An 8-month-old boy presented with periods of apnea occurring several times daily. Initial clinical and radiologic evaluations could not identify a cause for these episodes. Video electroencephalographic monitoring indicated interictal high-voltage θ and δ activity over the right frontotemporal area. A short ictal run of δ activity over the same region was registered during a brief apneic spell. Cranial magnetic resonance imaging revealed a lesion anteromedial in the right temporal lobe. After epilepsy surgery, anatomopathologic examination indicated meningioangiomatosis. This case demonstrates that a focal lesion in the temporal lobe can present during infancy with apnea as sole seizure manifestation, without other clear clinical evidence.