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Chronic immune sensory polyradiculopathy (CISP): First juvenile case description.

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Title: Chronic immune sensory polyradiculopathy (CISP): First juvenile case description.
Authors: Sotgiu, Stefano (AUTHOR), Minutolo, A. (AUTHOR), Carta, A. (AUTHOR), Puseddu, G. (AUTHOR), Doneddu, P. E. (AUTHOR), Nobile-Orazio, E. (AUTHOR)
Source: Neurological Sciences. 2021, Vol. 42 Issue 1, p333-336. 4p. 1 Chart, 1 Graph.
Subjects: Polyradiculopathy, Polyneuropathies, Peripheral nervous system, Neural conduction, Tibial nerve, Somatosensory evoked potentials
Abstract: In its typical presentation, chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) occurs more often in old males as a progressive/recurrent motor and sensory nerve dysfunction with tendon areflexia. However, CIDP has also atypical clinical presentations, including pure sensory neuropathies, among which chronic immune sensory polyradiculopathy (CISP) accounts for only 0.5% of all CIDP, with no juvenile cases reported as yet. A 17-year-old girl presented for a progressive sensory ataxia and hands clumsiness. Diffuse tendon areflexia and hypokinaesthesia were observed. Motor and sensory nerve conduction studies were normal. F-waves were normal in median nerves and elongated in tibial nerves. H-reflex and somatosensory evoked potentials (SSEP) were absent. CSF normal cellularity with hyperproteinorrachia was found. Paraneoplastic, metabolic, and paraproteinemic neuropathies were excluded. A diagnosis of CISP has been made based on the presence of pure sensory symptoms in a polyneuropathic distribution, normal peripheral nerve conduction studies, and two supportive criteria (SSEP and CSF). Our paper describes the first CISP case in the pediatric age. We confirm SSEP and CSF as useful complementary tests for this diagnosis also at this age and suggest that clinicians should consider CISP in the spectrum of sporadic sensory ataxias of the pediatric age. We also suggest that in the presence of normal F-wave and peripheral motor nerve conduction, an absent H-reflex can further substantiate SSEPs in the diagnosis of CISP. Intravenous immunoglobulins were rapidly effective and safe. [ABSTRACT FROM AUTHOR]
Copyright of Neurological Sciences is the property of Springer Nature and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
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  Data: Chronic immune sensory polyradiculopathy (CISP): First juvenile case description.
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  Data: <searchLink fieldCode="AR" term="%22Sotgiu%2C+Stefano%22">Sotgiu, Stefano</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Minutolo%2C+A%2E%22">Minutolo, A.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Carta%2C+A%2E%22">Carta, A.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Puseddu%2C+G%2E%22">Puseddu, G.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Doneddu%2C+P%2E+E%2E%22">Doneddu, P. E.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Nobile-Orazio%2C+E%2E%22">Nobile-Orazio, E.</searchLink> (AUTHOR)
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  Data: <searchLink fieldCode="JN" term="%22Neurological+Sciences%22">Neurological Sciences</searchLink>. 2021, Vol. 42 Issue 1, p333-336. 4p. 1 Chart, 1 Graph.
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  Data: <searchLink fieldCode="DE" term="%22Polyradiculopathy%22">Polyradiculopathy</searchLink><br /><searchLink fieldCode="DE" term="%22Polyneuropathies%22">Polyneuropathies</searchLink><br /><searchLink fieldCode="DE" term="%22Peripheral+nervous+system%22">Peripheral nervous system</searchLink><br /><searchLink fieldCode="DE" term="%22Neural+conduction%22">Neural conduction</searchLink><br /><searchLink fieldCode="DE" term="%22Tibial+nerve%22">Tibial nerve</searchLink><br /><searchLink fieldCode="DE" term="%22Somatosensory+evoked+potentials%22">Somatosensory evoked potentials</searchLink>
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  Data: In its typical presentation, chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) occurs more often in old males as a progressive/recurrent motor and sensory nerve dysfunction with tendon areflexia. However, CIDP has also atypical clinical presentations, including pure sensory neuropathies, among which chronic immune sensory polyradiculopathy (CISP) accounts for only 0.5% of all CIDP, with no juvenile cases reported as yet. A 17-year-old girl presented for a progressive sensory ataxia and hands clumsiness. Diffuse tendon areflexia and hypokinaesthesia were observed. Motor and sensory nerve conduction studies were normal. F-waves were normal in median nerves and elongated in tibial nerves. H-reflex and somatosensory evoked potentials (SSEP) were absent. CSF normal cellularity with hyperproteinorrachia was found. Paraneoplastic, metabolic, and paraproteinemic neuropathies were excluded. A diagnosis of CISP has been made based on the presence of pure sensory symptoms in a polyneuropathic distribution, normal peripheral nerve conduction studies, and two supportive criteria (SSEP and CSF). Our paper describes the first CISP case in the pediatric age. We confirm SSEP and CSF as useful complementary tests for this diagnosis also at this age and suggest that clinicians should consider CISP in the spectrum of sporadic sensory ataxias of the pediatric age. We also suggest that in the presence of normal F-wave and peripheral motor nerve conduction, an absent H-reflex can further substantiate SSEPs in the diagnosis of CISP. Intravenous immunoglobulins were rapidly effective and safe. [ABSTRACT FROM AUTHOR]
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  Data: <i>Copyright of Neurological Sciences is the property of Springer Nature and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.)
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