Complications of circumcision in male neonates, infants and children: a systematic review

PubMed, African Healthline, LILACS and the Cochrane Central Register of Controlled Trials databases were searched with keywords and MeSH terms including infant/newborn/pediatric/child, circumcision, complications adverse events, Africa, Asia and Arabic. For example, we searched PubMed with the following search terms: "Circumcision, Male" [Mesh] AND "Infant, Newborn" [Mesh] AND ("Africa" [Mesh] OR "Asia" [Mesh]); "complications " [Subheading] OR "Intraoperative Complications" [Mesh] OR "Postoperative Complications" [Mesh]) AND "Circumcision, Male" [Mesh] AND ("Africa" [Mesh] OR "Asia" [Mesh]); ("Child" [Mesh] AND "Circumcision, Male" [Mesh]) AND ("Africa" [Mesh] OR "Asia" [Mesh]); ("Infant, Newborn" [Mesh] OR "Child" [Mesh]) AND ("Circumcision, Male" [Mesh] OR ("Circumcision, Male/adverse effects" [Mesh] OR "Circumcision, Male/complications" [Mesh] OR "Circumcision, Male/contraindications" [Mesh] OR "Circumcision, Male/mortality" [Mesh])); "Circumcision" [Mesh] "Circumcision, Male " [Mesh] AND "Arabic".

Searches were conducted on November 6^th 2007 and updated on February 14th 2009. There was no language restriction. We also searched reference lists of relevant papers, including a systematic review of complications of male circumcision in Anglophone Africa [17]. A total of 1349 published papers were identified through these searches. The abstracts of these papers were read and full copies of 223 papers with information on complications were obtained. Data were extracted by HW and NL into standardised forms in Access.

Infant and child circumcision is almost universal in the Arab world, and we conducted additional searches of the Arabic literature, including searches of relevant databases, book reviews in 10 key academic centres on Middle Eastern Studies and searches of the Hebrew University of Jerusalem libraries for Masters and PhD research thesis focused on male circumcision. Searches were conducted from June to August 2008. The Arabic literature searches identified 46 potentially relevant papers, of which six contained information on circumcision complications.

Hospital-based studies of circumcision-related complications are usually retrospective and record-based [9, 18, 19]. Complications in these studies are commonly recorded from discharge sheets, so tend to under-estimate the true frequency of complications because events occurring after discharge are not captured. Furthermore, not all post-operative complications will be seen again at the same hospital. We therefore present results separately for prospective and retrospective studies. Age at circumcision, and type of provider (medical or non-medical) were also thought a-priori to be associated with frequency of complications, and we present results stratified by these factors. We define neonatal as age up to 28 days, infant as 28 days-11 months, and child as 12 months-12 years. Many studies included boys circumcised at a range of ages. We included studies in which the mean or median age at circumcision was age 12 years or younger.

We identified 16 prospective studies of complications following neonatal and infant circumcision, from 12 countries [9‐11, 21‐33] (Table 1). Of these, most used the Plastibell [11, 22, 23, 25‐28], with others using the Gomco clamp [21, 24, 30, 32], freehand circumcision [9, 31], or a combination of methods [27, 29, 33].

The median frequency of any adverse event was 1.5% (range 0-16%), and median frequency of any serious adverse event was 0% (range 0-2%). Nine studies reported no serious adverse events, but three studies reported that 1-2% of boys had a serious complication [10, 27, 29]. One, a Canadian study of 100 neonates circumcised in 1961/1962 using the Gomco clamp or Plastibell reported one severe infection requiring antibiotics and one severe meatal ulcer [29]. Less severe complications were reported in a further 13 boys in this study. The other two studies with serious complications were from Nigeria. In one, among 141 boys circumcised in 3 hospitals in southeast Nigeria, complications were assessed at a 6 week post-operative visit or if they presented earlier with any complication [27]. Three boys (2.1%) had a urethral laceration. The most common complications in this study were minor including bleeding (9%) and meatal stenosis (3.5%). Complications were substantially more common when circumcision had been performed freehand (27% excluding incomplete circumcision) rather than using the Plastibell (8%), and when performed by midwives (19%) rather than doctors (7%). Moreover, among the doctors, the reported frequency of complications at the public (University Teaching) hospital was 1.6%, compared to 20% at private hospitals where the level of training and supervision is lower. A much higher frequency (90%) was seen at the mission hospital, which acts as a referral centre for complicated circumcisions. Three circumcisions had been performed by a traditional birth attendant, and all three had resultant complications (one bleeding, one incomplete circumcision, and one urethral fistula). The other study was among 322 infants attending a welfare clinic in Ibadan [10], in which there were 2 cases of amputation of the glans penis and one buried (trapped) penis. Overall in this study, complications were reported in 9.3% of boys, with a further 11% having excess residual foreskin. The most common complication was excessive loss of foreskin (n = 16; 5%). Unusually, no cases of bleeding, wound infection, or haematoma were reported in this study. The method used was not reported for the majority of infants, and complications were most frequent when the procedure was performed by nurses rather than doctors or traditional circumcisers (data not given).

Of the remaining 13 studies, five reported adverse events in 0.3% or fewer boys [9, 22, 26, 30, 32], four in around 2% [11, 21, 23, 25], and the remaining four studies reported adverse events in up to 16% of boys [24, 28, 31, 33]. The studies with highest frequency of complications are from Pakistan and the United Kingdom (UK). The study from Pakistan reports on 200 infants circumcised under local anaesthesia at a Military Hospital using either the freehand or bone-cutter method (a forceps-guided method which does not shield the glans) [31]. Bleeding (defined as requiring more than an application of a pressure bandage) was reported in 9% of boys, and 7% had a local infection of the skin and mucosa. In the UK study, 1129 infants were circumcised by nurses using the Plastibell under local anaesthesia [28], and overall 125 (11.1%) of infants required some degree of follow-up, with complications seen in 5.5%. The most common complication involved the Plastibell ring device itself (3.6%), which is left on after the procedure and normally takes 7-10 days to fall off. The problems included delayed separation of the ring, incomplete separation of the ring, or the ring becoming stuck on the penile shaft. In all cases, the ring was removed without need of anaesthesia and the authors report this removal was quick, simple and atraumatic.

Three studies reported substantial variation in complication frequencies by age or circumcision method. For example, a US study of circumcision by the Gomco clamp stratified by age at circumcision and found no complications in 98 boys circumcised neonatally, but that 12/32 (30%) of infants aged 3-8.5 months had postoperative bleeding requiring suture repair [24]. These 32 boys were circumcised under general anaesthesia and no complications from the general anesthesia were reported. In another study, complications were seen more frequently using the Plastibell (12/381; 3.1%) than the sleeve technique (4/205; 1.95% [33]).

A further ten studies on neonatal/infant circumcision were retrospective hospital-record based studies (Table 2). Five of these were from the USA, two from Pakistan, one each from Israel, Oman and Turkey. Reported frequency of complications were slightly lower than for the prospective studies, with five studies finding very low frequencies (≤0.6%) [19, 34‐37] and four in the range 2-4% [38‐41]. The study reporting the highest proportion (4% in neonates, 10% in infants) included late complications (most commonly foreskin adhesions (7.8%)), with 3 cases (1.3%) of meatitis and 3 requiring circumcision revision (1.3%) [42]. As with prospective studies in neonates and infants, few serious adverse events were reported (<0.2% in all studies except among infants in one US study, where 3/230 (1.3%) of infants required circumcision revisions [42]).

We identified ten prospective studies of complications in children aged one year old or older following circumcision by medically trained providers (Table 3) [43‐52]. The median frequency of any adverse event was 6% (range 2-14%), and median frequency of any serious adverse event was 0% (range 0-3%). Adverse events were seen most commonly among boys circumcised mainly for medical, rather than religious or cultural, reasons. In one of these studies, among boys circumcised in the UK mainly for phimosis, 4/140 boys (2.8%) required acute re-admission to hospital [49] and the frequency of any adverse events was 6.4%. In the other, a Danish study of boys circumcised mainly for balanitis or phimosis, 1/43 (2.3%) boys required re-operation following Plastibell circumcision [52]. Of the other studies, in which boys were circumcised mainly for non-medical reasons, two studies reported any adverse event in about 2% of boys [43, 50], three were 2-5% [47‐49], and higher frequencies (7-14%) were seen in studies from the Netherlands [51], India [44], Iran [45] and Turkey [46]. Complications included bleeding, infection, meatal stenosis and problems with the Plastibell device. The study from the Netherlands reported on complications among 94 Muslim boys circumcised under local anaesthesia outside the hospital. Of these, 13 were seen again because of bleeding (n = 4), infection (n = 2) or swelling (n = 7) [51], excluding the two cases of mild bleeding the frequency of complications was 12%. The Indian study was also small (n = 15) and reported 2 cases of minor wound separation which did not require further surgical intervention [44]. The study from Iran was an RCT in 394 boys, in which 13 (3.3%) boys developed meatal stenosis and 26 (6.6%) had infections at the circumcision site, and 43 (10.9%) had post-circumcision bleeding. Complications were significantly less frequent among boys who parents were randomised to use a lubricant (petroleum jelly) on the circumcision site [45]. Finally, the Turkish study reports complications following a hospital-based mass circumcision exercise, in which 700 boys were circumcised over 5 days. Excluding the cases of bleeding stopped by simple compression, 8% of boys had a complication, most commonly infection (2.7%) and inadequate foreskin removal accompanied by secondary phimosis (2.1%).

Adverse events in 11 retrospective studies tended to be less frequent than for the prospective studies, probably due to under-ascertainment of complications. Most studies reported no serious adverse events (Table 4), but one [53] reported that 2/79 (2.5%) boys required circumcision revisions following circumcision by the Plastibell device. Frequencies of any adverse event varied from 0.3% in a study from Nigeria (5 minor complications reported out of 1563 circumcisions in the hospital over a 15 year period [7]) to 12% (15/129) in South Africa (mostly bleeding, haematoma and infection) and 17.5% (28/160) among boys circumcised with a new disposable device (the Shenghuan Disposable Minimally Invasive Circumcision Anastomosis Device) in China (mainly mild oedema (10%) but also moderate oedema and 2 cases of infection).

Table 5 summarizes the five studies of complications following circumcision by non-medically trained providers. In these studies, frequencies of adverse events are generally higher, and complications more serious, even including penile amputation [7]. A high frequency of complications was seen in a retrospective study from Turkey of 407 boys circumcised at two traditional mass circumcision events [54]. The mean age of the boys at time of circumcision was 7 years (range 1-14 years) and the procedure had taken place in non-sterile conditions by unlicensed providers. Overall, complications were seen in 73% of boys, with the most common complications being wound infection (14%), subcutaneous cysts (14%), bleeding which needed suturing (12%), and haematoma (6%). Five boys (1.3%) developed a urinary infection requiring hospitalisation and intravenous antibiotics. A further 12% of boys were deemed to have incomplete circumcision. In addition, 3 patients with (contra-indicated) hypospadias had been circumcised indicating inadequate screening of the boys.

The retrospective study from the Philippines interviewed 114 males aged 13-51 (mean age 25.9 years), of whom 94% reported having been circumcised below the age of 14 years. Most (68%) had been circumcised by non-medical personnel, and 60% of participants reported post-circumcision complications (inflammation and swelling) to their circumciser, and 4 (3.5%) reported profuse bleeding [55]. In contrast, in a household-based study in southwest Nigeria, respondents reported very few complications (2.8%) following circumcision, mainly by traditional providers [56]. Among 750 child circumcisions, there were 12 cases reported of excessive bleeding, 6 infections, 2 cases of tetanus and one death. The authors report that, although they include the death, there was insufficient information to be certain it was caused by circumcision. A study from Iran reported a late-phase complication frequency of 2.7% following traditional circumcision and a further 5% had excessive residual foreskin. This was similar to circumcisions performed by urologists or surgeons (2.8%), but lower than for GPs/paediatricians (6.1%) or paramedical personnel (9.1%). The authors argue that this is because traditional circumcisers in Iran are experienced and paramedical personnel do not receive effective training.