Background: Neuroimaging is pivotal in the evaluation of epilepsy. We present a case of incidental mesencephalic hypertrophy resembling a double brainstem in a patient with drug-dependent epilepsy. Case Presentation: A 16-year-old girl was hospitalized for generalized tonic-clonic status epilepticus following antiepileptic treatment discontinuation. Diagnosed with epilepsy at age 12, she had been prescribed levetiracetam (250 mg twice daily) but discontinued therapy for 4 months. Family history was unremarkable for similar conditions. Clinical examination revealed short stature, trisomy-like facies, bilateral hypoplasia of the fourth and fifth fingers, and pes planus. Mild cognitive impairment was noted, with no sensorimotor deficits. Contrast-enhanced brain computed tomography (CT) demonstrated a pseudo-double brainstem secondary to mesencephalic hypertrophy curving anteriorly between a pituitary adenoma and the pons. The patient was managed per institutional status epilepticus protocol, with no seizure recurrence. At discharge, lamotrigine (5 mg/kg/day) was initiated alongside counseling to improve treatment adherence. Conclusions: Epilepsy coexisting with congenital malformations necessitates comprehensive neuroimaging and genetic assessment. In resource-limited settings such as Madagascar, pedigree analysis and consistent follow-up are critical to optimizing care.
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