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• W2212346502 abstract "A 70-year-old woman presented with a 8-month history of severe chronic headache with pain attacks lasting 1-2 hours, associated with bilateral tinnitus. Headaches were characterized by gravative pain, across the forehead and over the vertex, and partially improved during a 6-month period treatment with dexamethasone. She had a history of chronic myeloid leukemia. Neurological examination was normal. Brain magnetic resonance imaging (MRI) depicted diffuse leptomeninges thickening with gadolinium enhancement (Figure) and T2-hyperintense lesion on the frontal regions (Figure). Angio-MRI showed superior sagittal sinus subocclusion (Figure). Routine blood tests, urine, and cerebrospinal fluid (CSF) analysis were all normal. Body and brain-positron emission tomography (PET) excluded a secondary localization of limphoproliferative disease. A single-spot opening measurement and 1-hour monitoring of the CSF pressure (CSFP) were both elevated, respectively, 206 and 247 mmH2O (normal value: 65-200). A lumbar tap with removal of 30 mL of CSF was performed and CSFP was measured at the site of puncture afterward, showing a normal value. Headache gradually resolved over the following days. Repeated MRI at 1 week showed reduced thickness and enhancement of the affected dura mater and disappearance of the right frontal lesion (Figure). Over a 6-month interval, the patient was still pain free and a new MRI evaluation showed resolution of parenchymal lesion, further reduction in the pachymeningeal thickening and enhancement (Figure). Hypertrophic cranial pachymeningitis (HCP) is a rare fibrosing inflammatory process that involves the dura mater. A number of pathological entities may produce HCP including granulomatous diseases, tumors, en plaque meningioma, dural carcinomatosis, and infectious agents. Often the etiology remains unclear and HCP is labeled as “idiopathic” (IHCP). The main clinical features is chronic headache whereas cranial nerve lesions, cerebellar symptoms, epileptic seizures, papilledema, and visual loss occur more rarely.1 The diagnosis of IHCP is based on neuroimaging of thickened and enhancing dura mater.1 The headache in HCP is generally ascribed to dural inflammation as most studies have not documented any increased intracranial pressure (ICP).1 Dural sinus occlusion has been rarely encountered in IHCP.2 Here we describe the case of a patient affected by IHCP associated with increased ICP and sinus subocclusion with concomitant parenchymal lesion, drastically improving after CSF removal. It is likely that our patient's headache was caused by a combined effect of pachymeningitis as well as increased ICP, as documented by high CSFP and MRI evidence of sinus subocclusion and parenchymal signal changes, reflecting possible venous hypertension. Dural venous sinus stenosis and increased ICP represent a rare complication of IHCP.2 Once elevated ICP is established, it may play a significant physiopathological role in the maintenance of both IHCP and headache. Nevertheless, the increased ICP is a reversible condition: a single tap test may shift the high pressure values toward a normal state, restoring a stable balance between CSF and venous outflow pressure, but at lower level.3 This led to the parallel resolution of both headache and parenchymal lesion on MRI, probably due to previous sinus subocclusion. Corticosteroid therapy has shown temporary effectiveness in alleviating symptoms of IHCP. Immunomodulators are often needed and, in refractory cases, surgical exploration for decompression is required.3 Our patient showed meaningful and long-lasting clinical and neuroradiological improvement, after normalization of intracranial pressure. High ICP and sinovenous abnormalities should be considered as a treatable consequence of IHCP. Tap test could represent a recommended therapeutic option in refractory cases of IHCP with increased ICP. Axial T1-weighted contrast-enhanced MRI images at baseline (A); coronal fluid attenuated inversion recovery (FLAIR) T2-weighted MRI images at baseline (B); Angio-MRI image at baseline (C);. coronal FLAIR T2-weighted MRI images 1 week after lumbar tap (D); axial T1-weighted contrast-enhanced MRI images 6 months after lumbar tap (E)." @default.
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• W2212346502 date "2015-12-21" @default.
• W2212346502 modified "2023-09-29" @default.
• W2212346502 title "A case of headache and idiopathic hypertrophic cranial pachymeningitis drastically improved after CSF tapping" @default.
• W2212346502 cites W1986066693 @default.
• W2212346502 cites W2011753884 @default.
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• W2212346502 doi "https://doi.org/10.1111/head.12735" @default.
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