Number
106 2
Fig. 5. Full-term Notice massive segment. Death
Spinal
male infant delivered epidural hemorrhage 48 hours after birth.
grossly normal but random, played arachnoid congestion sue edema and focal malacia lateral gray columns.
small sections with prominent in the dorsal
by elective in rachidian
distisand
Comment
The patients described show close similarities. In all 6, death was ascribed to intrarachidian traumatic hemorrhage with the assumption that the visceral congestion and the lung changes, mainly edema, atelectasis, and hyaline membrane disease, were secondary manifestations of cardiorespiratory distress of central origin. In 4 of these 6 cases (Case Nos. 2, 3, 4, 6)) the fatal outcome was in some way predictable. In Case No. 2, for instance, the infant weighed 760 grams and with or without the intrarachidian hemorrhage did not have much chance for survival. All infants died from 9 to 48 hours from birth after developing signs and symptoms of progressively worsening respiratory depression with shallow respiration, apnea, cyanosis, or pallor. The only valuable information that can be obtained from the history of these 4 cases is that 3 of the infants were premature, that 2 of them were the product of a cesarean section, and that one had lodged in the uterus for 2 weeks following rupture of the membranes while the mother was febrile, for undisclosed causes. As stressed by other investigators, prematurity is a well-estab-
repeat cesarean space extending
injury
section. from
and
neonatal
Normal cervical
death
277
pregnancy. to lumbar
lished contributing factor of traumatic spinal injury. Case No. 1 requires separate consideration. Death came unexpectedly 3 days after birth. The baby was at term of normal lzeight , gestation and delivery had been uneventful, and no premonitory signs of distress were detected from the time of birth to the time he was found dead in the crib. It is generally acknowledged that the clinical diagnosis of neonatal spinal cord and brain stem injury is difficult and that diagnosis can be missed entirely unless the location of possible damage is looked for by special methods (oxygen myelography) .ll The circumstances attending the death of the infant of Case No. 5 are even more puzzling. He was found dead in the crib, 6 weeks after term delivery by cesarean section; the indication for this procedure was a rising anti-D titer which required repeated exchange transfusions. After discharge from the hospital, the infant was doing well, except for a drop in hemoglobin to a range of 8.2 to 8.8 Gm. In the absence of any other suitable explanation, one wonders about possible links between the intraspinal hemorrhage and the drop in hemoglobin levels. Assuming that the injury took place in the perinatal period, in the light of the postmortem findings, two other points deserve consideration : one is the apparent lack of neurological manifestations in the period in-
278
Walter
and
Tedeschi Amer.
tervening between birth and death, and the other is the long-term survival of the infant (Group 3 of Towbin classification). Since serially cut, whole brain, whole spine sections were not carried out in the present study, a quantitative and topographic assessment of central nervous system lesions is of no avail. Probably in the central nervous system more than in other organ systems, there is a large gap between the amount of injury required to abolish or to prevent function and the amount required to cause damage of a type that one might expect to recognize either grossly or under the microscope. vertical compression, and Hyperflexion, hyperextension are the most common mechanisms by which the spine can be injured. This applies to perinatal trauma as well as to the accidental trauma which may occur later in life. Nonetheless, the circumstances operating in perinatal trauma are almost of an entirely different nature than those related to mechanical trauma taking place after birth. The substrate differs in plasthickness, and tensile ticity, elasticity, strength. The large head and weak neck of the infant make him particularly vulnerable to the mechanisms of injury. Perinatal trauma apparently can be so mild as to escape recognition until functional deficit becomes apparent or evidence of damage is found unexpectedly at postmortem examination. In every one of the cases here reported, the record was carefully reviewed and the interrogated to determine obstetricians whether or not these deliveries were more
January 15, 1970 J. Obstet. Gynec.
traumatic than usual. Apparently they were not. Similarly, in the cases delivered by cesarean section, the incisions in the abdominal wall and uterus were adequate and there was no “tight squeeze.” The physiologic hypoxia associated with the neonatal period makes the nervous tissue and the walls of the blood channels especially vulnerable to the forces of external trauma. Fetal hypoxia causes venous congestion and increased venous and cerebral spinal fluid pressure. As the brain stem becomes injured, depression of the respiratory centers and pulmonary complications furthe manifestations of ther accentuate anoxia, giving rise to a vicious cycle and to an overlapping of traumatic and anoxic changes. Should anoxia alone have been responsible for the rachidian hemorrhage, one would expect to find similar changes of comparable severity in the viscera, but this was not the case in our observations, nor in similar observations by others. Assuming that trauma was the primary cause of the rachidian injury, the time sequence cannot be established and it remains undetermined whether the trauma occurred in the uterus, at the time of delivery, or even in the postnatal period. In an age in which so much effort is being directed to the prevention of disease, observation such as the ones here presented call for the application of all possible precautionary measures in the handling of the infant in the perinatal period, espccially of the premature infant who is particularly susceptible to traumatic spinal injury.
REFERENCES
E.: Dublin J. Med. Sci. 10: 419, 1. Kennedy, 1836. C. W.: Amer. J. Dis. Child. 19: 473, 2. Burr, 1920. 3. Crothers, B.: Amer. J. Med. Sci. 165: 94, 1923. 4. Ford, F. R.: Arch. Neurol. Psychiat. 14: 742, 1925. R. N.: Surg. Gynec. Obstet. 37: 5. Pierson, 802, 1923. 6. Crothers, B., and Putnam, M. Cl.: In Ford, F. R., Crothers, B., and Putnam, M. C., ed-
itors: tem,
7. 8. 9. 10. 11.
Birth Iniuries of Central Nervous SysPart II,- Medical Monographs, Baltimore, 1927, The Williams & Wilkins Company, vol. II. Towbin, A.: Arch. Path. 77: 620, 1964. Stoltzenberg. F.: Klin. Wschr. 48: 1741. 1911. Melchior, 7. Cl., and Tygstrup, I.:’ Acta Paediat. 52: 171, 1963. Knowlton, R. W.: J. Obstet. Gynaec. Brit. Comm. 45: 834, 1938. Hellstrijm, B. O., and Sallmander, U.: J. A. M. A. 204: 1041, 1968.
of six cases
CARL
E.
WALTER,
LUKE
G.
TEDESCHI,
Framingham,
M.D. M.D.
Massachusetts
Six out of 8 consecutive infants at this hospital displayed at postmortem examination intrarachidian hemorrhage and concurrent spinal cord and brain stem injury. Since not until recently has examination of the rachidian space been made part of the routine autopsy in stillborn and newborn infants, this series, although impressive, may reflect a chance occurrence and does not necessarily express the actual incidence with which the lesion takes place. In the absence of any other suitable explanation, this occurrence focuses attention on the need of lasting consideration of the problem of trauma as a cause of neonatal death and crippling of neurological functions. Assuming that in these 6 cases the rachidian injury was traumatic in origin, in every instance the trauma had apparently been so mild as to escape recognition. Predisposing factors, including the role of the physiologic hypoxia associated with the neonatal period, are given consideration.
u
N DER
THE
TERM
Of
“SpiIlal
sudden death of the infant after labor. Another possibility is that the infant survives for a short period, after showing manifestations of respiratory depression, spinal shock, and hyaline membrane disease. Third, the infant may survive with transient or permanent signs and symptoms of paralysis and spasticity. Respiratory depression and hypoxia may concurrently lead to devastation of the cerebral cortex with ultimate blighting of mentation and motor deficit, as seen in cerebral palsy. Damage to the brain stem or to the nuclei of cranial nerves and spinal nerve root lesions may in turn be responsible for paralysis of the diaphragm and random deficit of cranial nerves.? Patterns of damage at the postmortem table lack specificity and bear the unpredictable imprint of any injury related to trauma. Vertebral fracture and dislocation with transectional injury of the cord have been described by early investigators but are rarely seen nowadays. Epidural, subdural, and intradural hemorrhage, of varying severity and distribution, occasionally dural tears, arachnoid congestion, and subarach-
a&IO-
plexy,” Kennedy,l in 1836, called attention to spinal trauma at birth as a cause of lethal injury to the cord and brain stem. In the course of time, several other investigators have substantiated the observation.g-6 Renewed interest on the subject has been elicited by the work of Towbin. Among the causes of spinal cord injury at birth, he stresses the effects of excessive traction, flexion, torsion of the vertebral column, causing stretch injury, and compression of the spinal cord and brain stem structures; contributing factors include intrauterine fetal malposition, such as brow, face, breech presentation, dystocia, prematurity, primiparity, precipitate delivery, intraspinal vascular occlusive phenomena, and vertebral and foramen magnum malformations in the fetus. The outcome of the trauma cannot be predicted. One possibility is stillbirth, or
From the Department of Pathology, Framingham Union Hospital, and the Boston University Medical Center. 272
Volume Number
106 2
noid hemorrhage are the most startling findings. The cervical and upper thoracic regions are most commonly involved, and not infrequently the entire length of the spinal canal shows heavy accumulation of blood. The hemorrhage often extends outward into the intervertebral structures and infiltrates the soft tissues which envelop the spinal column. Damage to the vertebral bones and tears of joint capsules and ligaments may be easily overlooked, but they may occur as actually demonstrated in sevrral cases of Stoltzenberg’s* series. The longitudinal continuity of the cord is usually preserved (nontransectional segmental injury), but intrinsic lesions almost invariably occur, primarily vascular congestion and thrombosis, with focal hemorrhage, malacia, and acute neuronal damage which are responsible for crippling of neurological functions. Fibrous thickening of the meninges, meningeal adhesions, hemosiderin deposits, and reactive gliosis are the expected sequalae, and they have been recorded in infants who survived, for a length of time, the injury sustained at birth.G In combination with spinal trauma, stretch injury of the brain stem is not uncommon, with laceration of the cerebellar peduncles, cerebellar hemispheres, and other structures about the fourth ventricle. Former obstetric techniques, with wide indications for version and extraction of the fetus in face and breech presentations, were held responsible to a great extent for trauma to the spinal cord. However, not infrequently, as in some of the cases of Crothers and Putnam,G no evidence could be found to the effect that the infant had sustained undue trauma at birth. The concept that abnormal intrauterine position from longitudinal to transverse (flying fetus), may result in hyperextension of the fetal head and eventually in spinal trauma is also well documented.1° If one of the major goals of pathologic observation is to establish preventive measures: the message by Hellstrom and Sallmanderll that hyperestension of the head can be demon-
Spinal
injury
and
neonatal
death
273
strated roentgenologically after labor has started, and that permanent injuries resulting from this position can probably be avoided by timely cesarean section, represents a step in the right direction. The clinical diagnosis of neonatal spinal cord and brain stem injury is often missed. Furthermore, since the spinal cord is not routinely examined at postmortem examination and thus the injury may escape detection, no reliable data are available on the actual incidence of neonatal spinal trauma. A fair estimate is that from 10 to 33 per cent of the deaths occurring in the neonatal period are due to damage of spinal structures. The finding of spinal cord injury in 6 out of 8 consecutive postmortem examinations in newborn infants at this hospital establishes a situation impressive enough to warrant this communication. Not until recently, approximately one year ago, when we came across the first case here reported, was the spinal cord routinely included in the postmortem examination of stillborn and newborn infants. Thus in this series, as in those of others, it remains undetermined how many other deaths previously ascribed to hyaline membrane disease or to other conditions might have actually turned out to be due to rachidian trauma, had the spinal cord been examined. With increasing awareness of the problem and the inclusion of spinal cord examination as part of the routine autopsy, more realistic figures can soon be expected. Report of cases Case No. 1 (A-67-192). The patient was a well-developed, apparently healthy, 3,730 gram, term male infant. Gestation and delivery were uneventful. Three days after birth, he was found dead in the crib. Review of the medical record and questioning of the attending physician and nursery personnel failed to provide any clue as to the unexpected death. A postmortem examination was performed, and death was attributed to cardiorespiratory collapse secondary to spinal cord injury. The heart chambers were dilated, the lungs edematous, and the liver, spleen, and kidneys were congested. The brain showed marked edema with
274
Walter
and Tedeschi
.Irne~-.
Fig. 1. Epidural hemorrhage in the cervical and upper thoracic segment. Case No. old mature male infant; normal gestation and delivery. Sudden death.
flattening of convolutions but no apparent herniations of brain parts. Opening of the spinal canal by the anterior route showed epidural hemorrhage in the cervical and upper thoracic segment (Fig. 1). Smaller amounts of blood were present also in the subdural spinal space, and the arachnoid membranes were congested with focal hemorrhages. The microscopic study was limited to small histologic sections from the grossly altered portions of cord. Prominent features included acute neuronal damage, focal areas of malacia and hemorrhage, and diffuse tissular congestion and edema. These alterations were most severe in the upper cervical segment but to a lesser extent they were present also in the brain stem. No fractured vertebrae were detected, and capsules and ligaments were apparently intact. Case No. 2 (A-67-200). The patient was a premature male infant of 33 weeks’ gestational age and 760 grams in weight. He was delivered vaginally in cephalic presentation. The mothcl was febrile and the membranes had ruptured 2 weeks previously. After birth the infant had periods of respiratory depression with apnea and cyanosis. A generalized systolic murmur was present on the precordial region. Evidence of respiratory depression gradually increased and death occurred 9 hours post partum. Autopsy showed cardiac dilatation, disseminated pulmonary atelectasis, congestion of liver and spleen, and prominent cerebral edema. Anterior opening of the spinal canal showed epi-
Janual-y J. Obstet.
15. 19ill Gyncc.
3-day-
dural and, to a lesser extent, subdural hemorrhage extending from the upper cervical down to the lumbar segments. The blood was freshly clotted. The arachnoid was congested and the hemorrhagic process extended into the soft tissues surrounding the spinal column. Dural tears and damage to vertebral bones, joint caps&c, and ligaments could not be shown. The longitudinal continuity of the cord was preserved hut intrinsic lesions could be demonstrated by the microscopic sections, primarily focal hemorrhage and malacia, most prominent in the brain stem and upper cervical region. Case No. 3 (A-68-3). The patient was a premature female infant, 6 pounds 4 ounces in weight, of a gestational age of 35% weeks. Delivery was uncomplicated, with cephalic presentation. Respiratory distress was present at birth, with shallow breathing, periods of apnea, and tachycardia. Respiratory depression became increasingly severe, and death occurred the following day. Postmortem examination showed cardiac dilatation, pulmonary edema with focal areas of atelectasis, and congestion of liver and spleen. Anterior opening of the spinal canal revealed large amounts of freshly clotted blood filling the epidural space from the cervical down to thr lumbar segments. Subdural blood clots were preqent in the cervical region and at this level the arachnoid was congested and hemorrhagic. The hemorrhage extended into the soft tissues surrounding the vertebrae and microscopically into
Volume Number
106 2
Spinal
injury
and
neonatal
death
275
Fig. 2. Large amounts of freshly clotted blood on the epidural surfaces of the cervical, thoracic and lumbar spinal cord. Case No. 4, premature male infant delivered by cesarean section at the eighth month of gestation because of placenta previa. Death 40 hours after delivery.
the spinal nerve roots. The cord was edematous and congested with acute neuronal damage and focal areas of hemorrhage and malacia, most prominent in the upper cervical segment. Case No. 4 (A-68-51). A male infant, 4 pounds 2 ounces, was delivered by cesarean section at the eighth month of gestation because of placenta previa. Incisions, of both the abdominal and uterine walls, were adequate. From birth he showed shallow respiration with periods of apnea and cyanosis. Respiratory depression became progressively severe, and death occurred 40 hours post partum. Postmortem examination showed cardiac dilatation and visceral congestion. The lungs were firm, poorly aerated, and microscopically showed hyaline membrane disease. The spinal canal contained large amounts of freshly clotted blood on the epidural surfaces of the cervical, thoracic, and lumbar spinal cord (Fig. 2). The soft tissues surrounding the cervical spine were infiltrated with blood, but vertebrae and ligaments were apparently intact. The arachnoid was congested, with focal hemorrhages in the cervical tract, the subjacent cord was also malacic, and microscopically showed acute neuronal damage, tissue edema and hemorrhage, and thrombosis of small branches of spinal artery. A spinal nerve at the point of exit from the dura showed perineural hemorrhage. Case No. 5 (A-68-112). This 6-week-old male infant was the third child of a mother who had had two previously uncomplicated pregnancies.
In the third pregnancy, a rising titer against anti-D was noted, and the mother underwent cesarean section. After birth the baby required several exchange transfusions which were well tolerated. While he was being followed at home after discharge from the hospital, an anemia was noted, with the hemoglobin falling in the range of 8.2 to 8.8 Cm. The baby otherwise appeared to be doing quite well, and no other significant abnormalities were noted. He was found dead in the crib. Postmortem examination revealed moderate to large amounts of soft, gelatinous, reddish brown clots on the epidural surfaces of the cervical and upper thoracic cord. Microscopically, diffuse epidural hemorrhage was noted (Fig. 3 ) . The arachnoid at several points of thr cervical segment was lifted by blood clots (Fig. 4) and the cord itself was edematous with acute neuronal damage, particularly pronounced in the dorsal and lateral gray columns. Viscera were congested and the lungs showed scattered areas of atelectasis. Case No. 6 (A-66-121). This 2-day-old white male, fulI-term infant, was delivered by elective repeat cesarean section. The pregnancy was normal in all respects. The baby was vigorous initially with an Apgar rating of 9 out of a possible 10. The birth weight was 6 pounds 11 ounces, and the initial physical examination was completely within normal limits. Approximately one-half hour after delivery, the infant developed respiratory distress and was
276
Walter
and
Tedeschi
Amel-.
Fig. 3. Microscopic view of epidural delivered close to term by cesarean crib 6 weeks after delivery.
Fig.
4. Massive
collection
of blood
hemorrhage (cervical segment). section because of rising anti-D
in
the subarachnoid
noted to have a dusky color. The cyanosis decreased after the administration of oxygen. Rapid, shallow respirations with substernal retractions and fine, crepitant rgles were heard bilaterally. A portable chest film taken 8 hours after delivery showed normal-appearing heart and lungs. However, 24 hours after delivery the infant was noted to have increased respiratory distress and a fever of 100.4O F. He was given Kanamycin and Ampicillin and administration
space.
Same
January 15: 19io J. Obstet. Gynec.
Case No. 5, male infant titer. Found dead in the
case as Fig.
3.
of oxygen was continued. The baby’s condition remained essentially unchanged and he died 48 hours after birth. Autopsy showed severe bilateral atelectasis of the lungs with hyaline membranes and focal superimposed acute pneumonia. From the cervical down to the lumbar level, the spinal dura was coated by an irregular layer of clotted blood (Fig. 5). Lesser amounts of blood were present also in the subdural space. The spinal cord was
Volume Number
106 2
Fig. 5. Full-term Notice massive segment. Death
Spinal
male infant delivered epidural hemorrhage 48 hours after birth.
grossly normal but random, played arachnoid congestion sue edema and focal malacia lateral gray columns.
small sections with prominent in the dorsal
by elective in rachidian
distisand
Comment
The patients described show close similarities. In all 6, death was ascribed to intrarachidian traumatic hemorrhage with the assumption that the visceral congestion and the lung changes, mainly edema, atelectasis, and hyaline membrane disease, were secondary manifestations of cardiorespiratory distress of central origin. In 4 of these 6 cases (Case Nos. 2, 3, 4, 6)) the fatal outcome was in some way predictable. In Case No. 2, for instance, the infant weighed 760 grams and with or without the intrarachidian hemorrhage did not have much chance for survival. All infants died from 9 to 48 hours from birth after developing signs and symptoms of progressively worsening respiratory depression with shallow respiration, apnea, cyanosis, or pallor. The only valuable information that can be obtained from the history of these 4 cases is that 3 of the infants were premature, that 2 of them were the product of a cesarean section, and that one had lodged in the uterus for 2 weeks following rupture of the membranes while the mother was febrile, for undisclosed causes. As stressed by other investigators, prematurity is a well-estab-
repeat cesarean space extending
injury
section. from
and
neonatal
Normal cervical
death
277
pregnancy. to lumbar
lished contributing factor of traumatic spinal injury. Case No. 1 requires separate consideration. Death came unexpectedly 3 days after birth. The baby was at term of normal lzeight , gestation and delivery had been uneventful, and no premonitory signs of distress were detected from the time of birth to the time he was found dead in the crib. It is generally acknowledged that the clinical diagnosis of neonatal spinal cord and brain stem injury is difficult and that diagnosis can be missed entirely unless the location of possible damage is looked for by special methods (oxygen myelography) .ll The circumstances attending the death of the infant of Case No. 5 are even more puzzling. He was found dead in the crib, 6 weeks after term delivery by cesarean section; the indication for this procedure was a rising anti-D titer which required repeated exchange transfusions. After discharge from the hospital, the infant was doing well, except for a drop in hemoglobin to a range of 8.2 to 8.8 Gm. In the absence of any other suitable explanation, one wonders about possible links between the intraspinal hemorrhage and the drop in hemoglobin levels. Assuming that the injury took place in the perinatal period, in the light of the postmortem findings, two other points deserve consideration : one is the apparent lack of neurological manifestations in the period in-
278
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and
Tedeschi Amer.
tervening between birth and death, and the other is the long-term survival of the infant (Group 3 of Towbin classification). Since serially cut, whole brain, whole spine sections were not carried out in the present study, a quantitative and topographic assessment of central nervous system lesions is of no avail. Probably in the central nervous system more than in other organ systems, there is a large gap between the amount of injury required to abolish or to prevent function and the amount required to cause damage of a type that one might expect to recognize either grossly or under the microscope. vertical compression, and Hyperflexion, hyperextension are the most common mechanisms by which the spine can be injured. This applies to perinatal trauma as well as to the accidental trauma which may occur later in life. Nonetheless, the circumstances operating in perinatal trauma are almost of an entirely different nature than those related to mechanical trauma taking place after birth. The substrate differs in plasthickness, and tensile ticity, elasticity, strength. The large head and weak neck of the infant make him particularly vulnerable to the mechanisms of injury. Perinatal trauma apparently can be so mild as to escape recognition until functional deficit becomes apparent or evidence of damage is found unexpectedly at postmortem examination. In every one of the cases here reported, the record was carefully reviewed and the interrogated to determine obstetricians whether or not these deliveries were more
January 15, 1970 J. Obstet. Gynec.
traumatic than usual. Apparently they were not. Similarly, in the cases delivered by cesarean section, the incisions in the abdominal wall and uterus were adequate and there was no “tight squeeze.” The physiologic hypoxia associated with the neonatal period makes the nervous tissue and the walls of the blood channels especially vulnerable to the forces of external trauma. Fetal hypoxia causes venous congestion and increased venous and cerebral spinal fluid pressure. As the brain stem becomes injured, depression of the respiratory centers and pulmonary complications furthe manifestations of ther accentuate anoxia, giving rise to a vicious cycle and to an overlapping of traumatic and anoxic changes. Should anoxia alone have been responsible for the rachidian hemorrhage, one would expect to find similar changes of comparable severity in the viscera, but this was not the case in our observations, nor in similar observations by others. Assuming that trauma was the primary cause of the rachidian injury, the time sequence cannot be established and it remains undetermined whether the trauma occurred in the uterus, at the time of delivery, or even in the postnatal period. In an age in which so much effort is being directed to the prevention of disease, observation such as the ones here presented call for the application of all possible precautionary measures in the handling of the infant in the perinatal period, espccially of the premature infant who is particularly susceptible to traumatic spinal injury.
REFERENCES
E.: Dublin J. Med. Sci. 10: 419, 1. Kennedy, 1836. C. W.: Amer. J. Dis. Child. 19: 473, 2. Burr, 1920. 3. Crothers, B.: Amer. J. Med. Sci. 165: 94, 1923. 4. Ford, F. R.: Arch. Neurol. Psychiat. 14: 742, 1925. R. N.: Surg. Gynec. Obstet. 37: 5. Pierson, 802, 1923. 6. Crothers, B., and Putnam, M. Cl.: In Ford, F. R., Crothers, B., and Putnam, M. C., ed-
itors: tem,
7. 8. 9. 10. 11.
Birth Iniuries of Central Nervous SysPart II,- Medical Monographs, Baltimore, 1927, The Williams & Wilkins Company, vol. II. Towbin, A.: Arch. Path. 77: 620, 1964. Stoltzenberg. F.: Klin. Wschr. 48: 1741. 1911. Melchior, 7. Cl., and Tygstrup, I.:’ Acta Paediat. 52: 171, 1963. Knowlton, R. W.: J. Obstet. Gynaec. Brit. Comm. 45: 834, 1938. Hellstrijm, B. O., and Sallmander, U.: J. A. M. A. 204: 1041, 1968.