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Idiopathic diaphragmatic paralysis—Satisfactory improvement of inspiratory muscle function by inspiratory muscle training

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Abstract

Daily inspiratory muscle strength and endurance training (IMT) was performed in a 44-year-old patient with idiopathic bilateral diaphragmatic paralysis (BDP) in addition to nocturnal non-invasive ventilation (NIV). After 4 months of training inspiratory muscle function improved satisfactorily whereas phrenic nerve latency remained pathological. Due to the improvement of inspiratory muscle capacity nocturnal NIV could be stopped without inducing nocturnal respiratory insufficiency.

Introduction

Bilateral diaphragmatic paralysis (BDP) is known to decrease ventilatory capacity causing ventilatory failure (Kreutzer et al., 1978). Non-invasive ventilation (NIV) has been shown to be an effective treatment modality to prevent ventilatory failure, especially during nighttime, in BDP (Celli, 2002). Inspiratory muscle training (IMT), on the other hand, has been shown to improve ventilatory capacity in many neurological and non-neurological disorders with impaired inspiratory muscle function (Kössler et al., 2001, Wanke et al., 1994). Therefore we hypothesized that IMT may improve ventilatory capacity in BDP thus avoiding the need of performing NIV.

Section snippets

Subject

A 44-year-old non-smoking male subject was referred to our hospital because of severe dyspnoe especially in the supine position.

Evaluation of ventilatory parameters and respiratory muscle capacity

Lung function tests consisted of spirometry and plethysmography (Jäger, Würzburg, Germany). Results were compared with predicted normal values from the European Respiratory Society (Quanjer et al., 1993). In addition to inspiratory vital capacity (VCin), maximal inspiratory pressure (MIP) and the 12 s maximal voluntary ventilation (12-s MVV) served as parameters for the

Protocol

Inspiratory muscle strength and endurance training was performed daily in addition to nocturnal NIV. The inspiratory muscle training device used was the Respifit S, which was described in detail previously (Reiter et al., 2006). Lung function and inspiratory muscle function evaluation in addition to measurement of nocturnal gas exchange and phrenic nerve evaluation was performed at the begin, 4, 5 and 7 months after start of the observation NIV was stopped in the fifth month of the observation

Results

At the begin of the observation period the lung function test showed a severe reduction of inspiratory vital capacity with further impairment in the supine position. After 4 months of IMT in addition to nocturnal NIV inspiratory vital capacity in the sitting and supine position increased by 9.6% and 9.5%, respectively.

MIP and 12-s MVV increased by 61% and 70%, respectively, thus indicating an improvement of inspiratory muscle capacity (Table 1). In the fifth month of observation a further

Discussion

NIV, especially during night, is a well established treatment modality in the respiratory management of diaphragm paralysis. Symptoms and outcomes are known to be improved in patients with BDP by treatment with NIV (Celli, 2002). In this study we could demonstrate an improvement in inspiratory muscle capacity in a patient with BDP performing a specific IMT. As phrenic nerve investigation remained pathological we may speculate that IMT induces increased non-diaphragmatic inspiratory muscle

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