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Letter to the Editor
Spontaneous spinal epidural haematoma and nonaneurysmal subarachnoid haemorrhage in a patient with eosinophilic granulomatosis with polyangiitis
Hematoma epidural espinal espontáneo y hemorragia subaracnoidea no aneurismática en paciente con granulomatosis eosinofílica con poliangitis
A. Lázaro Romero
Corresponding author
albalazaroromero@gmail.com

Corresponding author.
, A. Carilla Sanromán, L. Horna Cañete, M. Serrano Ponz
Hospital Universitario Miguel Servet, Zaragoza, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Eosinophilic granulomatosis with polyangiitis &#40;EGPA&#41; is a type of systemic necrotising vasculitis affecting small- and medium-sized vessels&#44; which belongs to the spectrum of anti-neutrophil cytoplasmic antibody &#40;ANCA&#41;&#8211;associated vasculitis&#46; Disease progression presents 3 phases&#44; which may or may not be sequential&#58; an allergic phase &#40;asthma&#44; rhinitis&#44; and nasal polyposis&#41;&#44; an eosinophilic phase &#40;eosinophilia in peripheral blood or lung or gastrointestinal tissues&#41;&#44; and a vasculitic phase&#44; which may involve multiple organs&#44; including the peripheral and&#44; rarely&#44; the central nervous systems &#40;CNS&#41;&#46; In 1990&#44; the American College of Rheumatology created a set of diagnostic criteria &#40;asthma&#44; eosinophilia &#62; 10&#37; in peripheral blood&#44; mono- or polyneuropathy&#44; non-fixed pulmonary infiltrates&#44; paranasal sinus abnormalities&#44; and extravascular eosinophilia&#41;&#44; with diagnosis being established in patients meeting 4 of the 6 criteria&#59; these criteria have sensitivity of 85&#37; and specificity of 99&#46;7&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Treatment includes corticosteroids and such immunosuppressants as cyclophosphamide in refractory cases&#46; Current series suggest a clear improvement in survival rates&#44; increasing from 70&#37; to 90&#37; at 5 years&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Cardiac involvement is the main cause of death in patients with EGPA&#44; followed by brain haemorrhage&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 54-year-old man diagnosed with EGPA due to history of severe cortico-dependent asthma&#44; paranasal polyposis&#44; eosinophilic dermatitis&#44; peripheral eosinophilia&#44; and recently diagnosed axonal sensory polyneuropathy&#46; He was treated exclusively with prednisone&#44; dosed at 5&#160;mg&#47;day&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was assessed at the emergency department due to a 48-h history of spontaneous&#44; sudden-onset low back pain&#44; developing into progressive weakness of the left leg&#44; paraesthesia in the left thigh&#44; pollakiuria&#44; and constipation&#46; Physical examination identified no fever&#44; and arterial blood pressure was 157&#47;111&#160;mm Hg&#46; Neurological examination revealed proximal weakness &#40;3&#47;5&#41; in the left leg&#44; hypoaesthesia at the L1 level&#44; exaggerated left patellar and Achilles reflexes&#44; and left Babinski sign&#46; At baseline&#44; laboratory results &#40;glycaemia&#44; ions&#44; liver and kidney function&#41; and coagulation were normal&#46; A complete blood count revealed leukocytosis &#40;20&#160;000 cells&#47;&#956;L&#59; normal range&#58; &#60; 10&#160;200&#41; and eosinophilia &#40;1300&#160;cells&#47;&#956;L &#91;14&#46;8&#37;&#93;&#59; normal range&#44; 500 &#91;&#60; 5&#37; of total&#93;&#41;&#46; A chest radiography displayed no abnormalities&#46; A thoracolumbar MRI study &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41; identified epidural haemorrhages at the T8-L1 and S1-S2 levels&#59; emergency T12-L1 laminectomy and evacuation of the haemorrhages were performed&#46; A spinal cord arteriography performed 24&#160;hours after the procedure revealed normal findings&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">On the third day after admission&#44; the patient presented a sudden decrease in the level of consciousness&#59; a head CT scan &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41; detected a spontaneous subarachnoid haemorrhage &#40;SAH&#41;&#44; classed as grade IV on the Fisher scale&#46; A subsequent CT angiography identified no relevant alterations&#46; A brain arteriography performed 36&#160;hours later yielded normal findings&#46; The patient died on day 6 after admission&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Our patient meets diagnostic criteria for EGPA of 3 years of progression&#59; given the recent diagnosis of polyneuropathy&#44; it may be classified as being in the vasculitic phase&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">CNS involvement is reported in 6&#37;-10&#37; of cases of EGPA&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> with ischaemic stroke and brain haemorrhages being particularly common&#59; multiple strokes may occur&#44; and stroke may be the first sign of the disease&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#8211;12</span></a> Spinal haemorrhages are much rarer&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13&#44;14</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The largest series of haemorrhagic complications involving the CNS in patients with EGPA is probably that reported by Sabio et al&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Of 28 patients diagnosed with EGPA&#44; 14 &#40;50&#37;&#41; presented spontaneous SAH&#44; 13 &#40;46&#37;&#41; presented intraparenchymal haemorrhage&#44; 5 &#40;17&#46;9&#37;&#41; presented intraventricular haemorrhage&#44; and 3 &#40;10&#46;7&#37;&#41; presented spinal haematomas&#46; Ross et al&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> report 6 cases of EGPA with spinal complications&#44; including 4 with subdural or subarachnoid haemorrhage&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Our patient initially presented a spontaneous spinal epidural haematoma &#40;SSEH&#41;&#44; as observed in the neuroimaging study and confirmed during surgery&#44; and subsequently a spontaneous SAH&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">It has been suggested that spontaneous SAH in the context of EGPA may be explained both by vasculitis&#44; with disruption of the internal elastic lamina&#44; and by rupture of the post-stenotic dilatation caused by granuloma formation in small vessels&#46; The cause of SSEH is undetermined in 40&#37; of cases&#46; Some authors propose that it may be caused by venous plexus involvement&#44; whereas others consider it to originate in the epidural arteries&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In our patient&#44; the fact that SSEH occurred in the context of EGPA&#44; almost simultaneously with spontaneous SAH&#44; suggests vasculitis as the most probable cause&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a> To our knowledge&#44; this is the first case of SSEH in a patient with EGPA&#46; The simultaneous presentation of spontaneous SAH in the brain is also unusual&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0055" class="elsevierStylePara elsevierViewall">This study has received no specific funding from any public&#44; commercial&#44; or non-profit organisation&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Consent</span><p id="par0060" class="elsevierStylePara elsevierViewall">Consent was not obtained from the patient due to his death&#46; The final author certifies the effort made to search for family contacts and the complete anonymisation of patient data&#46;</p></span></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A&#41; T1-weighted MRI study of the spine &#40;sagittal plane&#41;&#44; showing epidural haemorrhage of the thoracolumbar spine&#46; Loculi with greater posterior blood pooling are observed at the T12-L1 and S1-S2 levels&#46; The morphology of the spinal cord is normal&#46; B&#41; Non-contrast head CT scan showing perimesencephalic&#44; intraventricular&#44; and cerebellar subarachnoid haemorrhage &#40;Fisher grade IV&#41;&#46;</p>"
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Article information
ISSN: 21735808
Original language: English
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