Epidural haematoma after dural puncture in a parturient with neurofibromatosis

M. D. Esler1,2, J. Durbridge1,3 and S. Kirby1

1Department of Anaesthesia and Intensive Care, Hammersmith Hospital, Du Cane Road, London W12 0HS, UK.Present addresses: 2Department of Anaesthesia, British Columbia’s Women’s Hospital, Oak Street, Vancouver V6H 3N1, Canada. 3Magill Department of Anaesthesia, Chelsea and Westminster Hospital, Fulham Road, London SW10 9NH, UK*Corresponding author

Accepted for publication: July 24, 2001


    Abstract
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 Abstract
 Introduction
 Case report
 Discussion
 References
 
A case of epidural analgesia in a parturient with neurofibromatosis (von Recklinghausen’s disease) complicated by dural puncture and epidural haematoma is described and the management of the case is discussed. The case emphasizes the need for antenatal assessment of parturients with neurofibromatosis in order that the necessary investigations can be arranged and informed consent for analgesia and anaesthesia can be obtained.

Br J Anaesth 2001; 87: 932–4

Keywords: anaesthesia, obstetric; anaesthetic techniques, epidural; complications, neurofibromatosis; complications, epidural haematoma; complications, dural puncture; complications, neurological


    Introduction
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
Neurofibromatosis is an autosomal dominant condition with variable expressivity and an incidence of 1 in 3000 live births. The classical disease is characterized by multiple neurofibromata of peripheral nerves and cutaneous pigmentation patches (café au lait spots). In the only previous case report of regional analgesia in neurofibromatosis, the condition was identified, the parturient was investigated antenatally and the clinical course was uncomplicated.1 However, when the condition is not recognized the parturient may be exposed to considerable risk from attempts to provide regional analgesia.


    Case report
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 Abstract
 Introduction
 Case report
 Discussion
 References
 
A 41-yr-old nulliparous woman at 39 weeks gestation and in established labour requested epidural analgesia. She had booked late (at 20 weeks) and her antenatal care was unremarkable. She gave no medical history of note, but on examination by the anaesthetic trainee multiple pedunculated skin lesions were noted over her trunk, face and limbs (Fig. 1). She said she had been diagnosed with a skin condition, the name of which she could not recall. However, she was told it was of little significance. She gave no family history and her medical notes were unavailable. Her cervix was 4 cm dilated.



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Fig 1 View of the parturient’s back, demonstrating multiple pedunculated skin neurofibromata.

 
After routine verbal consent, an epidural catheter was sited uneventfully at L5/S1 level, avoiding passage through any of the skin lesions. An effective epidural block was obtained with a low-dose bupivacaine/fentanyl solution. Two boluses were given over the next 3 h, but then analgesia became ineffective as the catheter had migrated out of the epidural space and so was removed. Vaginal examination revealed an 8 cm dilated cervix, and at maternal request attempts were made to resite the epidural catheter. On location of the epidural space at the L3/4 level, the catheter fed into an epidural vein. Blood continued to be aspirated after catheter withdrawal so the catheter was removed. Then at the L4/5 level, inadvertent Tuohy needle dural puncture occurred so senior assistance was requested.

The medical notes had been obtained and revealed a diagnosis of neurofibromatosis. After review of the case and in the light of the complications already experienced by the patient, a combined spinal–epidural (CSE) was performed uneventfully by the consultant anaesthetist at the L2/3 level. This gave rapid relief and was effective with epidural boluses. After a further 6 h, the patient’s cervix was fully dilated but the fetal head was high. The obstetric team, informed of the dural puncture, allowed limited pushing in the second stage. The fetal head remained high, so after a trial of forceps in the operating theatre, a Caesarean section was performed uneventfully under epidural anaesthesia. The second stage was prolonged to 3.5 h as the obstetric team were occupied with other cases.

Review the following morning was unremarkable, the patient having mobilized with no headache or back pain. She was given diclofenac for postoperative pain, and did not receive heparin thromboprophylaxis. On the second postoperative day, she developed urinary incontinence and was recatheterized. Neurological examination revealed upgoing plantar reflexes bilaterally, but no other abnormal neurology. In particular, perianal sensation and anal sphincter tone were normal. An urgent magnetic resonance imaging (MRI) scan was performed, which showed an epidural haematoma at the L4/5 level. The haematoma was not impinging sufficiently on the dural sac to cause cauda equina compression. Neurosurgical review confirmed that no intervention was indicated.

The patient was reassured, and her urinary incontinence resolved by the third postoperative day. Follow-up cerebral and spinal MRI scan after 4 weeks revealed resolution of the epidural haematoma and no neurofibromata. The patient was advised that if she should become pregnant again, early obstetric and anaesthetic consultation should occur.


    Discussion
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 Abstract
 Introduction
 Case report
 Discussion
 References
 
The clinical presentations of neurofibromatosis and some anaesthetic implications have been reviewed recently.2 While this patient had no neurological symptoms, spinal tumours have been identified in 40% of patients with asymptomatic classical neurofibromatosis.3 The majority of tumours arise laterally at the intervertebral foramen but are at risk of direct trauma from an epidural needle if they extend towards the midline.4

It has been suggested that a potential complication after dural puncture could be rapid decompression below a spinal or intracranial tumour, causing spinal cord injury or even death, particularly in the presence of raised intracranial pressure.1 2 MRI or computed tomography scanning of the brain and spinal cord is recommended late in pregnancy, as tumours may grow significantly during gestation.5

This condition, in common with many neurological conditions, may be viewed as a relative contraindication to regional analgesia. A risk–benefit analysis should be made, thorough neurological examination recorded and detailed informed consent gained before proceeding. Alternative methods of pain relief, including nitrous oxide/oxygen and systemic opiate analgesia, should be considered.

This patient ultimately required Caesarean section for failure to progress, and it is unlikely that epidural analgesia influenced this outcome.6 Alternative anaesthesia for operative delivery would have included general anaesthesia. While neurofibromatosis has been associated with abnormal responses to neuromuscular blocking drugs, this has recently been contested.7 8 Airway problems have also been described,9 but this parturient had a clinically normal cervical spine, unremarkable airway assessment and no history of general anaesthetic complications.

This parturient booked late but otherwise had routine antenatal care. We were unaware of her diagnosis until her medical notes were obtained, by which time less than fully informed consent for epidural analgesia had been taken, and the bloody tap and dural puncture had already been made. Further clinical examination revealed several café au lait spots and the clinical diagnosis was not in doubt.

Consideration was given to passing an intrathecal catheter via the Tuohy needle after dural puncture, but at the time this was against department practice. The CSE technique was performed to give rapid relief to the parturient, who was increasingly distressed. Any additional leak from a 27 gauge Whitacre spinal needle puncture was not considered significant in comparison with the 16 gauge Tuohy needle puncture. Recent reports of conus damage from spinal anaesthesia administered at levels above Tuffier’s line suggest that CSE at the L2/3 level exposes the parturient to risk from this complication, however, and should be avoided.10

Parturients with pre-existing neurological conditions require neurological examination after regional blockade. The upgoing plantar reflexes elicited and bladder dysfunction led to urgent MRI investigation. No cause for these findings was identified but an epidural haematoma was found at the L4/5 level. While clinical identification of spinal levels may be inaccurate,11 this was thought to be the level of the dural puncture.

As many as 80–85% of parturients with dural puncture experience postdural puncture headache12 13 but our patient remained headache-free, perhaps because of the ‘blood patch’ effect of the epidural haematoma. Previous retrospective review of one unit’s dural punctures showed no protective effect of bloody tap in relation to the development of postdural puncture headache.14 Other work suggests that bloody tap is usually associated with only a small volume of epidural blood, not adhering to the thecal sac.15 In our case, impingement on the dural sac by the haematoma was seen at the level of the dural puncture, and hence an ‘auto-blood patch’ effect may have occurred.

The upgoing plantar reflexes remain unexplained and, because this parturient presented to the anaesthetist in labour, a full baseline neurological examination was not performed. This again emphasizes the need for good communication between obstetric and anaesthetic staff so that these patients can be identified, assessed and investigated before the onset of labour.

Bladder dysfunction after extended labour is well recognized. After a long second stage with a high fetal head, lumbosacral plexus neuropraxia seems a possible cause in this case as recovery was rapid. Maternal obstetric palsies are commonly attributed to regional techniques16 and, as Bromage has reiterated, it is vital that practising anaesthetists become skilled and active in bedside neurological diagnosis.17

While all the problems experienced by this parturient in association with our regional anaesthetic techniques would have occurred in the absence of neurofibromatosis, our diagnosis of her condition, albeit late, heightened our awareness of potential problems. This re-emphasizes the fact that thorough assessment of parturients with neurological conditions antenatally is vital so that analgesia and anaesthesia can be planned and, ideally, informed consent can be gained in advance. Experience of obstetrical anaesthesia assessment clinics suggests they may facilitate this,18 and in such a way adverse outcomes for parturients may be prevented.


    References
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 Abstract
 Introduction
 Case report
 Discussion
 References
 
1 Dounas M, Mercier FJ, Lhuissier C, Benhamou D. Epidural analgesia for labour in a parturient with neurofibromatosis. Can J Anaesth 1995; 42: 420–4[Abstract]

2 Hirsch NP, Murphy A, Radcliffe JJ. Neurofibromatosis: clinical presentations and anaesthetic implications. Br J Anaesth 2001; 86: 555–64[Abstract/Free Full Text]

3 Thakkar SD, Feigen U, Mautner VF. Spinal tumours in neurofibromatosis type 1: an MRI study of frequency, multiplicity and variety. Neuroradiology 1999; 41: 625–9[ISI][Medline]

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5 Ginsburg DS, Hernandez E, Johnson JWC. Sarcoma complicating von Recklinghausen disease in pregnancy. Obstet Gynecol 1981; 58: 385–7[Abstract]

6 Clark A, Carr D, Loyd G, Cook V, Spinnato J. The influence of epidural analgesia on cesarean delivery rates: a randomized, prospective clinical trial. Am J Obstet Gynecol 1998; 179: 1527–33[ISI][Medline]

7 Baraka A. Myasthenic response to muscle relaxants in von Recklinghausen’s disease. Br J Anaesth 1974; 46: 701–3[ISI][Medline]

8 Richardson MG, Setty GK, Rawoof SA. Responses to nondepolarizing neuromuscular blockers and succinylcholine in von Recklinghausen neurofibromatosis. Anesth Analg 1996; 82: 282–5

9 Crozier WC. Upper airway obstruction in neurofibromatosis. Anaesthesia 1987; 42: 1209–11[ISI][Medline]

10 Reynolds F. Logic in the safe practice of spinal anaesthesia. Anaesthesia 2000; 55: 1045–46[ISI][Medline]

11 Broadbent CR, Maxwell WB, Ferrie R, Wilson DJ, Gawne-Cain M, Russell R. Ability of anaesthetists to identify a marked lumber interspace. Anaesthesia 2000; 55: 1122–26[ISI][Medline]

12 Colonna-Romano P, Shapiro BE. Unintentional dural puncture and prophylactic epidural blood patch in obstetrics. Anesth Analg 1989; 69: 522–3[ISI][Medline]

13 Brownridge P. The management of headache following dural puncture in obstetric patients. Anaesth Intens Care 1983; 11: 4–16[ISI][Medline]

14 Khan KJ, Stride PC, Cooper GM. Does a bloody tap prevent postdural puncture headache? Anaesthesia 1993; 48: 628–9[ISI][Medline]

15 Beards SC, Jackson A, Holland J, Horsman EL. Does a bloody tap prevent postdural puncture headache? [letter; comment] Anaesthesia 1993; 48: 1111–2

16 Loo CC, Dahlgren G, Irestedt L. Neurological complications in obstetrical regional anaesthesia. Int J Obstet Anesth 2000; 9: 99–124[ISI]

17 Bromage PR. Neurological complications of subarachnoid and epidural anaesthesia. Acta Anaesthesiol Scand 1997; 41: 439–44[ISI][Medline]

18 Rosaeg OP, Yarnell RW, Lindsay MP. The obstetrical anaesthesia assessment clinic: a review of six years experience. Can J Anaesth 1993; 40: 346–56[Abstract]





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