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Case report
Extensive extradural pneumorrhachis related to obstetric analgesia
Neumorraquis extradural extenso relacionado con analgesia obstétrica
M.J. Garcia-Cebriána, I.M. Fontan-Atalayab, J. Garcia-Pereza, B. Fernandez-Torresa,c,
Corresponding author
bartolome@us.es

Corresponding author.
a Servicio de Anestesiología y Reanimación, Hospital Virgen Macarena, Seville, Spain
b Servicio de Obstetricia y Ginecología, Hospital Virgen Macarena, Seville, Spain
c Departamento de Cirugía, Facultad de Medicina, Universidad de Seville, Spain
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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">Pneumorrachis is a rare phenomenon defined as the presence of intraspinal air&#46; From a descriptive&#44; clinical&#44; and therapeutic perspective&#44; it is important to differentiate between intradural &#40;subdural or subarachnoid&#41; and extradural &#40;epidural&#41; pneumorrachis&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Aetiologically&#44; pneumorrachis can be classified as traumatic&#44; iatrogenic&#44; and non-traumatic &#40;also called spontaneous&#41;&#46; Most published cases involve traumatic pneumorrachis due to injury to the head&#44; neck&#44; thoracic&#44; lumbar spine&#44; or pelvis&#46; Non-traumatic pneumorrachis has been attributed to respiratory complications &#40;COVID-19&#41; and increased intrathoracic pressure &#40;vomiting&#44; Valsalva&#44; asthmatic crisis&#44; positive pressure ventilation&#44; drugs&#41;&#44; and also to a number of other conditions&#44; such as pneumothorax&#44; abscesses&#44; tumours&#44; foreign bodies&#44; radiotherapy&#44; intestinal perforation&#44; exercise&#44; and cardiopulmonary resuscitation&#46; Although iatrogenic causes are associated with anaesthesia procedures &#40;neuraxial anaesthesia&#44; nasotracheal intubation&#44; blood patch&#41;&#44; they have also been described in the setting of spinal surgery&#44; pulmonary surgery&#44; or insertion of chest tubes&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Two systematic reviews on spontaneous<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and traumatic<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> pneumorrachis have recently been published&#44; but to our knowledge no reviews on iatrogenic pneumorrachis have yet been published&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">We report the case of an obstetric patient with very extensive&#44; large-volume extradural pneumorrachis diagnosed in the immediate postpartum period due to the appearance of pain with atypical characteristics&#46; In this report&#44; we aim to establish the clinical-radiological relationship and define the most appropriate diagnostic&#44; preventive&#44; and therapeutic measures for extradural pneumorrachis&#46; Written informed consent was obtained from the patient for the publication of this case report and radiological images&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 37-year-old woman&#44; height 165&#160;cm&#44; weight 80&#160;kg &#40;BMI 29&#46;4&#160;kg&#47;m<span class="elsevierStyleSup">2</span>&#41;&#44; ASA II&#44; with a history of caesarean section&#44; was admitted for labour&#46; This was her second full-term pregnancy&#46; The anaesthesiology service was called at 6 a&#46;m&#46; for obstetric analgesia&#44; and after assessing the risks and benefits&#44; decided to administer epidural analgesia&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">To identify the epidural space&#44; the anaesthesiologist&#44; on one of his first shifts after qualifying as a specialist&#44; initially used the loss of resistance technique with saline&#44; trying both the medial and lateral approaches at various levels&#46; Being unable to locate the space&#44; he switched to the air technique&#44; and after several attempts was able to place the catheter 7&#160;cm from the skin at L3-L4&#46; He did not consider performing the technique under ultrasound guidance or consulting with an on-call anaesthesiologists&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">After administration of a test dose &#40;10&#160;mg 0&#46;25&#37; bupivacaine&#41; and the initial dose &#40;7&#46;5&#160;mg 0&#46;125&#37; bupivacaine&#41;&#44; at the anaesthesiologist&#8217;s discretion&#44; the patient was given a PCEA for continuous infusion of 0&#46;0625&#37; bupivacaine&#160;&#43;&#160;fentanyl 1&#160;&#181;g&#47;mL at 8&#160;ml&#47;h&#44; with 5&#160;ml boluses and a lock out time of 20&#160;min&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Analgesia was satisfactory&#44; and after 14&#160;h the patient underwent instrumental delivery with forceps and suture of vaginal tears with good pain management&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">The epidural catheter was removed in the immediate postpartum period and the patient was transferred to the ward&#46; Three hours after delivery&#44; she was evaluated by the gynaecologist for intense pain in the perineum and hypogastrium that did not respond to analgesic treatment&#46; As the gynaecological examination and abdominal and transvaginal ultrasound had been normal&#44; we requested an urgent CT angiography scan to rule out bleeding not evidenced in the tests performed&#44; given the lack of availability of emergency MRI in our hospital&#46; The report from the radiology department did not mention any bleeds&#44; but described &#34;air bubbles along the right margin of the entire spinal canal in the epidural space&#44; from D9 &#40;first vertebra included in the study&#41; to the right hemisacrum&#34;&#44; &#8220;a bubble in the posterior mediastinum adjacent to the descending aorta&#8221;&#44; and &#8220;bubbles extending adjacent to the iliopsoas and obturator internus muscules and into the right paravertebral muscle&#8221; &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1&#8211;3</a>&#41;&#46; The gynaecology service contacted the duty anaesthesiologist&#44; who verbally ordered a wait and see approach&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">The patient remained hospitalized due to persistent pain in the perineal area&#44; and 3 days after performing the CT angiography scan the on-duty anaesthesiology team were called again to assess the clinical and radiological correlation&#46; The patient denied headache&#44; low back pain&#44; pain in the lower limbs&#44; paraesthesia or motor deficit&#44; and was able to walk unaided&#46; Her bowel movements were normal&#44; although she reported difficulty urinating&#44; with slight incontinence and a sensation of a full bladder that required effort to empty&#46; The neurological examination did not show motor&#44; sensory&#44; or cranial nerve abnormalities&#46; Given the situation&#44; the team decided to administer conservative treatment with no further diagnostic or therapeutic measures&#46; The patient was discharged the day after our assessment&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">In our patient&#44; the use of air to identify the epidural space immediately raised our suspicion of iatrogenic pneumorrachis&#46; However&#44; in the differential diagnosis of pneumorrhachis&#44; valsalva maneuvers associated with continued pushing during prolonged expulsion should also be considered as a possible cause&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">In extradural pneumorrachis&#44; the air usually collects in the posterior epidural space where the connective tissue is weaker than in the anterior vascular network&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This typical distribution was not observed in our patient&#44; as air accumulated mainly on the right side&#46; Given the existence of bubbles in the musculature on the right side&#44; we believe that this situation is possibly conditioned by a lateral approach to the epidural space&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The clinical profile of extradural and intradural pneumorrachis usually differs&#46; While intradural pneumorrachis is an indicator of a serious underlying pathology&#44; generally traumatic&#44; extradural pneumorrachis rarely presents clinical signs and is usually an incidental radiological finding&#46; The only symptom reported by our patient was persistent&#44; severe pain&#44; with no neurological deficits&#46; Although extradural pneumorrachis is rarely symptomatic in itself&#44; being generally associated with discomfort and pain or even neurological deficits&#44; intraspinal air can act as a space-occupying lesion and compress the nerves of the spinal canal&#44; causing severe neurological symptoms&#46; The following clinical manifestations of pneumorrachis secondary to epidural catheter placement have been described&#58; meningeal irritation&#44; radicular pain&#44; unilateral lower extremity weakness&#44; cauda equina syndrome&#44; paraplegia&#44; and tetraplegia&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4&#8211;6</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">In our patient&#44; although the volume of air was significant&#44; it did not cause any neurological symptoms&#46; Similar cases of extensive asymptomatic epidural pneumorrachis with no motor&#44; sensory or cranial nerve deficits on neurological examination have been described<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>&#59; however&#44; some cases involving very small amounts of air<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> have been associated with monoplegia&#44; hypoesthesia and sphincter dysfunction<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> and even paraplegia that required immediate diagnosis and treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> We were unable to find any explanation of this discrepancy between air volume and symptomatology in the literature&#44; although it has been suggested that the location and distribution of air within the spinal canal depends on the site of air dissection&#44; the rate and volume of intraspinal air injection&#44; the size of the intraspinal space&#44; and the position of the patient&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">A Cochrane review found that the air and saline loss of resistance techniques were equally effective in locating the intradural space and equally likely to cause complications&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Nevertheless&#44; the air technique evidently increases the risk of extradural pneumorrachis and pneumocephalus&#44; particularly in cases such as ours where a considerable amount of air is injected&#44; the technique is performed by an inexperienced anaesthesiologist&#44; and difficulties are encountered locating the extradural space&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The amount of air that can be safely injected remains unclear&#44; but some experts suggest that less than 2&#160;ml of air is sufficient&#44; and that complications occur when more than 3&#160;ml or repeated boluses are administered in complicated blocks&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Loss of resistance to saline is safer&#44; but symptomatic pneumorrachis with nerve compression has also been described with this technique&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and even cases of accidental injection of air in the epidural space via a permanent epidural catheter have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Extradural pneumorrachis is probably an underdiagnosed radiological finding&#46; Ultrasound&#44; increasingly used in anaesthesiology&#44; cannot detect pneumorrachis&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Although the technique of choice for reliable&#44; rapid detection of pneumorrachis is CT&#44;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;5</span></a> it can be difficult to differentiate between intradural and extradural air&#46; Magnetic resonance imaging gives a far more sensitive and extensive diagnosis&#44;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;5</span></a> and should be used when there are signs of spinal cord compression or to rule out coexisting aetiologies&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Nitrous oxide must be avoided when administering general anaesthesia in a patient with pneumorrachis&#44; because it can expand the accumulated air&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a> High concentrations of inspired oxygen can facilitate reabsorption of air from the subdural space&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">There are as yet no guidelines on the management of pneumorrachis&#46; After the initial CT scan&#44; follow-up should consist primarily of symptom observation&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Serial imaging is not required unless there is a risk of spinal cord compression&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">Trapped air is usually reabsorbed spontaneously within a few days&#44; so from a neurological point of view&#44; conservative treatment is a good option in stable patients&#46; Treatment measures such as bed rest&#44; analgesics&#44; dexamethasone&#44; administration of high concentrations of inspired oxygen&#44; and hyperbaric oxygen therapy have been described&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;5</span></a> In some severe&#44; symptomatic cases&#44; percutaneous decompression of epidural space using a Tuohy needle<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;5</span></a> or surgical decompression with laminectomy can be performed&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">Oxygen therapy&#44; mainly used to treat pneumocephalus&#44; can also be effective in extradural pneumorrachis because it can wash out pulmonary nitrogen and create a gradient in which nitrogen in the extradural air bubble diffuses toward the lungs via blood&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Oxygen can be administered via non-rebreather masks and high-flow nasal cannulas&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9&#44;10</span></a> A case in which hyperbaric oxygen therapy<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> was used to avoid irreversible deficits in a patient with symptomatic epidural pneumorrachis has also been reported&#46; All symptoms resolved after a single session&#44; and some authors believe hyperbaric oxygen therapy should be the first-line treatment when there is evidence of neurological impairment&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0100" class="elsevierStylePara elsevierViewall">In conclusion&#44; diagnosis of extradural pneumorrachis can be challenging for the anaesthesiologist&#44; and this complication must always be borne in mind when using air technique to identify the epidural space&#46; Even in asymptomatic cases&#44; the best approach is to administer high-flow oxygen therapy for the first few hours and closely monitor the patient&#8217;s neurological status&#44; so it may be advisable to admit the patient to an anaesthesiology care unit&#46; Symptomatic patients should be evaluated as soon as possible by a multidisciplinary team comprising an anaesthesiologist&#44; a neurologist&#44; a neurosurgeon and an interventional radiologist to determine whether the patient should be treated with hyperbaric oxygen therapy or percutaneous or surgical decompression&#46;</p></span><span id="sec0011" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0031">Conflict of interest</span><p id="par0051" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Iatrogenic extradural pneumorrhachis is a rare clinical entity&#44; but anesthesiologists should be aware of this possibility when using the air technique for the identification of epidural space&#46; Although in most published cases extradural pneumorrhachis is asymptomatic&#44; relevant neurological consequences have been described&#44; such as meningeal irritation&#44; radicular pain&#44; unilateral lower extremity weakness&#44; cauda equina syndrome&#44; paraplegia&#44; and tetraplegia&#46;We describe a very extensive extradural pneumorrachis &#40;T9-S1&#41;&#44; related to obstetric analgesia&#44; in a patient with severe and atypical perineal pain after forceps-assisted delivery&#46; Our aim is to synthesize and organize the available scientific evidence&#44; analyzing preventive measures and summarizing the most appropriate diagnostic&#44; follow-up and therapeutic techniques for symptomatic conditions&#44; among which high concentrations of inspired oxygen&#44; hyperbaric oxygen therapy and percutaneous or surgical decompression have been described&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El neumorraquis extradural iatrog&#233;nico es una entidad cl&#237;nica poco frecuente&#44; pero los anestesi&#243;logos debemos ser conscientes de esta posibilidad cuando se utiliza la t&#233;cnica con aire para la identificaci&#243;n del espacio epidural&#46; Aunque en la mayor&#237;a de los casos publicados el neumorraquis extradural es asintom&#225;tico&#44; se han descrito consecuencias neurol&#243;gicas relevantes&#44; como irritaci&#243;n men&#237;ngea&#44; dolor radicular&#44; debilidad unilateral de extremidades inferiores&#44; s&#237;ndrome de cola de caballo&#44; paraplejia y tetraplejia&#46;Describimos un neumorraquis extradural muy extenso &#40;T9-S1&#41;&#44; relacionado con la analgesia obst&#233;trica&#44; en una paciente con dolor perineal intenso y at&#237;pico tras un parto asistido con f&#243;rceps&#46; Nuestro objetivo es sintetizar y organizar la evidencia cient&#237;fica disponible&#44; analizando las medidas preventivas y resumiendo las t&#233;cnicas diagn&#243;sticas&#44; de seguimiento y terap&#233;uticas m&#225;s adecuadas ante cuadros sintom&#225;ticos&#44; entre las que se han descrito altas concentraciones de ox&#237;geno inspirado&#44; oxigenoterapia hiperb&#225;rica y descompresi&#243;n percut&#225;nea o quir&#250;rgica&#46;</p></span>"
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