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CA Langford

Elisabeth A. M. Cornelissen

JD Akikusa

Lianne M. Geerdink

Linda Koster-Kamphuis

M Rottem

Michèl A. Willemsen

Nicole C. A. J. van de Kar

English

PubMed

Geerdink, L.M.

Koster-Kamphuis, L.

Cornelissen, E.A.M.

Willemsen, M.A.A.P.

Kar, N.C. van de

Radboud Repository

Unexplained hypothermia and bradycardia in two pediatric patients with Wegener's granulomatosis

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LETTER TO THE EDITORS
Unexplained hypothermia and bradycardia in two
pediatric patients with Wegener’s granulomatosis
Lianne M. Geerdink & Linda Koster-Kamphuis &
Elisabeth A. M. Cornelissen & Michèl A. Willemsen &
Nicole C. A. J. van de Kar
Received: 29 June 2010 /Revised: 21 July 2010 /Accepted: 26 July 2010 /Published online: 22 August 2010
# The Author(s) 2010. This article is published with open access at Springerlink.com
Sirs,
We report remarkable symptoms of Wegener’s granuloma-
tosis (WG), which might occur more often than reported so
far. Two patients (13 and 16 years old) with acute renal
failure due to WG developed simultaneous hypothermia
(<36.5˚C) and bradycardia (<60 bpm) during the first weeks
of treatment with intravenous pulse methylprednisolone
(IPM), cyclophosphamide (CYC), hemodialysis (HD), and
therapeutic plasma exchange (TPE). In both patients, rapidly
progressive pauci-immune glomerulonephritis was confirmed
by renal biopsy, and antineutrophil cytoplasmic antibody
(ANCA)-serology was positive. They received 3-day IPM
therapy (1 g/1.73 m2), followed by prednisolone and
cyclophosphamide (CYC) (2 mg/kg/day). Hemodialysis
and TPE were started immediately. Soon after onset of
treatment, hypothermia (34.9˚C) and bradycardia (40 bpm)
were observed (Fig. 1). Electrocardiogram showed a sinus
bradycardia; endocarditis was excluded. Serum electrolyte
and calcium levels were within reference ranges; thyroid
function was normal. There were no signs of infection.
Funduscopy showed no papilledema or renal vascular
abnormalities, and neurological examination was normal.
Cerebral magnetic resonance imaging (MRI) showed no
signs of cerebral granulomas or vasculitis.
In one patient, hypothermia and bradycardia remained
asymptomatic and resolved spontaneously over 4 weeks. The
other patient developed generalized seizures. Additional
diagnostics (cerebral computed tomography and cerebrospinal
fluid) showed no abnormalities. This patient also recovered
spontaneously over several weeks.
We have not been able to explain these conspicuous
findings. To the best of our knowledge, hypothermia has not
been reported as an adverse effect of treatment with IPM or
CYC. Bradycardia in IPM patients, however, has been
described [1], but remarkably, in both our patients, brady-
cardia continued for weeks after IPM treatment. In CYC-
treated patients, arrhythmias have been described, but only
when high doses exceeding 144 mg/kg were administered [2].
In only 4–9% of the WG patients is the central nervous
system (CNS) involved at onset [3], possibly due to
contiguous extension of sinus granulomas. In our patients,
no granulomas were seen by cerebral imaging. Neurological
abnormalities may also occur as a direct consequence of
small-vessel cerebral vasculitis. However, in one patient,
hypothemia and bradycardia were the only neurologic
abnormalities. Transient involvement of the CNS by vascu-
litis at the level of the hypothalamus causing hypothermia
and secondary bradycardia alone is unlikely, but it cannot be
excluded. In conclusion, unexplained, self-limiting, pro-
longed hypothermia and bradycardia can occur during the
first weeks of treatment of WG. The pathogenesis and
clinical relevance needs to be addressed in further studies.
Open Access This article is distributed under the terms of the
Creative Commons Attribution Noncommercial License which per-
mits any noncommercial use, distribution, and reproduction in any
medium, provided the original author(s) and source are credited.
L. M. Geerdink (*) : L. Koster-Kamphuis :
E. A. M. Cornelissen :N. C. A. J. van de Kar
Department of Paediatric Nephrology,
Radboud University Nijmegen Medical Centre,
PO Box 9101, 6500 HB Nijmegen, The Netherlands
e-mail: lgeerdink@gmail.com
M. A. Willemsen
Department of Paediatric Neurology,
Radboud University Nijmegen Medical Centre,
Nijmegen, The Netherlands
Pediatr Nephrol (2011) 26:325–326
DOI 10.1007/s00467-010-1635-6
References
1. Akikusa JD, Feldman BM, Gross GJ, Silverman ED, Schneider R
(2007) Sinus bradycardia after intravenous pulse methylprednisolone.
Pediatrics 119:778–782
2. Langford CA (1997) Complications of cyclophosphamide therapy.
Eur Arch Otorhinolaryngol 254:65–72
3. Rottem M, Fauci AS, Hallahan CW, Kerr GS, Lebovics R,
Leavitt RY, Hoffman GS (1993) Wegener granulomatosis in
children and adolescents: clinical presentation and outcome. J
Pediatr 122:26–31
Fig. 1 Body temperature (°C)
and heart rate (bpm) in patient
1 with rapidly progressive
glomerulonephritis due to
Wegener’s granulomatosis (WG)
during the first weeks of
treatment. Normal range in
grey. bpm beats per minute,
IPM intravenous pulse
methylprednisolone, TPE
therapeutic plasma exchange,
HD hemodialysis
326 Pediatr Nephrol (2011) 26:325–326


Unexplained hypothermia and bradycardia in two pediatric patients with Wegener’s granulomatosis

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Unexplained hypothermia and bradycardia in two pediatric patients with Wegener’s granulomatosis

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