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CUTANEOUS NEONATAL HERPES SIMPLEX INFECTION ASSOCIATED WITH RITUAL CIRCUMCISION

2000; Lippincott Williams & Wilkins; Volume: 19; Issue: 3 Linguagem: Inglês

10.1097/00006454-200003000-00025

1532-0987

Lorry G. Rubin, Philip Lanzkowsky,

Genital Health and Disease

Neonatal herpes simplex infection is a potentially life-threatening infection occurring in infants younger than 4 to 6 weeks of age. Disease classically takes one of three forms: localized skin, eye, or mucous membrane infection; disseminated infection; or central nervous system infection. The virus is most commonly acquired during delivery through contact with infected secretions from the maternal genital tract. Postnatally acquired neonatal infection occurs rarely. Sources of postnatal transmission consist of maternal lesions including breast1 or oral lesions, other adults with physical contact with the baby including hospital personnel caring for the neonate2 and possibly infant to infant in the nursery.3 We report two neonates with cutaneous herpes simplex infection involving the genital area with an onset several days after ritual circumcision. Both procedures were performed by the same individual; part of the ritual involved oral contact with the penis of the neonate after removal of the foreskin. Case reports. Infant 1. In 1988 Infant 1 was born by normal vaginal delivery after a 37-week gestation with a birth weight of 3.3 kg. Prenatal history was unremarkable. There was no maternal or paternal history of oral or genital herpetic infection. The infant was breast-fed and had an unremarkable perinatal course. At 8 days of age his ritual circumcision was performed by a mohel, an individual trained to perform circumcision as part of a religious ceremony. Four days later he developed erythema, swelling and a purulent yellow discharge from the glans of the penis and fever to 103°C. At 13 days of age he was admitted to Schneider Children's Hospital where physical examination showed a nontoxic appearing infant with a weight of 2.8 kg. Abnormal findings were limited to the penis which appeared erythematous with a yellow discharge. Total leukocyte count was 32 500/mm3 with a differential count of 64% polymorphonuclear leukocytes, 10% band forms, 10% lymphocytes, 10% monocytes and 6% atypical lymphocytes. Chest radiograph was normal. Cerebrospinal fluid (CSF) examination showed 3 leukocytes and 60 erythrocytes/mm3. Blood, CSF and a swab of the penile discharge were cultured and the patient was treated with ampicillin, nafcillin and gentamicin. Blood culture was negative, CSF grew Staphylococcus haemolyticus and the penis culture grew Staphylococcus epidermidis and Escherichia coli. Fever resolved within 48 h. Pustules were noted on the buttocks and papulovesicular lesions adjacent to the scrotum on the third hospital day. On the fifth hospital day a Tzanck preparation obtained from a vesicular lesion near the scrotum showed multinucleated giant cells; intravenous acyclovir was started at 30 mg/kg/day for treatment of presumptive herpes simplex infection. Herpes simplex virus (HSV) type 1 was isolated from skin lesions in the anterior perineum and buttocks and from a rectal swab. Papulovesicular lesions also appeared on the scrotum and penis. Eye examination was normal. Acyclovir was continued for 14 days with resolution of lesions. Maternal serology for HSV type 1, obtained near the end of the infant's hospitalization, was negative. At 10 years of age the child is normal and has not had recurrent herpetic lesions. Infant 2. In 1998 Infant 2 was born by normal vaginal delivery to a 22-year-old gravida 1 para 1 woman after an unremarkable prenatal course. There was no maternal or paternal history of oral or genital herpetic lesions. The infant was breast-fed and discharged at 2 days of age. At 8 days of age his ritual circumcision was performed by the same mohel who circumcised Infant 1. At 11 days of age swelling of the penis was noted; 2 days later low grade fever developed, and a bullous lesion was noted on the penis. He was given two daily intramuscular doses of ceftriaxone. Blisters developed on his penis, perineal area and right ankle. On hospitalization physical examination showed a well-appearing infant with a normal examination except for bullous lesions with erythematous borders on the base of the penis, buttocks and both extremities. Eye examination was normal. Blood count was normal for age. Alanine aminotransferase (ALT) was normal; aspartate aminotransferase (AST) was minimally elevated (66 IU/l, normal <43 IU/l). Cerebrospinal fluid cell examination showed 7 leukocytes and 83 erythrocytes/mm3; glucose and protein were normal for age. Computerized tomography of the head without contrast enhancement was normal. Bacterial cultures of a swab of the penis, blood, urine and CSF were sterile. A Tzanck preparation of a bullous lesion, obtained on the fifth hospital day, showed “cellular changes consistent with herpes viral effect”; viral culture grew HSV type 1. Serology for HSV-1 and HSV-2 by enzyme immunoassay was negative. He was treated with a 14-day course of intravenous acyclovir and his lesions resolved. Three weeks later he developed new vesicular lesions on the medial aspect of his right ankle at the site of previous lesions and was referred to Schneider Children's Hospital. He was otherwise well. CSF examination was normal and viral culture and PCR for herpes simplex was negative. Complete blood count was normal, but ALT and AST were 1.5 and 2.5 times the upper limit of normal, respectively. He was treated with a 21-day course of oral acyclovir (60 mg/kg/day) with prompt resolution of the cutaneous lesions. He had persistent mild elevations of ALT and AST that were normal when tested at 8 months of age. At 8 months of age he had recurrent cutaneous lesions in the genital region but was otherwise well. Maternal serology obtained when the infant was 10 weeks old was positive for HSV-1 and negative for HSV-2 by enzyme immunoassay. The mohel who performed both circumcisions had no history of oral herpetic infection. He reported having performed more than 1000 circumcisions. As a routine part of the ritual, after excising the foreskin the mohel had oral contact with the bleeding penis. Discussion. Although in 90% of cases of neonatal herpes simplex infection the source is the maternal genital tract and the majority of woman transmitting herpes simplex to their neonates during delivery have asymptomatic infection, these infants probably acquired HSV infection postnatally. There was no current or past history of genital lesions in the infants' mother, and it is unlikely that either woman had multiple previous sexual partners, a risk factor for genital herpes. The negative HSV serology in the mother of Infant 1 strongly suggests that his infection was postnatally acquired from someone other than his mother. Both infants were infected with type 1 strains and only 25% of neonatal HSV cases are due to type 1 strains.4 Furthermore, only ˜25% of first episodes of genital herpes and 2% of episodes of reactivated genital herpes are due to type 1 strains,5 making it less likely maternal genital infection was the source of the infants' viruses. There are several lines of circumstantial evidence suggesting that the mohel who performed the circumcision was the source of the virus. The penis is an uncommon site of cutaneous neonatal HSV infection despite the fact that circumcision is performed on the majority of infant boys born in the United States. In both babies no caretaker had a current or past history of lesions compatible with HSV infection. In both babies the initial lesions were in the genital area and symptoms and signs occurred 2 and 4 to 8 days after circumcision, timing within the estimated incubation period of 2 to 12 days.6 The same individual performed the circumcision on both infants. Significantly, as part of the circumcision ritual performed on each infant, after excising the foreskin the mohel had oral contact with the penis and infants' blood. Although the mohel had no history of oral herpetic lesions, the most plausible origin of neonatal infection was virus shed subclinically in oral secretions of the mohel spread to the penis during orogenital contact. Excretion of HSV in the saliva of asymptomatic adults is common. Hatherley et al.7 performed 4 weekly cultures of saliva for HSV on asymptomatic personnel in an obstetric hospital. HSV was recovered from 9.6% of 384 asymptomatic personnel; virus was recovered from 2 or more of the cultures from 2.6% of personnel. Douglas and Couch8 performed cultures of saliva for herpes simplex from 10 HSV-seropositive volunteers 3 times a week for 5 months. Six of the 10 individuals had at least 1 positive culture in the absence of herpetic lesions. Infrequent shedding could explain why additional infants with neonatal HSV infection after circumcision by this individual have not been identified. Identity by molecular typing of viral isolates from the mohel and the infants would be necessary to prove the presumed source of transmission. The mohel declined to be tested serologically or by culture for HSV but has agreed to refrain from having oral-genital contact when performing ritual circumcisions in the future. In Jewish tradition and by Jewish law, babies are circumcised 8 days after birth during a ritual known as bris. Circumcisions are performed by a mohel, an individual who has received specific training in the procedure. As part of the ritual as performed in Orthodox Judaism, the Mohel sucks the circumcised penis to “draw blood,” an act known as Mezizah. Many Orthodox ritual circumcisers (mohelim) have modified this procedure to avoid direct oral contact with the infant's blood or penis using a pipette-like device. Oral-genital or oral-blood contact is omitted from the bris performed by nonorthodox mohelim as well as mohelim certified by the Bris Milah Board of New York. Certification by the Bris Milah Board is required to perform circumcisions in New York hospitals. Transmission of infectious agents from mohelim to neonates has been previously documented. In 1946 Lewis reviewed 89 cases of primary tuberculosis of the penis. Seventy-two of the cases occurred in neonates after ritual circumcisions that included Mezizah.9 Interestingly he also commented that Mezizah had been practically eliminated from the bris ritual by the beginning of the 20th century. Mezizah with direct oral-genital contact is still practiced in certain Orthodox Jewish communities. Lewis also reported that syphilis and diphtheria have also been transmitted through this act. Other unusual medical complications resulting from Jewish religious practices have been reported.10 Mezizah with direct oral-genital contact has potentially important implications for transmission of infectious agents. In addition to concerns about transmission of infection to neonates, blood-borne pathogens such as hepatitis B, hepatitis C and HIV may be transmitted from an asymptomatic but infected neonate to the mohel during Mezizah. To prevent transmission of blood-borne infectious agents from neonates to mohelim and prevent herpes simplex and other pathogens from being transmitted from mohelim to neonates, public health officials and leaders of the Jewish community should act to modify the part of the circumcision ritual that involves direct oral contact with the blood and penis of neonates.