Trismus-pseudocamptodactyly syndrome with bilateral hypoplastic mandibular condyles and shallow mandibular fossa: A case report

Trismus-pseudocamptodactyly syndrome (TPS) (OMIM#158300), also known as Hecht syndrome, is a rare autosomal dominant distal arthrogryposis. Bilateral hyperplasia of the coronoid processes has been reported to cause trismus. In this study, we report a case of TPS definitively diagnosed on the basis of clinical signs in a patient who had bilateral hypoplastic mandibular condyles and shallow mandibular fossa; TPS in such patients have never been reported in the literature. On the basis of the present case, we propose that a hypoplastic mandibular condyle and shallow mandibular fossa are also clinical features of TPS.     

Bilateral coronoid hyperplasia. Report of a case

A case of bilateral coronoid hyperplasia is presented. This is a developmental abnormality that occurs primarily in males and is characterized by progressive, asymptomatic limitation of mandibular movements. The condition is usually detected shortly after the age of puberty. A diagnosis is usually made on the basis of the history and the radiographic findings. Surgical removal of the elongated coronoid processes, along with good postoperative range-of-motion exercises, will restore full mandibular movement.     

A case of reformed coronoid process and mandibular angle after coronoidectomy and anglectomy for masticatory muscle tendon-aponeurosis hyperplasia

Masticatory muscle tendon-aponeurosis hyperplasia is a newly defined disease entity in Japan, in which limited mouth opening results from limited masticatory muscle extension caused by hyperplasia of the tendon and aponeurosis. Recently, we encountered a rare case of reformed bilateral coronoid process and mandibular angle after coronoidectomy and anglectomy. A 19-year-old female with gradually progressive trismus had been diagnosed with this disease. Under general anesthesia, she received coronoidotomy of the bilateral coronoid process and anglectomy. At 1 week postoperatively, trismus was improved. At 6 months postoperatively, panoramic imaging showed the initiation of bilateral reformed coronoid process and mandibular angle. At 3 years postoperatively, computed tomography (CT) and three-dimensional reconstructed CT (3D-CT) demonstrated reformed coronoid process and mandibular angle. However, the maximum mouth opening length was not decreased. She continued to perform mandibular condyle movement exercise every day. Panoramic imaging is a very useful examination for evaluating bone reformation after coronoidectomy and anglectomy for masticatory muscle tendon-aponeurosis hyperplasia. In particular, CT and 3D-CT demonstrated reformed coronoid process and mandibular angle. Postoperative mouth-opening training is an important prognostic factor to prevent the recurrence of trismus in this disease.     

Bilateral coronoid hyperplasia: report of two cases

Bilateral hyperplasia of the coronoid processes of the mandible is an uncommon condition that has been reported in 24 patients.^1–3 The coronoid processes are usually normally shaped,^4,5 and have been classically described by Shira and Lister as “mere overgrowths of bone.”⁶ The process has been diagnosed as a developmental hyperplasia, and the etiology has remained unknown.^1,2,5,7 The most common symptom of bilateral coronoid hyperplasia is limitation of mandibular opening without visible facial deformity.^1–3,5,8 The previously reported cases of bilateral coronoid hyperplasia have all been reported in males.^1–3 This has led some authors to speculate that the condition is sex-linked. This paper describes two new cases, one being the first reported in a female.     

Bilateral coronoid hyperplasia resulting in severe limitation of mandibular movement. Report of a case

A case of bilateral coronoid hyperplasia with limitation of mandibular motion in a female patient has been presented. Her symptoms began to develop around the age of 44. We believe that this is the first case to be reported in a female. Also, in all other reported cases symptoms began to develop around the onset of puberty. The literature has been reviewed with specific reference to the etiology of bilateral coronoid hyperplasia. This case brings the total number of reported cases to twenty-two. We hope that if similar cases are identified in female patients the practitioners will report them promptly. Only in this way will we be in a better position to understand the etiology of bilateral coronoid hyperplasia with special reference to heredity.     

Bilateral hyperplasia of the coronoid processes: clinical report

Monolateral or bilateral hyperplasia of the coronoid processes of the mandible is a rare disorder resulting in reduction of mouth opening because of the unnatural contact of the coronoid process with the zygomatic bones. The authors describe two cases of bilateral hyperplasia of the coronoid processes that were successfully treated.     

Hyperplasia of the coronoid process in patients with ankylosing spondylitis (Bechterew disease)

Although involvement of the temporomandibular joint in patients with ankylosing spondylitis (AS, Bechterew disease) has been described previously, hyperplasia of the mandibular coronoid process in those patients has not been reported yet. Case notes were studied, and records were made of age, sex, clinical symptoms, radiography, and treatment in all patients with a confirmed diagnosis of coronoid hyperplasia presenting at the Department of Oral and Maxillofacial Surgery, University of Bonn, between 1995 and 2007. Sixteen cases of coronoid hyperplasia were recruited, of which 12 were bilateral and 4 were unilateral. Four patients had AS, 3 of them were HLA-B27-positive. Temporomandibular joint symptoms are frequently seen in patients with AS. Nevertheless, it must be considered that a limitation of jaw mobility in those patients might also be caused by an elongation of the mandibular coronoid process.     

An unusual cause of trismus.

This paper reports a case of limited mandibular movement caused by the rare condition of bilateral coronoid hyperplasia. Dental surgeons should be aware of the possibility of this condition when encountering patients with movement problems in the mandible.     

Bilateral hyperplasia of the mandibular coronoid processes: a report of two cases

Two cases of bilateral coronoid hyperplasia of the mandible are presented. Consistent with other reported cases, the patients were men whose onset of symptoms correlated with the onset of puberty. One case was unusual in that bilateral coronoidectomies did not relieve the restriction of mandibular movement. Additional masseter muscle and fascial surgery was required to provide unrestricted mandibular motion.     

Limitation of mandibular opening due to enlarged coronoid processes

The causes of limited mandibular opening are summarized. Coronoid process enlargement in five patients is discussed; two cases of bilateral coronoid hyperplasia and a unilateral coronoid-zygomatic ankylosis case underwent successful surgery. One of these bilateral patients exhibited bone regrowth a year later.     

Iperplasia bilaterale del processo coronoideo in pazienti pediatrici

ObjectivesTo present an international literature review on the coronoid hyperplasia in pediatric patients, describing the etiology, the diagnosis and the therapeutic options.Materials and methodsA systematic revision of the international literature has been done on Medline database (www.ncbi.nlm.nih.gov/pubmed). Keywords chosen were: “bilateral coronoid hyperplasia”, “coronoid process”, “temporomandibular joint ankylosis”, “coronoid hyperplasia and treatment”, “pediatric patients”.ResultsAmong the pathogenetic factors, there are: temporalis muscle hyperactivity, genetic inheritance, hormonal stimulus, temporomandibular joint dysfunctions, traumas and persistent coronoid cartilage growth centre. The main symptoms are: mandibular hypomobility, alterations in protrusion and in lateral mandibular movements and muscular dystonia. Orthopantomography, magnetic resonance imaging and computed tomography are the diagnostic gold standards. The most widely surgical treatment used is coronoidectomy.ConclusionsOn the scientific evidences present in literature it's possible to conclude that, being coronoid hyperplasia a frequent pediatric condition, an early diagnosis is important to restore stomatognathic multifunctions as soon as possible.     

Coronoid process hyperplasia: an unusual cause of mandibular hypomobility

A large number of disorders affecting the masticatory system can cause restriction of mouth opening. The most common conditions related to this problem are those involving the temporomandibular joint (TMJ) and the masticatory muscles, when facial pain also is an usual finding. Congenital or developmental mandibular disorders are also possible causes for mouth opening limitation, although in a very small prevalence. Coronoid process hyperplasia (CPH) is an example of these cases, characterized by an excessive coronoid process growing, where mandibular movements become limited by the impaction of this structure on the posterior portion of the zygomatic bone. This condition is rare, painless, usually bilateral and progressive, affecting mainly men. Diagnosis of CPH is made based on clinical signs of mouth opening limitation together with imaging exams, especially panoramic radiography and computerized tomography (CT). Treatment is exclusively surgical. This paper presents a case of a male patient with bilateral coronoid process hyperplasia, initially diagnosed with bilateral disk displacement without reduction, and successfully treated with intraoral coronoidectomy. It is emphasized the importance of differential diagnosis for a correct diagnosis and, consequently, effective management strategy.     

Gap coronoidotomy for management of coronoid process hyperplasia of the mandible

Patients with coronoid process hyperplasia of the mandibular area are rare. The treatment of this disease is to increase the patient's mouth opening by surgery. There are various, but controversial, methods to treat it. We present a modified (gap) coronoidotomy procedure in detail and compare it with other conventional methods to treat coronoid process hyperplasia.     

An excessive coronoid hyperplasia with suspected traumatic etiology resulting in mandibular hypomobility

There are multiple theories as to the causes of coronoid process hyperplasia of the mandible, including trauma, temporalis muscle hyperactivity, hormonal stimulus, and genetic inheritance. The excess growth of the coronoid process can cause impingement on the zygomatic processes and may result in mandibular hypomobility. A case of an excessive unilateral coronoid hyperplasia with suspected traumatic etiology, which was successfully treated by coronoidectomy and postoperative physiotherapy, is presented. The patient was a 21-year-old man whose maximum mouth opening was 23 mm. The attachments of the temporalis muscle were stripped and the coronoid process was accessed using the Al-Kayat and Bramley approach. The coronoid process was then resected via an intraoral pathway. One week after surgery, physiotherapy was started and the maximum mouth opening had increased to 38 mm. In the case presented, a coronoidectomy with postoperative physiotherapy for treatment of coronoid process hyperplasia produced satisfactory results in the correction of coronoid-malar interference.     

Accessory mandibular condyle at the coronoid process

Coronoid process hyperplasia is a rare cause of mandibular hypomobility. It can result from temporalis muscle hyperactivity, trauma, and neoplasia, but often is idiopathic. Enlargement of the coronoid process leading to pseudojoint formation with the zygomatic arch is known as Jacob's disease. It results most commonly from an osteochondroma of the coronoid process. This is the first reported case of a non-neoplastic accessory mandibular condyle located at the coronoid process articulating with the zygoma.     

Mandibular coronoid hyperplasia in pediatric patients

Bilateral coronoid hyperplasia is a relatively rare condition in the pediatric population and yet may be an unrecognized cause of limited mouth opening in children. There are multiple theories as to the causes of the hyperplasia, which include temporalis hyperactivity, hormonal stimulus, and genetic inheritance. The resulting excess growth of the coronoids results in impingement on the zygomatic processes leading to mandibular hypomobility. The diagnosis is confirmed with plain films and computed tomography scans. Treatment involves bilateral coronoidectomies to relieve impingement on the zygoma. Postoperative physical therapy is crucial for success; the therapy focuses on maintaining the mouth opening achieved at the time of surgery. Outcome reports have been variable despite good physical therapy, suggesting that the exact pathology of the condition is not well understood.     

Imaging modality correlations of an odontogenic keratocyst in the nevoid basal cell carcinoma syndrome: a family case report

Multiple maxillary and mandibular cysts are principle features of nevoid basal cell carcinoma syndrome (NBCCS; Gorlin-Goltz syndrome). We present a family case report of NBCCS with odontogenic keratocyst where the findings on plain films, CT, clinical, and histopathologic examinations are compared and analyzed. The systemic manifestations included frontal bossing, odontogenic keratocyst, ectopic calcification in 1 patient, and bifid rib in 1 patient. CT examination displayed aspects of bone morphology not visible on the plain films. Odontogenic keratocyst diagnosis was confirmed by histopathological examination. The features identified by these combined clinical, imaging, and histologic findings are helpful in identifying an NBCCS patient, distinguishing keratocyst from others cysts or neoplasic lesions, and can therefore influence surgical management. NBCCS is a rare autosomal dominant cancer predisposition syndrome, which is important to recognize when a patient has multiple odontogenic keratocysts, because lifelong monitoring is essential for patient management.     

Levandoski panographic analysis in the diagnosis of hyperplasia of the coronoid process

The Levandoski panographic analysis of routine panoramic radiographs was developed to improve the diagnosis of hyperplasia of the coronoid process. The ratio of the length of the coronoid process to the condylar process in the Levandoski panographic analysis has a linear relationship with that in the cephalometric analysis (r = 0.75, n = 59). The ratio in patients with hyperplasia of the bilateral coronoid process was significantly greater than that of the control on both sides (P < 0.01). Although the minimum value of ratio in the patients was 1.15, the maximum value of the controls was 1.07. These results show that the Levandoski panographic analysis is useful in the evaluation of hyperplasia of the coronoid process in adults. When a patient complains of limited mouth-opening and the ratio is more than 1.1, further investigations should be made to verify the diagnosis of hyperplasia of the coronoid process.     

Surgical access to bilateral coronoid hyperplasia using the bicoronal flap

The use of the bicoronal flap in the surgical treatment of a case of bilateral coronoid hyperplasia is presented. The surgical approach to large lesions of the coronoid process is discussed.