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Secondary hydrocephalus communicans after traumatic upper cervical spine injuries with leakage of cerebrospinal fluid is a rare and hardly described complication. A case of a 75-year-old woman sustained a type II dens axis without other injuries, especially without evidence of a hydrocephalus in the primary CT scan. Dorsal atlanto-axial fusion was performed. Postoperative drainage was prolonged and positive for β2-transferrin. Wound revision with an attempt to seal the leakage was not successful. Secondary CT scans of the brain were performed due to neurological deterioration and showed a hydrocephalus with typical EEG findings. No anatomical reason for a circulative obstruction was found in the CT scan. After application of a ventriculo-peritoneal shunt the neurological status improved and the patient could be discharged to neurological rehabilitation.
Fractures of the dens axis frequently occur in older patients after falls resulting in head trauma. Death due to respiratory depression is not uncommon in this injury. Normally, after surgical stabilisation of a dens axis fracture, the patients recover quickly and can quickly resume daily activities. Two approaches to stabilise a dens axis-fracture are used by the spinal surgeons. The ventral approach stabilises the dens axis with one or two screws set from the corpus to the apex of the axis with high risk of pseudarthrosis development. The dorsal approach aims at atlanto-axial fusion and can be performed in different techniques.1 Traumatic dural leakage associated with upper cervical fractures occurs rarely and has been described only in a post mortem study.2 Cerebrospinal fluid (CSF) leaks after spine surgery are well documented and are among the most commonly reported complications, occurring in approximately 15% of patients undergoing spine surgery.3 Usually, postoperative leakage of CSF leads to subcutaneous fluid accumulation at the operation site, but has no obstructive effect on liquor circulation, whereas the development of a hydrocephalus after cervical spine fractures, especially in association with pre-existing malformations, malignancies or inflammations, as a complication is known.4 The development of a hydrocephalus communicans (a combined form of hydrocephalus occlusivus and hydrocephalus maleresorbtivus) after fractures of the upper cervical spine in healthy older patients5 has never been reported to the best of our knowledge. This surprising complication was successfully managed by a ventriculo-peritoneal (VP) shunt leaving the occipito-nuchal liquor accumulation in situ. The aim of this case report is to introduce a new complication after high cervical spine injuries with leakage of CSF.
A 75-year-old, initially healthy female patient was admitted to the emergency department6 after having sustained an Anderson Type II7 dens axis fracture (figure 1A) with a cerebral concussion. A CT scan of the scull and brain upon admission showed no signs of hydrocephalus. The concomitant injuries included serial fractures of the ribs and a knee injury. The stabilisation of the dens axis fracture was carried out via a posterior approach using the Harms technique1 (figure 1B). The wound drainage was prolonged and had a clear yellow colour. A positive 2-transferrin test8 showed the fluid to be CSF. To estimate the size of the accumulated CSF, an MRI was performed which showed a subcutaneous CSF accumulation at the operation site. Secondary revision of the wound and application of Duraseal (Confluent Surgical, Waltham, Massachusetts, USA) to seal off the spinal canal from the surrounding muscles 12 days later was not successful. This was probably due to more ventral location of the dural tear. However, the wound remained dry (figure 2) and did not warrant further revisions. After several weeks a therapy-resistant arterial hypertension occurred with systolical values more than 180 mm Hg. The patient progrediently became disoriented and had a dysarthria. A CT scan of the brain performed 4 weeks after the accident showed a ventricular dilatation (figure 3B) with signs of elevated ventricular pressure. A hydrocephalus communicans occurred and a VP shunt (Hakim Codman Ventile; Cilag AG, Switzerland, 70 mm H2O pressure limitation) was applied. The CSF diagnostics showed no evidence of an infection. After normalisation of the CSF pressure the patient's neurological signs normalised, and the control CT scan performed 5 days later showed almost normally configured ventricles (figure 3C). The orientation and dysarthria significantly improved after application of the VP shunt. The patient was discharged in a neurological rehabilitation centre after 49 days.
A whole-body CT scan performed upon admission of the patient revealed a Type II dens axis fracture4 and serial rib fractures. No other injuries of the head, spine or torso were found. A conventional x-ray of the left knee revealed a high index of suspicion for a medial tibial plateau fracture.
After dorsal stabilisation, the CT scan of the cervical spine showed a proper position of the dens axis. The osteosythesis material was in the right place according to the Harms technique.1
A T2-weighted MRI was performed to define the origin of the cervical fluid collection. It showed a T2 hyperintense signal alteration ranging from C1 to C6 (approximately: 2.8×5.1×6.8 cm) in the subcutaneous and muscular tissue, corresponding to a possible accumulation of CSF. Normal presentation of the myelon was observed. A beginning disco-ligamentary protrusion of the cervical disci was seen, without neuro foraminal or spinal narrowing.
β2-transferrin analysis was routinely performed by isoelectrical focusing and immuno-fixation of β2-transferrin by a commercially available kit (Hydragel; Sebia, Bioparks Evry, France). No quantification was given; the samples were analysed only qualitatively.
A conventional EEG was performed after installation of a VP shunt to evaluate the presence of an epileptic status despite the fact that the patient remained somewhat somnolent; no epilepsy-like activity was observed. Immediately after the operation a small locus with θ-δ-activity was observed temporally on the left side, according to a residual focus after elevated intracranial pressure. A follow-up EEG showed vanished θ-δ-activity temporally left 24 h later, but showed a curve of somnolence.
The CSF was repetitively negative for microorganisms analysed by conventional cytospins and Gram stains. There were repetitively less leucocytes. Conventional bacteriological cultures revealed no bacterial growth. Conventional wide-spectral bacterial PCR detected no bacterial DNA in CSF. The analysis for Borrelia burgdorferi revealed negative results, as well for IgG as for IgM. Treponema pallidum was also negative. These tests were performed by particle agglutination with a commercially available kit.
The initial stabilisation of the Type II dens axis fracture was performed using the Harms technique.1 The decision for a dorsal spondylodesis (Harms technique) was made due to a high development of pseudarthrosis using ventral corporo-dental screws.10 Posterior spondylodesis was completed by adding DBX (DBX, Demineralized Bone Matrix; Synthes GmbH, Solothurn, Switzerland). There was no suspicion for a CSF leakage intraoperatively. The operative procedure was uneventful and the patient did well postoperatively. High secretion amount from the drainage position and the confirmation of β2-transferrin positivity led to operative revision and application of Duraseal (Confluent Surgical, Waltham, Massachusetts, USA) 12 days later. The direct operative approach could not prevent further CSF leakage. A VP shunt was positioned on the right temporal side after confirming the diagnosis of a hydrocephalus. The shunt adaptations were performed routinely according to patient's clinical status and CT follow-up.
Clinical and CT follow-up was performed weekly for 4 weeks and showed a normalisation of the ventricle size accompanied by increasing vigilance of the patient. At discharge from the clinic the patient could walk with crutches. Social interaction was without any deficits. The patient was almost fully reintegrated and independent of any help after 6 weeks; only a routine adaptation of pressure limit of the VP shunt was necessary.
Upper spinal fractures in older patients are not uncommon. The trauma mechanism mainly consists of a hyperextension of the neck after a fall. Usually these fractures come along with other injuries such as cerebral concussion or fractures of the extremities. At this cervical level (C2) the patient usually dies when there is a dislocation leading to any neurological manifestation. Dura mater is mostly torn in atlanto-axial fractures as earlier reported in a post mortem study.2 The operative approach leaves a cavitation for fluid accumulation. The Harms technique1 was used to stabilise this fracture without exposing the dural sac. Even if the CSF fistula would have been caused by the initial operative procedure there would not be any traumatic reason to develop a hydrocephalus. The genesis of a hydrocephalus communicans was obviously slow and took a few days. To discuss whether Duraseal or DBX could be the reason for CSF circulation disturbances remains hypothetical, due to a pressure gradient outward the intradural space. Persistent flow out of the dural space inhibits any material translocation towards CSF space. However, the inhibition of re-absorption of CSF after upper traumatic spine injuries cannot be explained as yet. Hydrocephalus as a result of spinal pathology is rare and has been reported with spinal tumours where the pathogenesis may be the disruption of the CSF flow, impaired absorption due to elevated CSF protein content, tumour infiltration into basal cisterns or compression of the spinal venous plexus. The pathogenesis after trauma may be related to the high level of the injury (C2), thus leading to blood accumulation from the site of the injury in the basal cisterns and consequently impairing the absorption of CSF by the arachnoid villi across the hemispheres, which in turn leads to a communicating hydrocephalus, a combined form of the malabsorptive and occlusive hydrocephalus. Several treatment modalities have been recommended for spinal CSF leakage, but only sufficient closure with cadaveric dura or muscle flap can withstand the high hydrostatic pressure.11–13 Some surgeons favour re-operation with direct repair or application of fibrin sealant on the dural defect.14 Others suggest continuous closed CSF drainage and/or permanent shunting.14 The continuous spinal drainage could lower the CSF pressure gradient across the leak and enhance spontaneous closure of the defect. The application of Duraseal was not successful in this case (figure 2). This is indicated by a persistent accumulation of CSF in the occipito-nuchal area and in the second wound drainage suggesting a fistula in the ventral area, a surgically inaccessible location. Whenever a pseudomeningocele9 or CSF leak occurs, one should consider the possibility of concurrent hydrocephalus. If this complication is encountered early, an externalised lumbal drain may be sufficient to treat this complication.
However, till date, the development of a hydrocephalus communicans cannot be rationally explained after a fracture of the dens axis or operative injury of the dural sac. There are only academic approaches to explain this pathogenesis without any evidence in medical literature to the best of our knowledge.
Hydrocephalus may develop after high cervical fractures accompanied with neurological deterioration in the further course.
Competing interests None.
Patient consent Obtained.