features include renal involvement, initial lung involvement,
cardiac involvement, positive baseline ANCA and persistently
elevated ANCA.
4
Relapse rate can be decreased by intensive
treatment within the first 6 months of disease,
4
prompting
change of therapy in this patient when poor prognostic factors
persisted. Case series and open label trials document im-
pressive response to rituximab, including two prospective
studies with complete response in 9/10 and 10/10 patients,
respectively.
5,6
This case demonstrates the existence of cardiac involvement
in paediatric WG, associated with a poor prognosis. During the
course of this 14-year-old girl’s illness, treatment was intensified
in response to the persistence of poor prognostic factors for
disease. Despite this, this patient sadly died. Although a post-
mortem was not carried out, the temporal existence of myo-
carditis associated with clinical deterioration in the absence of
infection, suggests that the presence of cardiac involvement was
instrumental in this girl’s collapse and death.
It is important to consider the possibility of sub-clinical
cardiac disease in paediatric WG. Recognition of cardiac involve-
ment should prompt aggressive treatment, although optimum
management remains unclear.
Acknowledgements
The authors would like to thank the patient’s family for allow-
ing us to publish this article.
We would also like to thank Dr Gordan Gladman, Consultant
Cardiologist Royal Liverpool Children’s Hospital, for his help
with this report.
Dr Flora McErlane
1
Dr Liza J McCann
2
1
Specialist Registrar Paediatric Rheumatology
2
Consultant Paediatric Rheumatologist
Department of Rheumatology
Alder Hey Children’s NHS Foundation Trust
Eaton Road, West Derby
Liverpool, UK
References
1 Frosch M, Foell D. Wegener granulomatosis in childhood and
adolescence. Eur. J. Pediatr. 2004; 163: 425–34.
2 Morelli S, Gurgo di Castelmenardo AM, Conti F, Sgreccia A, Alessandri
C, Bernardo ML. Cardiac involvement in patients with Wegeners
granulomatosis. Rheumatol. Int. 2000; 19: 209–12.
3 Oliveira GH, Seward JB, Tsang TS, Specks U. Echocardiographic findings
in patients with Wegener granulomatosis. Mayo Clin. Proc. 2005; 80:
1435–40.
4 Koldingsnes W, Nossent J. Baseline features and initial treatment as
predictors of remission and relapse in Wegener’s granulomatosis.
J. Rheumatol. 2003; 30: 80–8.
5 Stasi R, Stipa E, Del Poeta G, Amadori S, Newland AC, Provan D. Long
term observation of patients with anti-neutrophil cytoplasmic
antibody-associated vasculitis treated with rituximab. Rheum 2006; 45:
1432–6.
6 Keogh KA, Ytterberg SR, Fervenza FC, Carlson KA, Schroeder DR,
Specks U. Rituximab for refractory Wegener’s granulomatosis: report
of a prospective open-label pilot trial. Am. J. Respir. Crit. Care Med.
2006; 173: 180–7.
23 December 2008
Dear Editor,
STREPTOCOCCUS PNEUMONIAE: UNCOMMON CAUSE OF FATAL
NEONATAL SEPSIS
Neonatal Streptococcus pneumoniae infection is a recognised but
uncommon cause of neonatal sepsis. In a retrospective analysis,
the incidence of neonatal pneumococcal sepsis was 0.06/1000
live births and other reports suggest that 1–8% of culture posi-
tive neonatal sepsis is caused by S. pneumoniae.
1,2
The clinical
course closely resembles early onset group B Streptococcal
infection making it difficult to differentiate these two entities
clinically. However, the case fatality rate with S. pneumoniae
infections has been reported as 14.3–35% and should be con-
sidered as a cause of fulminant non-responsive sepsis.
2,3
We report a case of a term infant who deteriorated rapidly and
succumbed shortly after birth as a result of severe S. pneumoniae
sepsis.
A 4586-g male neonate was born by NVD at term following
an uneventful pregnancy. The mother was GBS negative with
no known gestational diabetes and no identifiable risk factors
for sepsis. Prophylactic antibiotics were not administered during
labour. The neonate breathed spontaneously at birth but was
soon observed to be dusky so supplemental oxygen was com-
menced. His Apgar scores were 7/1 and 7/5, respectively. At
6 min of age, he became apnoeic and bradycardic, requiring
intubation and aggressive resuscitation. He was admitted to the
NICU and commenced on conventional ventilation with high
pressures being required. Despite volume expansion and pres-
sors, he remained bradycardic with undetectable blood pressure.
He failed to respond to all supportive measures and died 80 min
after delivery. Because of time restraints, antibiotic therapy was
not administered during resuscitation.
Investigations revealed Hb: 109 g/L, WCC: 12.6 ¥ 10
9
/L, Plate-
lets: 151 ¥ 10
9
/L and CRP: <2 mg/L. His chest X-ray was normal.
A single blood culture was sterile.
However, S. pneumoniae was isolated from swabs taken from
the foetal and maternal sides of the placenta. Post-mortem
samples grew S. pneumoniae in the lung tissue and gastric secre-
tions. The cultured S. pneumoniae was susceptible to penicillin.
S. pneumoniae (Pneumococcus) is a gram positive coccus sur-
rounded by a polysaccharide capsule. Pneumococcal infections
are a major public health problem and cause significant mor-
bidity and mortality worldwide. Perinatal infections with S.
pneumoniae are rare but again cause significant morbidity and
mortality.
1,2
Pneumococcus is a rare inhabitant of the female
genital tract.
3
Some workers recommend pre-emptive treat-
ment for pneumococcal sepsis where S. pneumoniae is isolated
from the genital tract samples,
4
although standard screening
methods for GBS do not favour its isolation. Geelen and col-
leagues reported the largest series of seven cases and discussed
features of additional cases reported in the literature.
5
They
hypothesised that the majority of infants are colonised during
delivery and present in the first 48 h of life with a mortality of
about 50%. Early onset sepsis had a higher mortality than late
onset sepsis.
2
Some reports suggest that early onset pneumo-
coccal sepsis maybe an increasing problem especially in devel-
oping countries,
3
and this has been attributed to an increase in
Letters to the Editor
Journal of Paediatrics and Child Health 45 (2009) 684–687
© 2009 The Authors
Journal compilation © 2009 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
686