Chronic inflammatory demyelinating polyneuropathy: Plasmapheresis or cyclosporine can be good treatment options in refractory cases

Kim, Woo Joong; Shim, Young Kyu; Choi, Sun Ah; Kim, Soo Yeon; Kim, Hunmin; Lim, Byung Chan; Choi, Jieun; Kim, Ki Joong; Chae, Jong Hee

doi:10.1016/j.nmd.2019.06.010

Cited by 8 publications

(11 citation statements)

References 45 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…No other significant AEs following AZA or MTX were observed. Our data are in accordance with the study by Kim et al (38); although limited by the small size of the group, they provide a new option for treatment-resistant patients, including the administration of AZA, MTX, RTX, cyclosporine, and plasmapheresis to refractory patients. Thus, in order to implement these treatments, early referral to experienced neuromuscular centers may be crucial for the choice of an appropriate intervention to prevent irreversible axonal damage.…”

Section: Discussionsupporting

confidence: 92%

Pediatric CIDP: Diagnosis and Management. A Single-Center Experience

et al. 2021

View full text Add to dashboard Cite

Background: Chronic inflammatory demyelinating polyneuropathy (CIDP) is a rare acquired polyneuropathy that especially among youngest children should be differentiated with hereditary neuropathies. Even though upon diagnosis treatment options are similar in children and adults, diagnostic challenges are faced in the pediatric population.Methods: We conducted a retrospective analysis of clinical symptoms, nerve conduction study results, modes of treatment, and final outcome in 37 children aged 3.5–17 years with a final diagnosis of CIDP (18 girls, 19 boys). We established three groups of patients based on age at onset of CIDP: 0–4, 4–13, and 13–18 years. Follow-up ranged from 10 to 222 months.Results: In our analysis, 19/37 patients (51.4%) had an atypical presentation: distal variant of CIDP in 12/37 patients (32.4%) and pure motor variant of CIDP in 5/37 patients (13.5%), and one patient had a pure sensory variant (1/37, 2.7%). Furthermore, 3/37 patients (8.1%) had additional concurring symptoms, including involuntary movements of face muscles (1/37, 2.7%) or hand tremor (2/37, 5.4%). During the follow-up, 23/37 patients (62.2%) received intravenous immunoglobulin (IVIg); 22/37 patients (59.5%) received steroids, 6/37 patients (16.2%) received IVIg and steroids, and 12/37 patients (32.4%) received immunosuppressive drugs, mostly azathioprine, but also methotrexate and rituximab. One patient was treated with plasmapheresis. Complete remission was achieved in 19/37 patients (51.4%) with CIDP in its typical form. Remission with residual symptoms or minimal deficit was observed in 4/37 patients (10.8%), whereas 14/37 patients (37.8%) remain on treatment with gradual improvement.Conclusion: Childhood CIDP may occur in its typical form, but even ~50% of children can present as an atypical variant including distal, pure motor, or pure sensory. Most children have a good prognosis; however, many of them may require long-term treatment. This highlights the importance of an early diagnosis and treatment for childhood CIDP.

show abstract

Section: Discussionsupporting

confidence: 92%

Pediatric CIDP: Diagnosis and Management. A Single-Center Experience

et al. 2021

View full text Add to dashboard Cite

show abstract

“…Our study results indicate that serial grip strength measurements are a feasible and objective way to assess motor strength improvement in children with CIDP receiving immunotherapy. In our study, the participant demographics matched those of previous published pediatric CIDP case series in terms of gender distribution and the wide age range at diagnosis 1,2,7–9,16–21 . A few studies reported more males 2,18 .…”

Section: Discussionsupporting

confidence: 68%

Outcome measures in pediatric chronic inflammatory demyelinating polyradiculoneuropathy

Guttikonda,

Ahmad,

Goyal

et al. 2024

Muscle and Nerve

View full text Add to dashboard Cite

Introduction/AimsObjective outcome measures in children undergoing treatment for chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) are lacking. The aim of the study was to record serial grip strength and motor nerve conduction studies to assess interval change.MethodsThis was a retrospective review of 16 children (8 females and 8 males; median age, 9.7 years; interquartile range, 6–13 years) with CIDP followed at a tertiary children's hospital from 2013 to 2021. Subjects were treated with intravenous immunoglobulin (IVIG). Right and left grip strength measurements were obtained at each clinic visit using a handheld dynamometer. Annual right median motor nerve conduction study data were recorded during the study period.ResultsMean duration of follow‐up was 2.9 years. Grip strength (right: 0.19 kg/month, p < 0.001; left 0.23 kg/month, p < 0.001) and median F‐wave latencies (−0.23/month, p = 0.015) showed significant improvement over time. Akaike information criterion showed time + IVIG frequency <21 days as best fit for grip strength and distal compound muscle action potential amplitude.DiscussionOur study results indicate serial grip strength measurements are a feasible and objective way to assess motor strength improvement in children with CIDP receiving immunotherapy.

show abstract

“…Data from the paediatric studies were inconsistent, with McMillan et al 57 reporting a higher proportion of responders among IVIg- than TPE-treated patients; Kim et al 56 found that TPE and IVIg were similarly efficacious, but TPE was likely a more favoured option for IVIg- and corticosteroid-refractory CIDP. These studies also reported different clinical experiences with TPE administration.…”

Section: Resultsmentioning

confidence: 99%

Comparison of IVIg and TPE efficacy in the treatment of neurological disorders: a systematic literature review

Pinto

Sèze

Jacob

et al. 2023

Ther Adv Neurol Disord

View full text Add to dashboard Cite

Background: Intravenous immunoglobulin (IVIg) and therapeutic plasma exchange (TPE) are among the main immunotherapies for neurological disorders. Their benefit is greatest in immune-mediated conditions, but their distinct efficacy cannot be simply explained. Objectives: This review aimed to systematically identify studies comparing the efficacy of TPE and IVIg treatments for selected autoimmune neurological disorders and identify optimal therapies for each condition. Data Sources and Methods: PubMed, MEDLINE and Embase databases were searched for original publications from 1990 to 2021. Additional publications were identified via expert recommendations. Conference abstracts older than 2017, review articles and articles without information on TPE and IVIg comparison in title and abstract were excluded. Risks of bias were descriptively addressed, without a meta-analysis. Results: Forty-four studies were included on Guillain–Barré syndrome (20 studies – 12 adult, 5 paediatric, 3 all ages), myasthenia gravis (11 studies –8 adult, 3 paediatric), chronic immune–mediated polyradiculoneuropathy (3 studies –1 adult, 2 paediatric), encephalitis (1 study in adults), neuromyelitis optica spectrum disorders (5 studies –2 adult, 3 all ages) and other conditions (4 studies – all ages). TPE and IVIg were mostly similarly efficacious, measured by clinical outcomes and disease severity scores. Some studies recommended IVIg as easy to administer. TPE procedures, however, have been simplified and the safety has been improved. TPE is currently recommended for management of neuromyelitis optica spectrum disorder relapses and some myasthenia gravis subtypes, in which rapid removal of autoantibodies is crucial. Conclusion: Despite some limitations (e.g. the low evidence levels), this review provides an extensive 30-year-long overview of treatments for various conditions. Both IVIg and TPE are usually comparably efficacious options for autoimmune neurological disorders, with few exceptions. Treatment choices should be patient-tailored and based on available clinical resources. Better designed studies are needed to provide higher-level quality of evidence regarding clinical efficacy of TPE and IVIg treatments.

show abstract

Chronic inflammatory demyelinating polyneuropathy: Plasmapheresis or cyclosporine can be good treatment options in refractory cases

Cited by 8 publications

References 45 publications

Pediatric CIDP: Diagnosis and Management. A Single-Center Experience

Pediatric CIDP: Diagnosis and Management. A Single-Center Experience

Outcome measures in pediatric chronic inflammatory demyelinating polyradiculoneuropathy

Comparison of IVIg and TPE efficacy in the treatment of neurological disorders: a systematic literature review

Contact Info

Product

Resources

About