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Clinical features of idiopathic inflammatory polymyopathy in the Hungarian Vizsla.

Tauro A, Addicott D, Foale RD, Bowman C, Hahn C, Long S, Massey J, Haley AC, Knowler SP, Day MJ, Kennedy LJ, Rusbridge C - BMC Vet. Res. (2015)

Bottom Line: The average of inbreeding coefficient (CoI) of 16.3% suggests an increased expression of a Dog Leukocyte Antigen Class II haplotype, leading to an increased disease risk.The prognosis remains guarded, as treatment can only manage the disease.Recurrence of clinical signs and perceived poor quality of life are the most common reasons for humane euthanasia.

View Article: PubMed Central - PubMed

Affiliation: Fitzpatrick Referrals, Halfway Lane, Eashing, Godalming, GU7 2QQ, Surrey, UK. Annat@Fitzpatrickreferrals.co.uk.

ABSTRACT

Background: A retrospective study of the clinicopathological features of presumed and confirmed cases of idiopathic inflammatory polymyopathy in the Hungarian Vizsla dog and guidelines for breeding.

Results: 369 medical records were reviewed (1992-2013) and 77 Hungarian Vizslas were identified with a case history consistent with idiopathic inflammatory polymyopathy. Inclusion criteria were: group 1 (confirmed diagnosis); histopathology and clinical findings compatible with an inflammatory polymyopathy and group 2 (probable diagnosis); clinical findings compatible with a polymyopathy including dysphagia, sialorrhea, temporal muscle atrophy, elevated serum creatine kinase (CK) activity, and sufficient clinical history to suggest that other neuromuscular disorders could be ruled out. Some group 2 dogs had muscle biopsy, which suggested muscle disease but did not reveal an inflammatory process. The mean age of onset was 2.4 years; male dogs were slightly overrepresented. Common presenting signs were dysphagia, sialorrhea, masticatory muscle atrophy, and regurgitation. Common muscle histopathological findings included degenerative and regenerative changes, with multifocal mononuclear cell infiltration with lymphoplasmacytic myositis of variable severity. A positive response to immunosuppressive treatment supported an immune-mediated aetiology. The mean age at death and survival time were 6.4 and 3.9 years, respectively. Recurrence of clinical signs and aspiration pneumonia were common reasons for euthanasia.

Conclusions: Diagnosis of Vizsla idiopathic inflammatory polymyopathy can be challenging due to lack of specific tests, however the presence of dysphagia, regurgitation and masticatory muscle atrophy in this breed with negative serological tests for masticatory muscle myositis and myasthenia gravis, along with muscle biopsies suggesting an inflammatory process, support the diagnosis. However, there is an urgent need for a more specific diagnostic test. The average of inbreeding coefficient (CoI) of 16.3% suggests an increased expression of a Dog Leukocyte Antigen Class II haplotype, leading to an increased disease risk. The prognosis remains guarded, as treatment can only manage the disease. Recurrence of clinical signs and perceived poor quality of life are the most common reasons for humane euthanasia.

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Sialorrhoea in VIP.
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Fig1: Sialorrhoea in VIP.

Mentions: Seventy-seven of the 369 Hungarian Vizsla dogs (Tables 1 and 2) were determined to have a case history consistent with Vizsla idiopathic inflammatory polymyopathy (VIP) (Group 1, n = 24 dogs; Group 2, n = 53 dogs including eight dogs with biopsy-confirmed non-inflammatory muscle disease). Two cases were shown to have acetylcholine receptor antibodies (consistent with myasthenia gravis) and they were excluded from the study. Eight of the 77 affected cases were from outside Europe: three were from USA, one was from New Zealand, and four were from Australia. The mean age of onset was 2.4 years (range 0.2–10.3 years). Male dogs were slightly overrepresented (entire male:neutered male:entire female:neutered female 26:24:9:17). In one case the gender was unknown. The most common presenting signs in both groups (Tables 1 and 2) were dysphagia (i.e. pharyngeal phase of deglutition) (Additional file 1: Video 1) (90% of all dogs; group 1: 83% and group 2: 92%), difficulty in eating and drinking (i.e. oral phase of deglutition) (Additional file 2: Video 2) (90% combined; group 1: 87% and group 2: 91%), sialorrhea (87% of all dogs; group 1: 87% and group 2: 87%) (Figure 1) and regurgitation (79% of all dogs; group 1: 79% and group 2: 79%). Pain on opening the jaw was reported in 12% of all dogs (group 1: 4% and group 2: 15%). Masticatory muscle atrophy (i.e. masseter, temporalis and pterygoid muscles) (Figure 2) was present in 84% of all dogs (group 1: 83% and group 2: 85%), and 21% of all dogs had restricted jaw motility (group 1: 12% and group 2: 25%). Masticatory muscle atrophy either appeared early in the course of the disease, or had a more insidious onset, progressing slowly over several months or years (Figure 3). Enophthalmos, if present, was secondary to atrophy of the pterygoid muscle. Generalised muscle atrophy (43% of all dogs; group 1: 38% and Group 2: 45%), exercise intolerance (35% of all dogs; group 1: 46% and group 2: 30%), generalised weakness (30% of all dogs; group 1: 38% and group 2: 26%), and lameness (19% of all dogs; group 1: 21% and group 2: 19%) were less common signs. Three cases had dysuria (two in group 1 and one in group 2). The owners described a “stop-start flow” as if the dogs had difficulty maintaining a urine stream. The underlying pathophysiology for this was not ascertained.Table 1


Clinical features of idiopathic inflammatory polymyopathy in the Hungarian Vizsla.

Tauro A, Addicott D, Foale RD, Bowman C, Hahn C, Long S, Massey J, Haley AC, Knowler SP, Day MJ, Kennedy LJ, Rusbridge C - BMC Vet. Res. (2015)

Sialorrhoea in VIP.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4414416&req=5

Fig1: Sialorrhoea in VIP.
Mentions: Seventy-seven of the 369 Hungarian Vizsla dogs (Tables 1 and 2) were determined to have a case history consistent with Vizsla idiopathic inflammatory polymyopathy (VIP) (Group 1, n = 24 dogs; Group 2, n = 53 dogs including eight dogs with biopsy-confirmed non-inflammatory muscle disease). Two cases were shown to have acetylcholine receptor antibodies (consistent with myasthenia gravis) and they were excluded from the study. Eight of the 77 affected cases were from outside Europe: three were from USA, one was from New Zealand, and four were from Australia. The mean age of onset was 2.4 years (range 0.2–10.3 years). Male dogs were slightly overrepresented (entire male:neutered male:entire female:neutered female 26:24:9:17). In one case the gender was unknown. The most common presenting signs in both groups (Tables 1 and 2) were dysphagia (i.e. pharyngeal phase of deglutition) (Additional file 1: Video 1) (90% of all dogs; group 1: 83% and group 2: 92%), difficulty in eating and drinking (i.e. oral phase of deglutition) (Additional file 2: Video 2) (90% combined; group 1: 87% and group 2: 91%), sialorrhea (87% of all dogs; group 1: 87% and group 2: 87%) (Figure 1) and regurgitation (79% of all dogs; group 1: 79% and group 2: 79%). Pain on opening the jaw was reported in 12% of all dogs (group 1: 4% and group 2: 15%). Masticatory muscle atrophy (i.e. masseter, temporalis and pterygoid muscles) (Figure 2) was present in 84% of all dogs (group 1: 83% and group 2: 85%), and 21% of all dogs had restricted jaw motility (group 1: 12% and group 2: 25%). Masticatory muscle atrophy either appeared early in the course of the disease, or had a more insidious onset, progressing slowly over several months or years (Figure 3). Enophthalmos, if present, was secondary to atrophy of the pterygoid muscle. Generalised muscle atrophy (43% of all dogs; group 1: 38% and Group 2: 45%), exercise intolerance (35% of all dogs; group 1: 46% and group 2: 30%), generalised weakness (30% of all dogs; group 1: 38% and group 2: 26%), and lameness (19% of all dogs; group 1: 21% and group 2: 19%) were less common signs. Three cases had dysuria (two in group 1 and one in group 2). The owners described a “stop-start flow” as if the dogs had difficulty maintaining a urine stream. The underlying pathophysiology for this was not ascertained.Table 1

Bottom Line: The average of inbreeding coefficient (CoI) of 16.3% suggests an increased expression of a Dog Leukocyte Antigen Class II haplotype, leading to an increased disease risk.The prognosis remains guarded, as treatment can only manage the disease.Recurrence of clinical signs and perceived poor quality of life are the most common reasons for humane euthanasia.

View Article: PubMed Central - PubMed

Affiliation: Fitzpatrick Referrals, Halfway Lane, Eashing, Godalming, GU7 2QQ, Surrey, UK. Annat@Fitzpatrickreferrals.co.uk.

ABSTRACT

Background: A retrospective study of the clinicopathological features of presumed and confirmed cases of idiopathic inflammatory polymyopathy in the Hungarian Vizsla dog and guidelines for breeding.

Results: 369 medical records were reviewed (1992-2013) and 77 Hungarian Vizslas were identified with a case history consistent with idiopathic inflammatory polymyopathy. Inclusion criteria were: group 1 (confirmed diagnosis); histopathology and clinical findings compatible with an inflammatory polymyopathy and group 2 (probable diagnosis); clinical findings compatible with a polymyopathy including dysphagia, sialorrhea, temporal muscle atrophy, elevated serum creatine kinase (CK) activity, and sufficient clinical history to suggest that other neuromuscular disorders could be ruled out. Some group 2 dogs had muscle biopsy, which suggested muscle disease but did not reveal an inflammatory process. The mean age of onset was 2.4 years; male dogs were slightly overrepresented. Common presenting signs were dysphagia, sialorrhea, masticatory muscle atrophy, and regurgitation. Common muscle histopathological findings included degenerative and regenerative changes, with multifocal mononuclear cell infiltration with lymphoplasmacytic myositis of variable severity. A positive response to immunosuppressive treatment supported an immune-mediated aetiology. The mean age at death and survival time were 6.4 and 3.9 years, respectively. Recurrence of clinical signs and aspiration pneumonia were common reasons for euthanasia.

Conclusions: Diagnosis of Vizsla idiopathic inflammatory polymyopathy can be challenging due to lack of specific tests, however the presence of dysphagia, regurgitation and masticatory muscle atrophy in this breed with negative serological tests for masticatory muscle myositis and myasthenia gravis, along with muscle biopsies suggesting an inflammatory process, support the diagnosis. However, there is an urgent need for a more specific diagnostic test. The average of inbreeding coefficient (CoI) of 16.3% suggests an increased expression of a Dog Leukocyte Antigen Class II haplotype, leading to an increased disease risk. The prognosis remains guarded, as treatment can only manage the disease. Recurrence of clinical signs and perceived poor quality of life are the most common reasons for humane euthanasia.

Show MeSH
Related in: MedlinePlus