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Long-standing "stiff-man" syndrome: a particular form of disseminated inflammatory CNS disease?

Sir: In this journal we recently reported on neurophysiological and neuropharmacological findings in a patient with the stiff man syndrome followed over 20 years.' This patient has now been re-examined and has a new latent paresis of the left leg with slight lateralization of the deep tendon reflexes, but normal plantar responses. Multimodality evoked potentials were normal and reflex-electromyography demonstrated unchanged abnormal cutaneo-muscular reflexes. However, magnetic resonance imaging (MRI) revealed numerous small diameter (< 1 cm) signal-enhancements at spin echo times of 60 and (less distinct) 120ms, located on both sides exclusively in the frontal and parietal white matter (fig left). In contrast to earlier examinations, moreover, evidence for local IgG-synthesis and positive oligoclonal IgG bands were found in the cerebrospinal fluid (CSF), associated with a normal cell count and total protein as well as albumin. Immunological investigations revealed an increased borrelia-IgG only in serum, but not in CSF. Since 1979 we have followed another patient whose clinical picture and course resembles those of this patient; since 1978 she has suffered from severe stiffness of the trunk and proximal limb muscles which increases with exteroceptive stimuli. Stiffness is superimposed by acute and painful spontaneous myoclonic attacks of the trunk muscles. Muscle stiffness and spasms are completely abolished by diazepam or clonazepam. Symptoms return however, when this medication is interrupted. Repeated examinations of her CSF, cranial computed tomography, and multimodality evoked potentials have been normal. On recent examination, however, MRI showed periventricular white matter lesions (fig right), and oligoclonal banding was found in her CSF. The fact that the diagnosis "stiff man" syndrome has to be revised (after a 10-20 years follow-up with numerous clinical, neurophysiological, CSF, and CCT-investigations) raises several questions. First of all, it underlines that such a (purely descriptive) diagnosis cannot be made appropriately on Letters