Literature DB >> 4041956

Anaesthetic implications of nemaline rod myopathy.

M Cunliffe, F A Burrows.   

Abstract

Nemaline rod myopathy is an inherited congenital myopathy first described in 1963. Affected patients characteristically present in infancy with a non-progressive hypotonia and symmetrical muscle weakness. The disease affects all skeletal muscles including the diaphragm with sparing of cardiac and other muscle. Facial dysmorphism is common as are skeletal deformities, including kyphosis, scoliosis and pectus excavatum. We present two sisters with nemaline rod myopathy and their anaesthetic management for scoliosis surgery. Facial dysmorphism was a feature of both cases. Preoperatively, both patients demonstrated poor respiratory function on pulmonary function testing. Both cases were successfully managed using controlled ventilation and inhalational anaesthetic agents, avoiding muscle relaxants. Postoperatively, there were no respiratory complications. Although one case report describes the use of succinylcholine and pancuronium in a patient with nemaline rod myopathy, we feel that neuromuscular blocking agents should be avoided where possible and only used with careful monitoring.

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Year:  1985        PMID: 4041956     DOI: 10.1007/bf03010806

Source DB:  PubMed          Journal:  Can Anaesth Soc J        ISSN: 0008-2856


  15 in total

1.  NEMALINE MYOPATHY; A SECOND CASE.

Authors:  W K ENGEL; T WANKO; G M FENICHEL
Journal:  Arch Neurol       Date:  1964-07

2.  CONGENITAL NONPROGRESSIVE MYOPATHY. CENTRAL CORE DISEASE AND NEMALINE MYOPATHY IN ONE FAMILY.

Authors:  A K AFIFI; J W SMITH; H ZELLWEGER
Journal:  Neurology       Date:  1965-04       Impact factor: 9.910

3.  Nemaline myopathy. A fatal case.

Authors:  I S Kolin
Journal:  Am J Dis Child       Date:  1967-07

4.  Nemaline myopathy: report of a fatal case, with histochemical and electron microscopic studies.

Authors:  S A Shafiq; V Dubowitz; H de C Peterson; A T Milhorat
Journal:  Brain       Date:  1967-12       Impact factor: 13.501

5.  Nemaline myopathy.

Authors:  P Hudgson; D Gardner-Medwin; J J Fulthorpe; J N Walton
Journal:  Neurology       Date:  1967-12       Impact factor: 9.910

6.  Neuromuscular blockade in a patient with nemaline myopathy.

Authors:  S O Heard; R F Kaplan
Journal:  Anesthesiology       Date:  1983-12       Impact factor: 7.892

7.  Malignant hyperthermia and central core disease in a child with congenital dislocating hips.

Authors:  G D Eng; B S Epstein; W K Engel; D W McKay; R McKay
Journal:  Arch Neurol       Date:  1978-04

8.  Nemaline myopathy. A long-term clinicopathologic study of affected mother and daughter.

Authors:  I J Hopkins; J R Lindsey; F R Ford
Journal:  Brain       Date:  1966-06       Impact factor: 13.501

9.  Nemaline myopathy. The origin of nemaline structures.

Authors:  N K Gonatas; G M Shy; E H Godfrey
Journal:  N Engl J Med       Date:  1966-03-10       Impact factor: 91.245

10.  LIGHT AND ELECTRON MICROSCOPIC STUDIES OF "MYOGRANULES" IN A CHILD WITH HYPOTONIA AND MUSCLE WEAKNESS.

Authors:  P E CONEN; E G MURPHY; W L DONOHUE
Journal:  Can Med Assoc J       Date:  1963-11-09       Impact factor: 8.262

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  2 in total

Review 1.  Nemaline myopathy: current concepts. The ENMC International Consortium and Nemaline Myopathy.

Authors:  K N North; N G Laing; C Wallgren-Pettersson
Journal:  J Med Genet       Date:  1997-09       Impact factor: 6.318

2.  Mutation update: the spectra of nebulin variants and associated myopathies.

Authors:  Vilma-Lotta Lehtokari; Kirsi Kiiski; Sarah A Sandaradura; Jocelyn Laporte; Pauliina Repo; Jennifer A Frey; Kati Donner; Minttu Marttila; Carol Saunders; Peter G Barth; Johan T den Dunnen; Alan H Beggs; Nigel F Clarke; Kathryn N North; Nigel G Laing; Norma B Romero; Thomas L Winder; Katarina Pelin; Carina Wallgren-Pettersson
Journal:  Hum Mutat       Date:  2014-12       Impact factor: 4.878

  2 in total

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