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Headache and Inflammatory Disorders of the Central Nervous System

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Headache and Inflammatory Disorders of the Central Nervous System Neurol Sci (2004) 25:S148–S153 DOI 10.1007/s10072-004-0275-7 HEADACHES ATTRIBUTED TO NON-VASCULAR INTRACRANIAL DISORDERS L. La Mantia • A. Erbetta Headache and inflammatory disorders of the central nervous system Abstract The subcommittee of the International Headache Introduction Society for headache classification (ICHD-II) has recently recognised that secondary headaches may occur in patients The subcommittee of the International Headache Society affected by inflammatory diseases (ID) of the central ner- for headache classification (ICHD-II) has recently under- vous system (CNS), classifying them among the headaches lined that secondary headaches may occur in patients attributed to non-vascular intracranial disorders. The aim of affected by inflammatory diseases (ID) of the central ner- the study was to verify the association between headache vous system (CNS). Secondary headaches are recognised in and inflammatory non-infectious diseases of the CNS, by a 4 main non-infectious IDs: neurosarcoidosis (sec 7.3.1), review of the literature data on the topic, integrated by per- aseptic (non-infectious) meningitis (7.3.2), other non-infec- sonal cases and data. Secondary headaches may occur in tious ID (7.3.3) and lymphocytic hypophysitis (7.3.4) [1]. four main disorders: neurosarcoidosis (sec 7.3.1), aseptic The aim of the study was to verify the association (non-infectious) meningitis (7.3.2), other non-infectious ID between headache and ID of the CNS, by a review of the (7.3.3) and lymphocytic hypophysitis (7.3.4). Headache literature data on the topic, with the focus on neurosar- and/or primary headaches are frequently reported in coidosis, Behçet’s syndrome (BS), acute disseminated patients with neurosarcoidosis (30%), Behçet’s syndrome encephalomyelitis (ADEM) and multiple sclerosis (MS). (BS) (55%) and acute disseminated encephalomyelitis (45–58%). Recent data show a high incidence of headache also in multiple sclerosis (MS) (58%) (not mentioned in ICHD-II). The association between headache and inflam- Methods matory dysimmune diseases of the CNS, in particular BS and MS, might suggest a pathogenetic relationship. References were retrieved by electronic data base using the fol- lowing key words: headache, migraine, tension headache, sar- Key words Headache • Inflammatory disease • Central ner- coidosis, Behçet syndrome, ADEM and MS. The literature data were analysed in terms of incidence and characteristics of vous system • Neurosarcoidosis • Behçet’s syndrome • headache and integrated by personal cases. The review was per- Multiple sclerosis formed following the ICHD-II classification. Results of the review on secondary headache to CNS inflammatory non-infectious diseases L. La Mantia (౧) Headache attributed to neurosarcoidosis MS Centre National Neurological Institute “C. Besta” Sarcoidosis (NS) is a multisystem granulomatous disease Via Celoria 11, I-20133 Milan, Italy of unknown cause, most commonly affecting young adults. e-mail: [email protected] Involvement of the CNS is clinically evident in 5% and A. Erbetta silent in 10% of the cases with systemic sarcoidosis: it may Neuroradiological Department occur at presentation in 10–30% of patients and more rarely National Neurological Institute “C. Besta”, Milan, Italy is strictly confined to the CNS. Sarcoid lesions are non- L. La Mantia, A. Erbetta: Headache in inflammatory diseases of the CNS S149 caseating epithelioid granulomas. Intracranial lesions, Table 1 Neurosarcoidosis. Clinical signs and symptoms of CNS [3] detectable by magnetic resonance imaging (MRI), appear as nodular or diffuse leptomeningeal thickening, mimicking Symptoms Percentage of affected cases various forms of meningitides, neoplastic seeding including carcinomatous and/or intra-parenchymatous lesions. MRI Cranial nerve palsies 50 Headache 30 findings have been included in the diagnostic criteria [2]. Seizures 10 Pituitary dysfunction 10 Clinical aspects with focus on headache Sensory and motor deficits 10 Neuropsychological deficits 10 Cerebellar symptoms 10 Any part of the CNS may be involved, resulting in a wide spec- Hydrocephalus 5 trum of clinical symptoms: headache is one of the most com- Meningitis 5 mon neurological symptoms (see Table 1) [3]. Different kinds of headache are likely to occur, in relation to neuropathologic involvement: focal lesions, meningitis, cranial nerve palsies. to trigeminal or optic nerve involvement. Migraine has been No typical characteristics of headache are known: however, also reported [7]. detailed information on these aspects is occasionally reported in the published studies. Occipital radiating frontally Case report intractable headache with nausea and visual disturbances have been reported in patients with isolated supratentorial tumour- A 56-year-old woman came to our observation for a painful like lesions [4]. Diffuse or bifrontal pain is a more typical paresis of the third right cranial nerve, neuropapillitis and tran- symptom of leptomeningeal involvement, associated or not to sient trigeminal neuralgia on the right side. MRI showed a papilledema [5, 6]. Other forms of cranial pain may be related granulomatous lesion of the right cavernous sinus (Fig. 1). a bc d e Fig. 1a–e Axial T1-weighted images (a, b) after contrast medium administration (c, d). The inflammatory process fills the right cavernous sinus. The enlarged cavernous sinus contains abnormal soft tissue isointense to grey matter (a, b), and enhancing after contrast medium administration (c, d). The CT scan shows abnormal soft tissue density in the orbital apex and gives more informa- tion about the actual extension of the granuloma (e). These features are com- monly seen in meningioma or lym- phoma too. Clinical history allows fur- ther precision in differential diagnosis S150 L. La Mantia, A. Erbetta: Headache in inflammatory diseases of the CNS Steroid treatment allowed pain remission within 3 months, but tions. Autoimmune response to myelin basic protein trig- without effect on the neurological deficits. MRI showed a sig- gered by infection or immunisation is considered to be a nificant reduction of the lesion after therapy. The diagnostic possible aetiologic factor. The diagnosis is based on clini- work-up (angiotensin converting enzyme level, chest CT scan, cal, MRI and CSF findings [8]. BAL, gallium scintigraphy) allowed the diagnosis of probable neurosarcoidosis [2]. Clinical aspects with focus on headache Clinical features include sudden onset of multifocal neuro- logical disturbances such as bilateral optic neuritis, aphasia, motor and sensory deficits, ataxia, movement disorders and Headache attributed to other non-infectious inflammatory signs of acute meningoencephalomyelopathy with diseases meningism, decreased level of consciousness and seizures. Headache is a frequent symptom at onset, occurring in Headache can be associated with but is not usually a pre- 45–58% of cases [9, 10]: it is usually severe, worsening with- senting or dominant symptom of systemic lupus erythe- in a few days, associated with vomiting, photophobia and matosus (SLE), antiphospholipid antibody syndrome, Vogt- meningism. Koyanagi-Harada syndrome [1]. The headaches reported in demyelinating disorders of the CNS, such as ADEM, BS Case report and MS, have been analysed in the following sections. This 22-year-old woman was affected, 7 months before her hospitalisation in our institute, by an acute episode charac- Acute disseminated encephalomyelitis terised by headache, confusional state and ataxia. Brain MRI revealed disseminated encephalomyelitis (Fig. 2), confirmed ADEM is an uncommon, acute, demyelinating disorder of by cerebral biopsy. She was treated with high dosages of the CNS, typically occurring after infections or vaccina- steroids with partial recovery. She complained of persistent a b c d Fig. 2a–d Axial proton-density images (a, b) show large and scattered white matter lesions involving both hemispheres. Dense and irregular enhancement of por- tions of lesions is evident on T1-weighted images after gadolinium (c, d). Although there are large lesions, the mass effect is not prominent L. La Mantia, A. Erbetta: Headache in inflammatory diseases of the CNS S151 frontal headache (chronic post-intracranial disorder Headache is a frequent manifestation of the disease, its headache). CSF examination was within normal limits. incidence ranging from 4 to 64% [12–16] (Table 2). It is the most common initial symptom of favourable prognostic Behçet’s syndrome value [15]. According to Evans [17], different types of headache may occur in BS, as in the general population, as BS is a chronic multisystem T-cell mediated disorder, associ- related to specific neurological involvement and also to ocu- ated with HLA–B5 and B 51 antigens; vasculitis is the under- lar manifestations. lying pathogenetic process, which can be seen in the retina, brain and subcutaneous tissue. In 1990 the International Study Case report Group for BS published the diagnostic criteria [11]. This 47-year-old woman was affected by right uveitis asso- ciated with fronto orbital pain, and by oral and genital Clinical aspects with focus on headache ulcers from 7 years. She complained of recurrent episodes Neurological involvement is reported in 22–50% of the cases of paraparesis and parestesias of the legs, gradually wors- (NeuroBehçet). Three main patterns have been identified: 1. ening from 2 years. CSF revealed oligoclonal bands. Brain Parenchymal CNS (65%) (brain stem syndrome, diffuse and cervical spinal cord MRI showed demyelinating
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