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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 11  |  Issue : 2  |  Page : 62-68

Coronoid Hyperplasia (Jacob’s Disease) as a Rare Cause of Restricted Mouth Opening: a Case Report


1 Department of Oral and Maxillofacial Surgery, Dental Implant Research Center, School of Dentistry, Isfahan University of Medical Science, Isfahan, Iran
2 Department of Oral and Maxillofacial Radiology, School of Dentistry, Kermanshah University of Medical Science, Kermanshah, Iran
3 Department of Oral and Maxillofacial Surgery, School of Dentistry, Isfahan University of Medical Science, Isfahan, Iran

Date of Submission26-Jul-2019
Date of Decision17-Dec-2019
Date of Acceptance14-Feb-2020
Date of Web Publication10-Jun-2020

Correspondence Address:
Assistant Professor Maryam Ghazizadeh
Department of Oral and Maxillofacial Radiology, School of Dentistry, Kermanshah University of Medical sciences, Kermanshah
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/denthyp.denthyp_61_19

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  Abstract 


Introduction: Restricted mouth opening (MO) is associated with the problems in intra/extra-articular components such as trauma to the articular bone component especially at the early ages, chronic displacement of the articular disk, tumors, condylar anomalies, coronoid hyperplasia and hyperactivity of jaw muscles. A pseudo-joint can form between the zygoma and the hyperplastic coronoid process (Jacob’s disease). Pseudo-joint restricts MO, it causes multiple problems for patients, and finally it decreases the quality of their or patients’ lives. Therefore, early diagnosis and treatment are very important. Case Report: Described here is a 28-year-old man that due to trauma in childhood, his coronoid process became hyperplastic. The pseudo-joint formed between coronoid process and zygomatic bone interfere with MO. Discussion: The intraoral approach offers direct access without the risk of facial nerve injury or scars on the face

Keywords: Coronoid hyperplasia, jacob’s disease, temporomandibular joint


How to cite this article:
Samandari MH, Shalamzari ME, Ghazizadeh M, Navaei H. Coronoid Hyperplasia (Jacob’s Disease) as a Rare Cause of Restricted Mouth Opening: a Case Report. Dent Hypotheses 2020;11:62-8

How to cite this URL:
Samandari MH, Shalamzari ME, Ghazizadeh M, Navaei H. Coronoid Hyperplasia (Jacob’s Disease) as a Rare Cause of Restricted Mouth Opening: a Case Report. Dent Hypotheses [serial online] 2020 [cited 2020 Jul 16];11:62-8. Available from: http://www.dentalhypotheses.com/text.asp?2020/11/2/62/286350




  Introduction Top


Growth and development of the temporomandibular joint (TMJ) depends on the full development of the mandible. Although TMJ is one of the most common sites for traumatic injuries, it is usually disregarded during the checkup. Unilateral trauma to the condyle and coronoid during the early ages of life leads to the mouth opening restriction and facial asymmetry in adulthood.[1]

One of the reasons for restricted mouth opening (MO) is an abnormal increase in the length of coronoid. The long coronoid interferes with the medial or temporal surface of the zygomatic bone which decreases the infratemporal space. This space is required for mandibular rotation and translation. Therefore, MO and lateral movements are restricted.[2],[3] Coronoid hyperplasia was first described in 1835 by Von Langenback and was published in 1963 by Row.[4] He described it as an abnormal process of coronoid then categorized it into two types of developmental or tumoral. The developmental type is usually bilateral while the tumoral type is usually unilateral. French anatomist Jacob reported the first case of Jacob disease in 1989 forming a pseudo-joint between the coronoid process and the zygomatic arch.[5]

This report presents a case of Jacob’s disease as a consequence of trauma, which caused limitation in mouth opening.


  Case Report Top


A 28-year-old man suffering from joint pain during mouth opening referred to the Oral and Maxillofacial Surgery Department of Isfahan University of Medical Sciences. The patient had no history of using drugs, alcohol, tobacco or cigarette. The patient reported a jaw trauma at the age of 10. Clinical examination didn’t show any obvious facial asymmetry. Maximal interincisal distance was 25 mm [Figure 1]. Temporomandibular joint examination did not report any tenderness or joint click.
Figure 1 The patient’s limitation in mouth opening.

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Then panoramic radiography and CT scan with 3D reconstruction were obtained. On the images, there was an elongation of the left coronoid process, and an opaque mass at the tip of the coronoid was observed [Figure 2]. The opaque mass had the typical mushroom-shaped appearance extended to the left of the zygomatic arch root, and they were very close together.
Figure 2 Patient’s panoramic radiography (high) and CT scan (low). Arrow in all the images shows the bony process.

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Regarding the MO limitation and the need to receive dental services, the surgical operation was done. Due to the MO limitation, the patient’s intubation was done through the nose using fiber optic. The patient’s anesthesia was performed by injecting Midazolam, Fentanyl, Nasonal, Morphine and Atracurium.

After palpation of the hyperplastic coronoid process intraorally, an intra-oral incision of size 2-cm was performed to achieve the coronoid region. The coronoid was completely exposed by removing mucosal tissue, muscle and periosteum [Figure 3]. Two weeks after surgery, the MO limitation was improved, and there was not any special problem for the patient.
Figure 3 Bone process of the left coronoid removed during surgery.

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  Discussion Top


A literature review revealed that 62 cases of Jacob’s disease (20 female and 42 male) have been reported since 1943 [Table 1].[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26],[27],[28],[29],[30],[31],[32],[33],[34],[35],[36],[37],[38],[39],[40],[41],[42],[43],[44],[45],[46],[47],[48],[49],[50],[51],[52],[53],[54],[55],[56],[57],[58],[59],[60],[61] In the reported cases, the patients aged in the range of 5–73 with an average of 30 years. The majority of patients were diagnosed before the age of 30.
Table 1 Summary of the reported cases of Jacob’s disease

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The average of maximum MO was 0–25 mm. Among reported cases, eight cases were bilateral and the rest of them were unilateral. 19 cases were treated by extraoral surgery method, 29 cases were treated by intraoral surgery method and five cases were treated by applying both methods. Only one case was treated by the method called Sphenopalatine ganglion block.[54] Physiotherapy has an important role in preventing relapse of the condition. However, one relapsed case was reported.[38] Since 1984, computed tomography has been used to diagnose the Jacob’s disease, and gradually the combination of two radiography methods (panoramic and CT) played an important role in diagnosing of that. After exploring the literature, we found the present case is the first reported Jacob’s disease from Iranian population.

Coronoid hyperplasia restricts anterior and posterior movements of the mandibular condyle.[62] There are different theories explaining the causes of coronoid hyperplasia, such as trauma, hyperactivity of temporal muscle, hormonal stimulus and genetic. This excessive growth of the coronoid leads to the mandible movement restriction due to the compression of the coronoid appendage to the zygomatic bone.[63]

Jacob’s disease is a rare condition of pseudo-joint formation between hyperplastic coronoid and the internal side of the zygomatic bone.[64] Patients with Jacob’s disease usually have a painless progressive reduction in MO.[45],[46],[48] All of the signs related to this disease are similar to the coronoid hyperplasia that involves advanced and asymptomatic limitation of mouth opening without occlusion abnormality. Therefore, these two disorders must be distinguished.

Although the radiological diagnosis might seem simple, because of very few numbers of existed cases, coronoid hyperplasia and Jacob’s disease are usually ignored by the clinician. Although panoramic image is adequate for diagnosing, it only creates a two-dimensional image. Three-dimensional CT (3DCT) can reconstruct images in coronal, sagittal and oblique planes. [53],[57],[61],[65] Levandoski analysis is usually used for diagnosing coronoid hyperplasia. in panoramic images. In this analysis, a line is drawn from the lowest part of mandibular angle to the highest limit of mandibular condyle and top of the coronoid. The height of normal coronoid is shorter than condyle (between 12 and 13 mm) but in hyperplastic size, it is more than 20 mm.[66]

Treatment of Jacob’s disease is done by surgery with prolonged Physiotherapy especially in cases of MO limitation. The standard surgical treatment removes coronoid-zygoma interference by coronoidectomy with the intraoral approach. However, coronal and submandibular approaches have been used.[4] Torre et al.[38], reported a 16-year-old boy with Jacob disease. In their case, due to lack of compliance with physiotherapy, MO limited to 25 mm 4 months after intraoral coronoidectomy. Zarembinski et al, reported a case with cluster headache and temporal muscle tenderness because of bilateral coronoid hyperplasia with pseudo-joint. it was temporary treated by Sphenopalatine ganglion block and injection of 1 mL of 0.25% bupivacaine and 1 mL of dexamethasone (10 mg/mL).[54] The intraoral approach offers direct access without the risk of facial nerve injury or scars on the face, but if the exostosis is bulbous and large, an extraoral approach may be necessary.[40],[44],[55]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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