Neonatal compartment syndrome Two cases of forearm
compartment
syndrome
in neonates
are presented.
The compartment
syndrome may be initiated before actual delivery and may appear in an advanced stage. In both children good clinical results were achieved when standard guidelines for managing compartment syndrome
and established
partment
syndrome
Volkmann’s
contracture
should be considered
unable to move an extremity.
S.C. Kline, MD, No$itk,
Vu., and J. Russell
w
Case reports Case 1. The patient was an otherwise healthy neonate delivered with moderate difficulty caused by brachypelvic disproportion. He was the firstborn child of a healthy mother who had an uncomplicated pregnancy. Hand surgery consultation was requested to evaluate a swollen extremity (Fig. I, A) 2 hours after birth. The child was not moving his left extremity. The forearm was tense and swollen, especially on the anterior side, and skin blistering was present. The tips of the thumb, long, ring. and small fingers were necrotic, while
Received for publication April 16. 1991; accepted in revised
form
June 8. 1991.
No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: Samuel C. Kline, MD, Hand Surgery Associates, P.C., 6161 Kempsville Circle, Suite 210. Norfolk, VA 23502.
256
THE JOURNAL
OF HAND
SURGERY
Although
diagnosis
uncommon,
com-
of the neonate who is
(J HAND SURC 1992;17A:256-9.)
hen a neonate is not moving an extremity, the clinician must consider a wide range of possible diagnoses. Often there will be none of the typical signs of infection, such as fever; nor will an examination reliably demonstrate tenderness or painful range of motion. ‘. ’ The main differential diagnosis is between trauma and infection. Perinatal trauma, including brachial plexus injuries, spinal cord injuries, clavicle and humerus fractures, and a wide range of soft tissue injuries, may result in a motionless limb? Infectious processes, including osteomyelitis and septic arthritis, may also be manifest as a motionless limb although many of the other signs of infection are absent.’ We present two cases of perinatal compartment syndrome and believe that this uncommon problem should be considered in the evaluation of a motionless limb of a neonate.
3/l/32613
were applied.
in the differential
Moore,
MD, Luth-ville,
Md.
the remainder of the hand was pulseless and swollen. Three hours after delivery a forearm fasciotomy and carpal tunnel release was performed in the nursery to restore circulation to the hand. For this procedure, local anesthesia was used. After fasciotomy the tense, discolored muscle of the forearm compartment bulged through the incision, and wrist pulses were restored. Postoperatively the child did well. The fingertips healed with minimal scarring. and the patient demonstrated return of sensibility and motor function. Fig. I. B shows the clinical results at 3 months of age. Scar revision may be necessary if contracture develops in the future. Case 2. An otherwise healthy neonate was born via an uncomplicated vaginal delivery. He was the firstborn child of a healthy mother whose pregnancy was complicated by gestational diabetes. Retrospectively, the mother reported having had lower abdominal pain for approximately 2 weeks before delivery. Several hours after delivery a surgery evaluation was obtained concerning full-thickness pressure necrosis of the skin of the left forearm. Full-thickness skin loss with peripheral areas of healing and tiexion contracture of the wrist and fingers were noted. Clinical photographs at 2 days of age are shown in Fig. 2, A and B. The child had no spontaneous finger or wrist motion. Circulation to the hand was judged to be adequate; therefore conservative management of the hand was undertaken. Silver sulfadiazine (Silvadene) cream was used to prevent bacteria1 superinfection, and the mother was instructed in passive stretching exercises. The skin healed uneventfully in 3 months’ time. Wrist motion returned at 2 months. followed by finger motion at approximately 5 months. By the age of 8 months. the child was using his left hand. Fig. 2, C and D shows the clinical result at 10 months. when the flexion contracture had resolved and the patient demonstrated good motion of his fingers and wrist. Appearance of skin wrinkles indicative of sensory reinnervation was first noted at 13 months of age. Nonoperative management yielded good cosmetic and functional results in this infant. Discussion
These two cases illustrate the spectrum of compartment syndrome in the neonatal period. One previous
Vol. 17A. No. 2 March 1992
Neotutal
cotnpurtment
sydrottw
257
Fig. 1. Case 1. A, Involved extremity at 2 hours of age. In addition to swelling and blistering 01 the forearm. pulses were absent and circulatory compromise of the digits was evident. B, Clinical results 3 months later, after emergency forearm fasciotomy. The patient demonstrated good return of motor function.
report’ documented a similar problem in a newborn who eventually required fasciotomy and nerve decompression at 7 days of age. Skin necrosis was noted at birth and was followed by progressive pain and flexion deformity, which prompted a delayed hand surgery consultation and decompression. The authors thought this was an unusual manifestation of amniotic band syndrome, although birth trauma and the wrapping of the umbilical cord around the arm were not completely
ruled out. Multiple-limb compartment syndrome in a neonate with sclerema neonatorum attributed to noncompliant skin, hypotension, and sepsis has also been reported.’ In addition, neonatal compartment syndrome after arterial catheterization injury has also been described .’ In our Case 1 the compartment syndrome could have resulted from a difficult delivery; however, digital tip necrosis suggested that the process was initiated before
258
Kline and Moore
The Journal of HAND SURGERY
Fig. 2. Case 2. A and B, Involved extremity at 2 days of age. Full-thickness skin loss with marginal reepithelialization as well as flexion contracture of the wrist and digits was present. C and D, Clinical result 10 months later, after conservative management with Silvadene and passive stretching exercises. The patient had good return of sensory and motor function by the age of 18 months.
the time of delivery. Difficulty with fetal descent and delivery may have resulted in compression trauma of the limb, which produced an acute compartment syndrome similar to trauma later in life.‘.” Vascular compromise of the hand was a strong indication for immediate fasciotomy; compartment pressures were not thought to be necessary. In Case 2 the syndrome appears to have occurred in utero before delivery, since the skin was necrotic at birth with evidence of healing and the flexion contractures of the wrist and fingers had already been established. Early surgical intervention for delayed presentation of Volkmann’s contracture is not recommended.“. I2 Nerve function gradually improved, so decompression was not necessary. The flexion contracture resolved with passive stretching, although plasticity, less scar formation, and rapid growth of a newborn should also be credited. The cause of intrauterine compartment syndrome in this child is not known. No stigmata of constriction band syndrome were apparent in any extremity; nor has the process occurring in this patient been noted in any other patient with amniotic band syndrome in our review of the literature.13-“j We believe that Volkmann’s ischemic contracture in this
child resulted from some form of intrauterine pressure, perhaps entrapment of the arm during the later stages of pregnancy and descent. Wrapping of the umbilical cord causing pressure necrosis is unlikely without evidence of fetal distress from umbilical cord compression. B,irth trauma or a primary vascular problem also cannot be eliminated. In the treatment of these newborn infants, we have applied accepted guidelines’* for the management of older children with Volkmann’s ischemic contracture. Emergency fasciotomy was performed in the first case when there was evidence of acute compartment syndrome with vascular compromise of the hand. In the second case established Volkmann’s contracture was present. The overlying skin was a necrotic eschar with evidence of healing around the margins, and a fixed flexion contracture of the wrist and fingers had already been established. The vascular supply to the hand was not compromised. Passive exercises were successful in stretching the contracture, and nerve function was restored, so further surgery to release the contracture or entrapped nerves was not necessary. Compartment syndrome can occur in the neonatal period. It may be the result of trauma during delivery
Vol. 17A. No. 2 March 1992
or the consequence of an intrauterine pressure injury. After birth, other unusual circumstances have also been documented to precipitate compartment syndrome in the newborn; these include arterial catheterization injury’ and sclerema neonatorum.5 Compartment syndrome should be considered in the differential diagnosis of a neonate who is not moving an extremity, and management should proceed according to guidelines estabIished for older patients. We thank Drs. E.F. Shaw Wilgis and Charles Hamlin for allowing us to include their patients in this report.
REFERENCES 1. Avery ME, First LR. Pediatric medicine. Baltimore: Williams & Wilkins, 1989: 193-6. 2. Feigin RD. Ahramson SL, Edwards MS. Postnatal bacterial infections. In: Fanaroft AA. Martin RJ. Neonatalperinatal medicine; diseases of the fetus and infant. St. Louis: CV Mosby, 1987:763-98. 3. Mangurten HH. Birth injuries. In: Fanaroft AA, Martin RJ. Neonatal-perinatal medicine; diseases of the fetus and infant. St. Louis: CV Mosby, 1987:317-59. 4. Tsur H, Yaffie B, Engel Y. Impending Volkmann’s contracture in a newborn. Ann Plast Surg 1980;5:317-20. 5. Christiansen SD, DeSain S, Polito AR, Slack MR. Ischemit extremities due to compartment syndrome in a septic neonate. J Pediatr Surg 1983;18:641-3. 6. Garrett RC, Kerstein MD. Compartment syndrome in the newborn. South Med J 1987:80:533-4.
Neonatui compurtment
syndrwne
259
7. Matsen FA, Winquist RA, Krugmire RB. Diagnosis and management of compartment syndromes. J Bone Joint Surg 1980;62A:286-91. syn8. Mubarak SJ, Hargens AR. Acute compartment dromes. Surg Clin North Am 1983;63:539-65. 9. Newmeyer WL, Kilgore ES. Volkmann’s ischemic contracture due to soft tissue injury alone. J HAND SURC~ 1976:1:221-7. IO. Roland SA. Fasciotomy: the treatment of compartment syndrome. In: Green DP. Operative hand surgery, 2nd ed. New York: Churchill Livingstone, 1988:591-607. Il. Tsuge K. Treatment of established Volkmann’s contracture. J Bone Joint Surg 1975;57A:925-9. 12. Tsuge K. Management of established Volkmann’s contracture. In: Green DP. Operative hand surgery. 2nd ed. New York: Churchill Livingstone. 1988:59 l-607. 13. Dobyns JH. Congenital ring syndrome (congenital constriction band syndrome). In: Green DP. Operative Hand Surgery. 2nd ed. New York: Churchill Livingstone. 1988503-9. 14. Miura T. Congenital constriction band syndrome. J HAND SlJRG 1984:9A:82-8. 15. Patterson TJS. Congenital ring constrictions. Br J Plast Surg 1961;14:1-31. 16. Temtamy SA, McKusick VA. Digital and other malformations associated with congenital ring constrictions. Birth Defects 1978;14:547-57.
compartment
syndrome
in neonates
are presented.
The compartment
syndrome may be initiated before actual delivery and may appear in an advanced stage. In both children good clinical results were achieved when standard guidelines for managing compartment syndrome
and established
partment
syndrome
Volkmann’s
contracture
should be considered
unable to move an extremity.
S.C. Kline, MD, No$itk,
Vu., and J. Russell
w
Case reports Case 1. The patient was an otherwise healthy neonate delivered with moderate difficulty caused by brachypelvic disproportion. He was the firstborn child of a healthy mother who had an uncomplicated pregnancy. Hand surgery consultation was requested to evaluate a swollen extremity (Fig. I, A) 2 hours after birth. The child was not moving his left extremity. The forearm was tense and swollen, especially on the anterior side, and skin blistering was present. The tips of the thumb, long, ring. and small fingers were necrotic, while
Received for publication April 16. 1991; accepted in revised
form
June 8. 1991.
No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: Samuel C. Kline, MD, Hand Surgery Associates, P.C., 6161 Kempsville Circle, Suite 210. Norfolk, VA 23502.
256
THE JOURNAL
OF HAND
SURGERY
Although
diagnosis
uncommon,
com-
of the neonate who is
(J HAND SURC 1992;17A:256-9.)
hen a neonate is not moving an extremity, the clinician must consider a wide range of possible diagnoses. Often there will be none of the typical signs of infection, such as fever; nor will an examination reliably demonstrate tenderness or painful range of motion. ‘. ’ The main differential diagnosis is between trauma and infection. Perinatal trauma, including brachial plexus injuries, spinal cord injuries, clavicle and humerus fractures, and a wide range of soft tissue injuries, may result in a motionless limb? Infectious processes, including osteomyelitis and septic arthritis, may also be manifest as a motionless limb although many of the other signs of infection are absent.’ We present two cases of perinatal compartment syndrome and believe that this uncommon problem should be considered in the evaluation of a motionless limb of a neonate.
3/l/32613
were applied.
in the differential
Moore,
MD, Luth-ville,
Md.
the remainder of the hand was pulseless and swollen. Three hours after delivery a forearm fasciotomy and carpal tunnel release was performed in the nursery to restore circulation to the hand. For this procedure, local anesthesia was used. After fasciotomy the tense, discolored muscle of the forearm compartment bulged through the incision, and wrist pulses were restored. Postoperatively the child did well. The fingertips healed with minimal scarring. and the patient demonstrated return of sensibility and motor function. Fig. I. B shows the clinical results at 3 months of age. Scar revision may be necessary if contracture develops in the future. Case 2. An otherwise healthy neonate was born via an uncomplicated vaginal delivery. He was the firstborn child of a healthy mother whose pregnancy was complicated by gestational diabetes. Retrospectively, the mother reported having had lower abdominal pain for approximately 2 weeks before delivery. Several hours after delivery a surgery evaluation was obtained concerning full-thickness pressure necrosis of the skin of the left forearm. Full-thickness skin loss with peripheral areas of healing and tiexion contracture of the wrist and fingers were noted. Clinical photographs at 2 days of age are shown in Fig. 2, A and B. The child had no spontaneous finger or wrist motion. Circulation to the hand was judged to be adequate; therefore conservative management of the hand was undertaken. Silver sulfadiazine (Silvadene) cream was used to prevent bacteria1 superinfection, and the mother was instructed in passive stretching exercises. The skin healed uneventfully in 3 months’ time. Wrist motion returned at 2 months. followed by finger motion at approximately 5 months. By the age of 8 months. the child was using his left hand. Fig. 2, C and D shows the clinical result at 10 months. when the flexion contracture had resolved and the patient demonstrated good motion of his fingers and wrist. Appearance of skin wrinkles indicative of sensory reinnervation was first noted at 13 months of age. Nonoperative management yielded good cosmetic and functional results in this infant. Discussion
These two cases illustrate the spectrum of compartment syndrome in the neonatal period. One previous
Vol. 17A. No. 2 March 1992
Neotutal
cotnpurtment
sydrottw
257
Fig. 1. Case 1. A, Involved extremity at 2 hours of age. In addition to swelling and blistering 01 the forearm. pulses were absent and circulatory compromise of the digits was evident. B, Clinical results 3 months later, after emergency forearm fasciotomy. The patient demonstrated good return of motor function.
report’ documented a similar problem in a newborn who eventually required fasciotomy and nerve decompression at 7 days of age. Skin necrosis was noted at birth and was followed by progressive pain and flexion deformity, which prompted a delayed hand surgery consultation and decompression. The authors thought this was an unusual manifestation of amniotic band syndrome, although birth trauma and the wrapping of the umbilical cord around the arm were not completely
ruled out. Multiple-limb compartment syndrome in a neonate with sclerema neonatorum attributed to noncompliant skin, hypotension, and sepsis has also been reported.’ In addition, neonatal compartment syndrome after arterial catheterization injury has also been described .’ In our Case 1 the compartment syndrome could have resulted from a difficult delivery; however, digital tip necrosis suggested that the process was initiated before
258
Kline and Moore
The Journal of HAND SURGERY
Fig. 2. Case 2. A and B, Involved extremity at 2 days of age. Full-thickness skin loss with marginal reepithelialization as well as flexion contracture of the wrist and digits was present. C and D, Clinical result 10 months later, after conservative management with Silvadene and passive stretching exercises. The patient had good return of sensory and motor function by the age of 18 months.
the time of delivery. Difficulty with fetal descent and delivery may have resulted in compression trauma of the limb, which produced an acute compartment syndrome similar to trauma later in life.‘.” Vascular compromise of the hand was a strong indication for immediate fasciotomy; compartment pressures were not thought to be necessary. In Case 2 the syndrome appears to have occurred in utero before delivery, since the skin was necrotic at birth with evidence of healing and the flexion contractures of the wrist and fingers had already been established. Early surgical intervention for delayed presentation of Volkmann’s contracture is not recommended.“. I2 Nerve function gradually improved, so decompression was not necessary. The flexion contracture resolved with passive stretching, although plasticity, less scar formation, and rapid growth of a newborn should also be credited. The cause of intrauterine compartment syndrome in this child is not known. No stigmata of constriction band syndrome were apparent in any extremity; nor has the process occurring in this patient been noted in any other patient with amniotic band syndrome in our review of the literature.13-“j We believe that Volkmann’s ischemic contracture in this
child resulted from some form of intrauterine pressure, perhaps entrapment of the arm during the later stages of pregnancy and descent. Wrapping of the umbilical cord causing pressure necrosis is unlikely without evidence of fetal distress from umbilical cord compression. B,irth trauma or a primary vascular problem also cannot be eliminated. In the treatment of these newborn infants, we have applied accepted guidelines’* for the management of older children with Volkmann’s ischemic contracture. Emergency fasciotomy was performed in the first case when there was evidence of acute compartment syndrome with vascular compromise of the hand. In the second case established Volkmann’s contracture was present. The overlying skin was a necrotic eschar with evidence of healing around the margins, and a fixed flexion contracture of the wrist and fingers had already been established. The vascular supply to the hand was not compromised. Passive exercises were successful in stretching the contracture, and nerve function was restored, so further surgery to release the contracture or entrapped nerves was not necessary. Compartment syndrome can occur in the neonatal period. It may be the result of trauma during delivery
Vol. 17A. No. 2 March 1992
or the consequence of an intrauterine pressure injury. After birth, other unusual circumstances have also been documented to precipitate compartment syndrome in the newborn; these include arterial catheterization injury’ and sclerema neonatorum.5 Compartment syndrome should be considered in the differential diagnosis of a neonate who is not moving an extremity, and management should proceed according to guidelines estabIished for older patients. We thank Drs. E.F. Shaw Wilgis and Charles Hamlin for allowing us to include their patients in this report.
REFERENCES 1. Avery ME, First LR. Pediatric medicine. Baltimore: Williams & Wilkins, 1989: 193-6. 2. Feigin RD. Ahramson SL, Edwards MS. Postnatal bacterial infections. In: Fanaroft AA. Martin RJ. Neonatalperinatal medicine; diseases of the fetus and infant. St. Louis: CV Mosby, 1987:763-98. 3. Mangurten HH. Birth injuries. In: Fanaroft AA, Martin RJ. Neonatal-perinatal medicine; diseases of the fetus and infant. St. Louis: CV Mosby, 1987:317-59. 4. Tsur H, Yaffie B, Engel Y. Impending Volkmann’s contracture in a newborn. Ann Plast Surg 1980;5:317-20. 5. Christiansen SD, DeSain S, Polito AR, Slack MR. Ischemit extremities due to compartment syndrome in a septic neonate. J Pediatr Surg 1983;18:641-3. 6. Garrett RC, Kerstein MD. Compartment syndrome in the newborn. South Med J 1987:80:533-4.
Neonatui compurtment
syndrwne
259
7. Matsen FA, Winquist RA, Krugmire RB. Diagnosis and management of compartment syndromes. J Bone Joint Surg 1980;62A:286-91. syn8. Mubarak SJ, Hargens AR. Acute compartment dromes. Surg Clin North Am 1983;63:539-65. 9. Newmeyer WL, Kilgore ES. Volkmann’s ischemic contracture due to soft tissue injury alone. J HAND SURC~ 1976:1:221-7. IO. Roland SA. Fasciotomy: the treatment of compartment syndrome. In: Green DP. Operative hand surgery, 2nd ed. New York: Churchill Livingstone, 1988:591-607. Il. Tsuge K. Treatment of established Volkmann’s contracture. J Bone Joint Surg 1975;57A:925-9. 12. Tsuge K. Management of established Volkmann’s contracture. In: Green DP. Operative hand surgery. 2nd ed. New York: Churchill Livingstone. 1988:59 l-607. 13. Dobyns JH. Congenital ring syndrome (congenital constriction band syndrome). In: Green DP. Operative Hand Surgery. 2nd ed. New York: Churchill Livingstone. 1988503-9. 14. Miura T. Congenital constriction band syndrome. J HAND SlJRG 1984:9A:82-8. 15. Patterson TJS. Congenital ring constrictions. Br J Plast Surg 1961;14:1-31. 16. Temtamy SA, McKusick VA. Digital and other malformations associated with congenital ring constrictions. Birth Defects 1978;14:547-57.
