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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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Vol. 30. Núm. 5.
Páginas 228-232 (septiembre - octubre 2019)
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Vol. 30. Núm. 5.
Páginas 228-232 (septiembre - octubre 2019)
Case Report
Spontaneous intracranial hypotension due to sacral diverticula: Two-case history and a pocket-sized review
Hipotensión intracraneal espontánea por divertículos sacros: historia de dos casos y revisión de bolsillo
Visitas
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Francesco Cultrera
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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Abstract

Spontaneous intracranial hypotension is an increasingly recognized cause of atypical, secondary headaches. Nevertheless, its clinical and imaging spectrum is far from an exhaustive definition, ranging from straightforward cases with unambiguous findings and prompt response to treatment to more challenging ones, requiring advanced, more complex imaging and targeted therapies. We describe two unusual cases as a cue to draw a literature-based, practical approach to the management of the syndrome.

Keywords:
Headache
Spontaneous intracranial hypotension
CSF leak
Imaging
Epidural blood patch-management
Resumen

La hipotensión intracraneal espontánea es cada vez más reconocida como causa de cefalea secundaria atípica. Su espectro clínico y de imágenes está lejos de ser exhaustivamente definido, y varía desde casos simples con hallazgos inequívocos y pronta respuesta al tratamiento a los más desafiantes, que requieren imágenes avanzadas y terapias dirigidas. Describimos 2 casos como una señal para dibujar un enfoque práctico basado en la literatura para el manejo de este síndrome.

Palabras clave:
Cefalea
Hipotensión intracraneal espontánea
Fuga de fluido cerebroespinal
Imágenes
Parche de sangre epidural

Artículo

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