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Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat.

ABSTRACT: Case summary:An 8-year-old spayed female domestic shorthair cat was referred for trismus of progressive onset, which had started at least 1 month previously. The patient presented with weakness, anorexia, chronic bilateral purulent nasal discharge and concurrent reduced nasal airflow. Upon physical examination, painful mouth opening, bilateral swelling of the temporal areas, with an inability to open and close the mouth completely, were apparent. A vertical mandibular range of motion (vmROM) of 22 mm was noted. Complete blood count, biochemistry, electrolytes and various serology tests were unremarkable. CT revealed multiple mineralised lesions within the masticatory muscles. Histopathological features were consistent with those seen in the human disease fibrodysplasia ossificans progressiva (FOP). Supportive treatment did not improve the vmROM, eventually resulting in a 13 mm open bite, and total inability to close and open the mouth. Cardiac arrest occurred at the induction of an anaesthesia procedure aiming to perform tracheostomy and nasal lavage. Despite emergency tracheotomy and cardiac resuscitation, humane euthanasia was elected by the owners. Post-mortem molecular investigations highlighted a heterozygous deletion, compatible with a splicing site mutation in ACVR1, which is also associated with FOP in humans. Relevance and novel information:This is the first report in the veterinary literature of FOP-like disease selectively affecting the masticatory muscles. This condition is associated with a poor prognosis, as no medical or surgical treatment has currently proven to be of any prophylactic or curative benefit. Although rare, FOP-like disease should be included in the differential diagnosis of trismus in the cat. Any further muscle injury should be avoided.

SUBMITTER: Guzu M

PROVIDER: S-EPMC6448120 | biostudies-literature | 2019 Jan-Jun

REPOSITORIES: biostudies-literature

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Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat.

Guzu Michel M Gaillot Hugues A HA Rosati Marco M Nicolier Alexandra A Hennet Philippe R PR

JFMS open reports 20190101 1

<h4>Case summary</h4>An 8-year-old spayed female domestic shorthair cat was referred for trismus of progressive onset, which had started at least 1 month previously. The patient presented with weakness, anorexia, chronic bilateral purulent nasal discharge and concurrent reduced nasal airflow. Upon physical examination, painful mouth opening, bilateral swelling of the temporal areas, with an inability to open and close the mouth completely, were apparent. A vertical mandibular range of motion (vm ...[more]

PMID: 30984411

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Project description:BACKGROUND:Myositis ossificans describes a heterotopic bone formation within a muscle. Thereby myositis ossificans is classified in two different groups: myositis ossificans progressiva (MOP) which describes a genetic autosomal dominant rare disease and myositis ossificans traumatica (MOT). The exact pathogenesis of MOT is unclear. The aim of this article was to analyse and interpret the existing literature reporting MOT of masticatory muscles and compare the results with our own clinical experience with MOT. Risk-factors, etiology, clinical features, diagnostic imaging, as well as different treatment options were evaluated and recommendations for the prevention, diagnosis, and therapy of MOT of the masticatory muscles were given. METHODS:Following the PRISMA-Guidelines, a systematic search within the PubMed/Medline database with a view to record literature of MOT of the masticatory muscles was performed. Furthermore, the database of our own clinic was screened for cases of MOT. RESULTS:In total, 63 cases of MOT of the masticatory muscles which were reported in English-based literature were included in this study. Overall, 25 female and 37 male patients could be analysed whereas one patient's gender was unknown. Complication of wisdom-tooth infection (n =?3) as well as the results of dental procedures like dental extraction (n =?7), mandibular nerve block (n =?4), periodontitis therapy (n =?1) were reported as MOT cases. From the 15 reported cases that appeared after dental treatment like extraction or local anesthesia the medial pterygoid (n =?10) was the most affected muscle. Hereof, females were more affected (n =?9) than males (n =?6). The most reported clinical symptom of MOT was trismus (n =?54), followed by swelling (n =?17) and pain (n =?13). One clinical case provided by the authors was detected. CONCLUSIONS:Dental procedures, such as local anesthesia or extractions, may cause MOT of the masticatory musculature. Demographical analyses demonstrate that females have a higher risk of developing MOT with respect to dental treatment. The most important treatment option is surgical excision. Subsequent physical therapy can have beneficial effects. Nevertheless, a benefit of interpositional materials and drugs as therapy of MOT of the masticatory muscles has not yet been proven. Myositis ossificans progressiva has to be excluded.

Dataset Information

Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat.

Publications

Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat.

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