Acta Neurochir (Wien) (2008) 150: 845–846 DOI 10.1007/s00701-008-1508-y Printed in The Netherlands
Brief Report of Special Case Bilateral vocal cord palsy due to brain-stem cavernoma – a diagnostic dilemma! Z. Ali, H. Prabhakar, G. P. Rath, D. Singh Department of Neuroanaesthesiology, All India Institute of Medical Sciences, New Delhi, India Received 19 June 2007; Accepted 20 December 2007; Published online 21 May 2008 # Springer-Verlag 2008
Summary Vocal cord palsy may result from varied causes. However, cord palsy resulting from a brain-stem cavernoma has never been reported. We report a patient with vocal cord palsy in a 30-year-old male resulting from the brain-stem lesion. The patient became symptomatic each time the lesion bled and improved gradually when the bleeding resolved. Repeated insults on the brain-stem produced permanent cord palsy. Although a rare presentation, brain-stem cavernoma may be considered in the aetiology of cord palsy. Keywords: Respiratory distress; brain-stem cavernoma; vocal cord palsy.
Introduction Among intracranial vascular malformations the cavernous malformation is one of the rarest, with a necropsy incidence of between 0.39 and 0.53% [4]. With the introduction of magnetic resonance imaging (MRI), cavernous malformations are now diagnosed more often [3]. Between 10 and 23% are located in the posterior fossa with most being in the pons [1]. If these lesions bleed they can cause severe functional disturbances or even
Correspondence: Dr. Hemanshu Prabhakar, CN Center, Department of Neuroanaesthesiology, 7th floor, All India Institute of Medical Sciences, New Delhi 110029, India. e-mail:
[email protected]
death. Vocal cord palsy as a result of medullary cavernoma is unusual and can be easily missed. Clinical details A 30-year-old male was admitted to our emergency department with respiratory distress and stridor. The trachea was immediately intubated and ventilation assisted. Bilateral abductor vocal cord palsy was diagnosed on direct laryngoscopy at the time of intubation. Following intubation and assisted ventilation the patient’s condition became stable. MRI revealed multiple cavernous angiomas with a large lesion on the right side of the medulla (18 mm in diameter) with a variegated appearance due to recurrent bleeds (Fig. 1). He was scheduled for an elective midline suboccipital craniectomy and complete microsurgical excision of the tumour. The past history was significant with a focal neurological deficit 15 years previously diagnosed as acute demyelinating encephalomyelitis and treated with steroids. He recovered after one week. Five years ago, he presented with stridor of acute onset for which tracheostomy was performed and he recovered a week later. On the present admission, the surgical and anaesthetic course was uneventful. On the first post-operative day, extubation of trachea was attempted but as the patient continued having stridor, he was reintubated and tracheostomy planned. He was referred to the otorhinolaryngologist who planned vocal cord lateralisation as the definite treatment.
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Fig. 1. Magnetic resonance image of a well defined focal lesion in the pons (black arrow) showing heterogenous signal on T2 weighted image corresponding to subacute and chronic haemorrhages
Z. Ali et al.: Bilateral vocal cord palsy due to brain-stem cavernoma
He had developed similar symptoms earlier for which a tracheostomy was required. The symptoms resolved with time, possibly as the bleed was minor leading to resolution of the blood clot. Also, the previous neurological deficit which was misdiagnosed as acute demyelinating neuropathy could have been the result of compression of the pyramidal tracts in the medulla due to a bleed from the cavernoma. The effects due to compression again resolved over time as the bleed was small and the definitive diagnosis was again missed. Our explanation on the causation of these symptoms were confirmed when the MRI showed numerous bleeds of varying ages in the medulla (Fig. 1). Brainstem cavernomas have been reported with initial ENT manifestations such as, hearing loss, sudden tinnitus and facial paraesthesine [2]. Ours is probably the first example of a brainstem cavernoma with vocal cord palsy and respiratory distress. The patient was misdiagnosed twice because of the atypical presentation. We suggest that brain-stem cavernoma should also be included in the aetiology of vocal cord palsy, even though it may be a rare occurrence.
Discussion The characteristic symptoms and signs of a brainstem cavernoma occur after haemorrhage. The first haemorrhage is followed in most patients by relatively mild signs and symptoms such as vertigo, headache, and hemihypoaesthesia. In patients with repeated haemorrhages worsening of neurological deficits with progressive gait disturbances, cranial nerve paresis and long tract disturbances occur. Our patient presented with respiratory symptoms associated with stridor and hoarseness possibly due to bleeding from the angioma in the medulla leading to nuclear and infranuclear vagal palsy.
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