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

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...

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Main Authors: Michel Guzu, Hugues A Gaillot, Marco Rosati, Alexandra Nicolier, Philippe R Hennet
Format: Article
Language:English
Published: SAGE Publishing 2019-04-01
Series:Journal of Feline Medicine and Surgery Open Reports
Online Access:https://doi.org/10.1177/2055116919839857
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spelling doaj-ffe30f5d4b8746c0bdab93300cf6f3cd2020-11-25T02:58:17ZengSAGE PublishingJournal of Feline Medicine and Surgery Open Reports2055-11692019-04-01510.1177/2055116919839857Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair catMichel Guzu0Hugues A Gaillot1Marco Rosati2Alexandra Nicolier3Philippe R Hennet4ADVETIA Specialists Referral Hospital, Vélizy-Villacoublay, FranceADVETIA Specialists Referral Hospital, Vélizy-Villacoublay, FranceSection of Clinical and Comparative Neuropathology, Centre for Clinical Veterinary Medicine, Ludwig-Maximilians-Universität, Munich, GermanyVet Diagnostics, Lyon, FranceADVETIA Specialists Referral Hospital, Vélizy-Villacoublay, FranceCase 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.https://doi.org/10.1177/2055116919839857
collection DOAJ
language English
format Article
sources DOAJ
author Michel Guzu
Hugues A Gaillot
Marco Rosati
Alexandra Nicolier
Philippe R Hennet
spellingShingle Michel Guzu
Hugues A Gaillot
Marco Rosati
Alexandra Nicolier
Philippe R Hennet
Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
Journal of Feline Medicine and Surgery Open Reports
author_facet Michel Guzu
Hugues A Gaillot
Marco Rosati
Alexandra Nicolier
Philippe R Hennet
author_sort Michel Guzu
title Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
title_short Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
title_full Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
title_fullStr Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
title_full_unstemmed Bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
title_sort bilateral fibrodysplasia ossificans affecting the masticatory muscles and causing irreversible trismus in a domestic shorthair cat
publisher SAGE Publishing
series Journal of Feline Medicine and Surgery Open Reports
issn 2055-1169
publishDate 2019-04-01
description 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.
url https://doi.org/10.1177/2055116919839857
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