Mandibular Movement Restoration in a Child with Bilateral Coronoid Hyperplasia: A Case Report

The coronoid process is an anatomical part of the mandible that serves as the attachment for the temporalis muscle, buccinator muscle, and the anterior part of the masseter muscle. In classic anatomy, it is described as a sharp triangular-shaped structure in extension of the anterior border of the mandibular ramus. In reality, this structure shows great morphological variety, including hook-shaped type, triangular type, or rounded type. In the study of Lalitha and Sridevi 1 , the majority (73.9%) of mandibles had the same type of coronoid process on both sides, while 26.1% of the cases showed different types on the two sides. Allometric variation can be established in mandibular shape in humans, with taller individuals having superoinferiorly taller rami with more anteriorly-oriented and higher coronoid processes and a corresponding deeper sigmoid notch 2 .Mandibular coronoid process hyperplasia (MCPH) is an uncommon congenital or developmental condition that is characterized by a macroscopic increase in the size of the coronoid process with a normal histologic structure of the bone. MCPH can exist as a uni-or bilateral condition and causes a slow and progressive reduction of mouth opening. Restricted mouth opening results from impingement of the coronoid process on the medial surface of the zygomatic arch [3][4][5] . Unilateral MCPH can involve facial asymmetry with deviation toward the affected side 5 . Langenbeck was the first to report MCPH in 1853, and the first case of restricted mouth opening due to coronoid process enlargement was reported in 1899 by Jacob 3 . Jacob's disease refers to the condition where the coronoid process creates a new joint with the zygomatic process.The pathogenesis of MCPH remains unclear. Several factors might be associated with development of MCPH 6,7 . History of facial trauma and, in particular, zygomatic arch trauma is thought to be a contributing factor in some cases [8][9][10][11]

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A case of bilateral coronoid process hyperplasia in a child with suspected VACTERL-H syndrome

Takeuchi

Tanaka

Sawada

et al. 2018

Jpn. J. Oral. Maxillofac. Surg.

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VACTERL syndrome is defined by the presence of the following specific anomalies: vertebral anomalies, anal atresia, cardiac abnormalities, tracheo-esophageal fistula, renal anomalies, and limb abnormalities. VACTERL-H is a syndrome with at least three of the aforementioned features plus hydrocephalus, and the prognosis is poor. We describe the case of a girl who was suspected to have VACTERL-H syndrome. The patient had microglossia and jaw deformity with bilateral hyperplasia of the mandibular coronoid process. Because bilateral coronoid hyperplasia is a rare condition in the pediatric age, it may be an unrecognized cause of limited mouth opening. We report on a 9-year-old girl who clinically met the diagnostic criteria for VACTERL-H syndrome. She was referred to our department because of limited mandibular movement. CT imaging confirmed the presence of jaw deformity with bilateral coronoid process hyperplasia. Bilateral coronoidotomy was performed through an intraoral approach to improve limited mandibular movement. Mouth opening exercises were started 3 days after surgery, and after 1 year, mouth opening was preserved at an adequate level. : VACTERL-H syndrome (VACTERL-H 症候群) ，coronoid process hyperplasia (筋突起過形成) ， microglossia (小舌症) ，jaw deformity (顎変形症) ，limited mandibular movement (開口障害) 1) 聖隷浜松病院歯科口腔外科 (主任：竹内啓人部長) 2) 聖隷浜松病院矯正歯科 (主任：澤田紘美医長) 3) 九州歯科大学顎口腔機能矯正学分野 (主任：川元龍夫教授)

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Mandibular Movement Restoration in a Child with Bilateral Coronoid Hyperplasia: A Case Report

Cited by 3 publications

References 9 publications

Coronoid hyperplasia: A review

Coronoid hyperplasia: A review

A systematic review of treatment and outcomes in patients with mandibular coronoid process hyperplasia

A case of bilateral coronoid process hyperplasia in a child with suspected VACTERL-H syndrome

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