Atraumatic fetal cervical spinal cord injury and cruciate paralysis David J. Berck, MD, MPH, George M. Mussalli, MD, and Frank A. Manning, MD Background: Congenital cervical spinal cord injury usually is attributed to intrapartum mechanical trauma. Case: A fetal cervical spinal cord hemorrhage presented as congenital isolated upper extremity (“cruciate”) paralysis and muscle atrophy after an atraumatic delivery. Timely neonatal diagnosis and surgery resulted in complete recovery. Conclusion: Paralysis may result from atraumatic spinal cord injuries occurring in the antepartum period. (Obstet Gynecol 1998;91:833– 4. © 1998 by The American College of Obstetricians and Gynecologists.)
Fetal cervical spinal cord injury is a rare complication of pregnancy. In almost all reported cases, the damaging insult appears to have occurred in the intrapartum period, usually in association with either breech presentation, extreme deflexion attitude of the cephalic presentation, or delivery by mid forceps.1 Cervical spinal cord injury in the fetus usually presents with profound flaccid quadriplegia and paralytic apnea. Cruciate paralysis, manifest as isolated paralysis of the upper limbs with normal lower limb function, is the result of a spinal cord lesion at the cervical medullary junction at the point of the decussation of the corticospinal tracts. There are at least six case reports in the neurology literature in which an antenatal origin of spinal paralysis was suspected but unproven.2– 4 This case report describes the obstetric and neonatal course in an infant who exhibited overt cruciate paralysis at birth. Case A 19-year-old woman, gravida 1, para 0, presented at 39 weeks’ gestation with a blood pressure of 140/90. She had no headache, visual changes, abdominal pain, or other symptoms of preeclampsia. Her prenatal course was unremarkable, with no environmental or drug exposures. At 38 weeks, she complained of decreased, though present, fetal activity. A nonstress test performed at this time was reactive. Her past medical and surgical history was unremarkable. Apart from
From the Division of Maternal Fetal Medicine, Department of Obstetrics & Gynecology, Columbia-Presbyterian Medical Center, New York, New York.
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hypertension, the physical examination was normal. The cervix was long and closed. The fetal heart rate tracing was reactive at 140 beats per minute, with normal long- and short-term variability. Her laboratory evaluation revealed 21 protein on urine dip, with normal hematocrit, platelets, coagulation profile, and liver function tests. Labor was induced with oxytocin infusion after cervical ripening with prostaglandin gel. The fetal heart rate tracing was reactive, with normal short- and long-term variability throughout labor. After cervical dilation arrested at 8 cm, a cesarean delivery was performed through a Pfannenstiel skin incision and a generous low transverse hysterotomy. The fetal vertex was guided easily through the hysterotomy and delivered atraumatically with fundal pressure. The time interval from hysterotomy to cord clamping was one minute. The remainder of the surgery and maternal postoperative course were uncomplicated. The neonate was received from the operating table by a neonatologist. Apgar scores were 8 and 8 at one and five minutes, respectively, with 2 points lost for movement. At the time of birth, the arms were noted to be completely immobile. Upper extremity reflexes were absent, although sensation was intact. Marked atrophy of the upper extremities was noted. Palmar creases were within normal limits, and skin dimpling was absent. The remainder of the neonatal examination, including the head, neck, diaphragm, trunk, and lower extremities was normal. Extensive laboratory evaluation revealed no evidence of maternal or neonatal coagulopathy. A magnetic resonance image of the head and neck, obtained at 12 hours of life, revealed an extensive intramedullary cord lesion extending from the medulla through T2, with varying blood products consistent with hyperacute and subacute ages, indicating that the lesion was at least 3 to 4 days old (Figure 1). There was no evidence of delivery trauma. A computed tomography (CT) was confirmatory. On the second day of life, a C3-5 laminectomy was performed to evacuate the hematoma. A biopsy of the hematoma capsule revealed a low-grade astrocytoma, although reactive gliosis could not be completely ruled out pathologically. Postoperatively, the neonate gradually regained complete range of motion of both upper extremities. At 6 weeks of age, 5/5 right upper extremity strength and 4/5 upper extremity strength were present, with normal range of motion and reflexes bilaterally.
Comment In the infant described here, it is evident that cruciate paralysis was the result of a bleed into the substance of the spinal cord at and around the cervicomedullary junction. This was confirmed by CT scan shortly after birth and on day 2 of life by direct operative visualization. The hematoma identified at laminectomy was located centrally and was adjacent to the corticospinal tracts. The precise etiology of the intramedullary hemorrhage is unknown. The most likely etiology is a neoplasm, a vascular defect such as an arteriovenous malformation, or an aneurysm of the anterior spinal
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upper limb paralysis with severe atrophy and abnormal dermatoglyphics implies that the insult must have occurred early in gestation.2– 4 The failure to identify a specific lesion in the spinal cord after birth most likely was the result of spontaneous resorption of the hematoma in utero. The current case is unusual in that the pathophysiology of the lesion was confirmed and prompt intervention resulted in clinical improvement.
References 1. Menticoglou SM, Perlman M, Manning FA. High cervical spinal cord injury in neonates delivered with forceps: Report of 15 cases. Obstet Gynecol 1995;86:589 –94. 2. Darwish H, Sarnat H, Archer C, Brownell K, Kotagal S. Congenital cervical spinal atrophy. Muscle Nerve 1981;4:106 –10. 3. Hageman G, Ramaekers VT, Hilhorst BGJ, Rozeboom AR. Congenital cervical spinal muscular atrophy: A non-familial, nonprogressive condition of the upper limbs. J Neurol Neurosurg Psychiatry 1993;56:365– 8. 4. Philpot J, Muntoni F, Skellett S, Dubowitz V. Congenital symmetrical weakness of the upper limbs resembling brachial plexus palsy: A possible sequel of drug toxicity in first trimester of pregnancy? Neuromuscul Disord 1995;5:67–9.
Address reprint requests to: Figure 1. Contrast-enhanced axial magnetic resonance image of the cervical spine taken at roughly 12 hours of life. The arrow points to an intramedullary hemorrhage, which extends from the medulla to T2.
artery. The evidence at birth of atrophy of the muscles indicates that the insult predated the intrapartum period and was not related to birth trauma. The precise timing of the insult is unknown, but the maternal perception of a change in the nature and frequency of fetal movement one week before delivery may offer some insight. In previously reported cases, the presence at birth of
Pyoderma gangrenosum following cesarean delivery Ursula A. Steadman, MD, Tracy E. Brennan, MD, Lauren A. Daman, MD, and Stephen L. Curry, MD Background: Pyoderma gangrenosum is a rare, chronic inflammatory disease that can develop after minor trauma or surgery. Most reports of this condition developing after abdominal surgery have been reported following ostomy From the Department of Obstetrics and Gynecology, Hartford Hospital, Hartford, Connecticut.
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David J. Berck, MD Department of Obstetrics and Gynecology Division of Maternal-Fetal Medicine Columbia-Presbyterian Medical Center 633 West 168th Street, PH-16 New York, NY 10032 E-mail: [email protected]
Received April 28, 1997. Received in revised form August 5, 1997. Accepted August 5, 1997. Copyright © 1998 by The American College of Obstetricians and Gynecologists. Published by Elsevier Science Inc.
formation. A number of systemic diseases have been associated with this disorder, most commonly inflammatory bowel disease, though 40 –50% of cases have no associated systemic disease. Case: A 32-year-old woman developed spiking fever and purulent appearing wound discharge with a rolled erythematous border after cesarean delivery. Her condition worsened despite various regimens of intravenous antibiotics and local treatment. Pyoderma gangrenosum was diagnosed. Treatment with steroids resolved the lesion, and associated systemic diseases were not found. Conclusion: Consideration of the diagnosis of pyoderma gangrenosum in cases of apparent wound infections unresponsive to antibiotics should result in early diagnosis and treatment of this condition. (Obstet Gynecol 1998;91:834 – 6.
Obstetrics & Gynecology