Note:
Here a black and white photograph of the perineum of an infant is displayed. A catheter is emerging from the urethra. The leads of an ECG machine are also visible. The scrotum and penis are swollen and discolored.
Pediatrics, Volume 64, Issue 3: Pages 301-303, 1979.
The lack of valid medical indications has caused routine neonatal circumcision to fall into disrepute in recent years.1-3 However, despite the substantial arguments against continuation of this therapy, it ranks as one of the most common surgical procedures.3,4
Significant infection is rarely solely attributed to circumcision because a clear-cut cause and effect relationship is difficult to establish. There have been reports of newborns5-7 who have been circumcised and then developed life threatening infection within several days of this procedure. However, the fact that in these infants infection has occurred at an age when the incidence of infection is high due to other factors complicates interpretation of these reports.
We report a 6-week-old infant in whom the relationship between circumcision and sepsis was unequivocal.
CASE REPORT
On admission T. H. was the 6-week-old product of a 32-week twin gestation born to a 26-year-old multiparous mother. Birth weight was 2,160 gm (4 lb, 12 oz). His early course was complicated by transient tachypnea requiring oxygen therapy for 2 1/2 days and hyperbilirubinemia requiring phototherapy for four days. He was discharged from the nursery at 12 days of age with appropriate weight gain. At 6 weeks of age he was circumcised in the office of the mother's physician. Within 24 hours he became irritable and anorectic. The scrotum developed a purpuric discoloration and the base of the penile shaft became red and firm. There was no fever, however, and the mother telephoned her physician who recommended aspirin therapy. When marked lethargy developed, the infant was taken to a local hospital where he was noted to be moribund. After intubation and stabilization he was transferred to Hermann Hospital, University of Texas Medical School at Houston. Initial physical exam revealed an ashen gray, hypotonic infant with a pulse rate of 178 beats a minute, temperature of 34.7 C (rectally), and blood pressure of 103/58 mm Hg. He required a respirator to maintain ventilation. He was obtunded with occasional spontaneous limb movements. The only other pertinent findings were the genitalia. The shaft of the penis and surrounding tissue were firm and erythematous and the scrotum had a bluish discoloration (figure). Ampicillin, methicillin, and gentamycin were begun. Initial laboratory data showed: hemoglobin, 8.6 gm/dl; hematocrit, 25.7%; white blood cell count 5,000/cu mm with 94% lymphocytes, 2% segmented neutrophils, 2% stab cells, 1% myelocytes, 1% eosinophils (the infant subsequently developed a brisk neutrophilic leukocytosis). The platelet count was 166,000/ cu mm. The urinalysis showed proteinuria, hematuria, and pyuria. An initial arterial blood gas determination while the infant was receiving 100% oxygen revealed a pH of 6.87, Po2 of 53 mm Hg. CSF contained 105 leukocytes/cu mm, all of which were mononuclear. CSF glucose was 2 mg/dl, CSF protein was 1,210 mg/dl. The Gram strain showed bacteria. The infant's subsequent hospital course was complicated by shock, generalized seizures, hypocalcemia, renal failure, disseminated intravascular coagulation, arrhythmias, and death. Blood, urine, and CSF cultures grew group B beta-hemolytic Streptococcus.
Note:
Here a black and white photograph of the perineum of an infant is displayed. A catheter is emerging from the urethra. The leads of an ECG machine are also visible. The scrotum and penis are swollen and discolored.
Figure Perineum of the infant shortly after circumcision
DISCUSSION
Because circumcision was delayed in this child until 6 weeks of age, the relationship between the operation and his subsequent infection were clear-cut, unlike the situation in some previous reports. Estimates of the frequency of infection vary widely although it is generally stated that serious infection is a rare event. The interpretation of what constitutes an infected circumcision is problematic because the wound often has some exudate and erythema without other definite signs of infection. Even if the wound looks uninfected, it is possible that it may serve as a portal of entry for bacteria. Tooth brushing, proctoscopy, and a variety of other "innocuous" procedures causing a break in the epithelial barriers have been documented to cause bacteremia. It would not be surprising if circumcision also caused bacteremia. This, coupled with the natural tendency of physicians not to ascribe a poor outcome to an elective procedure, may have led to gross underestimates of the frequency of life threatening infectious complications. In one retrospective series of more than 900,000 male births, there were no deaths attributed to infection.8 The incidence of neonatal sepsis is estimated at 1:300 to 1:1,600 live births. With the usual male predominance (approximately 2:1), one might have expected between 700 and 3,900 in this large study. Since the rate of circumcision during the years of study was 61%, there must have been many males who were circumcised and who became septic but in whom the two events were not judged to have been related.
Despite the marked male predominance, few reviews of neonatal sepsis mention how many infants were circumcised. Although one series suggests that circumcision is a major cause of sepsis in newborn male infants9 most make no conclusion regarding a relationship. The marked excess of sepsis in male infants has been assumed to reflect the biologic inferiority of a single dose X chromosome. It has been noted that two X chromosomes cause greater genetic heterogeneity for X-linked genes involved in immunoglobulin synthesis,10 thymic function, and glucose-6-phosphate dehydrogenase related phagocytic cell formation.11 Although differences in infection susceptibility do exist in both man and experimental animals,12 seldom is the female advantage more exaggerated than in the neonatal period. It would seem that a reasonable explanation for some of these infections in male infants is that they routinely have the integrity of their integument violated in a moist, warm, contaminated area. Much has been made of the umbilicus as a portal of entry for bacteria, partly because of its proximity to the perineum. However, few fears have been voiced that an iatrogenic perineal wound may pose a risk of potentially greater importance.
The University of Texas Medical School, Houston
Reprint requests to (T.G.C.): The University of Texas Medical School, 6431 Fannin, 228 Freeman Building, Houston, TX 77030.
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