Primary Ciliary Dyskinesia in the Dog

Morrison, Wallace B.; Wilsman, Norman J.; Fox, Leslie E.; Farnum, Cornelia E.

doi:10.1111/j.1939-1676.1987.tb01989.x

Cited by 34 publications

(39 citation statements)

References 25 publications

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“…The results from the mucociliary clearances and ultrastructural analysis of the respiratory cilia have been reported [2,8,11]. Briefly, there was no mucociliary clearance in the CCD dogs and the ultraslructural analy sis of respiratory cilia in the CCD dogs were similar to those found in people with CCD.…”

Section: Resultsmentioning

confidence: 59%

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Communicating Hydrocephalus in Dogs with Congenital Ciliary Dysfunction

Daniel¹,

Edwards²,

Harvey³

et al. 1995

Dev Neurosci

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Magnetic resonance imaging (MRI) and radionuclide ventriculography were performed in 5 dogs with congenital ciliary dysfunction (CCD) and 3 normal dogs. Ventricular and brain dimensions and volumes, and CSF flow rates were measured or calculated from the MR images and radionuclide clearance. All CCD dogs had hydrocephalus based on previously published criteria of a percent vertical brain dimension (PVBD) greater than 14%. The PVBD was significantly larger (p = 0.001) in the dogs with CCD (mean ± SD) 33.00 ± 5.42% than in normal dogs 11.07 + 0.61 %. The ventricular volume was significantly larger (p = 0.021) in CCD dogs 10,841 ± 4,127 mm³ compared to the volume measured in normal dogs 3,069 ± 1,167 mm³. The CSF flow rate was not significantly different (p = 0.876) between CCD dogs (253.00 ± 147.25 mm³/h) and normal dogs (267.667 ± 47.61 ram³/h). This suggests that the ventricular dilation in CCD dogs is not due to impedance of CSF flow from the ventricular system by dysfunctional ependymal cilia.

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Section: Resultsmentioning

confidence: 59%

“…Congenital ciliary dysfunction (CCD) has been reported in man, rat, mouse, pig and dog [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20]. Unlike other species with CCD, affected dogs are a nearly exact model of the human disease.…”

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confidence: 99%

Communicating Hydrocephalus in Dogs with Congenital Ciliary Dysfunction

Daniel¹,

Edwards²,

Harvey³

et al. 1995

Dev Neurosci

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“…A more recent discovery was the presence of normal ciliary ultrastructure in human beings with ICS.4 Ciliary lesions reported in dogs with ICS include outer dynein arm deficien~y,~,~,~ microtubular transposition,* random orie n t a t i~n ,~.~ and partial microtubular deficiency. 6 The finding of normally oriented, structurally complete respiratory cilia in this Chow Chow dog with KS demonstrates that the diagnosis of ICS in dogs, like human beings, cannot always be established by ultrastructural evaluation ofcilia. In most men with ICS and in this dog, the ultrastructural image of the spermatozoal flagellum mirrors that seen in respiratory cilia.…”

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confidence: 99%

Kartagener's Syndrome in a Chow Chow Dog with Normal Ciliary Ultrastructure

Edwards

Kennedy²,

Toal³

et al. 1989

Vet Pathol

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A 2-year-old male Chow Chow dog was presented with chronic nasal discharge and cough first seen at 8 months of age. Abnormal physical findings included bilateral mucopurulent nasal discharge and diffuse increase in auscultable lung sounds. A generalized bronchopneumonia with alveolar consolidation in dependent portions and situs inversus totalis were detected radiographically. Bronchiectasis was seen with contrast bronchography, and lesions consistent with a serous rhinitis were present on nasal radiographs. Severe dilatation of the lateral brain ventricles was detected with computed tomography (Fig. 1). Blood analysis revealed a mature neutrophilia and normal numbers of lymphocytes. Serum concentration of IgM (1 08 mg/dl; normal, 100-200 mg/dl) was normal, but concentrations of IgA (220 mg/dl; normal, 40-160 mg/dl) and IgG (2,400 mg/dl; normal, 1,000-2,000 mg/ dl) were increased. Humoral responses to distemper viral and parvoviral vaccinations were normal. Test results of neutrophil function including Staphylococcus aureus ingestion, iodination, antibody dependent cell-mediated cytotoxicity, and chemotaxis were comparable to same-day results obtained with neutrophils from clinically normal dogs, but nitroblue tetrazolium reduction was 129-1669'0 of control values. A transtracheal wash was described cytologically as a purulent exudate and on culture yielded Pasteurella multocida and Streptococcus zooepidemicus. Decreased numbers (1 28,00O/ejaculate; normal, > 200,00O/ejaculate) and progressive motility (< 15%; normal, > 80%) of spermatozoa were present on semen analysis. No tracheal mucociliary clearance was detected in the dog at 6 or 7 years of age using 99 "'Tc-labeled macroaggregated albumin and gamma camera imaging (normal, 6.9 ? 0.5 mm/min).I0 Beat frequencies of 7.2, 7.7, and 16.9 Hz (normal, 4-17 Hz) were measured photometrically on cilia in nasal turbinate biopsy specimens obtained at 2 and 6 years of age. When video tape images of the cilia (30 framedsec) were analyzed, a multiplanar motion, indistinguishable from that of cilia from clinically normal dogs, was seen. When particulate matter in the media, mostly red blood cells, contacted the cilia it was consistently accelerated in a uniform, linear direction over the surface of the biopsy specimen (-2 mm diameter.). When viewed with transmission electron microscopy, nasal cilia displayed normal orientation and axonemal structure (Figs. 2, 3). Analysis of 50 ciliary cross sections from nine areas indicated that 92% of cilia had both outer and inner dynein arms on peripheral microtubular doublets. Only 8% displayed loss of one to three outer dynein arms, and a single cilium (Fig. 3) lacked three peripheral microtubules. Computer enhancementS of a ciliary cross section (Fig. 4)

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“…In dogs the predominant clinical signs are usually respiratory in nature, resulting from impaired airway clearance of mucus, and typically include chronic nasal discharge and recurrent lower respiratory tract infections from shortly after birth. [1][2][3][4][5][6][7] In human medicine the diagnosis of PCD is confirmed by light microscopic assessment of ciliary beating, generally limited to determination of ciliary beat frequency, and identification of cilial structural defects by electron microscopy. 9 In classic PCD, ciliary beating is reduced or completely absent and a variety of ultrastructural cilial defects can be present.…”

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confidence: 99%

“…8 Specific dog breeds reported to be affected include the English Springer Spaniel, Old English Sheepdog, Bichon Frise, Golden Retriever, English Setter, Shar Pei, Dalmatian, Chow Chow, Rottweiler, Doberman, Newfoundland and Dachshund. [1][2][3][4][5][6][7] Ciliated cells are present in several different anatomic locations, including the respiratory tract, genital tract, middle ear and brain, and PCD can cause a variety of clinical signs referable to these body systems. In dogs the predominant clinical signs are usually respiratory in nature, resulting from impaired airway clearance of mucus, and typically include chronic nasal discharge and recurrent lower respiratory tract infections from shortly after birth.…”

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confidence: 99%