Neonatal Fournier’s gangrene;pattern and predisposing factors in a tertiary health facility in Southern Nigeria

Introduction

Necrotizing fasciitis is a fulminant and rapidly progressive infection of the subcutaneous tissues. Commonly involving synergistic infection by both aerobic and anaerobic organisms, these infections trigger a cascade of pathological processes leading to thrombosis of subcutaneous blood vessels, ischaemia and necrosis of involved tissues and overlying skin. ¹

Though the occurrence of this pathological condition in the external genitalia and perineal region had been reported by Baurienne in 1764 and Avicenna in 1877, it was popularized in 1883 by Alfred Fournier who recognized that this was not a sexually acquired infection.^2,3 Fournier’s gangrene (FG) is by far more common in adults than in children. It is considered a rare occurrence in neonates. ⁴ Most reports on FG are tilted towards the adult population, and most of what is known of the clinical presentation and predisposing factors relates to them.^5–8 The consequence of this in neonates is a low index of suspicion and delayed recognition of this deadly entity. The scenario is worsened in an environment where a significant number of births are conducted by traditional birth attendants or poorly trained caregivers, mostly under squalid circumstances. We reviewed our records to assess the pattern of Fournier’s gangrene among neonates in our service with a view to identifying any predisposing factors, review the literature and make recommendations on prevention, early identification and treatment.

Patients and methods

Ours is a five-year retrospective review of cases of neonatal Fournier’s gangrene (NFG) seen in our service. The study was approved by the University of Port Harcourt Teaching Hospital Ethics Committee. The medical case notes of neonates who were diagnosed with FG from April 2016 to March 2021 were retrieved. Data obtained included health facility of birth, mode of delivery, symptoms and signs at presentation, investigation results, treatment and outcome. Data were then collated and subjected to simple descriptive statistical analysis.

Results

Sixteen neonates were diagnosed with FG during the period under review. They comprised 13 males.(M:F = 4.3:1). Fourteen were delivered outside our centre, but two within. Their age at presentation ranged from 8 to 27 days with a median of 22. Delivery was conducted by trained and qualified care givers in eight cases, by traditional birth attendants in five but the status of the caregiver was not established in three. The mode of delivery was vaginal in all; eight were by breech while six were cephalic and two unrecorded. Nine had a patch of necrotic tissue on the genitalia or perineum at the time of presentation (Figure 1), five had an area of erythema or induration while one had sloughing of the entire perineum with the rectum hanging freely, at the time of presentation. Fever and tachycardia were common (Table 1). The extent and site of involvement varied, with the scrotum and perineum being most commonly involved (Table 2). The circumstances we identified that may be contributory to the development of FG are shown in Table 3. There were no cases of urethral catheterization, or any form of instrumentation, but one case was related to circumcision. Investigation results of swab microbiology were obtained in eleven and the most common organisms isolated were Staph aureus, E. coli and Ps. aeruginosa. Only in three were up to three organisms isolated. Random blood sugar results were <2.5 mmol/L in 3/14 at presentation but there were no cases of hyperglycaemia. There was a history of massaging of babies’ perineum with hot water in five and application of a native concoction in two (Table 3)

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Figure 1.

Fifteen day old with gangrene of entire perineum.

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Table 1.

Clinical presentation of patients.

Clinical presentation	Patients
Fever	12
Jaundice	4
Scrotal skin oedema	10
Subcutaneous emphysema	2
Perineal erythema	9
Perineal gangrenous patch	6
Tachycardia (>120 beats/min)	13

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Table 2.

Site and extent of fourniers gangrene in the perineum.

Site	Patients (n = 16)
Penis only	2
Scrotum only	4
Perineum (sparing the genitalia)	1
Penis  +  perineum	1
Vulva  +  perineum	3
Scrotum and perineum	5

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Table 3.

Potential risk factors identified.

Potential risk factor	Patients
Breech delivery	8
Hot water massaging	5
Native concoction application	2
Circumcision	1
Neonatal asphyxia/ sepsis	3

Antibiotics and wound debridement were the mainstay of treatment (Figure 2). One debridement was done in eleven cases, but was repeated in the remaining three. Flap reconstruction was only required in one baby; healing was impressively quick in the remainder, needing only six weeks maximum following debridement (Figure 3). Four died of overwhelming sepsis and one from acute kidney injury within 24 h of admission.Two babies were septicaemic at presentation, and could not be debrided before their demise. One died just as we were trying to secure vascular access on arrival at our children emergency room. The other baby was very pale on arrival and died before we could obtain blood and secure theatre space.

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Figure 2.

Debridement for scrotal gangrene.

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Figure 3.

Six weeks post debridement.

Discussion

Neonatal Fournier’s gangrene (NFG) is not uncommon in our practice in Southern Nigeria. Encountering an average of three cases per year and given its high mortality, it is a clinical entity to look out for. Reports by other researchers in Nigeria have either combined the data of neonates and other children or reviewed only adults; the scenarios are not the same, and are not comparable.^8–10 Whilst omphalitis was the commonest predisposing factor in one series, ¹¹ we did not witness this in our series, and this was not seen in a series from Pakistan. ¹² The suggested aetiology essentially relates to compromised immunity, trauma or circumstances either introducing a nidus of infection or enhancing its spread. Culture results are notoriously variable.^9,12,13 Anaerobic culture may be difficult in some environments, and techniques of sampling may be suboptimal. We intend to investigate this further.

All neonates are known to have a developing but suboptimal immunity at birth but not all develop NFG. This means, therefore, that some additional factors must come into play to result in NFG. Identifying such additional factors would raise an index of suspicion and facilitate diagnosis and intervention. In our study, there were no cases of hyperglycaemia but two patients were hypoglycaemic at presentation. Whilst HIV infection has been reported to be a factor in FG, ¹⁴ none of our patients was so infected. Obviously this remains as a potent cause of immune compromise.

On the other hand, half of our patients were delivered by breech. Viewed against the fact that <10% of vaginal deliveries are normally by breech, this must be a significant factor in our study.^15,16 There is clearly an increased risk of perineal bruising and trauma with breech delivery. The delivery of babies by poorly trained birth attendants and poor hygienic circumstances may well be important factors for NFG. Rarely, unusual factors such as neonatal malignancy have been identified.^17–19

The wide variety of presentations makes early diagnosis of NFG difficult. Those who presented with scrotal oedema, perineal surgical emphysema, or erythema eventually progressed to loss of overlying skin despite broad spectrum antibiotic coverage. Scrotal erythema which resolves may actually be cases only of cellulitis and not necessarily NFG. Real cases of NFG invariably progress to skin loss.

The mortality rate among our patients is 31%, which is far higher than in adults. Inevitably it is a question of natural immunity. Unfortunately, we could not correlate survival with a gangrene severity index. ²⁰ Two of our patients were in such clinical state on presentation that they died before we could conclude arrangements to debride their gangrenous tissues. Early presentation clearly has an influence on mortality.

NFG is thus one of the most serious surgical infections encountered in our neonates. Those who do not come at an advanced stage have varied systemic and local clinical features, making early diagnosis difficult. Our study is inevitably of a small number of patients; any sign of inflammation in the perineal or genital skin of neonates delivered by breech should be very closely followed.