Umbilical gangrene in the newborn

Umbilical Gangrene in the Newborn By R.J. Stunden, R.A. Brown, H. Rode, A.J.W. Millar, and S. Cywes Rondebosch, Repubfic o f South Africa 9 Six neonates are described with a gangrenous omphalitis, a disease not reported for many years, They had many features in c o m m o n including previous good health. In t w o , the disease was preceded by the application of tobacco ash to the umbilical stump. Clinically there was gangrene of the umbilicus with a blood-stained discharge and extensive cellulitis radiating into the abdominal wall. Despite aggressive therapy including excision of the affected area, the systemic effects of the local disease progressed rapidly until all of the first five infants w h o presented, died. Microbiologically there was a polybacterial infection involving a wide variety of organisms particularly E coil and Clostridia species. There appeared to be an appropriate response by the infants to an acute bacterial infection, and t w o patients investigated showed no evidence of cellular or humoral immunodeficiency. The pathology was an infection causing local tissue necrosis with a vasculitis and microabscess formation, which involved adjacent structures by direct extension. The severe systemic symptoms w e r e attributable to an endotoxemia or exotoxemia. The evolution of therapy for gangreneous omphalitis in the newborn, successful in the sixth patient, is discussed. 9 1988 by Grune & Stratton. Inc. INDEX W O R D S : Omphalitis; umbilical gangrene.

I N O R U M B I L I C A L SEPSIS is still common even in advanced countries, and may be endemic in less developed regionsY 2 Postnatal Staphylococcal colonization is frequent and may not be entirely innocent with omphalitis, a familiar manifestation of Staphylococcal disease in the newbornJ Umbilical infection resulting in neonatal tetanus is still a common cause of death in India 3 and Northern Natal, South Africa but other Clostridial infections in the newborn are rare. 4 This may be surprising since Clostridia are normally found in the female genital tract, and appear in the neonatal intestine at an early age. Recent reports of neonatal clostridial infections are listed. 5-8 Although neonatal omphalitis is not uncommon 4'7"9'1~and may be fatal, 4'1~ we can find no recent reference to a uniformly lethal form of umbilical gangreneJ 1

M

MATERIALS AND METHODS Six patients with gangrene of the umbilicus have been admitted to the Red Cross War Memorial Children's Hospital in as many years (Fig 1). The presenting features are summarized in Table 1. All of the neonates had healthy parents and in only one instance had there been a complication of pregnancy, preeclamptic toxemia. One was born by cesarian section (not for a perinatal complication), the remainder by normal vertex delivery (NVD). Both sexes, and both black and white children were affected. All weighed more than 3.2 kg at birth, and only one had a congenital anomaly apart from a patent ductus arteriosus. They all had had an uncomplicated perinatal period and appeared healthy until developing umbilical sepsis. They all presented between the seventh and ninth days of life with a short history of umbilical sepsis and spreading perinmbilical edema or eellulitis (Figs 2 and 3). There was an obvious predisposing factor to sepsis in two with the application of ground tobacco ash from a pipe into the umbilical stump (see addendum).

RESULTS

The hematologic investigations on admission, treatment and outcome of the six neonates are summarized in Table 2. In patients 5 and 6, blood taken on admission showed that white cell function as measured by lymphocyte transformation to mitogens was reduced by 50%. Total T cells, T helper, and T suppressor, as well as B cell counts, were in the normal range for both patients. Phagocyte cell function in bacteriocidal assay was normal. Complement levels were normal. Plasma immunoglobulin levels were in or above the normal range (values of patient 5 followed by those of patient 6): IgG 7.94/7.78 g / L (normal range 2.70 to 7.50 g/L), IgM 0.26/0.26 g / L (normal range 0.12 to 0.87 g / L ) and IgA 0.53/0.19 g / L (normal range 0.06 to 0.58 g/L). Table 3 lists the microorganisms isolated from an umbilical pus swab, an aspirate of the anterior abdominal wall, blood culture, and from a postmortem peritoneal aspirate where taken. The macroscopic findings and histopathology of excised specimens and at postmortem are summarized in Table 4. DISCUSSION

From the Department of Paediatric Surgery, Institute of Child Health, Red Cross War Memorial Children's Hospital and University of Cape Town, Rondebosch, Republic of South Africa. Address reprint requests to S. Cywes, Department of Paediatric Surgery, Institute of Child Health, Red Cross War Memorial Children's Hospital and University of Cape Town, Rondeboseh, Cape 7700, Republic of South Africa. 9 1988 by Grune & Stratton, Inc. 0022-3468/88/2302-0008503.00/0 130

A distinction must be made between omphalitis in general and the umbilical gangrene of these patients. Omphalitis is often ascribed to a single organism, particularly Staphylococcus aureusJ 2 It may be associated with local pus, cellulitis of the anterior abdominal wall, or extension from the umbilicus via fetal remnants into the circulation, liver or genitourinary Journal of Pediatric Surgery, Vol 23, No 2 (February), 1988: pp 130-134

UMBILICAL

GANGRENE

IN T H E N E W B O R N

C e l l u l i t i s of t h e a n t e r i o r a b d o m i n a l

F i g 1.

a gangrenous

131

wall emanating from

I n v e r t e d lateral plain r a d i o g r a p h y of a n i n f a n t w i t h N o t e t h e n o r m a l intestinal gas p a t t e r n , but w a l l s e c o n d a r y to gross t h i c k e n i n g of t h e a n t e r i o r a b d o m i n a l Fig 2.

umbilicus.

umbilical

gangrene.

cellulitis.

systems. However, unless there are established complications at the time of presentation, the outcome using appropriate intravenous antibiotics and local therapy is normally favorable and rapid. This is in marked contrast to umbilical gangrene. At presentation to the hospital the infants often appeared deceptively well though two were pyrexial. Cardinal features included umbilical gangrene with a blood-stained discharge, and abdominal distension with no other apparent cause. Pyrexia, tachycardia, and paralytic ileus were not prominent early, but soon developed with the rapid progress of the disease. As can be seen from Table 2, all the infants had a normal hemoglobin level at initial examination, were mounting a white cell response appropriate to an acute infection, and had platelets in adequate numbers. White cell function and immune competence were

normal in patients 5 and 6, excluding an immunodeficiency syndrome as an etiological factor. The striking feature microbiologically (Table 3) is the ubiquity of multiple organisms; an unusual occurrence in infections of otherwise healthy neonates. The most common organisms isolated were Escherichia coli (5), Clostridium sp including those of the gas gangrene group (4), Staphylococcus epidermidis (2), and Streptococcusfaecalis (2). Bacteriemia was not a major feature of this disease, although peripheral circulatory failure suggestive of endotoxemia developed in all. All showed a falling platelet count, inferring absorption of toxins. The significant discrepancy between high bacterial counts of local swabs and

T a b l e 1. N e o n a t a l Umbilical G a n g r e n e : F e a t u r e s a t P r e s e n t a t i o n Patient Sex Race(black/white) Pregnancycomplication Birth mode Birth weight (kg)

1

Congenit~ anomalies Predisposing factor Age at presentation(d) Duration of symptoms (hr)

M B No NVD 3.5 No None 7 12

Symptoms

Refusedfeeds, abdomi-

2 F W No LSCS 3.4 No None 7 24 Umbilical redness

nal swelling, vomited

4

5

M B No NVD 3.9 No None 9 48 Abdominal distension

3

F B Pet NVD 3.2 No Ash 7 24 Poor feeding, abdomihal swelling

M B NO NVD 3.4 No Ash 7 48 Umbilical redness, abdominal swelling

M B No NVD 3.6 VSD, PAS None 8 48 Umbilical sepsis, abdominal swelling

6

Distension. ascites Pus discharge,(]an-

Distension Blood-stained dis-

Oister~sion Blood-stained dis-

DistensiOn Blood-stained discharge,gangrene

once Signs Abdomen Umbilicus

Cenulitis

Otherfeatures

Irritable Distension Blood-stained discharge, inverted umbilicus, gangrene Abdominal wall

Pneumonia

irritable, pyrexia Distension, ascites Blood-stained discharge, gangrene Abdominal wall, flanks, chest,

perineum Pleuraleffusion

Lethargy,jaundice

grene Abdominal wall,

perineum pleuraleffusion

charge,gangrene Abdominal wall, perineum

Pyrexia

charge,gangrene Abdominal wall

Abdominal wall

Situs inversus VSD PAS

Abbreviations: PET, preeclamptictoxemia; NVD, normal vertex delivery; LSCS, lower segment cesarian section; VSD, ventricularseptal defect; PAS, pulmonaryartery stenosis.

132

STUNDEN

Fig 3 .

Gangrene

of the

umbilicus

with

surrounding

cellulitis

(detail).

infrequent isolation of organisms from blood cultures suggest that the systemic effects resulted from endotoxins and exotoxins. The infants often appeared to respond to treatment initially, only to later follow a rapid downhill course (Table 2). All those who died did so within five days of admission, three within 24 hours, despite intensive supportive measures. This included broad spectrum antibiotic therapy (Benzyl penicillin, aminoglycoside, and Metronidazole), aggressive surgical excision of the affected abdominal wall, platelet transfusion, and the use of inotropic agents and ventilatory support were indicated. The clinical course was remarkably consistent after the appearance of abdominal distention. Tachycardia and tachypnea were followed by peripheral circulatory failure and oliguria. Convulsions in one patient, and jitteriness in two were suggestive of Table 2. Patient

Hemoglobin on admission (g/dL) (1) White cell count on admission (1 /d-) (1) Platelet count on admission (1 pL) (1} Treatment

Progress

Neonatal

1

Umbilical Gangrene: 2

ET AL

tetanus, but no pathognomonic features were present. Leakage of protein from the vascular space ensued, together with a fall in platelet count with or without evidence of DIC. Those infants who had a postmortem examination appeared well nourished and had no congenital anomalies apart from a patent ductus arteriosus, a normal anatomic finding in the first 2 weeks of life. Histologically, there was gangrene of the umbilicus with cellulitis radiating into the abdominal wall, even as far as the axillae and perineum. Microabscesses were evident within the subcutaneous tissue. The gangrene extended into the underlying muscles, and in one case affected the peritoneum underlying the rectus abdominis with local involvement of the adjacent adherent small intestine. A vasculitis was present, associated with fibrin microthrombi. The effects of the disease were more widespread than this, attested by a stress reaction in the thymus and adrenal glands. FUTURE MANAGEMENT

The disease is predominantly a local infection with severe systemic effects. Treatment has advanced with each new patient and the most recent one is the only survivor. The first was managed nonoperatively, with the use of intravenous antibiotics. Umbilical excision was performed in the second, but only when she was already desperately ill; the third was moribund soon after arrival in hospital. The next patient received much earlier aggressive supportive treatment, but he died before surgery was undertaken. The penultimate patient was operated on as an urgent procedure, with wide excision of the affected anterior abdominal wall; he was ventilated electively to reduce the work of breathing and to maximize tissue oxygenation. The sixth, who survived, was also elecInvestigation,

3

Treatment,

and Progress

4

5

6

20,6

18,7

15,6

22,5

17,2

13.4

45,900

28,300

24,800

54,900

14,000

15,700

275,000

315,000

554,000

370,000

389,000

292,000

Intravenous antibiotics, umbilical dressing

Intravenous antibiotics, excision of umbilicus

Intravenous antibiotics, drainageof pleural effusion, wide excision of umbilicus

Intravenous antibiotics, umbilical dressing, died before surgery

Intravenous antibiotics, wide excision of urnbilicus and abdomihal wall, including muscle, Silastic patch

Renal failure, died aged 8d

Convulsion, DIC, died aged 10 d

Died aged 14 d

DIC, died aged 8 d

Endotoxemia, DIC, died aged 8 d

Intravenous antibiotics, antitetanus IgG, wide excision of umbilicus and abdominal wall, wound irdgation with hydrogen peroxide, penicillin injected into remaining abdominal wall. Postoperative paralysis and IPPV. Second operation 48 hr affer admission. Split skin graft to abdominal wall after 3 wk Alive

Abbreviations: IPPV, intermittent positive pressure ventilation; DIC, disseminated intravascular coagulopathy.

UMBILICAL GANGRENE IN THE NEWBORN

133

Table 3.

Patient

Umbilical Swab

Microbiology

Peritoneal Culture (Postmortem)

Blood Culture

Abdominal Wall Aspirate

1

Proteus vulgaris Staph aureus

Not performed

No growth

2

E coil Klebsiella sp Streptococcus faecalis

Streptococcus faecalis

Micrococcus sp

3

CIostridia sp

No growth

No growth

Streptococcus group D Staph epidermidis

4

E coil Clost.~lium (gas gangrene group) ~-haemolytic Streptococcus

E coil

No growth

No g r o w t h

Nonhemolytic Strepto-

coccus Bacteroides fragilis E coil Staph aureus Streptococcus faecalis Peptococcus Clostridium sp

Diphtheroids

5

6

E coil Klebsiella oxytoca Proteus vulgaris Streptococcus faecalis

Ctostridium (gas gangrene group) E coli Peptostreptococcus Streptococcus group A Streptococcus milleri Acinetobacter anitratus Clostrt~lium (gas gangrene group) E coil

Staph epidermidis

Not performed

No growth

/~-haemolytic

Streptococcus not group A

Clostridial endotoxin levels can be m e a s u r e d , a n d the use of a n t i s e r u m should be investigated.

tively p a r a l y z e d a n d ventilated. A n t i t e t a n u s i m m u n o globulin was a d m i n i s t e r e d on presentation. A s soon as swabs had been taken and m a j o r resuscitation commenced, the anterior a b d o m i n a l wall was widely excised b a c k to h e a l t h y tissue, into which penicillin was injected. T h e wound was i r r i g a t e d with h y d r o g e n peroxide. A n e x c h a n g e transfusion was effectively p e r f o r m e d p e r o p e r a t i v e l y by p e r m i t t i n g free bleeding at the wound m a r g i n s for some t i m e before c o m p l e t i n g hemostasis, and then infusing fresh blood. Platelet consumption occurs with the a b s o r p t i o n of endotoxin from the p l a s m a , and platelet transfusion was p e r f o r m e d if the count fell below 20 • I 0 9 / L . Inotropic agents were ineffective in the first five patients, and were not used in the sole survivor. T h e use of h y p e r b a r i c oxygen in C l o s t r i d i a l disease is controversial, and p r o b a b l y c o n t r a i n d i c a t e d in this age group.

Table 4. Patient

1

Macroscopic Umbilicalgangrene, abdominal

wall cellulitis Adjacent localizedperitonitis Interstitial pneumonia Kidneys: acute tubular necrosis

Microscopic Thymus: stressreaction

Subcutaneouscellulitis

Neonatal

ACKNOWLEDGMENT We would like to thank Professor D. Beatty for performing and interpreting the white cell function and immunologic studies, Dr C. Sinclair-Smith for reviewing the histologic specimens, and Dr D. Hanslo for assisting with the microbiology.

ADDENDUM The application of substances to the umbilicus of neonates has been prevalent for centuries, and includes the use of soil, herbs, and human or animal excreta. In some tribes of Southern Africa, dried white wood ash was widely used as an umbilical dessicant long before the introduction of baby powder. After the mass migration of the rural population to cities, the ash from coal fires was used as a substitute since the availability of wood declined. In homes where there may be no fireplaces, tobacco is now replacing coal as the source of ash,

Umbilical Gangrene:

Pathology

2

3

4

Umbilicalgangrene,abdominal wall cellulitis, chestwall and

Umbilicalgangrene, abdomi-

Umbilicalgangrene, ab-

nal wall cellulitis, perineal

perineal oedema, ascites edema Patchy pneumonia, pleuraleffu- Adjacentlocalized peritonitis,

sion Liver: punctate hemorrhages, patent ductus arteriosis Adrenals: lipid depleted

Subcutaneous cellulitis, microabscessformation and vasculitis

5

Umbilicalgangrene, abdominal wall cellulitis, dominalwall ceUulitis perineal cellulitis

6

Umbilicalgangrene, abdominalwall cellulitis

local musclenecrosis

Thymus: stressreaction Adrenals:lipid depleted

Subcutaneous cellulitis, urnbilical vasculitis

Thymus: stressreactions Adrenals:lipid depleted Vegetable matter ambedded in umbilicus

SubcutaneousceUulitis

Vegetablematter embeddad in umbilicus

Subcutaneouscellulitis and vasculitis

Subcutaneouscellulitis and microabscess formation

134

STUNDEN ET AL

REFERENCES

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7. Isenberg AN: Clostridium welchii infection. A clinical evaluation. Arch Surg 92:727-731, 1966 8. Walker SH, Macaraeg El: Neonatal sepsis due to Clostridium perfringens. MD State Med J 22:61-62, 1973 9. Dinari G, Haimov H, Geiffman M: Umbilical arteritis and phlebitis with scrotal abscess and peritonitis. J Pediatr Surg 6:176, 1971 10. Halliday HL: A case of gross neonatal omphalitis in the United Kingdom. Ulster Med J 43:120-122, 1974 11. Von Reuss AR: The Diseases of the Newborn. London, Bale, Sons & Danielsson Ltd, 1920, pp 431-435 12. Johnson JD, Malachowski NC, Vosti KL, et al: A sequential study of various modes of skin and umbilical care and the incidence of Staphylococcal colonization and infection in the neonate. Pediatrics 58:354-361, 1976