Meningitis‐retention syndrome (MRS) is characterized by aseptic meningitis and acute urinary retention without other apparent focal neurological deficits or magnetic resonance imaging (MRI) abnormalities [1, 2]. MRS has been speculated to represent a very mild form of acute disseminated encephalomyelitis (ADEM), although definitive evidence is lacking [1, 2]. Here, we report two pediatric cases of MRS with high serum anti–myelin oligodendrocyte glycoprotein (MOG) IgG titers, suggesting that a subset of MRS may represent a limited phenotype within MOG antibody‐associated disease (MOGAD).
Case 1 involved a 10‐year‐old boy (Figure 1A). On Day 1, he developed fever, followed by progressive voiding difficulty on Day 2. By Days 5–6, he was unable to void spontaneously. On Day 5, he developed abdominal pain, and ultrasonography revealed marked bladder distension due to urinary retention. He was admitted on Day 6, and urethral catheterization drained 830 mL of urine. He also presented with headache, nausea, constipation, and neck stiffness. MRI of the brain and spine revealed no abnormalities (Figure 1B,C). MRS was diagnosed, and he was managed without immunotherapy. His fever and constipation resolved on Day 17. Because repeated catheter removal trials were unsuccessful, an indwelling urinary catheter was maintained for 18 days. Urinary retention resolved by Day 23. The serum anti‐MOG IgG titer was positive at 1:256 and decreased to 1:16 over 5 months without relapse.
FIGURE 1.
Clinical courses and spinal MRI findings of the two patients. (A) Clinical course of Case 1. (B, C) Sagittal T2‐weighted spinal MRI and axial T2‐weighted MRI at the thoracolumbar spinal cord level in Case 1. (D) Clinical course of Case 2. (E, F) Sagittal T2‐weighted spinal MRI and axial T2‐weighted MRI at the thoracolumbar spinal cord level in Case 2. No abnormalities were identified in the spinal cord, conus medullaris, or cauda equina in either patient.
Case 2 involved a 17‐year‐old male adolescent (Figure 1D). On Day 1, he developed fever and was diagnosed with influenza B on Day 2. Although his fever subsided on Day 8, it recurred on Day 11. On Day 14, he developed urinary difficulty and worsening headache and was admitted on Day 15. Ultrasonography demonstrated bladder distension, and intermittent catheterization drained 500 mL of urine. Because urinary retention persisted, an indwelling urinary catheter was placed. MRI of the brain and spine revealed no abnormalities (Figure 1E,F). MRS was diagnosed based on the clinical features. Given the preceding infection, an immune‐mediated mechanism was suspected, and intravenous methylprednisolone (1000 mg/day) was administered for 3 days. His fever subsided on Day 17. After 12 days of catheterization, the first catheter removal trial was successful, and urinary retention resolved by Day 26. The serum anti‐MOG IgG titer was positive at 1:256. Oral prednisolone was tapered over 3 months, and he remained relapse‐free during 3 months of follow‐up.
In both cases, neurological examination showed no impairment of consciousness, motor or sensory deficits, or abnormalities of deep tendon reflexes. Sacral sensation and anal reflexes were preserved. Brain and spinal MRI revealed no abnormalities suggestive of spinal cord lesions, nerve root involvement, or occult spinal dysraphism. Cerebrospinal fluid analysis revealed mononuclear pleocytosis (32 and 268/mm3, respectively) with normal protein (30 and 34 mg/dL) and glucose (64 and 61 mg/dL) levels, and PCR tests for herpes simplex virus and varicella‐zoster virus were negative.
MRS has traditionally been regarded as a benign, self‐limiting disorder without clear structural abnormalities [1, 2]. Although immunotherapy such as steroid pulse therapy may shorten the clinical course, its efficacy remains uncertain [1, 2]. Urodynamic studies, when performed, demonstrate an areflexic detrusor, suggesting dysfunction of the sacral autonomic nervous system, similar to acute spinal shock [1, 2]. Despite these characteristic findings, MRS remains a diagnosis of exclusion, and its etiology has not been fully elucidated [1, 2]. Nevertheless, elevated cerebrospinal fluid myelin basic protein levels have been reported in some patients with MRS, suggesting that MRS may represent a mild autoimmune inflammatory demyelinating disorder [1, 2].
Recent studies have expanded the clinical spectrum of MOGAD, which includes ADEM, myelitis, optic neuritis, and aseptic meningitis [3, 4]. MOGAD can also involve the spinal cord or sacral autonomic pathways even in the absence of overt MRI abnormalities [5]. Notably, both of our cases showed no abnormalities on MRI of the brain or spine, despite clear clinical features of MRS and high serum anti‐MOG IgG titers. The presence of MOG antibodies in these patients suggests that some cases classified as MRS may represent a limited phenotype within the MOGAD spectrum. The differing clinical courses—spontaneous recovery in one case and response to immunotherapy in the other—may reflect variability within this spectrum [3, 4].
Pediatric cases of MRS are limited, and previous reports have not evaluated MOG antibody status [2]. To our knowledge, this is the first report of MRS associated with anti‐MOG antibodies. Our cases support the hypothesis that MOG‐antibody‐mediated inflammation can affect meningeal and sacral autonomic pathways, leading to urinary retention.
There are several limitations. Urodynamic studies were not performed. The number of cases is small, and false positivity of MOG antibodies cannot be excluded. The absence of MRI abnormalities also limits anatomical confirmation of lesion sites.
Despite these limitations, our findings suggest that MOG antibody‐associated mechanisms may underlie a subset of cases previously diagnosed as MRS, particularly in pediatric patients. Recognition of this association may influence diagnostic evaluation, including antibody testing and therapeutic strategies. Further studies are warranted to clarify whether MRS should be redefined within the expanding clinical spectrum of MOGAD.
Author Contributions
A.I. and H.I. wrote the manuscript. H.I. and K.F. provided medical management for the patients. Y.N. and K.F. critically reviewed the manuscript. All authors have read and approved the final version of the manuscript.
Consent
Written informed consent was obtained from the patients and their parents for publication.
Conflicts of Interest
The authors declare no conflicts of interest.
References
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