Neonatal Herpes Infection Associated With Direct Orogenital Suction During Ritual Jewish Circumcision

To the Editor—In a letter to the Editor of the Journal of the Pediatric Infectious Diseases Society that was first published online on January 28, 2015, Dr. Berman et al [ 2 ] responded to a recent systematic review of neonatal herpes simplex virus type 1 (HSV-1) infection associated with direct orogenital suction (DOS) during ritual Jewish circumcision [ 2 ]. This practice is also known as metzizah b'peh . The letter from Dr. Berman et al contains several inaccuracies and denies the significant risk of neonatal herpes HSV infection associated with DOS, which involves direct contact between the mouth of an adult and the infant penis immediately after removal of the foreskin.

Herpes simplex virus shedding in oral secretions occurs frequently, intermittently, and often without symptoms among those who are infected [ 3 ]. The practice of DOS facilitates transmission of HSV from a religious circumciser who is shedding the virus in oral secretions to the mucosa of a susceptible infant. The presence of HSV-1 lesions on the genitalia and associated dermatomes in babies who present with neonatal HSV after DOS is consistent with direct viral inoculation at the circumcision site. The timing of infection is also consistent with transmission via DOS. The neonatal HSV incubation periods cited by Dr. Berman et al [ 1 ] refer to infants who acquire HSV perinatally and are not relevant to transmission of HSV by direct oral-genital inoculation. Indeed, the median number of days between circumcision and the appearance of HSV lesions (median 8, range 5–20) cited in the New York City Department of Health and Mental Hygiene's (NYC DOHMH) summary of their years-long investigation into 11 such cases is highly consistent with inoculation at the time of circumcision [ 4 ]. Furthermore, in the 3 years after their 2012 publication, the NYC DOHMH documented an additional 6 cases of neonatal HSV infection after DOS, also with incubation periods highly consistent with inoculation at the time of circumcision (median days from circumcision 8.5, range 5–12) [ 5 ].

Epidemiological data clearly support a causal relationship between DOS and neonatal HSV. A rigorous investigation by the NYC DOHMH of cases included in the 2012 Morbidity and Mortality Weekly Report (MMWR) identified no evidence of nosocomial HSV transmission, and the possibility of perinatal transmission from mother to baby was largely excluded. Despite the rarity of neonatal herpes infections (∼13 in 100 000 live births in NYC) [ 6 ], multiple case series describing neonatal herpes after DOS have documented clusters of cases among babies exposed to DOS by specific religious circumcisers. The cases described in the 2012 NYC DOHMH MMWR occurred among infants who lived within a very few zip codes and included a pair of twins circumcised by the same individual, as well as 2 brothers born 3 years apart, who were each circumcised 8 days after birth by the same religious circumciser [ 4 ]. Another report of 8 cases of neonatal HSV-1 infection after DOS among infants in Canada and Israel included 2 infants who were circumcised by 1 individual 5 years apart and 2 infants who were circumcised by another individual 5 weeks apart [ 7 ]. The mean number of days from circumcision to diagnosis of HSV-1 in this report was 7.25 ± standard deviation 2.5 days, which is consistent with the incubation period reported in the NYC DOHMH MMWR. In addition, a 2000 report in the Pediatric Infectious Disease Journal also identified 2 infants who were circumcised by the same individual 10 years apart (1 in 1988, 1 in 1998) and who were diagnosed with HSV-1 infection within 2 weeks of DOS [ 8 ].

Dr. Berman et al [ 1 ] make the unsubstantiated claim that there is an “absence of any attention” to cases of neonatal herpes not associated with DOS and that scrutiny of this practice will distract physicians from identifying neonatal herpes infections in babies who are not ultraorthodox Jewish males. This claim is contradicted by the preponderance of peer-reviewed literature and practice guidelines from professional medical organizations addressing neonatal HSV infection prevention and management. Moreover, some investigators have evaluated the utility of empiric acyclovir in all infants who present with an illness requiring systemic antibiotics in the first 21 days of life, because neonatal HSV may present with nonspecific symptoms and may be easily missed [ 9 ].

Seventeen laboratory-confirmed cases of HSV infection attributable to DOS have been reported to the NYC DOMH since 2000. Two of the 17 infants died, and at least 2 infants suffered brain damage [ 5 ]. In the case series by Gesundheit et al [ 7 ], 4 infants developed recurrent genital HSV lesions and 1 infant was diagnosed with central nervous system disease with neurological sequelae. Neonatal HSV, even when treated with early initiation of antiviral medications, may be fatal and result in seizures and devastating neurological complications. We therefore disagree with the suggestion of Leas and Umscheid [ 2 ] that prospective observational trials should be conducted to address the risk of DOS. This practice poses an avoidable and unnecessary risk of infection to the infant and is contradictory to the American Academy of Pediatrics' recommendation that circumcision should not include DOS [ 10 ]. Thus any such trial, even if observational, would be unethical.

We also strongly disagree with the assertion of Leas and Umscheid [ 2 ] and of Berman et al [ 1 ] that only DNA evidence can prove a causal relationship between DOS and neonatal HSV infection. DNA evidence is not necessary to document the risk of DOS. Direct orogenital suction poses an incontrovertible risk of transmission of HSV and other infections, and the practice violates the basic standards of infection control. The epidemiological data linking the practice of DOS to neonatal HSV infection are overwhelming. For all of these reasons, DNA evidence is not necessary to prove that DOS poses an unacceptable risk to infants. In many ultraorthodox Jewish communities, the practice of DOS has been modified through use of a glass pipette to reduce the risk of infection transmission [ 7 ], although there is no evidence that this practice reduces risk. As pediatricians, we advocate that DOS should never be performed.