Fluctuating hearing loss, episodic headache, and stroke with platelet hyperaggregability: Coexistence of auditory neuropathy and cochlear hearing loss

We encountered a 10-year-old girl with fluctuating sensorineural hearing loss, episodic headache, and white matter stroke. Strenuous exercise, febrile illness, and general anesthesia all temporarily worsened hearing. Audiologic findings were asymmetric: left-sided retrocochlear dysfunction consisten...

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Bibliographic Details
Published in:Brain & development (Tokyo. 1979) 2006, Vol.28 (1), p.55-59
Main Authors: Nobutoki, Tatsuro, Sasaki, Masayuki, Fukumizu, Michio, Hanaoka, Shigeru, Sugai, Kenji, Anzai, Yuki, Kaga, Makiko
Format: Article
Language:English
Subjects:
Adenosine Triphosphate - therapeutic use
Antithrombin III - metabolism
Audiometry - methods
Blood Platelets - metabolism
Child
Collagen - metabolism
Evoked Potentials, Auditory, Brain Stem - drug effects
Evoked Potentials, Auditory, Brain Stem - physiology
Female
Functional Laterality
Headache
Headache - blood
Headache - complications
Headache - pathology
Hearing Loss, Sensorineural - blood
Hearing Loss, Sensorineural - complications
Hearing Loss, Sensorineural - drug therapy
Hearing Loss, Sensorineural - pathology
Humans
Magnetic Resonance Imaging - methods
Peptide Hydrolases - metabolism
Platelet Aggregation - physiology
Platelet hyperaggregability
Sensorineural hearing loss
Stroke - blood
Stroke - complications
Stroke - pathology
Thromboxane B2 - metabolism
White matter
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Summary:We encountered a 10-year-old girl with fluctuating sensorineural hearing loss, episodic headache, and white matter stroke. Strenuous exercise, febrile illness, and general anesthesia all temporarily worsened hearing. Audiologic findings were asymmetric: left-sided retrocochlear dysfunction consistent with auditory neuropathy contrasted with cochlear hearing loss in the right ear. Platelets obtained during a headache-free period showed excessive responsiveness to collagen in vitro, while episodic elevations of thromboxane B 2 and thrombin–antithrombin III complex were noted in blood sampled during headache. Treatment of hyperaggregability of platelets with aspirin and antioxidant vitamins relieved headache, while adenosine triphosphate administration improved hearing thresholds. In this patient, hearing impairment and white matter strokes appeared to respectively related to impaired blood flow to the cochlea and white matter caused by platelet dysfunction triggered by physiologic stresses.
ISSN:0387-7604
1872-7131
DOI:10.1016/j.braindev.2005.03.012