Literature DB >> 30294725

Non-invasive pulmonary function test on Morquio patients.

Caitlin Doherty1, Francyne Kubaski2,3, Shunji Tomatsu3,4,5, Thomas H Shaffer3,6.   

Abstract

Morquio patients, in many cases, present with severe tracheal narrowing and restrictive lung problems making them susceptible to high mortality arising from sleep apnea and related complications. Tracheal obstruction with growth imbalance, short neck, adeno and tonsillar hypertrophy, large mandible, and/or pectus carinatum also contributes to the challenges in managing the airway with intubation and extubation due to factors intrinsic to Morquio syndrome. Taken together, these issues lead to serious respiratory distress and life-threatening complications during anesthetic procedures. Furthermore, patients with Morquio syndrome frequently cannot perform standard pulmonary function tests as a result of their distinctive skeletal dysplasia and chest deformity, thus making diagnosis of incipient pulmonary disease difficult. In many cases, conventional spirometry is too difficult for patients to complete, deriving from issues with cooperation or clinical circumstance. Therefore, it is an unmet challenge to assess pulmonary insufficiency with standard pulmonary function test (PFT) with minimal effort. Non-invasive PFT such as respiratory inductance plethysmography, impulse oscillometry system, and pneumotachography were described in Morquio patients as compared with spirometry. Findings from our previous study indicate that these non-invasive tests are a reliable approach to evaluate lung function in a larger range of patients, and provide valuable clinical information otherwise unobtainable from invasive tests. In conclusion, the present study describes the utility of non-invasive (PFT) to accommodate a broad range of patients including intolerance to effort-dependent PFT.

Entities:  

Keywords:  Impulse oscillometry system; Morquio syndrome; Non-invasive pulmonary function test; Pneumotachography; Respiratory inductance plethysmography

Year:  2017        PMID: 30294725      PMCID: PMC6171363     

Source DB:  PubMed          Journal:  J Rare Dis Res Treat


  27 in total

Review 1.  Dwarfs: pathophysiology and anesthetic implications.

Authors:  I D Berkowitz; S N Raja; K S Bender; S E Kopits
Journal:  Anesthesiology       Date:  1990-10       Impact factor: 7.892

2.  General anaesthesia in an adult patient with Morquio syndrom with emphasis on airway issues.

Authors:  Lajla Kadic; Jacques J Driessen
Journal:  Bosn J Basic Med Sci       Date:  2012-05       Impact factor: 3.363

3.  Airway proteoglycans are differentially altered in fatal asthma.

Authors:  Marcus de Medeiros Matsushita; Luiz Fernando Ferraz da Silva; Mario Adriano dos Santos; Sandra Fernezlian; Jasmijn A Schrumpf; Peter Roughley; Pieter S Hiemstra; Paulo Hilário Nascimento Saldiva; Thais Mauad; Marisa Dolhnikoff
Journal:  J Pathol       Date:  2005-09       Impact factor: 7.996

4.  Anesthetic care and perioperative complications of children with Morquio syndrome.

Authors:  Mary C Theroux; Tanvi Nerker; Colleen Ditro; William G Mackenzie
Journal:  Paediatr Anaesth       Date:  2012-06-27       Impact factor: 2.556

5.  Surgical Reconstruction for Severe Tracheal Obstruction in Morquio A Syndrome.

Authors:  Christian Pizarro; Ryan R Davies; Mary Theroux; Ellen A Spurrier; Lauren W Averill; Shunji Tomatsu
Journal:  Ann Thorac Surg       Date:  2016-10       Impact factor: 4.330

6.  Obstructive airway in Morquio A syndrome, the past, the present and the future.

Authors:  Shunji Tomatsu; Lauren W Averill; Kazuki Sawamoto; William G Mackenzie; Michael B Bober; Christian Pizarro; Christopher J Goff; Li Xie; Tadao Orii; Mary Theroux
Journal:  Mol Genet Metab       Date:  2015-09-21       Impact factor: 4.797

7.  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

8.  Molecular Genetics and Metabolism Report Long-term follow-up of post hematopoietic stem cell transplantation for Hurler syndrome: clinical, biochemical, and pathological improvements.

Authors:  Eriko Yasuda; William Mackenzie; Kristen Ruhnke; Tsutomu Shimada; Robert W Mason; Jozef Zustin; Paul L Martin; Mihir Thacker; Tadao Orii; Yoshimichi Sai; Shunji Tomatsu
Journal:  Mol Genet Metab Rep       Date:  2015-03

9.  Measuring lung function using sound waves: role of the forced oscillation technique and impulse oscillometry system.

Authors:  Bill Brashier; Sundeep Salvi
Journal:  Breathe (Sheff)       Date:  2015-03

10.  New Respiratory Inductive Plethysmography (RIP) Method for Evaluating Ventilatory Adaptation during Mild Physical Activities.

Authors:  Yann Retory; Pauline Niedzialkowski; Carole de Picciotto; Marcel Bonay; Michel Petitjean
Journal:  PLoS One       Date:  2016-03-23       Impact factor: 3.240

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  2 in total

Review 1.  Morquio B Disease. Disease Characteristics and Treatment Options of a Distinct GLB1-Related Dysostosis Multiplex.

Authors:  Nataliya Yuskiv; Katsumi Higaki; Sylvia Stockler-Ipsiroglu
Journal:  Int J Mol Sci       Date:  2020-11-30       Impact factor: 5.923

2.  Automated Assessment of Thoracic-Abdominal Asynchrony in Patients with Morquio Syndrome.

Authors:  Madhavi V Ratnagiri; Yan Zhu; Tariq Rahman; Mary Theroux; Shunji Tomatsu; Thomas H Shaffer
Journal:  Diagnostics (Basel)       Date:  2021-05-15
  2 in total

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