Short-lasting unilateral neuralgiform headache attacks with conjunctival injection and tearing (SUNCT) syndrome is a rare headache syndrome classified among the trigeminal autonomic cephalalgias. It is usually idiopathic, although infrequent secondary forms have been described. Recently the term short-lasting unilateral headache with cranial autonomic symptoms (SUNA) has been defined by the International Headache Society (ICHD-2) as similar to SUNCT with less prominent or absent conjunctival injection and lacrimation. We report two patients with paroxysmal orbito-fronto-temporal pains, phenotypically suggesting SUNA, occurring after traumatic head injury.
A 32-year-old lady was admitted with sudden-onset left-sided headache followed rapidly by left-sided in-coordination and weakness. Initial examination revealed right hemi-sensory loss to pain and temperature sparing the face, left hemi-facial numbness and left-sided Horner's syndrome with left facial and palatal weakness. There was marked in-coordination of the lower limbs, the patient having a wide-based stance and gait. She had brisk deep tendon reflexes and extensor plantar reflexes bilaterally.Magnetic resonance imaging of the brain ( Fig. 1) revealed left medullary infarct and a magnetic resonance angiogram demonstrated left vertebral artery dissection with involvement of the posterior cerebellar artery. Because she had had a mildly raised temperature on admission, a cerebrospinal fluid examination was performed, which showed no cells, normal protein and glucose. The patient was anticoagulated with warfarin.The patient continued to experience burning dysaesthesias involving the right side of the body, which was partially relieved by gabapentin (900 mg/day). Three to four weeks following the acute event she developed intermittent short-lasting left-sided headache lasting 5-300 s accompanied by left-sided ptosis, pupillary constriction as well as conjunctival injection. She was left with mild residual ptosis and miosis which got significantly worse during the attacks. However, there was no clear history of any excessive lacrimation. The headaches were mainly during the day, mainly involving the left orbito-frontal and parietal regions, happening at a frequency of three to five times daily. She denied having any triggers for these attacks. The truncal dysaesthesias improved significantly and were unrelated to the headaches.There was no past history or family history of headaches. The headaches partially responded to gabapentin (prescribed initially for right-sided dysaesthesia), later becoming resistant to it, at doses of up to 2700 mg/day. Other medications tried include pregabalin (up to 600 mg/day) and indomathacin (up to 150 mg/day), both of which had had a short-term effect. The headaches are currently well controlled (but with very infrequent relapses) using lamotrigine 100 mg/day in two divided doses. Attempts to reduce or withdraw the medication were resisted by the patient, fearing relapse of further attacks.
Introduction Brachial Neuritis also called as neuralgic amyotrophy is an idiopathic condition which involves the brachial plexus. It usually affects young to middle aged subjects. Its classical presentation involves painful progressive asymmetrical upper limb weakness with sensory abnormalities. Herein we present a rare form of brachial neuritis which started of after endurance exercise complicated by type 2 respiratory failure due to diaphragmatic weakness. Case Presentation This 55-year-old man presented with rapid onset interscapular pain radiating to both shoulders after strenuous endurance exercise on an exercise bike. He complained of breathlessness associated with the pain, on lying down. He was admitted to the acute respiratory ward for assessment. His worsened steadily and was not able to lie flat. He was unable to lift his arms above his shoulders and he described severe radicular in both arms. His Vital capacity was 1.6 liter on standing and 0.9 liter on lying flat (figure 1). Oxygen saturation was normal upright but dropped in the horizontal position, with significant hypoxia and mild acute carbon dioxide retention. EMG/NCS Nerve conduction studies showed reduced median compound muscle action potentials and reduced median and ulnar sensory action potentials bilaterally. Needle EMG showed widespread features of denervation in keeping with bilateral brachial plexus involvement. Management and Progress He was treated with two courses of Methyl prednisolone after which his symptoms started to plateau. He was still however unable to lie flat for an MRI scan of the brachial plexus. Discussion Neuralgic amyotrophy or Brachial neuritis is a distinct condition associated with acute onset shoulder pain, weakness and sensory symptoms. This condition was first described in 1896 in a man with bilateral shoulder weakness with winging of scapula. However, the first and most descriptive paper was from Parsonage and Turner in 1948 were they described 136 cases with a new name called the neuralgic amyotrophy the shoulder girdle syndrome. There are several conditions which were associated with brachial plexopathy. Classical Neuralgic amyotrophy or Brachial neuritis is a type of brachial plexopathy which is rare and idiopathic. Here we describe a very rare sub-type with diaphragmatic paralysis. A literature search revealed up to 70 similar case reports with diaphragmatic involvement. Clinically most of the cases had been described as our patient's presentation with some variations. Most interestingly only a single report, to our knowledge, described onset after exertion, as in our patient. Use of lying and standing vital capacity had been one of the useful investigations which helped in identifying the diaphragmatic failure. The neurophysiology, chest x ray and neuroradiological investigations may help in diagnosing the condition hence high index of suspicion is important. Our patient stopped deteriorating after 2nd course of methyl prednisolone, which may suggest an effect on corticosteroids. Generally long-term fu...
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