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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Spontaneous intracranial hypotension &#40;SIH&#41; is an increasingly recognized secondary headache syndrome&#46; It is caused by an occult leakage of CSF at the spinal level with loss of buoyancy effect and secondary descent&#47;stretching of upper nerve&#47;vascular structures&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#8211;5</span></a> The typical SIH-patient presents with orthostatic headache&#44; unequivocal findings on imaging and an eventually benign course with positive response to epidural autologous blood patch -EBP- usually into the lumbar space&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#8211;4&#44;6</span></a> There are&#44; conversely&#44; challenging cases with misleading clinico&#47;radiological features&#44; frustratingly refractory to conventional treatment requiring advanced imaging along with targeted therapies&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#44;4&#44;6&#8211;9</span></a> We report two SIH cases whose description is merged with the relative literature to offer a practical management paradigm&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Description of cases</span><p id="par0010" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">&#35;1</span>&#58; 40 y&#47;o woman&#46; Previous mild spinal trauma &#40;fall on back&#41;&#46; Worsening postural headache and tinnitus over two weeks&#46; Cranial CT&#58; bilateral SDH&#46; Brain MRI&#58; consistent with SIH&#46; Spinal MRI&#58; sacral meningeal pouches adjacent to an anterior CSF collection at S4&#46; An EBP &#40;18<span class="elsevierStyleHsp" style=""></span>ml blood<span class="elsevierStyleHsp" style=""></span>&#43;<span class="elsevierStyleHsp" style=""></span>4<span class="elsevierStyleHsp" style=""></span>ml iodinated contrast&#44; L4&#8211;L5 access&#41; was performed&#46; Post-procedural CT confirmed distribution of contrast medium at the expected leakage site&#46; The headache responded promptly&#46; Follow-up&#58; gradual resorption of subdural hematomas&#44; disappearance of leakage &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">&#35;2</span>&#58; 71 y&#47;o man&#46; Back pain after overstraining&#44; followed by persistent non-positional headache&#46; Cranial MRI&#58; bilateral SDH plus features of SIH&#46; Spinal MRI&#58; subdural blood with L5&#8211;S2 meningeal diverticula showing blood-fluid level&#46; Upon the hypothesis that CSF pooling at pouches could have caused SIH at first stage with blood acting as &#8220;spontaneous&#8221; patching only surgical evacuation of SDH was undertaken&#46; The headache recurred in two weeks along with subdural collections&#46; EBP was then performed &#40;L3&#8211;L4 access&#44; &#8776;40<span class="elsevierStyleHsp" style=""></span>ml blood&#41; followed by disappearance of orthostatic headache &#40;3&#8211;4 days&#41; and gradual resorption of SDH over two months &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">SIH is an increasingly recognized cause of secondary atypical headache&#46; Estimated annual incidence is 5&#47;100&#44;000 with female prevalence &#40;2&#58;1&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#44;3&#44;4</span></a> The most credited causative mechanism is an occult&#44; spontaneous CSF leak at the spinal level&#46; The impaired cushion effect leads to descent and sagging of cranial nervous elements&#44; stretching of pain-sensitive and vascular structures and venous congestion&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#8211;3</span></a> While such pathogenesis is largely recognized&#44; the anatomical substrate is less clear&#46; An underlying weakness of spinal meninges is suspected&#44; possibly related to connective tissue disorders&#46; Such fragility predisposes to defects&#8217; genesis &#40;tears&#44; pouches&#41; with CSF egress occurring spontaneously or after even trivial trauma &#40;about 1&#47;3 patients report mild accidents&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#8211;3</span></a> Alternative mechanisms have been recently advocated&#58; dural tears by bony spurs and direct CSF-venous fistulas&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">4&#44;5</span></a> Orthostatic headache is the hallmark of SIH&#46; With time&#44; however&#44; it may become chronic or even turn paradoxical &#40;worsened by recumbency&#41;&#46; Additional symptoms include&#58; neck pain&#47;stiffness&#44; hearing and visual disturbances&#44; facial pain and weakness&#44; pituitary dysfunction&#46; Severe brain displacement may lead to downward herniation and coma&#46; CSF opening pressure can be low or normal as well so&#44; once clinically suspected&#44; the diagnosis relies heavily on imaging&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#8211;4</span></a> Gd-brain MRI displays characteristic findings named after Schievink&#58; <span class="elsevierStyleItalic">subdural fluid collections&#44; enhancement of pachymeninges&#44; engorgement of veins&#44; pitutitary hyperemia and sagging of brain</span> &#40;acronym&#58; SEEPS&#41;&#46; Such features grossly reflect compensatory changes to CSF depletion&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">1&#8211;3&#44;7&#44;9</span></a> Dural enhancement is by far the most common &#40;&#8776;80&#37;&#41;&#46; Subdural hematomas occur in 20&#8211;45&#37; of SIH cases&#44; either &#8220;ab initio&#8221; or in the course of disease&#46; On the other hand&#44; they are common in the neurosurgical non-SIH population&#46; Clinical data &#40;headache is positional rather than continuous&#44; absent trauma history&#41;&#44; meningeal enhancement&#44; tendency to recur may direct diagnosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">10&#44;11</span></a> Noticeably&#44; cranial MRI findings may be incomplete or absent in up to 28&#37; of patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">4&#44;7&#44;8</span></a> As SIH is related to CSF leakage in the vertebral column&#44; spinal imaging follows&#46; MRI findings include epidural CSF collections &#40;most suggestive of SIH but often non-focal&#41;&#44; dilated epidural veins&#44; dural enhancement&#44; &#8220;falsely-localizing&#8221; CSF collections at C1-C2&#44; Chiari-like features and meningeal diverticula &#40;often multiple&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#8211;5&#44;7&#44;8&#44;10</span></a> These latter deserve additional consideration&#58; they have been shown to occur equally in healthy and SIH patients&#46; In a CT-myelography study&#44; Kranz et al&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">12</span></a> found no significant differences between two groups in terms of number&#44; size&#47;morphology and distribution of spinal diverticula&#46; Among 568 SIH patients undergoing advanced spinal imaging<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">5</span></a> meningeal diverticula were found in &#8776;42&#37; but in only &#8776;20&#37; could a leak be demonstrated&#46; Although larger&#44; irregular pouches might contribute to SIH by CSF pooling rather than leakage&#44; there&#39;s actually no evidence to support at first stage targeting therapies to diverticula unless direct leakage is demonstrated&#46;<a class="elsevierStyleCrossRefs" href="#bib0105"><span class="elsevierStyleSup">5&#44;12</span></a> Heavily T2-weighted sequences &#40;myelo-MRI&#41; can highlight the egress point in some cases&#46; Nevertheless&#44; spinal MRI&#44; although non-invasive and avoiding radiation exposure&#44; is rarely sufficient to identify the exact leakage site and rather considered a corroborate to brain imaging&#46;<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">7&#8211;9</span></a> To precisely localize the leakage site advanced imaging is warranted&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#44;6&#8211;9</span></a> There are no clear guidelines as to the best diagnostic modality&#58; CT-myelography has been for long &#8220;gold standard&#8221;&#58; most of leaks are visualized at cervical and thoracic spine&#46; Rarely CSF-venous fistulas can be visualized as well&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">4&#44;9</span></a> MRI-myelography &#40;after off-label intrathecal gadolinium&#41; has emerged as even more sensitive&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">2&#44;3&#44;7&#44;9&#44;13</span></a> Delayed imaging may be needed to highlight intermittent&#47;small volume leaks while rapid&#44; high-volume flows are best discovered by dynamic&#44; ultrafast CT-myelography or digital subtraction myelography &#40;DSM&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#44;4&#44;7&#44;9</span></a><span class="elsevierStyleSup">111</span>In-DTPA radionuclide cisternography may provide over-time &#40;up to 48<span class="elsevierStyleHsp" style=""></span>h&#41; direct and mostly indirect leakage visualization but its specificity and localizing value are questioned&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">4&#44;7&#44;9&#44;14</span></a> A review of investigation techniques and selection criteria has been offered by Kranz et al&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">9</span></a> After the diagnosis is confirmed a screening for connective tissue disorders is suggested&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> Many SIH cases follow a benign course&#44; a small subset &#40;8&#37;&#41; is self-limiting or responds to conservative measures &#40;bed rest&#44; hydration&#44; caffeine&#44; steroids&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">10</span></a> The injection of autologous blood into the epidural space is the mainstay of treatment&#46; The early effect is perhaps through volume replacement&#44; while epidural scarring and sealing of defects account for long-term results&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">2&#8211;4&#44;6&#44;8</span></a> When not pointed at the CSF egress site&#44; the procedure is usually performed at the lumbar level &#40;<span class="elsevierStyleItalic">blind EBP</span>&#41;&#58; yet&#44; a 36&#8211;90&#37; success rate may be expected &#40;directly correlating to blood volume and cumulative&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">6&#44;10&#44;11</span></a><span class="elsevierStyleItalic">Targeted</span> EBP is aimed to the leakage site thus involving its identification with advanced imaging&#46; Cho et al&#46;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">15</span></a> found targeted EBP more effective than blind after first injection &#40;87&#37; vs 52&#37; respectively&#41;&#46; However&#44; it may also be more demanding particularly when the target is in the upper spine &#40;cord compression&#44; limited blood volume&#44; use of radiological guidance&#41;&#46; The need for invasive imaging in order to precisely localize the leakage site should not be overlooked&#46; Conversely&#44; blind EBP takes advantage of a wider and safer lumbar epidural space and of blood spreading over multiple levels from the site of injection&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">6&#44;10&#44;11&#44;15&#44;16</span></a> Fibrin glue percutaneous patch and direct surgical repair &#40;clipping&#47;ligation of meningeal pouches&#44; repairing of rents along with removal of bony spurs&#41; are additional options of proven efficacy<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#44;5&#44;6&#44;8&#44;9&#44;16</span></a> once the leakage is localized&#46; Evacuation of intracranial subdural collections is advised in selected cases &#40;life threatening hematomas&#44; persistently symptomatic&#41; but generally fruitless as long as the underlying SIH is not addressed &#40;as in our case 2&#41;&#46; Additionally&#44; many subdural collections resolve spontaneously once the leakage is sealed&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">4&#44;10&#44;11</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">It must be underlined that a non-negligible percentage of patients may escape leakage detection even after exhaustive investigation&#58; EBP empirically aimed at larger dural pouches or via upper thoracic route and surgical exploration may be beneficial&#46;<a class="elsevierStyleCrossRefs" href="#bib0125"><span class="elsevierStyleSup">9&#44;15</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">A widely accepted algorithm for the management of SIH is still lacking&#46; A detailed literature review is beyond the scope of this paper but&#44; as an example&#44; features of recently reported cases are summarized in <a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#46; It can be speculated that the diversity of approaches reflects local experience and resources&#8217; availability&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">We tried to draw a stepwise management protocol that can be summarized as follows&#46; At first-tier SIH patients would receive non-invasive spinal imaging&#44; conservative measures and <span class="elsevierStyleItalic">blind</span> EBP &#40;targeted patch just in case of undoubtful leakage identification&#41;&#46; If the first injection is unsuccessful&#44; a repeated EBP with larger blood volume &#40;targeting a leak presumably too large or rostral to seal&#41; or via upper thoracic route should be considered&#46; Unremitting cases require second tier therapies &#40;targeted EBP and&#47;or fibrin glue injection&#44; direct surgical repair&#41; therefore involving advanced spinal imaging<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">2&#8211;4&#44;6&#44;9&#8211;11&#44;15&#44;16</span></a> &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">After a successful treatment&#44; some patients may experience an effect known as rebound intracranial hypertension &#40;headache exacerbated by lying down&#41; usually not severe and self-limiting&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#44;4</span></a> Recurrence of symptoms may be anticipated in probably less than 10&#37; cases&#58; an alternative mechanism &#40;i&#46;e&#46; undetected CSF-venous fistulas&#41; or different diagnosis should receive consideration&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0045" class="elsevierStylePara elsevierViewall">SIH is presumably still underdiagnosed and a greater awareness about this syndrome is the first step to proper recognition and timely management&#46; Since the course is often benign&#44; it can be suggested that patients might not undergo advanced spinal imaging and receive blind EBP as initial treatment&#46; Should this approach fail to provide relief&#44; second tier imaging and targeted therapies would get into the game&#46;</p></span></span>"
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            0 => "Cefalea"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Spontaneous intracranial hypotension is an increasingly recognized cause of atypical&#44; secondary headaches&#46; Nevertheless&#44; its clinical and imaging spectrum is far from an exhaustive definition&#44; ranging from straightforward cases with unambiguous findings and prompt response to treatment to more challenging ones&#44; requiring advanced&#44; more complex imaging and targeted therapies&#46; We describe two unusual cases as a cue to draw a literature-based&#44; practical approach to the management of the syndrome&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La hipotensi&#243;n intracraneal espont&#225;nea es cada vez m&#225;s reconocida como causa de cefalea secundaria at&#237;pica&#46; Su espectro cl&#237;nico y de im&#225;genes est&#225; lejos de ser exhaustivamente definido&#44; y var&#237;a desde casos simples con hallazgos inequ&#237;vocos y pronta respuesta al tratamiento a los m&#225;s desafiantes&#44; que requieren im&#225;genes avanzadas y terapias dirigidas&#46; Describimos 2 casos como una se&#241;al para dibujar un enfoque pr&#225;ctico basado en la literatura para el manejo de este s&#237;ndrome&#46;</p></span>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Case &#35;1&#58; imaging timelapse&#58; &#40;a&#8211;c&#41; STIR-MRI&#44; coronal views highlight multiple meningeal diverticula at the sacral level with a pre-vertebral CSF collection &#40;arrow&#41;&#46; &#40;d&#44; e&#41; CT-guided EBP&#58; contrast medium can be traced to the expected leakage site &#40;asterisk&#41;&#46; &#40;f&#41; Follow-up MRI &#40;STIR&#44; coronal view&#41;&#58; disappearance of CSF collection&#46;</p>"
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">A proposed algorithm for SIH management &#40;EBP<span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>epidural blood patch&#59; SDH<span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>subdural hematoma&#59; Y&#47;N<span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>yes&#47;no&#41;&#46;</p>"
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Case Report
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Autor para correspondencia
, Giorgio Lofrese, Maria Teresa Nasi
Neurosurgery Department, M. Bufalini Hospital, v.le Ghirotti 286, 47521 Cesena (FC), Italy

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