Meningitis retention syndrome: An unusual complication of viral meningitis
Correspondence Address: Source of Support: None, Conflict of Interest: None DOI: 10.4103/0028-3886.193811
Source of Support: None, Conflict of Interest: None
Meningitis retention syndrome (MRS) is a very rare syndrome mainly reported to be associated with herpes simplex virus type 2 (HSV-2) meningitis. The development of urinary retention in the context of meningitis and cerebrospinal fluid (CSF) pleocytosis, without any lumbosacral radiculomyelitis is known as meningitis retention syndrome (MRS). Only a few reports of MRS have been reported in literature.,,,,, A few cases have, however, been seen with meningitis caused by West Nile and HSV-1 virus meningitis, as well as with listeria and tuberculous meningitis. We report a case of MRS associated with HSV-1 meningitis.
A 20-year-old, unmarried male presented to the emergency department with complaints of severe throbbing headache, neck stiffness, and vomiting for 6 days. He had taken multiple analgesics for his headache with no relief. On examination, he was febrile with neck stiffness. Kernig's sign was positive, but mentation was normal. Fundus examination and non-contrast computed tomography head was normal. CSF analysis showed a white blood cell count of 180 cells per high power field, predominantly lymphocytes, with the glucose level being 44 mg/dl and the protein level being 75 mg/dl. The Gram's stain was negative. He was managed symptomatically for viral meningitis.
On the 4th day of admission, he developed urinary retention and was catheterized. Detailed examination revealed normal power and sensation in the lower extremities. Deep tendon reflexes were normal. Also, perianal sensation and anal tone were normal. He did not have genital lesions or a history of sexual contact.
The bladder scan revealed a postvoid urine volume of 450 ml. Brain contrast magnetic resonance imaging (MRI) showed mild meningeal enhancement [Figure 1], and spinal contrast MRI did not reveal any features suggestive of myelitis or radiculitis.
Due to the predominance of lymphocytes in the CSF and a negative Gram's stain, an empiric antiviral therapy was initiated with a 10-day course of intravenous acyclovir. Headache and vomiting subsided after 4 days, but he continued to have urinary retention and required urinary catheterization. After 1 week, CSF-polymerase chain reaction (PCR) herpes panel confirmed the presence of HSV-1 infection. The CSF testing was negative for HSV-2, cryptococcus, tuberculosis, and syphilis. The CSF was also negative for oligoclonal bands.
He showed recovery of bladder function after 10 days of onset of symptoms. A follow-up visit at 3 months confirmed the persistence of functional recovery of his bladder function.
HSV-1 is known for causing the common form of encephalitis, and less frequently, meningitis. In some other countries, however, HSV (type 2 more than type 1) is more commonly isolated as the causative agent for viral meningitis, although it is implicated still less frequently than the enteroviruses. HSV-1 is rarely found to be the etiological organism in MRS; most cases of MRS have been associated with HSV-2 meningitis.
We are reporting a very rare case suffering from HSV-1 meningitis, presenting with acute urinary retention that was consistent with MRS. His clinical presentation and radiographic studies were compatible with MRS. The diagnosis of HSV-1 meningitis was confirmed by serological and CSF studies. This syndrome is an under-recognized clinical constellation of aseptic meningitis and acute urinary retention, and carries a favorable prognosis. Most patients with MRS recover from urinary retention within 3 weeks after their recovery from meningitis, and it is currently considered as a self-remitting disease without sequelae. The initial three case reports in the literature were described by Sakakibara et al., in 2005. The patients had mononuclear pleocytosis in the CSF with an elevated protein and normal-to-near normal glucose levels. All viral studies were negative. MRI studies of the brain and spinal cord were normal, and the urinary retention resolved within 3 weeks.
The neurological axis for bladder control is said to involve the cortex, splenium of the corpus callosum, pons, spinal cord, and its parasympathetic nerves to the bladder., In MRS, the mechanism by which the meningitis causes urinary retention, without there being any evidence of sacral radiculomyelitis, is not clear. Detrusor hyporeflexia due to an unknown mechanism is regarded to be the cause of urinary retention.,
Several hypotheses have been put forward to explain the detrusor hypofunctioning and urinary retention in MRS, including spinal shock secondary to meningeal irritation, inflammation of tracts of the spinal cord (leading to upper neuron dysfunction), direct viral invasion, or the development of postinfectious acute disseminated encephalomyelitis (ADEM). In our patient, however, no lesion could be documented after screening the whole brain and spinal cord. Although an upper motor neuron involvement has been suggested as the implicating factor in MRS (which includes a combination of acute urinary retention and aseptic meningitis), till date only four cases have been reported to display the symptoms that are suggestive of myelitis.,
MRS has also been suggested to be a mild variant of ADEM, which is regarded to have a parainfectious or an autoimmune origin. Although MRS has been reported to follow a benign and self-remitting course (usually, a duration of 2 to 10 weeks), urgent management of the acute urinary retention is often necessary.,,
An inflammatory sacral radiculitis may still be the possible cause of this syndrome. Although, there has been no radiological evidence of inflammation in the cases described until now, electrophysiological changes in these cases have not been well studied.,,, A nerve conduction study or an electromyography (EMG), especially the bladder EMG, may show changes representative of active denervation in such cases; and, in future, may also show changes that aid in diagnosing the possible cause of retention in these patients.
Our patient had no symptoms or signs of encephalitis or myelitis and had a near-normal central nervous system imaging. A unique feature of our case was the relatively late development of retention and an early recovery of bladder function within 2 weeks, contrary to the 3–8 weeks reported in the literature., We hypothesize that treatment with acyclovir could have contributed to this good result. Acyclovir was started empirically before the availability of the result of the HSV-1 PCR assay. The case shows the importance of a close follow-up of patients with viral meningitis as some patients may behave atypically and may need an early definitive treatment. Our case also highlights the importance of recognizing MRS as a unique neuro-urological condition and a rare complication of viral meningitis. The lack of symptoms and a normal radiological imaging should not dissuade physicians from the pursuit of the diagnosis of a HSV-1 infection as the underlying cause of the urinary symptoms since early treatment with antiviral therapy may improve outcome.
We would like to thank the resident staff of the Department of Neurology, Sher-I-Kashmir Institute of Medical Sciences, Srinagar, Jammu and Kashmir, India.
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Conflicts of interest
There are no conflicts of interest.