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Osteochondroma of coronoid process: a rare etiology of jacob disease.

ABSTRACT: Jacob disease is a rare entity consisting of the formation of a pseudojoint between the inner surface of the zygoma and the coronoid process. This requires constant contact between the two implicated surfaces. It can be achieved by two mechanisms: one by an enlarged coronoid process and two by an anterior displacement of the coronoid process caused by a temporomandibular joint (TMJ) disorder. Although von Langenbeck described coronoid process hyperplasia in 1853, Oscar Jacob was the first author to describe the pathology in 1899. Since then, only a few cases have been published in the literature. The authors report a rare case of Jacob disease caused by an osteochondroma of the coronoid process, which is even less common, and review the literature.

SUBMITTER: Losa-Munoz PM

PROVIDER: S-EPMC4221146 | biostudies-other | 2014 Dec

REPOSITORIES: biostudies-other

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Project description:Vertical malocclusion is a developmental condition, resulting from complex interactions among multiple etiological factors during the growth period. As a tricky dentofacial deformity clinically, long-face (LF) morphology is characterized by excessive vertical facial growth with severe disarrangement of jaws and teeth. Since the improvement of LF patients on facial profile and occlusion is often difficult and lacks long-term stability, it becomes important to unravel the etiology of LF pattern formation for early prevention and treatment. In the current studies, we identified a transgenic mouse model that exhibited a dysplastic coronoid process and LF morphology. Although the mutant mice exhibited jaw structures and occlusion comparable to controls at birth, they all acquired typical LF morphology with anterior open bite during postnatal growth, resembling clinical features of the selected skeletal class III patients. Since the coronoid process provides an insertion site for the temporalis attachment, we examined the initial development and differentiation of the temporalis and found identical results in both control and mutant mice before E17.5 when the temporal muscle makes attachment to the coronoid process. However, thereafter, we observed altered orientation and reduced size of the cross-sectional area of the temporalis in mutant mice, which persisted to the weaning stage. Biomechanical analysis and simulation modeling further support the idea that altered morphology of the coronoid process may impair the efficiency of the vertical temporalis contraction and appears to correlate with LF formation. Consistently, we present evidence that a dysplastic mandibular coronoid process was also seen in some human patients with skeletal III LF morphology. Taken together, the results presented in this study establish an association of the craniofacial bony structures with vertical patterning, which will have implications in earlier prediction for clinical precaution and intervention.

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Osteochondroma of coronoid process: a rare etiology of jacob disease.

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