Headache arising from idiopathic changes in CSF pressure

doi:10.1016/S1474-4422(15)00015-0

The Lancet Neurology

Volume 14, Issue 6, June 2015, Pages 655-668

https://doi.org/10.1016/S1474-4422(15)00015-0 Get rights and content

Summary

New onset of sudden or progressive headache can have various causes, including disorders of intracranial pressure (ICP). Headache is the most common—and often the presenting—symptom of both intracranial hypertension and intracranial hypotension syndromes, which can be symptomatic or idiopathic. Despite the widespread availability of diagnostic tests, including ocular ophthalmoscopy, neuroimaging, and measurement of CSF pressure, delays in diagnosis or misdiagnosis of idiopathic intracranial hypertension and spontaneous intracranial hypotension remain common. If left untreated, idiopathic intracranial hypertension and spontaneous intracranial hypotension produce highly disabling headaches, and threaten vision, hearing, and in rare cases, brain function and life. To improve the diagnosis of idiopathic intracranial hypertension and spontaneous intracranial hypotension, changes in the overall diagnostic strategy for headaches will be necessary in most care centres. Improved understanding of CSF physiology and the mechanisms of idiopathic intracranial hypertension and spontaneous intracranial hypotension will guide the development of new treatments.

Introduction

New-onset headache can have various causes, including disorders of intracranial pressure (ICP).¹ Headache is the most common—and often the presenting—symptom of both intracranial hypertension and intracranial hypotension syndromes. These syndromes can be either symptomatic or idiopathic, with no clear identifiable cause. Most causes of intracranial hypertension are life-threatening (panel 1), so clinical investigations are always urgent, including those for patients presenting with isolated headaches. In symptomatic intracranial hypertension or hypotension, treatment of the underlying disease often results in improvement of headaches, the specific management of which is not addressed in this Review. Sometimes, the only identified cause of headache is an idiopathic change in CSF pressure: an increase in CSF pressure, in association with papilloedema, is used to define idiopathic intracranial hypertension, while a decrease in CSF pressure defines spontaneous intracranial hypotension. Diagnosis of these conditions can be challenging. Many cases present with isolated headaches, normal examination—except for papilloedema in the case of idiopathic intracranial hypertension—and normal brain imaging.

If left untreated, idiopathic intracranial hypertension and spontaneous intracranial hypotension can reduce quality of life and cause serious complications, including blindness for idiopathic intracranial hypertension and coma for spontaneous intracranial hypotension. In this Review, we describe the pathological mechanisms, clinico-radiological features, and management of headache arising from CSF pressure changes related to idiopathic intracranial hypertension and spontaneous intracranial hypotension.

Section snippets

Physiology of intracranial pressure and CSF

Pressure within the brain parenchyma is equal to the pressure in the intracranial extra-axial spaces, including the ventricular and subarachnoid spaces, which contain CSF. Normal CSF pressure is equal to ICP, varies from 60 to 250 mm H₂O in healthy adults, and fluctuates during the day because of many factors. According to the Monro-Kellie doctrine,² because bone is rigid, the volume of the intracranial and spinal canal spaces must remain constant: a volume change in one constituent must be

Pathological mechanisms of changes in CSF pressure

Volume increase of any intracranial constituent (eg, brain parenchyma, blood, or CSF) above the threshold of compensatory mechanisms will result in increased ICP (panel 1). Similarly, CSF hypovolaemia reduces ICP.⁵ Uncompensated ICP changes can cause headache through traction on pain-sensitive structures, including intracranial large blood vessels and nerves.

Potential pathological mechanisms of idiopathic intracranial hypertension have been reviewed previously.⁶ Classic mechanisms include

Suspected headache secondary to change in CSF pressure

Most patients presenting with headache as the chief complaint have a primary headache disorder, such as migraine or tension-type headache, the diagnosis of which relies on strict diagnostic criteria in the absence of any objective marker.¹ Secondary headache disorders manifest as new-onset headaches that arise in close temporal association with the underlying cause.¹ Secondary headache should be suspected in any patient without a history of primary headache who reports a de-novo headache and in

Measurement of CSF pressure

The gold standard for ICP measurement is placement of an intraventricular catheter connected to an external pressure transducer, which is an invasive procedure used in intensive care units.³¹ The classic, less invasive method to reliably estimate ICP is lumbar puncture (panel 2).31, 34, 38

Although the definition of normal CSF opening pressure is still debated, results from studies have shown that the upper limit of CSF opening pressure in healthy individuals should be regarded as 250 mm H₂O in

Characterisation and classification

Idiopathic intracranial hypertension is characterised by increased ICP of unknown cause,⁴⁶ in the absence of any intracranial process, venous sinus thrombosis, or meningeal process (panel 1). Patients with high ICP secondary to specific drugs or cerebral venous stenosis are still classified as having idiopathic intracranial hypertension. Suggestions have been made that the older term pseudotumour cerebri could be used again,³³ but we prefer the term idiopathic intracranial hypertension, which

Demographics, predisposing factors, and associated disorders

Spontaneous intracranial hypotension has been reported in patients aged from 2 to 86 years,81, 82 but occurrence peaks between 35 and 42 years and the disorder is most common in women.44, 82, 83, 84 Spontaneous intracranial hypotension is probably not as rare as is generally thought, as shown by the large number of patients (338) from the same institution who were retrospectively included in a study during a 9 year period.⁸⁵ Incidence is estimated to be two to five cases per 100 000 people per

Conclusions

Idiopathic changes in CSF pressure are rare causes of secondary headaches that are treatable. Despite the widespread availability of diagnostic tests, misdiagnosis of idiopathic intracranial hypertension and spontaneous intracranial hypotension remains common. If untreated, these disorders cause highly disabling headaches and threaten vision, hearing, and, in rare cases, brain function and life. Improvements in the overall strategy for the diagnosis of headaches will be needed to improve the

Search strategy and selection criteria

We identified references for this Review through searches of PubMed from Jan 1, 1955, to March 16, 2015, with the terms “idiopathic intracranial hypertension”, “pseudotumor cerebri”, “intracranial hypertension”, “papilledema”, “idiopathic intracranial hypertension without papilledema”, “spontaneous intracranial hypotension”, “CSF hypovolemia”, and “CSF leak”. We identified additional references by manually searching journals and relevant articles. We only reviewed articles published in English

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Pseudotumor cerebri in the paediatric population: clinical features, treatment and prognosis
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El síndrome de pseudotumor cerebri (SPTC) en pacientes prepuberales presenta características que lo diferencian respecto a su presentación en la etapa pospuberal. Nuestro objetivo es describir las características de los pacientes diagnosticados de SPTC pediátrico en nuestro centro y compararlas en función de su estado puberal.
Se incluyeron a los pacientes diagnosticados de SPTC en un hospital de tercer nivel entre los años 2006 y 2019 con edades comprendidas entre uno y 18 años que cumplieran los criterios diagnósticos actualizados del SPTC. Se clasificaron en función de su estado puberal y peso corporal. Posteriormente, se analizaron los datos de las punciones lumbares, estudios de neuroimagen, valoraciones oftalmológicas, así como el régimen terapéutico recibido a lo largo de su seguimiento.
Se recogieron 28 pacientes, 22 prepuberales y seis pospuberales, con edad media de 9,04 ± 2,86 años. El 83,3% de los pacientes pospuberales eran varones presentando sobrepeso/obesidad en el 66,7%. Eran varones el 27% de los pacientes prepuberales, de ellos asociaban sobrepeso el 31,8%. La sintomatología más frecuente fue cefalea (89,9%) y visión borrosa (42,9%). Todos los pacientes presentaron papiledema; un 21,4% de los casos presentaron parálisis del VI par. Se identificó un posible desencadenante en un 28,6%. El 19% presentaron recurrencia clínica, siendo todos ellos prepuberales. La resolución clínica completa se produjo en el 55,6% de los pacientes.
Pacientes con SPTC presentan menor prevalencia de obesidad en la etapa prepuberal, junto con un mayor porcentaje de etiologías secundarias y tasa de recurrencia que los pacientes pospuberales.
Pseudotumor cerebri (PC) in prepubertal patients displays certain characteristics that differentiate it from its presentation at the postpubertal stage. The aim of this study is to describe the characteristics of paediatric patients diagnosed with PC at our centre and to compare them according to their pubertal status.
We included patients aged between 1 and 18 years who were diagnosed with PC in a tertiary-level hospital between 2006 and 2019 and who met the updated diagnostic criteria for PC. They were classified according to body weight and pubertal status. Subsequently, we analysed results from lumbar punctures, neuroimaging studies, ophthalmological assessments, and treatments received during follow-up.
We included 28 patients, of whom 22 were of prepubertal age and 6 were of postpubertal age. The mean age (standard deviation) was 9.04 (2.86) years. Among the postpubertal patients, 83.3% were boys, 66.7% of whom presented overweight/obesity. In the group of prepubertal patients, 27% were boys, 31.8% of whom were overweight. The most frequent symptoms were headache (89.9%) and blurred vision (42.9%). All patients presented papilloedema, and 21.4% manifested sixth nerve palsy. Possible triggers were identified in 28.6% of cases. Nineteen percent of patients presented clinical recurrence, all of whom were prepubertal patients. Complete clinical resolution was achieved in 55.6% of patients.
Prepubertal patients with PC show lower prevalence of obesity, higher prevalence of secondary aetiologies, and higher recurrence rates than postpubertal patients.
Patient-reported symptomatology and its course in spontaneous intracranial hypotension – Beware of a chameleon
2024, Clinical Neurology and Neurosurgery
Although orthostatic headache is the hallmark symptom of spontaneous intracranial hypotension (SIH), patients can present with a wide range of different complaints and thereby pose a diagnostic challenge for clinicians. Our aim was to describe and group the different symptoms associated with SIH and their course over time.
We retrospectively surveyed consecutive patients diagnosed and treated for SIH at our institution from January 2013 to May 2020 with a specifically designed questionnaire to find out about their symptomatology and its course.
Of 112 eligible patients, 79 (70.5%) returned the questionnaire and were included in the analysis. Of those, 67 (84.8%) reported initial orthostatic headaches, whereas 12 (15.2%) denied having this initial symptom. All except one (98.7%) patients reported additional symptoms: most frequently cephalic pressure (69.6%), neck pain (68.4%), auditory disturbances (59.5%), nausea (57%), visual disturbances (40.5%), gait disturbance (20.3%), confusion (10.1%) or sensorimotor deficits (21.5%). Fifty-seven (72.2%) patients reported a development of the initial symptoms predominantly in the first three months after symptom onset. Age and sex were not associated with the symptomatology or its course (p > 0.1).
Although characteristic of SIH, a relevant amount of patients present without orthostatic headaches. In addition, SIH can manifest with non-orthostatic headaches at disease onset or during the course of the disease. Most patients report a wide range of associated complaints. A high degree of suspicion is crucial for an early diagnosis and targeted treatment.
Super resolution imaging reconstruction reveals that gold standard methods may not correctly conclude neural/brain functional recovery
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The status of cerebral perfusion and its restoration level play a vital role in the prognosis and clinical decision making of many neurosurgical diseases. As such, gold standard methods including CT, MR and ICP monitoring, which can indicate and measure cerebral perfusion and restoration, have been widely adopted to evaluate whether or not a patient has recovered from neurofunctional disabilities. This robust combination of methods, however, is confronted with a growing number of contradictions in recent years due to its inability to measure the status of cerebral reperfusion in microvasculature level, even though this has been shown to determine neurofunctional restoration as well or even better. To this date, nevertheless, we have very limited imaging methods that could evaluate human cerebral microperfusion both safely and accurately under most neurosurgical conditions. We herein report a new method of acquiring a patient’s cerebral microperfusion status noninvasively which could display the precise distribution of microvasculature in deep cerebral regions with a resolution of ∼30 µm, using everyday bed-side ultrasonography combined with a computerized super-resolution reconstruction algorithm. Using this imaging modality, we found that a patient’s cerebral microperfusion might not be improved by some routine administrations even though the gold standard method had yielded the opposite conclusions. Our imaging modality retains the safe, portable feature of ordinary ultrasonography while possesses the extraordinary super-resolution nature, which enables an efficient, precise diagnosis of cerebral perfusion. Most importantly, the super resolution nature of this method may also facilitate early-stage evaluation of a patient’s neurofunctional restoration level and avoid overoptimistic conclusions from conventional angiography or ICP monitoring.
Headache: concept, diagnosis, alarms signs, and additional examinations
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Headache is a common cause for consultation in all levels of care and a cause of disability for patients who suffer from it. There are common pathophysiological mechanisms for the pain and other specific mechanisms for certain types of headache. Nociceptor neurons have projections in the cerebral cortex that associate headache with affective and cognitive components. The International Classification of Headache Disorders, a consensus document which is periodically updated, offers diagnostic criteria for the main types of headache. Primary headaches are fundamentally diagnosed via a medical history; it is recommendable for it to be structured. There are multiple causes of secondary headache, some of which are banal and others which are due to life-threatening causes. Both alarm and caution signs have been proposed that help guide the diagnostic process of a patient with headache. Alarm signs are classified as those derived from the patient's personal or family medical history, the headache's characteristics, and the findings of the physical examination. A suspicion of secondary headache makes additional tests necessary. In emergency care, it is important to take into account a diagnostic algorithm in patients with «thunderclap headache».
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We show in this case report a non-congenital sphenoethmoidal meningoencephalocele causing rhinoliquoral fistula and spontaneous intracranial hypotension.
this 65-year-old woman presented with sporadic rhinoliquorrhoea associated with orthostatic headache, nausea and dizziness. Brain MRI revealed a small lesion of an ethmoidal sinus, which was successfully treated with endoscopic endonasal surgery. Histology confirmed the presence of meningoencephalic tissue positive for S100 protein on immunohistochemistry.
When dealing with lesions of the paranasal sinuses in contact with the anterior skull base, rhinoliquorrhoea presence suggests meningoencephalocele. In dubious cases, a proper workup, including a thorough clinical history and neurological examination, specific imaging, and a direct search of CSF-like markers, is essential to support the differential diagnosis. In such cases, a transnasal endoscopic surgical approach is recommended to obtain a final histological diagnosis and to perform eventual dural plastic surgery.
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ReviewHeadache arising from idiopathic changes in CSF pressure

Summary

Introduction

Section snippets

Physiology of intracranial pressure and CSF

Pathological mechanisms of changes in CSF pressure

Suspected headache secondary to change in CSF pressure

Measurement of CSF pressure

Characterisation and classification

Demographics, predisposing factors, and associated disorders

Conclusions

Search strategy and selection criteria

Rev Neurol (Paris)

Clin Neurol Neurosurg

Neurol Clin

Handb Clin Neurol

Rev Neurol (Paris)

Lancet Diabetes Endocrinol

J Clin Neurosci

Clin Neurol Neurosurg

The international classification of headache disorders, 3rd edition (beta version)

Cephalalgia

Treatment of alterations in CSF dynamics

Neurol Sci

A new look at cerebrospinal fluid circulation

Fluids Barriers CNS

A mathematical model of idiopathic intracranial hypertension incorporating increased arterial inflow and variable venous outflow collapsibility

J Neurosurg

Idiopathic intracranial hypertension headache

Curr Pain Headache Rep

Pseudotumor cerebri pathophysiology

Headache

Oligoclonal bands and increased cytokine levels in idiopathic intracranial hypertension

Cephalalgia

Elevated intracranial venous pressure as a universal mechanism in pseudotumor cerebri of varying etiologies

Neurology

Cerebral venography and manometry in idiopathic intracranial hypertension

Neurology

Idiopathic intracranial hypertension: the prevalence and morphology of sinovenous stenosis

Neurology

Pseudotumor cerebri: brief review of clinical syndrome and imaging findings

AJNR Am J Neuroradiol

Clinical course of idiopathic intracranial hypertension with transverse sinus stenosis

Neurology

Spontaneous spinal cerebrospinal fluid leaks

Cephalalgia

Lack of causal association between spontaneous intracranial hypotension and cranial cerebrospinal fluid leaks

J Neurosurg

Spontaneous low pressure, low CSF volume headaches: spontaneous CSF leaks

Headache

Dynamic CT myelography: a technique for localizing high-flow spinal cerebrospinal fluid leaks

AJNR Am J Neuroradiol

Detection of cerebrospinal fluid leakage: initial experience with three-dimensional fast spin-echo magnetic resonance myelography

Acta Radiol

MRI with intrathecal gadolinium to detect a CSF leak: a prospective open-label cohort study

J Neurol Neurosurg Psychiatry

Orthostatic headaches without CSF leak in postural tachycardia syndrome

Neurology

Orthostatic headache without CSF leak

Neurology

Spontaneous intracranial hypotension syndrome: a novel speculative physiopathological hypothesis and a novel patch method in a series of 28 consecutive patients

J Neurosurg

Treatment of spontaneous intracranial hypotension: evolution of the therapeutic and diagnostic modalities

Neurol Sci

Thunderclap headache

BMJ

Nonmydriatic ocular fundus photography among headache patients in an emergency department

Neurology

Nonmydriatic retinal photography in the evaluation of acute neurologic conditions

Neurol Clin Pract

Cerebrospinal fluid pressure in adults

J Neuroophthalmol

Diagnostic criteria for idiopathic intracranial hypertension

Neurology

Revised diagnostic criteria for the pseudotumor cerebri syndrome in adults and children

Neurology

Interpretation of lumbar puncture opening pressure measurements in children

J Neuroophthalmol

Effect of patient positioning on cerebrospinal fluid opening pressure

J Neuroophthalmol

Review
Headache arising from idiopathic changes in CSF pressure