CCDC39 is required for assembly of inner dynein arms and the dynein regulatory complex and for normal ciliary motility in humans and dogs.
Merveille, Anne-Christine ; ; et al
in Nature Genetics (2011), 43(1), 72-8
Primary ciliary dyskinesia (PCD) is an inherited disorder characterized by recurrent infections of the upper and lower respiratory tract, reduced fertility in males and situs inversus in about 50% of ... [more ▼]
Primary ciliary dyskinesia (PCD) is an inherited disorder characterized by recurrent infections of the upper and lower respiratory tract, reduced fertility in males and situs inversus in about 50% of affected individuals (Kartagener syndrome). It is caused by motility defects in the respiratory cilia that are responsible for airway clearance, the flagella that propel sperm cells and the nodal monocilia that determine left-right asymmetry. Recessive mutations that cause PCD have been identified in genes encoding components of the outer dynein arms, radial spokes and cytoplasmic pre-assembly factors of axonemal dyneins, but these mutations account for only about 50% of cases of PCD. We exploited the unique properties of dog populations to positionally clone a new PCD gene, CCDC39. We found that loss-of-function mutations in the human ortholog underlie a substantial fraction of PCD cases with axonemal disorganization and abnormal ciliary beating. Functional analyses indicated that CCDC39 localizes to ciliary axonemes and is essential for assembly of inner dynein arms and the dynein regulatory complex. [less ▲]Detailed reference viewed: 31 (3 ULg)
Lack of efficacy of low-dose spironolactone as adjunct treatment to conventional congestive heart failure treatment in dogs
; ; et al
in Journal of Veterinary Pharmacology & Therapeutics (2011)
Aldosterone plays an important role in the pathophysiology of heart failure. Aldosterone receptor blockade has been shown to reduce morbidity and mortality in human patients with advanced congestive left ... [more ▼]
Aldosterone plays an important role in the pathophysiology of heart failure. Aldosterone receptor blockade has been shown to reduce morbidity and mortality in human patients with advanced congestive left ventricular heart failure. This study was designed to assess the efficacy and tolerance of long-term low-dose spironolactone when added to conventional heart failure treatment in dogs with advanced heart failure. Eighteen client-owned dogs with advanced congestive heart failure due to either degenerative valve disease (n=11) or dilated cardiomyopathy (n=7) were included in this prospective, placebo-controlled, double-blinded, randomized clinical study. After initial stabilization including furosemide, angiotensin-converting enzyme inhibitors, pimobendan and digoxin, spironolactone at a median dose of 0.52 mg/kg (range 0.49-0.8 mg/kg) once daily (n=9) or placebo (n=9) was added to the treatment, and the dogs were reassessed 3 and 6 months later. Clinical scoring, echocardiography, electrocardiogram, systolic blood pressure measurement, thoracic radiography, sodium, potassium, urea, creatinine, alanine aminotransferase, aldosterone and aminoterminal atrial natriuretic propeptide were assessed at baseline, 3 and 6 months. Survival times were not significantly different between the two treatment groups. Spironolactone was well tolerated when combined with conventional heart failure treatment. [less ▲]Detailed reference viewed: 20 (2 ULg)
Clinical, bronchoscopic, histopathologic, diagnostic imaging, and arterial oxygenation findings in West Highland White Terriers with idiopathic pulmonary fibrosis.
; ; et al
in Journal of Veterinary Internal Medicine (2011)
BACKGROUND: Idiopathic pulmonary fibrosis (IPF) is a chronic, interstitial lung disease primarily affecting West Highland White Terriers (WHWTs). OBJECTIVE: To describe the clinicopathological and ... [more ▼]
BACKGROUND: Idiopathic pulmonary fibrosis (IPF) is a chronic, interstitial lung disease primarily affecting West Highland White Terriers (WHWTs). OBJECTIVE: To describe the clinicopathological and diagnostic imaging features in WHWTs with IPF. ANIMALS: Twelve WHWTs with IPF and 14 healthy control WHWTs. METHOD: Prospective study. Clinical signs and findings of physical examination, blood and arterial blood gas analyses, radiography, high-resolution computed tomography (HRCT), bronchoscopy and bronchoalveolar lavage (BAL) of IPF dogs were obtained and compared with controls. Histopathologic changes in IPF dogs were evaluated. RESULTS: Mean partial pressure of oxygen was significantly lower in IPF (mean ± SD, 65.5 ± 15.4 mmHg) than in controls (99.1 ± 7.8 mmHg, P<.001). The alveolar-arterial oxygen gradient was significantly higher in IPF (50.1 ± 17.3 mmHg) than in controls (17.5 ± 4.9 mmHg, P<.001). In HRCT, ground glass opacity (GGO) was detected in all IPF dogs, traction bronchiectasis in 4, and honeycombing in 1. Bronchoscopic airway changes were noted in all IPF dogs. On BAL fluid (BALF) cytology, the total cell count (TCC) was higher in IPF dogs, and the numbers but not the percentages of macrophages, neutrophils, and mast cells were increased. On histopathology, multifocal or diffuse interstitial fibrosis, type II pneumocyte hyperplasia, prominent intraalveolar macrophages, distortion of alveolar architecture, and emphysematous change were detected. CONCLUSION AND CLINICAL IMPORTANCE: IPF causes substantial hypoxemia. In HRCT, GGO is a consistent finding. IPF dogs have concurrent airway changes and an increase in BALF TCC. [less ▲]Detailed reference viewed: 16 (5 ULg)
A model of subclinical chronic bronchitis induced by repeted nebulisations of bacterial lipopolysaccharide
Krafft, Emilie ; ; Tual, Charlotte et al
in 20th ECVIM Meeting - Toulouse - France - 9-12 septembre 2010 (2010, September 09)Detailed reference viewed: 26 (3 ULg)
Procollagen type III amino terminal propeptide concentration in dogs with iodiopathic pulmonary fibrosis, chronic bronchitis and eosinophilic bronchopneumopathy
; Krafft, Emilie ; et al
in 20th ECVIM Meeting - Toulouse - France - 9-12 septembre 2010 (2010, September 09)Detailed reference viewed: 11 (1 ULg)
Prevalence of the mutation responsible for primary ciliary dyskinesia a large population of European and American old English sheepdogs
Merveille, Anne-Christine ; ; et al
in 20th ECVIM Meeting - Toulouse - France - 9-12 septembre 2010 (2010, September)Detailed reference viewed: 5 (0 ULg)
Respiratory functional assessment using barometric whole-body plethysmography in healthy growing and aging beagle dogs
Manens, Jefferson ; ; et al
in ACVIM Meeting - Anaheim, Californie - 9-12 juin 2010 (2010, June 09)Detailed reference viewed: 6 (1 ULg)
Elevation of procollagen type III amino terminal propeptide concentration in bronchoalveolar lavage fluid of dogs with idiopathic pulmonary fibrosis
; Krafft, Emilie ; et al
Poster (2010, June 09)Detailed reference viewed: 7 (2 ULg)
Feline bronchitis: What do we know ? What do we need?
in Proceedings of the 35th Annual World Small Animal Veterinary Association Congress (2010, June)Detailed reference viewed: 1 (1 ULg)
Serum endothelin-1 concentration in terrier dogs with idiopathic pulmonary fibrosis.
Krafft, Emilie ; ; et al
in Proceedings of the 28th ACVIM Forum (2010, June)Detailed reference viewed: 19 (7 ULg)
Description of original endoscopic findings and respiratory functional assessment using barometric whole-body plethysmography in dogs suffering from brachycephalic airway obstruction syndrome.
Bernaerts, Frederique ; ; Leemans, Jérôme et al
in Veterinary Journal (2010), 183(1), 95-102
The clinical features of brachycephalic airway obstructive disease in 11 brachycephalic dogs are described in this study. The respiratory strategy was assessed before (n=11) and after (n=6) surgery using ... [more ▼]
The clinical features of brachycephalic airway obstructive disease in 11 brachycephalic dogs are described in this study. The respiratory strategy was assessed before (n=11) and after (n=6) surgery using barometric whole-body plethysmography (BWBP), with the relationship between BWBP variables and the severity of the clinical signs assessed by the use of a respiratory score based on clinical, radiographic and endoscopic findings. Partial collapse of the left main bronchus was a common finding not previously described as part of the brachycephalic airway obstructive disease syndrome. Epiglottic cysts, laryngeal granulomas and nasopharyngeal turbinates in English Bulldogs were other previously unreported findings. No significant correlation between the respiratory score and any of the BWBP variables was detected. Compared to healthy dogs, brachycephalic dogs had a significantly lower Te/Ti ratio (expiratory time over inspiratory time), peak inspiratory flow (PIF) per kg bodyweight (BW), significantly higher peak expiratory flow (PEF) per kgBW, PEF/PIF, and enhanced pause. These variations are compatible with upper airway obstructions primarily in the extrathoracic airways. Following surgery, a significant decrease in PEF/PIF was detected. The study showed that BWBP could be used to characterise the respiratory strategy in brachycephalic dogs before and after surgery. [less ▲]Detailed reference viewed: 100 (26 ULg)
Efficacy of intrasinusal administration of bifonazole cream alone or in combination with enilconazole irrigation in canine sino-nasal aspergillosis: 17 cases.
Billen, Frédéric ; ; et al
in Canadian Veterinary Journal = Revue Vétérinaire Canadienne (2010), 51Detailed reference viewed: 35 (9 ULg)
Evaluation of the influence of age on pulmonary arterial pressure by use of right ventricular catheterization, pulsed-wave Doppler echocardiography, and pulsed-wave tissue Doppler imaging in healthy Beagles.
Mercier, Elise ; ; Sandersen, Charlotte et al
in American Journal of Veterinary Research (2010), 71(8), 891-7
OBJECTIVE: To assess the influence of age on pulmonary hemodynamics and hemorheological properties in healthy dogs. ANIMALS: 14 healthy Beagles. PROCEDURES: Dogs were placed in 2 age groups as follows ... [more ▼]
OBJECTIVE: To assess the influence of age on pulmonary hemodynamics and hemorheological properties in healthy dogs. ANIMALS: 14 healthy Beagles. PROCEDURES: Dogs were placed in 2 age groups as follows: young dogs (<or= 5 years old; n = 8) and old dogs (>or= 8 years old; 6). Hematologic characteristics, plasma total protein and fibrinogen concentrations, and blood viscosity were measured. Systolic time intervals of pulmonary blood flow were recorded by pulsed-wave Doppler echocardiography. Early (E') and late (A') diastolic myocardial velocities, isovolumic contraction velocity, and systolic myocardial velocity of the free tricuspid annulus were recorded by pulsed-wave tissue Doppler imaging (TDI). Dogs were anesthetized and pulmonary arterial pressures (PAP) and cardiac output were recorded with a pediatric thermodilution Swan-Ganz catheter. RESULTS: Hemorheological measurements were not different between the 2 groups. Systolic, mean, and diastolic PAP were higher in old dogs, compared with values in young dogs; this difference was attributed to a high pulmonary vascular resistance and low arterial compliance in old dogs. Systolic time intervals of pulmonary blood flow stayed unchanged. The A' wave of the free tricuspid annulus was increased in old, compared with that young dogs, and the E':A' ratio was decreased. Pulmonary vascular resistance was inversely correlated with compliance. CONCLUSIONS AND CLINICAL RELEVANCE: The age of dogs should be taken into account when interpreting pulmonary hemodynamic results and TDI variables of right ventricular diastolic function. [less ▲]Detailed reference viewed: 25 (8 ULg)