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complete or incomplete foramen transversarium. During the same period of embryogenesis, the neural arch proceeds directly backwards to a variable extent forming pedicles, articular facets and part of the lamina. A developmental defect in our cases could have occurred during or before this stage of embryonic development. The exact cause of the defect in our patients can not be known. There was no evidence of consanguinity or obvious teratogenic events in utero. The clinical and radiological features of the case reported here have remarkable similarities to the other two patients presented by us earlier. There was no history of a major trauma to the neck or head. In the earlier reported cases, there was a partial reduction of the dislocation after the application of traction. However, the patient in the present report did not tolerate traction and showed clinical symptomatic worsening. An attempt to fix the spine by interlaminar instrumentation after a partial reduction of the dislocation with traction had resulted in neurological worsening in our earlier reported case. Anterior transcervical decompression, which was followed up by a posterior fixation, had resulted in a satisfactory clinical improvement in the other patient. In the present case, a transoral decompression and plate and screw fixation resulted in an immediate and sustained clinical improvement. Although, there is a risk of infection following placement of a metal plate via a transoral route, the ease of exposure and decompression and fixation by this route can be employed to the advantage. We are not yet convinced whether a transoral or a transcervical route is superior or vice versa. However, further experience is mandatory to formulate the most appropriate treatment protocol for this complex anomaly and challenging therapeutic problem.
REFERENCES 1. Asakawa H, Yanaka K, Narushima K, Meguro K, Nose T. Anomaly of the axis causing cervical myelopathy. Case report. J Neurosurg 1999; 91: 121–123. 2. Davis D, Gutierrez FA. Congenital anomaly of the odontoid in children. A report of four cases. Childs Brain 1977; 3: 219–229. 3. Feilding JW, Hensinger RN, Hawkins RJ. Os odontoideum. J Bone J Surg A 1980; 62: 376–383. 4. Goel A, Bhatjiwale M, Desai K. Basilar invagination: a study based on 190 surgically treated patients. J Neurosurg 1998; 88: 962–968. 5. Kawano K, Uehara S, Nagata Y. A case of myelopathy due to a peculiar anomaly of the axis. No Shinkei Geka 1987; 15: 543–546 (Jpn, with Eng. abstract). 6. Koyama T, Tanaka K, Handa J. A rare anomaly of the axis: report of a case with shaded three-dimensional computed tomographic display. Surg Neurol 1986; 25: 491–494. 7. Mondschein J, Karasick D. Spondylolysis of the axis vertebra: a rare anomaly simulating hangman's fracture. AJR Am J Roentgenol 1999; 172: 556– 557. 8. Smoker WR. Craniovertebral junction: normal anatomy, craniometry, and congenital anomalies. Radiographics 1994; 14: 255–277. 9. Steinberg EL, Reider I, Barak S, Khermosh O, Wientroub S. Unilateral dysplasia of the superior articular facet of the axis: a case report. J Pediatr Orthop 1990; 10: 540–541. 10. Stevens JM, Chong WK, Barber C, Kendall BE, Crockard HA. A new appraisal of abnormalities of the odontoid process associated with atlanto-axial subluxation and neurological disability. Brain 1994; 117: 133–148. 11. Morizono Y, Sakou T, Maehara T. Congenital defect of posterior elements of the axis. Clin Orthop 1987; 216: 120–123. 12. Goel A, Gupta S, Laheri V. Congenital absence of posterior elements of axis: a report of two cases. Br J Neurosurg 1999; 13: 459–461. 13. Vangilder JC, Menezes AH, Dolan KD. Radiology of asymtomatic craniovertebral anomalies and abnormalities. In: Vangilder JC, Menezes AH (eds) The Craniovertebral Junction and its Abnormalities. Futura Publishing Co., New York 1987: 89. 14. Geipel P. Zur Kenntnis der spaltbildung des atlas und epistropheus. Fortschr abGebd Rontgenstrahlen 1935; 52: 533. 15. Bailey RE. The Cervical Spine. Lea and Febiger, Philadelphia 1974.
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16. Bailey DK. The normal cervical spine in infants and children. Radiology 1952; 59: 712–719. 17. Von Torklus D, Gehle W. The Upper Cervical Spine. Regional Anatomy, Pathology and Traumatology. A Systemic Radiological Atlas and Text Book. Grune and Stratton, New York 1972. 18. O'Rahilly R, Muller F, Meyer DB. The human vertebral column at the end of the embryonic period proper 2. The occipitocervical region. J Anatomy 1983; 136: 181–195.
Propionibacterium acnes causing delayed subdural empyema – a case report and review of literature S.R.A. Ghalayini MRCS, A.M. Likhith A. Golash FRCS(NS)
FRCS,
Department of Neurosurgery, Royal Preston Hospital, Preston, Lancashire, UK
Summary The authors report a patient who presented with a delayed subdural empyema caused by Propionibacterium acnes following excision of a meningioma. This organism should be suspected in all patients with delayed empyema especially in the presence of implants and immunosuppression. The main features of this pathogen is discussed. Surgical drainage and high-dose intravenous penicillin should be the recommended therapy. ª 2004 Published by Elsevier Ltd. Journal of Clinical Neuroscience (2004) 11(6), 677–679 0967-5868/$ - see front matter ª 2004 Published by Elsevier Ltd. doi:10.1016/j.jocn.2003.09.017
Keywords: subdural empyema, Propionibacterium acnes Received 6 June 2003 Accepted 2 September 2003 Correspondence to: S. Ghalayini MBBS, Department of Neurosurgery, Royal Preston Hospital, Preston, PR2 9HT Lancashire, UK. Tel.: +44-1772-716565; E-mail:
[email protected]
INTRODUCTION Delayed post-operative subdural empyema is a rare occurrence. The common organisms are Streptococcus milleri, Staphylococcus aureus and Staphylococcus epidermidis. Infection with Propionibacterium acnes (P. acnes) is usually seen in patients with prosthesis or with immunosuppression.1;2 Only a few cases of post-operative empyema with P. acnes following non-implant surgery have so far been reported.3 Such a case is reported with discussion of its main features and review of the literature. CASE REPORT A 55-year-old right-handed gentleman presented with a single tonic clonic seizure without any focal neurological deficits. A CT scan of his brain revealed a right frontal lobe lesion suggestive of a meningioma. He subsequently underwent a right frontal craniotomy and excision of the meningioma. One month following his surgery he was re-admitted with a history of two further seizures, persistent headache and lethargy. On neurological examination he had a GCS of 15/15 with some Journal of Clinical Neuroscience (2004) 11(6)
678 Ghalayini et al.
mild left sided weakness (MRC grade 4/5). He had a mild pyrexia (37.5 C) and an elevated ESR of 70 mm per hour. White cell count was within normal limits. CT scan of the brain revealed a subdural collection in the right frontal region at the site of the previous craniotomy with contrast enhancement of the periphery. There was evidence of free gas within the collection. (Figs. 1 and 2). He underwent exploration of the craniotomy for evacuation of this collection. During surgery purulent material was seen on either side of the artificial dura, which was fashioned during the earlier surgery from an attached pericranial flap. The pus was evacuated and the pericranium, along with the bone flap, was removed. Gram stain showed abundant white cells and Gram-
Fig. 1 Plain CT scan showing evidence of previous craniotomy and an underlying subdural collection with air.
positive bacilli. Cultures revealed the presence of P. acnes, which was sensitive to penicillin, ciprofloxacin and ceftriaxone. He was treated with a course of intravenous benzylpenicillin and ceftriaxone for 2 weeks followed by oral amoxycillin and ciprofloxacin for 6 weeks. He made a good recovery without any neurological deficits. CT scan at follow-up showed total resolution of the subdural empyema.
DISCUSSION P. acnes are gram positive, non-spore-forming bacilli that grow under anaerobic conditions. It requires prolonged culture and the pathogenicity is similar to that of Straphylococcus epidermidis.1;3 This is illustrated by the rather delayed and indolent course of illness in our patient. They are frequent inhabitants of sebaceous glands, hair follicles, mouth and upper respiratory tract. Although believed to be a benign commensal, P. acnes has been implicated in infections of orthopaedic and cardiac prostheses, surgical wounds and in cutaneous abscesses.3–5 There are also reports that it may be associated with chronic low-grade infection in the lumbar vertebral discs of patients with severe sciatica.6 After neurosurgical operations, empyema caused by P. acnes is rare and when seen is most commonly associated with the presence of foreign bodies such as shunt systems.1 Two cases of non-prosthetic subdural empyema were reported by Critchley and Strachan.3 They were secondary to burr hole evacuation of chronic subdural haematoma and had a prolonged course between the initial surgery and final diagnosis. The presence of free gas in the subdural space without communication with the outside atmosphere is a rare phenomenon seen in the presence of gas producing organisms like clostridium. In our case the wound had healed well and any residual air from the initial surgery would have been absorbed. This suggests that the gas seen on the CT scan was a result of the infection (most likely produced by the bacteria). Attached pericranium was used for duroplasty after the involved dura was removed hence no foreign material was used. The patient had received prophylactics pre-operative cephalosporin antibiotics as recommended for clean non-implant procedures. The pathogen was sensitive to penicillin and ceftriaxone and the patient responded well to surgical drainage and intravenous high-dose penicillin and cephalosporin. CONCLUSION This case serves to illustrate some of the main features of a subdural empyema due to P. acnes, especially the delayed presentation and relatively indolent course along with the rare phenomenon of intracranial gas production. This organism should be suspected when intracranial suppuration in seen following neurosurgical operation in patients with implants or immunosuppression. Surgical drainage with removal of implant material and high-dose intravenous penicillin is the recommended therapy. REFERENCES
Fig. 2 CT scan following injection of contrast showing peripheral enhancement of the subdural collection.
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1. Uzomi Y, Nawashiro H, Higo R et al. Post-operative subdural empyema due to Propionibacterium acnes. No Shinkei Geka 2002; 30(7): 731–733. 2. Chu RM, Tummala RP, Hall WA. Focal intracranial infections due to Propionibacterium acnes: report of three cases. Neurosurgery 2001; 49(3): 717–720. 3. Critchley G, Strachan R. Postoperative subdural empyema caused by Propionibacterium acnes – a report of two cases. Br J Neurosurg 1996; 10(3): 321–323. 4. Tunney MM, Patrick S, Gorman SP et al. Improved detection of infection in hip replacements. J Bone Joint Surg Br 1998; 80: 568–572.
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5. Brook I, Frazier EH. Infections caused by Propionibacterium species. Rev Infect Dis 1991; 13: 819–822. 6. Stirling A, Worthington T, Rafiq M, Lambert PA, Elliott TSJ. Association between sciatica and Propionibacterium acnes. Lancet 2001; 357(9273): 2024.
Preoperative visualization of microvascular compression of cranial nerve IX using constructive interference in steady state magnetic resonance imaging in glossopharyngeal neuralgia Hiroshi Karibe MD, Reizo Shirane Takashi Yoshimoto MD
MD,
Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Japan
Summary We report a case of successfully treated glossopharyngeal neuralgia, in whom preoperative magnetic resonance imaging (MRI) with a sequence of constructive interference in steady-state (CISS) precisely delineated microvascular compression to the glossopharyngeal nerve at its cisternal portion. A 70-year old female experienced paroxysmal and excruciating pain, starting in the right ear and descending to the tongue and throat. The pain was brief, burning and triggered by swallowing food and drinks. Preoperative CISS-MRI delineated a small vascular loop compressing the glossopharyngeal nerve at its cisternal portion. The patient was diagnosed with glossopharyngeal neuralgia, and then, microvascular decompression surgery was performed. Intraoperative inspection revealed that the glossopharyngeal nerve root was in contact with a small arterial loop at its cisternal portion and was deformed as demonstrated by preoperative CISS-MRI. The glossopharyngeal nerve was displaced using a piece of Teflon felt, freeing it from the arterial loop compression. The patient’s neuralgia resolved immediately after surgery. During a period of 2-years follow up, no recurrence of the neuralgia has occurred. The usefulness of CISS-MRI for the diagnosis of glossopharyngeal neuralgia is discussed. ª 2004 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2004) 11(6), 679–681 0967-5868/$ - see front matter ª 2004 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2003.05.011
Keywords: glossopharyngeal neuralgia, microvascular decompression, constructive interference in steady state magnetic resonance imaging Received 20 April 2003 Accepted 6 May 2003 Correspondence to: Hiroshi Karibe MD, Department of Neurosurgery, Tohoku University Graduate School of Medicine, 1-1, Seiryo-machi, Aoba-ku, Sendai 980-8574, Japan. Tel.: +81-22-717-7230; Fax: +81-22-717-7233; E-mail:
[email protected]
INTRODUCTION Glossopharyngeal neuralgia is a rare condition, characterized by attacks of spasmodic facial pain, described as sharp, shooting, ª 2004 Elsevier Ltd. All rights reserved.
stabbing, lasting from a few seconds to one minute, experienced in the throat and ear and usually precipitated by swallowing.9;10 Since microvascular compression to the nerve root has been hypothesized as a cause, analogous with trigeminal neuralgia,8 microvascular decompression has been proposed as a surgical treatment.8;9;12 Since the patterns of vascular compression are closely related to the result of microvascular decompression,14 preoperative assessment of microvascular compression to the glossopharyngeal nerve may be important to improve surgical results and to prevent unnecessary surgery. It has been reported that three-dimensional constructive interference in steady-state (CISS) magnetic resonance imaging (MRI) has a high resolution and excellent contrast between cranial nerves, small vessels and CSF, so can precisely and accurately delineate normal and abnormal neurovascular relationships in trigeminal neuralgia.15 Recently, it has also been demonstrated that this MRI sequence provides best resolution of the lower cranial nerves and their relationship to surrounding structures.11 These results suggest that CISS-MRI may be a powerful tool in delineating neurovascular relationship in cases with glossopharyngeal neuralgia. However, it is surprising that this MRI sequence has not been applied for the radiological diagnosis of glossopharyngeal neuralgia. In this case report, a case with glossopharyngeal neuralgia is reported, in which microvascular compression to the glossopharyngeal nerve has been diagnosed by CISS-MRI. CASE REPORT A 70-year-old woman experienced paroxysmal and excruciating pain since 1998, starting in the right ear and descending to the mandibular angle and to the tongue and throat. The pain was brief, burning, and triggered by swallowing food and drinks. At the beginning, the pain was infrequent – every 2 or 3 weeks the patient suffered for a minute a few times a day. But soon, the pain became more frequent, the patient suffering several times each day. The topography and characteristics of the pain led to the diagnosis of right glossopharyngeal neuralgia. Carbamazepine, 300 mg/day, was prescribed, giving good relief of pain. But this treatment had to be discontinued due to drug-induced liver dysfunction, causing the pain to recur. The patient was then referred and admitted to our department on 21st December 2000. At the time of admission, the general and neurological examination of the patient was normal, with no impairment in motor or sensory function. In particular, the cranial nerves were all normal, the gag reflex present and the blood pressure 140/80 mmHg. CT scan and MRI examination using post-contrast T1 and T2weighted pulse sequences with thick slices were considered normal with no evidence of a space occupying lesion. Additional MR imaging was performed using CISS pulse sequence. Parameters for CISS were TR: 12.25 ms, TE: 5.9 ms, flip angle: 70, FOV: 200 mm, rectangular FOV: 7/8, matrix: 224 512, number of partitions: 40, and acquisitions: 1. Then, axial images of 0.9 mm contiguous sections were obtained. Three-dimensional spoiled gradient recalled acquisition at the steady state (SPGR) with 0.9 mm thick sections was also obtained, after injection of 0.1 mmol/ kg of gadolinium-tetra-azacyclododecane-tetra-acetic acid. These MR images showed that a small vascular loop, which arose from the right posterior inferior cerebellar artery (PICA), had a superoposterior direction and described a large loop with an infero-lateral convexity. The vago-glossopharyngeal nerve root was in contact with the vascular loop and was deformed in its cisternal portion, as demonstrated by CISS image (Fig. 1). Microvascular decompression surgery was decided upon. The patient was operated on in the prone position, the head turned 45 Journal of Clinical Neuroscience (2004) 11(6)
