Case StudyLongitudinal extensive transverse myelitis with cervical epidural haematoma following dengue virus infection
Introduction
Dengue is the most common mosquito borne disease in the world with an estimated 50 million people affected by dengue each year.1 Dengue virus infection is known to be associated with neurological manifestations including encephalopathy, acute disseminated encephalomyelitis (ADEM), encephalitis, Guillain-Barre syndrome and transverse myelitis (TM).2, 3
Acute TM is a spinal cord syndrome with a relatively abrupt onset of motor, sensory, and sphincter disturbances that is usually attributed to an inflammatory demyelinating lesion. Approximately 20% of acute TM cases occur in children.4 Longitudinally extensive transverse myelitis (LETM) refers to a spinal lesion that extends at least three vertebral segments.5 TM is classified broadly as: (i) demyelination – including monofocal clinical isolated syndrome or part of multifocal demyelinating disease including ADEM or multiple sclerosis; (ii) associated with systemic connective tissue disease; (iii) infectious and (iv) idiopathic of which the majority are presumably parainfectious in aetiology.6
Spinal magnetic resonance imaging (MRI) plays a crucial role in making the diagnosis of TM. In addition, transcranial magnetic brain stimulation (TMS) used to measure central motor conduction time (CMCT) is also a useful non-invasive investigation to assess the integrity of the corticospinal tract. Prolonged CMCT has been demonstrated in patients with TM.7 There has only been one published report to date showing the utility of CMCT in a paediatric TM patient.8
TM associated with dengue infection is uncommon with only 10 published case reports to date of which only one was in a paediatric patient aged 14 years.9, 10, 11, 12, 13, 14, 15, 16, 17 Of these 10 published cases, LETM was even rarer with only 5 adult cases of LETM reported (age range 31–45-years old) and 2 of these having concomitant spinal epidural haematoma.9, 10, 11, 12 There were no reported paediatric cases of LETM or spinal epidural hematoma associated with dengue infection.
We present a 12-year-old girl with LETM and concomitant cervical epidural hematoma following dengue infection. We also show the utility of serial non-invasive TMS in this patient and summarise the reported cases of TM associated with dengue infection.
Section snippets
Case report
A previously healthy 12-year-old girl was transferred to our tertiary unit from a local hospital on day 9 of illness with acute lower limb paraparesis and dysesthesia of her chest wall. She was diagnosed as acute dengue fever at the local hospital after a 6-day history of pyrexia and myalgia. Her full blood count at day 6 showed haemoglobin of 18 g/dL (elevated haematocrit of 46.8), leucopenia (2.7 × 109/L), thrombocytopenia (32 × 109/L) and positive dengue IgM serology. There was no evidence
Discussion
This is the first reported paediatric case of dengue virus associated LETM and expands the parainfectious causes of paediatric LETM. Our case also reiterates the clinical importance of differentiating between TM and LETM as LETM is associated with a number of specific conditions which has important diagnostic, prognostic and therapeutic implications.18 The most frequent cause of LETM is neuromyelitis optica (NMO) which requires long-term immunosuppression to prevent relapses.18 In cases with a
Conclusion
Our case report is the first paediatric case of LETM with dengue infection and expands the parainfectious causes of paediatric LETM. Our report is also the first to illustrate that in post-dengue infection patients with concomitant spinal epidural haematoma surgical intervention may not always be required. Clinicians should be aware of parainfectious dengue-related LETM in children presenting with acute weakness beyond the acute febrile phase of dengue infection and consider prompt
Competing interests
All authors have nothing to declare.
Funding
All authors have nothing to declare.
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