Literature DB >> 2361382

The lungs and airways in achondroplasia. Do little people have little lungs?

D C Stokes1, M E Wohl, R A Wise, R E Pyeritz, D L Fairclough.   

Abstract

Achondroplasia is a unique model of the effects of skeletal dysplasia and dwarfism on the respiratory system. We measured chest dimensions, spirometry, lung volumes, maximal expiratory flow volume curves, nasal and airways resistance, closing volume, maximal inspiratory/expiratory pressures, and tracheal area by acoustic reflection in 12 healthy subjects with achondroplasia. Anterior-posterior thoracic diameter was mildly reduced in men. Vital capacity for all subjects was 108 percent +/- 18.6 percent (SD) of that predicted for achondroplastic subjects, but was reduced when compared with values for people of average stature that were predicted, based on either sitting height or thoracic height. The reduction was relatively greater in male than in female subjects. The RV/TLC and FRC/TLC ratios were normal. Other measurements were similar to those in average-statured adults. We conclude that achondroplasia results in a reduction in vital capacity out of proportion to what would be expected if these subjects had normal limb size. Although the lungs may be small, they are functionally normal, as are the airways.

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Year:  1990        PMID: 2361382     DOI: 10.1378/chest.98.1.145

Source DB:  PubMed          Journal:  Chest        ISSN: 0012-3692            Impact factor:   9.410


  6 in total

1.  Non-invasive pulmonary function test on Morquio patients.

Authors:  Francyne Kubaski; Shunji Tomatsu; Pravin Patel; Tsutomu Shimada; Li Xie; Eriko Yasuda; Robert Mason; William G Mackenzie; Mary Theroux; Michael B Bober; Helen M Oldham; Tadao Orii; Thomas H Shaffer
Journal:  Mol Genet Metab       Date:  2015-06-23       Impact factor: 4.797

2.  Distinct patterns of respiratory difficulty in young children with achondroplasia: a clinical, sleep, and lung function study.

Authors:  R C Tasker; I Dundas; A Laverty; M Fletcher; R Lane; J Stocks
Journal:  Arch Dis Child       Date:  1998-08       Impact factor: 3.791

3.  Enzyme replacement therapy for mucopolysaccharidosis VI: evaluation of long-term pulmonary function in patients treated with recombinant human N-acetylgalactosamine 4-sulfatase.

Authors:  Paul Harmatz; Zi-Fan Yu; Roberto Giugliani; Ida Vanessa D Schwartz; Nathalie Guffon; Elisa Leão Teles; M Clara Sá Miranda; J Edmond Wraith; Michael Beck; Laila Arash; Maurizio Scarpa; David Ketteridge; John J Hopwood; Barbara Plecko; Robert Steiner; Chester B Whitley; Paige Kaplan; Stuart J Swiedler; Karen Hardy; Kenneth I Berger; Celeste Decker
Journal:  J Inherit Metab Dis       Date:  2010-02-06       Impact factor: 4.982

Review 4.  Current knowledge of medical complications in adults with achondroplasia: A scoping review.

Authors:  Svein O Fredwall; Grethe Maanum; Heidi Johansen; Hildegun Snekkevik; Ravi Savarirayan; Ingeborg B Lidal
Journal:  Clin Genet       Date:  2019-04-22       Impact factor: 4.438

5.  Emergency anesthetic management of an achondroplastic elderly gravida with polytrauma.

Authors:  Abhay Tyagi; Christopher Dass; Nageswara Tangirala Rao; Kapil Dev Soni
Journal:  Int J Crit Illn Inj Sci       Date:  2019-12-11

6.  Physical fitness and activity level in Norwegian adults with achondroplasia.

Authors:  Olga Marieke de Vries; Heidi Johansen; Svein Otto Fredwall
Journal:  Am J Med Genet A       Date:  2020-12-31       Impact factor: 2.802

  6 in total

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