Intra-Uterine Gangrene Of The Forefoot: A Preliminary Report
Edward T. Haslam, M.D.
This is a preliminary report concerning a patient now under treatment at our clinic. This particular case presents some unusual features which we found of great interest.
The patient (A.B.) was born on December 20, 1962. Her mother, who was 27 years of age at the time, had had six previous pregnancies, with five children born alive and still living. She reported no previous maternal disease and no complications of this pregnancy. The duration of gestation was unknown. The duration of labor was four hours. She had a spontaneous rupture of her membranes, with delivery following ten minutes later. The child was a vertex presentation with an L.O.A. (Left Occiput Anterior) position and was delivered normally. The mother did not receive analgesics or anaesthesia and there were no maternal delivery complications.
The infant breathed spontaneously and appeared to be in good condition, except for a deformity of the left foot. The birth weight was four pounds, 12 1/2 ounces, or 2,130 grams. It was noted that the child had an early gangrene of the toes of the left foot. In addition, a severe inversion on the left foot was observed and evidence of cyanosis was noted in the premature nursery.
Dr. Ralph Platou, Professor of Pediatrics at Tulane University, examined A.B. and expressed the opinion that she had ectodermal dysplasia with gangrene. The Tulane University Surgical Department believed that the child's circulation was adequate to keep the gangrene from progressing in the absence of infection and that early treatment should
be conservative. She received penicillin and "streptomycin for one day and this treatment was then discontinued. Two days later, members of the Orthopaedic Service examined the child and noted that the gangrene was demarcating at midfoot level (Fig. 1 ).
A.B. was treated on a daily basis with cool pHisoHex soaks. On January 2, 1963, purulent drainage developed beneath the scar and treatment with Chloromycetin and penicillin was initiated and continued for a period of ten days. Cultures from the drainage revealed a staphylococcus aureus, coagulates positive, which was resistive to penicillin and sensitive in varying degrees to ethromy-cin, tetracyline, Chloromycetin, novobiocin and staphcyllin. Blood cultures were negative. It was my feeling that arterial pulsations were present in the feet, as well as in the popliteal spaces. However, they could not be palpated by all observers.
The gangrenous areas were debrided periodically without anaesthesia in the nursery and, eventually, a granulating would was obtained which showed no indication of deeper drainage or protruding bone.
A.B. was discharged from the premature
nursery on January 23, 1963, at the age of 34 days. Her weight was 3,010 grams. Plaster correction of the varus was begun on February 1, 1963 because of an increasing varus deformity and the apparent development of a constricting band at the medial aspect of the distal third of the tibia. It is interesting to note that there was no equinus tendency in the left foot and, in fact, it appeared to be slightly calcaneous. On February 15, 1963, it was noted that the stump had epithelialized, her weight gain was thought to be satisfactory, and she was discharged from the hospital (Fig. 4 ) .
A month later, the stump had healed satisfactorily (Fig. 5 ).* The child then was fitted with a 3-point pressure brace (Fig. 6 ) in an effort to correct her varus and to avoid the necessity of frequent changes of plaster. The brace was enlarged periodically and A.B. experienced a normal development. Subsequently, she underwent two periods of hospitalization, one caused by pneumonia and the other due to an occipital skull fracture caused by a fall from her crib.
The child was last seen in our clinic on December 4, 1963. At that time she was unable to walk, but was attempting to stand in a short leg brace attached to a shoe with a toe filler. The brace was equipped with a lateral compression strap. A medial heel and a malleolus pad were inserted within the shoe to prevent the development of additional varus (Fig. 7 ). She continues to wear the 3-point pressure brace at night.
Our latest X-rays reveal additional bony development within the foot and persistent abnormalities in the distal tibia and fibula. The present status of the distal tibial epiphysis is considered to be uncertain (Fig. 8 and Fig. 9 ).
I will not attempt to review the literature concerning a case of this type. It is well known that the majority of so-called congenital amputations are really cases of defective skeletal limb development but that cases of spontaneous intra-uterine amputation do sometimes occur. This case appears to fall in this category, and it is interesting to speculate what the foot would have looked like had the child been born at full term.
Our observations in this case unfortunately have not answered questions of etiology or pathogenesis. The initial appearance of the leg suggested defective development extending from the proximal tibia dis-tally, but the presence of arterial pulsations in the foot would eliminate arterial occlusion as a cause. However, the possibility of a temporary vasospasm or emboli affecting the arteries in the forefoot and causing a temporary spasm of the remaining vessels cannot be excluded. The constricting bands were not evident when the child was first seen. They appeared to develop later but have not been progressive or deep.
The stump is now theoretically capable of permitting effective ambulation with a single inside brace upright and a T-strap with the shoe equipped with a toe filler. It is not certain whether the distal tibial epiphysis will grow sufficiently to allow the child to function permanently as a partial foot amputee but there is nothing to be gained by amputating at a higher level at this time.
In summary, the case of a premature infant, weighing four pounds and 12 1/2 ounces, who was born with gangrene of the toes of her left foot, has been presented. The usual conservative treatment of dry gangrene has resulted in a stump which has functioned satisfactorily thus far. X-rays reveal some indication of possible damage to the distal tibial epiphysis and the eventual outlook is uncertain. Comments and observations from clinic chiefs and other orthopedists who have treated cases of this type would be welcomed.
Edward Haslam is Co-Clinic Chief, Juvenile Amputee Clinic, Crippled Children's Hospital, New Orleans, Louisiana
Glessner, James R., Jr., "Spontaneous Intrauterine Amputation", J. of Bone and Joint Surgery, Vol. 45-A, No. 2, p. 351, March 1963.