We present a 37-year-old male subject who presented with burning sensations in his hands and feet with generalised twitching of his limbs trunk and face. nerve hyperexcitability with spontaneous and continuous muscle fibre activity [1]. It has variously been called undulating myokymia Isaac’s syndrome and cramp-fasciculation syndrome [2]. It might be hereditary or acquired and there have been a variety of reported causes and associations. Only three cases have been however been attributed to infective causes in the literature. We present Jolkinolide B a case of acquired neuromyotonia following an upper respiratory tract infection and discuss the literature relevant to the case. Case presentation Our patient is a 37-year-old Caucasian man who presented with a fourteen-month history of a burning painful sensation in his hands and feet. This developed about two weeks after an upper respiratory tract infection. His symptoms were present all the time but were exacerbated by activity and stress. There was associated twitching of his hands and face as well as regular facial flushing and disturbed night sleep. He had no memory impairment or sweating disturbances. He has a past history of mild renal dysfunction and is on antibiotics for a persistent cellulitis over his left elbow. He does not smoke and takes only minimal alcohol. On examination he had generalised myokymia and fasciculations involving all parts of his face tongue trunk and all four limbs. Power coordination and reflexes were normal. He had impaired pain sensation to his ankles and wrists; other modalities of sensation were normal. Electromyography (EMG) revealed widespread continuous semi-rhythmic doublets and triplets of low frequency with interspersed silent periods. There were fasciculations in the right tibialis anterior and right opponens pollicis with some polyphasia in the tibialis anterior muscle. Motor and sensory nerve conduction velocities were normal. He tested negative for anti voltage gated potassium channel (VGKC) antibodies antinuclear antibodies and anti-acetylcholine receptor antibodies. Jolkinolide B He was also negative for the anti-neuronal antibodies anti Hu anti Yo and anti Ri. His creatinine kinase was mildly elevated at 474 U/L as was serum creatinine at 111-micromol/L. eGFR was mildly reduced at 67. Computerised tomography scan of his chest was normal. He was initially treated with gabapentin with no benefit. He had improvement in pain and sleep disturbance on 90 Jolkinolide B mg daily dose of amitriptyline. He also reported further improvement in sensory symptoms Mouse monoclonal to MCL-1 on Lamotrigine but not in motor symptoms. Discussion Our patient had most of the typical features of neuromyotonia. The major manifestations Jolkinolide B are cramps muscle twitching or stiffness pseudomyotonia paraesthesias numbness and hyperhydrosis [2]. The commonest feature is muscle twitching which is present in over 90% of cases; facial twitching is seen in a quarter of cases [1]. Our patient had both limb and facial involvement. Central nervous system features are Jolkinolide B seen in a quarter of cases of neuromyotonia and these include Jolkinolide B mood change sleep disorders hallucinations and personality changes [2]. Our patient however did not report central nervous system symptoms. The only risk factor for neuromyotonia in our patient appears to be the upper respiratory tract infection he had two weeks prior to onset of symptoms. The elbow cellulitis is unlikely to be responsible because there was no temporal relationship to the onset of Neuromyotonia. There have been case reports of neuromyotonia following spinal epidural abscess with septicaemia [3 4 and there is also a report of ocular neuromyotonia following cavernous sinus thrombosis due to mucormycosis [5]. In one of these cases antibodies to voltage-gated potassium channels were demonstrated during the infection and these disappeared after resolution of the infection [3]. Although these antibodies were negative in our patient they were only tested for after he had recovered from the respiratory tract infection. It is therefore possible that similar antibodies underlie the pathogenesis in our patient and other cases resulting from infective causes. Infections are however uncommon reported causes of neuromyotonia. More frequently reported are associations with autoimmune diseases including myasthenia gravis systemic lupus erythematosus.