Literature DB >> 26189194

Hereditary neuropathy with liability to pressure palsies in childhood: Case series and literature update.

Nicolas Chrestian1, Hugh McMillan2, Chantal Poulin3, Craig Campbell4, Jiri Vajsar5.   

Abstract

Hereditary Neuropathy with Liability to Pressure Palsy (HNPP) is a rare condition in childhood with a diverse range of clinical presentations. We analyzed the clinical presentation and electrophysiological data of 12 children with a confirmed PMP22 gene deletion and reviewed the published reports of HNPP in children and compared our data with the reports from the literature review. Peroneal palsy was the most common presentation (42%) followed by brachial plexus palsy in 25% of our cases. Nerve conduction studies were always suggestive of the diagnosis demonstrating 3 major patterns: multifocal demyelination at the area of entrapment, generalized sensory-motor polyneuropathy and a combination of the two first patterns in a vast majority (60%). Surprisingly, there was bilateral or unilateral electrophysiological entrapment of the median nerve at the carpal tunnel in all our patients. The clinical presentation of HNPP in childhood is heterogeneous and electrophysiological findings are helpful in establishing the diagnosis. Any unexplained mononeuropathy or multifocal neuropathy should lead to PMP22 gene testing to look for the deletion. Early diagnosis is important in order to facilitate appropriate genetic counseling and also for the appropriate care for these patients.
Copyright © 2015 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Childhood; HNPP; Hereditary; Mononeuropathies; PMP22 protein

Mesh:

Substances:

Year:  2015        PMID: 26189194     DOI: 10.1016/j.nmd.2015.06.463

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  6 in total

1.  Hereditary neuropathy with liability to pressure palsies (HNPP): Intrafamilial phenotypic variability and early childhood refusal to walk as the presenting symptom.

Authors:  Shani Karklinsky; Shir Kugler; Omer Bar-Yosef; Andreea Nissenkorn; Anat Grossman-Jonish; Irit Tirosh; Asaf Vivante; Ben Pode-Shakked
Journal:  Ital J Pediatr       Date:  2022-06-03       Impact factor: 3.288

2.  Nonrecurrent PMP22-RAI1 contiguous gene deletions arise from replication-based mechanisms and result in Smith-Magenis syndrome with evident peripheral neuropathy.

Authors:  Bo Yuan; Juanita Neira; Shen Gu; Tamar Harel; Pengfei Liu; Ignacio Briceño; Sarah H Elsea; Alberto Gómez; Lorraine Potocki; James R Lupski
Journal:  Hum Genet       Date:  2016-07-07       Impact factor: 4.132

3.  Hereditary Neuropathy With Liability to Pressure Palsies: A Single-Center Experience in Southern Brazil.

Authors:  Paulo José Lorenzoni; Cláudia Suemi Kamoi Kay; Cristiane Cavalet; Raquel C Arndt; Lineu Cesar Werneck; Rosana Herminia Scola
Journal:  Neurol Int       Date:  2016-09-30

Review 4.  Peripheral neuropathy and gastroenterologic disorders: an overview on an underrecognized association.

Authors:  Carlotta Spagnoli; Francesco Pisani; Francesco Di Mario; Gioacchino Leandro; Federica Gaiani; Gian Luigi De' Angelis; Carlo Fusco
Journal:  Acta Biomed       Date:  2018-12-17

5.  Neuralgic Amyotrophy with Concomitant Hereditary Neuropathy with Liability to Pressure Palsy as a Cause of Dropped Shoulder in a Child after Human Papillomavirus Vaccination: A Case Report.

Authors:  Hye-Chan Ahn; Do-Hoon Kim; Chul-Hyun Cho; Jun-Chul Byun; Jang-Hyuk Cho
Journal:  Children (Basel)       Date:  2022-04-07

6.  Hereditary neuropathy with liability to pressure palsy (HNPP): report of a family with a new point mutation in PMP22 gene.

Authors:  Carlo Fusco; Carlotta Spagnoli; Grazia Gabriella Salerno; Elena Pavlidis; Daniele Frattini; Francesco Pisani
Journal:  Ital J Pediatr       Date:  2017-10-27       Impact factor: 2.638

  6 in total

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