Ondine's curse secondary to unilateral medullary infarction has been reported. However, the mechanism remains unclear. This case of a patient with an infarction extending from the right side of the medulla oblongata to the spinal cord at the C1 level showed absence and decrease of respiratory movement on the right and left sides of the chest, respectively, during automatic respiration. The physiological respiratory findings implied that Ondine's curse due to unilateral medullary infarction could be caused by both the impaired regulation of automatic respiration and the disconnection of the projection to ipsilateral phrenic motoneurons.
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