Clinical Characteristics of Pruritus in Neurofibromatosis 1

Brenaut, E.; Nizery-Guermeur, Constance; Audebert‐Bellanger, Séverine; Ferkal, Salah; Wolkenstein, Pierre; Miséry, Laurent; Abasq‐Thomas, C.

doi:10.2340/00015555-2241

Cited by 32 publications

(34 citation statements)

References 14 publications

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“…On completion of the first part, patients answered the question ‘Do you have pruritus?’. If the answer was ‘yes’, they completed the second part of the questionnaire, about the characteristics of their pruritus . This part contained two validated scores of pruritus: the 5‐D itch scale and the ItchyQol [a pruritus‐specific quality of life (QoL) instrument] scale …”

mentioning

confidence: 99%

“…If the answer was 'yes', they completed the second part of the questionnaire, about the characteristics of their pruritus. 3,4 This part contained two validated scores of pruritus: the 5-D itch scale 5 and the ItchyQol [a pruritus-specific quality of life (QoL) instrument] scale. 6 The study protocol was approved by the ethics committee of the university hospital.…”

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confidence: 99%

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Itch and systemic sclerosis: frequency, clinical characteristics and consequences

et al. 2017

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confidence: 99%

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confidence: 99%

Itch and systemic sclerosis: frequency, clinical characteristics and consequences

et al. 2017

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“…Neuropathic pruritus is caused by pathology located at any point along the afferent pathway of the nervous system . This can arise due to pathology affecting the peripheral nervous system causing postherpetic neuropathy, brachioradial pruritus or notalgia paraesthetica, or due to lesions affecting pathways of the central nervous system, for example as a result of spinal cord tumours, neurofibromatosis type 1 or multiple sclerosis . Sensory symptoms including burning, paraesthesia, stinging and tingling can accompany neuropathic pruritus …”

Section: Investigation Of Generalized Pruritus Without Rash and Intermentioning

confidence: 99%

“…Nerve fibre compression can cause pruritus in the corresponding dermatome, and nerve fibre degeneration (such as small fibre neuropathy) can cause a localized or generalized pruritus. Small fibre neuropathy can occur in systemic diseases such as diabetes mellitus, Guillain–Barré syndrome, sarcoidosis, neurofibromatosis type 1 and HIV . Diabetic neuropathy can lead to a regional pruritus affecting the trunk .…”

Section: Investigation Of Generalized Pruritus Without Rash and Intermentioning

confidence: 99%

British Association of Dermatologists’ guidelines for the investigation and management of generalized pruritus in adults without an underlying dermatosis, 2018

et al. 2018

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“…Clinical research has demonstrated the prevalence of recurrent pain and bothersome skin sensations as salient symptoms in individuals with NF1 (Bardo‐Brouard et al, ; Brenaut et al, ). Speech difficulties have also been identified as significant neurogenic manifestations of NF1 (Alivuotila et al, ).…”

Section: Introductionmentioning

confidence: 99%

Speech difficulties and patient health communication mediating effects on worry and health‐related quality of life in children, adolescents, and young adults with Neurofibromatosis Type 1

Varni

Nutakki

Swigonski

2019

American J of Med Genetics Pt A

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The objective was to investigate the serial mediating effects of speech difficulties, patient health communication, and disease-specific worry in the relationship between neurofibromatosis (NF) symptoms (pain and skin symptoms) and total generic healthrelated quality of life (HRQOL) in children, adolescents, and young adults with NF Type 1 (NF1) from the patient perspective. The Speech, Communication, Worry, Pain, Skin Itch Bother, and Skin Sensations Scales from the Pediatric Quality of Life Inventory (PedsQL) NF1 Module and the PedsQL 4.0 Generic Core Scales were completed in a multi-site national study by 305 patients ages 5-25 years. A serial multiple mediator model analysis was conducted to test the hypothesized sequential mediating effects of speech difficulties, health communication, and worry as intervening variables in the association between NF1 symptoms and HRQOL. Symptoms predictive effects on total generic HRQOL were serially mediated by speech difficulties, patient health communication, and worry. In predictive analytics models utilizing hierarchical multiple regression analyses with age and gender demographic covariates, the pain, skin itch bother, and skin sensations multiple mediator models accounted for 61%, 59%, and 56% of the variance in generic HRQOL (p < .001), reflecting large effect sizes. Speech difficulties, patient health communication, and disease-specific worry explain in part the mechanism of symptoms predictive effects on total generic HRQOL in pediatric patients with NF1. Identifying NF1-specific predictors and serial mediators of total generic HRQOL in pediatric patients with NF1 from the patient perspective enables a patient-centered comprehensive care approach for children, adolescents, and young adults with NF1. K E Y W O R D S communication, health-related quality of life, neurofibromatosis, patient-reported outcomes, PedsQL, speech Abbreviations: HRQOL, health-related quality of life; NF1, Neurofibromatosis Type 1; PedsQL, Pediatric Quality of Life Inventory.The PedsQL is available at http://www.pedsql.org.

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Clinical Characteristics of Pruritus in Neurofibromatosis 1

Cited by 32 publications

References 14 publications

Itch and systemic sclerosis: frequency, clinical characteristics and consequences

Itch and systemic sclerosis: frequency, clinical characteristics and consequences

British Association of Dermatologists’ guidelines for the investigation and management of generalized pruritus in adults without an underlying dermatosis, 2018

Speech difficulties and patient health communication mediating effects on worry and health‐related quality of life in children, adolescents, and young adults with Neurofibromatosis Type 1

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