Literature DB >> 27000762

Multidisciplinary assessment of congenital insensitivity to pain syndrome.

Vugar Nabiyev1, Ateş Kara2, M Cemalettin Aksoy3.   

Abstract

BACKGROUND: Congenital insensitivity to pain and anhidrosis (CIPA) is a rare clinical condition characterized by the absence of normal subjective and objective responses to noxious stimuli in patients with intact central and peripheral nervous systems. CASE PRESENTATIONS: Two patients with CIPA are reported. The first patient was a 13-year-old girl who presented to our hospital with multiple joint destructions secondary to osteomyelitis. The second patient was a 10-year-old boy who presented with multiple hand lesions and right leg osteomyelitis. Our patients were treated with multiple debridements and intravenous antibiotics according to our hospital protocol.
CONCLUSION: Early recognition of the disease is important. The treatment for this condition is focused more on the prevention of bone injuries and joint infection, as opposed to a cure. There are no standard techniques or guidelines available to treat this rare disease. Overall, effective CIPA treatment is built around family education and patient training.

Entities:  

Keywords:  Anhidrosis; Congenital insensitivity to pain; Neuropathy; Pain insensitivity; Sensory

Mesh:

Substances:

Year:  2016        PMID: 27000762     DOI: 10.1007/s00381-016-3059-5

Source DB:  PubMed          Journal:  Childs Nerv Syst        ISSN: 0256-7040            Impact factor:   1.475


  22 in total

1.  Congenital insensitivity to pain with anhydrosis. A unique syndrome in two male siblings.

Authors:  A G SWANSON
Journal:  Arch Neurol       Date:  1963-03

2.  Congenital idiopathic inability to perceive pain: a new syndrome of insensitivity to pain and itch with preserved small fibers.

Authors:  Paola Sandroni; David P Martin; Barbara K Bruce; Jeffrey D Rome
Journal:  Pain       Date:  2006-03-09       Impact factor: 6.961

3.  Skeletal complications in congenital insensitivity to pain with anhidrosis: a case series of 14 patients and review of articles published in Japanese.

Authors:  Yasu Zhang; Nobuhiko Haga
Journal:  J Orthop Sci       Date:  2014-06-23       Impact factor: 1.601

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Authors:  C Bronfen; H Bensahel; J G Teule
Journal:  Chir Pediatr       Date:  1985

5.  Neurogenic joint disease secondary to congenital insensitivity to pain.

Authors:  F Gherlinzoni; G Gherlinzoni
Journal:  Ital J Orthop Traumatol       Date:  1982-12

Review 6.  Hereditary motor and sensory neuropathies: Understanding molecular pathogenesis could lead to future treatment strategies.

Authors:  Nivedita U Jerath; Michael E Shy
Journal:  Biochim Biophys Acta       Date:  2014-08-06

7.  Orthopedic aspects of congenital insensitivity to pain.

Authors:  T D Greider
Journal:  Clin Orthop Relat Res       Date:  1983 Jan-Feb       Impact factor: 4.176

8.  Pain insensitivity syndrome misinterpreted as inflicted burns.

Authors:  Gerbrich E van den Bosch; Martin G A Baartmans; Paul Vos; Jan Dokter; Tonya White; Dick Tibboel
Journal:  Pediatrics       Date:  2014-04-14       Impact factor: 7.124

9.  A novel lymphocyte signaling defect: trk A mutation in the syndrome of congenital insensitivity to pain and anhidrosis (CIPA).

Authors:  I Melamed; J Levy; R Parvari; E W Gelfand
Journal:  J Clin Immunol       Date:  2004-07       Impact factor: 8.317

10.  Update Review and Clinical Presentation in Congenital Insensitivity to Pain and Anhidrosis.

Authors:  L M Pérez-López; M Cabrera-González; D Gutiérrez-de la Iglesia; S Ricart; G Knörr-Giménez
Journal:  Case Rep Pediatr       Date:  2015-10-22
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  3 in total

1.  The Orthopedic Manifestations of Congenital Insensitivity to Pain: A Population-based Study.

Authors:  Maximillian Mifsud; Michelle Spiteri; Karl Camilleri; Matthew Bonello; Thomas Azzopardi; Massimo Abela
Journal:  Indian J Orthop       Date:  2019 Sep-Oct       Impact factor: 1.251

2.  Congenital Insensitivity to Pain with Anhidrosis: A Rare Entity.

Authors:  Priya Rajbansh; Mamta Yadav; Piyush Kumar; Anupam Das
Journal:  Indian Dermatol Online J       Date:  2020-03-09

3.  Population Study of Hand and Wrist Manifestations of Congenital Insensitivity to Pain.

Authors:  Michelle Spiteri; Maximillian Mifsud; Thomas Azzopardi; Henk Giele
Journal:  Hand (N Y)       Date:  2020-03-06
  3 in total

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