We present two patients with recurrent painful ophthalmoplegia startin
g in early childhood. Clinically, both patients fulfilled the criteria
for ophthalmoplegic migraine. In one case, magnetic resonance investi
gations were performed following the second attack, between the third
and fourth and during the fourth attack. The left third cranial nerve
was significantly thickened in its course from the brainstem through t
he prepontine cistern to the cavernous sinus during the attacks and mo
derately thickened between the attacks. In the second case, magnetic r
esonance imaging was performed during the 14th attack, when the oculom
otor nerve dysfunction was almost permanent, and the imaging demonstra
ted a swollen oculomotor nerve. Whether these findings are pathognomon
ic of ophthalmoplegic migraine awaits further reports using magnetic r
esonance imaging in infants showing recurrent painful ophthalmoplegia
of early onset.