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Nissrine Louhab,1 Nawal Adali,1 Mehdi Laghmari,2 Wafae El Hymer,2 Said Ait Ben Ali,2 Najib Kissani11Neurology Department, 2Neurosurgery Department, University Hospital of Mohammed the VIth, Cadi Ayyad University, Marrakech, MoroccoIntroduction: Spontaneous intracranial hypotension is an infrequent cause of secondary headache due to cerebrospinal fluid (CSF) hypovolemia.Objective: To describe a case of headache revealing spontaneous intracranial hypotension complicated by subdural hematoma following lumbar puncture.Observation: A 34-year-old man presented with acute postural headache. The first cerebral computed tomography scan was normal. Lumbar puncture showed hyperproteinorachy at 2 g/L with six lymphocytic cells. The headache became very intense. At admission, clinical examination was normal. Ophthalmological examination did not show any abnormalities. Encephalic magnetic resonance imaging (MRI) showed bilateral subdural hematoma with tonsillar descent simulating Chiari type I malformation. After surgical drainage and symptomatic treatment, the patient was discharged with no recurrence.Conclusion: Spontaneous intracranial hypotension is associated with simple clinical presentation, orthostatic headache, and characteristic MRI findings. Misdiagnosed, it leads to unnecessary procedures.Keywords: intracranial hypotension, headache, magnetic resonance imaging