• Yang Zhou and Chenglin Tong.
• Department of Neurology, Shaoxing Second Hospital, Shaoxing, Zhejiang, China.
• Medicine (Baltimore). 2024 Jul 26; 103 (30): e39090e39090.

RationaleSpontaneous intracranial hypotension (SIH) is a well-established condition typically presenting with disabling orthostatic headache. Corpus callosum agenesis (CCA) is one of the most common human brain malformations with a wide spectrum of associated malformations, chromosomal abnormalities, and clinical syndromes.Patient ConcernsA 53-year-old woman presented with recurrent orthostatic headache for about 1 month. The head computed tomography examination of the patient showed CCA and the initial pressure of subsequent lumbar puncture was only 5 centimeters cerebrospinal fluid. Magnetic resonance imaging examination confirmed CCA with gray matter heterotopia.DiagnosisThe final diagnose was SIH related headache with CCA.InterventionThe patient's symptom improved after oral hydration, intravenous fluids, and bed rest.OutcomeFavorable outcome was observed.LessonsAlthough this co-occurrence of SIH and CCA is probably coincidental, this finding suggests that the developmental malformations of the brain may lead to structural changes in brain tissue or disturbances in cerebrospinal fluid production and reflux, resulting in pathological changes over time.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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