Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature

Abstract Background Masticatory muscle tendon-aponeurosis hyperplasia (MMTAH) is a relatively newly identified clinical condition that manifests as trismus with a square-shaped mandible. Herein, we report a case of MMATH that was initially misdiagnosed for polymyositis due to trismus and simultaneou...

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Main Authors: Wataru Katagiri, Daisuke Saito, Satoshi Maruyama, Makiko Ike, Hideyoshi Nisiyama, Takafumi Hayashi, Jun-ichi Tanuma, Tadaharu Kobayashi
Format: Article
Language:English
Published: SpringerOpen 2023-05-01
Series:Maxillofacial Plastic and Reconstructive Surgery
Subjects:
Online Access:https://doi.org/10.1186/s40902-023-00386-6
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author Wataru Katagiri
Daisuke Saito
Satoshi Maruyama
Makiko Ike
Hideyoshi Nisiyama
Takafumi Hayashi
Jun-ichi Tanuma
Tadaharu Kobayashi
author_facet Wataru Katagiri
Daisuke Saito
Satoshi Maruyama
Makiko Ike
Hideyoshi Nisiyama
Takafumi Hayashi
Jun-ichi Tanuma
Tadaharu Kobayashi
author_sort Wataru Katagiri
collection DOAJ
description Abstract Background Masticatory muscle tendon-aponeurosis hyperplasia (MMTAH) is a relatively newly identified clinical condition that manifests as trismus with a square-shaped mandible. Herein, we report a case of MMATH that was initially misdiagnosed for polymyositis due to trismus and simultaneous lower limb pain, with literature review. Case presentation A 30-year-old woman had a history of lower limb pain after exertion for 2 years. Initial physical examination had been performed at the Department of General Medicine in our hospital. There was also redness in the hands and fingers. Although polymyositis was suspected, it was denied. The patient visited our department for right maxillary wisdom tooth extraction. Clinical examination revealed that the patient had a square-shaped mandible. The maximal mouth opening was 22 mm. There was no temporomandibular joint pain at the time of opening. Furthermore, there was awareness of clenching while working. Panoramic radiography revealed developed square mandibular angles with flattened condyles. Computed tomography showed enlarged masseter muscles with high-density areas around the anterior and lateral fascia. Magnetic resonance imaging also showed thickened tendons and aponeuroses on the anterior surface and inside bilateral masseter muscles. Finally, the patient was diagnosed with MMTAH. Bilateral aponeurectomy of the masseter muscles with coronoidectomy and masseter muscle myotomy was performed under general anesthesia. The maximum opening during surgery was 48 mm. Mouth opening training was started on day 3 after surgery. Histopathological examination of the surgical specimen showed that the muscle fibers were enlarged to 60 μm. Immunohistochemistry testing for calcineurin, which was associated with muscle hypertrophy due to overload in some case reports, showed positive results. Twelve months after surgery, the mouth self-opening and forced opening were over 35 mm and 44 mm, respectively. Conclusions Herein, we report a case of MMATH. Lower limb pain due to prolonged standing at work and overload due to clenching were considered risk factors for symptoms onset of MMATH.
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spelling doaj.art-ca476389ed1d499f8f567df8fa3348762023-05-07T11:08:37ZengSpringerOpenMaxillofacial Plastic and Reconstructive Surgery2288-85862023-05-014511810.1186/s40902-023-00386-6Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literatureWataru Katagiri0Daisuke Saito1Satoshi Maruyama2Makiko Ike3Hideyoshi Nisiyama4Takafumi Hayashi5Jun-ichi Tanuma6Tadaharu Kobayashi7Department of Oral and Maxillofacial Surgery, Gifu University Graduate School of MedicineDivision of Reconstructive Surgery for Oral and Maxillofacial Region, Faculty of Dentistry & Graduate School of Medical and Dental Science, Niigata UniversityOral Pathology Section, Department of Surgical Pathology, Niigata University HospitalDivision of Oral and Maxillofacial Radiology, Faculty of Dentistry & Graduate School of Medical and Dental Sciences, Niigata UniversityDivision of Oral and Maxillofacial Radiology, Faculty of Dentistry & Graduate School of Medical and Dental Sciences, Niigata UniversityDivision of Oral and Maxillofacial Radiology, Faculty of Dentistry & Graduate School of Medical and Dental Sciences, Niigata UniversityDivision of Oral Pathology, Faculty of Dentistry & Graduate School of Medical and Dental Sciences, Niigata UniversityDivision of Reconstructive Surgery for Oral and Maxillofacial Region, Faculty of Dentistry & Graduate School of Medical and Dental Science, Niigata UniversityAbstract Background Masticatory muscle tendon-aponeurosis hyperplasia (MMTAH) is a relatively newly identified clinical condition that manifests as trismus with a square-shaped mandible. Herein, we report a case of MMATH that was initially misdiagnosed for polymyositis due to trismus and simultaneous lower limb pain, with literature review. Case presentation A 30-year-old woman had a history of lower limb pain after exertion for 2 years. Initial physical examination had been performed at the Department of General Medicine in our hospital. There was also redness in the hands and fingers. Although polymyositis was suspected, it was denied. The patient visited our department for right maxillary wisdom tooth extraction. Clinical examination revealed that the patient had a square-shaped mandible. The maximal mouth opening was 22 mm. There was no temporomandibular joint pain at the time of opening. Furthermore, there was awareness of clenching while working. Panoramic radiography revealed developed square mandibular angles with flattened condyles. Computed tomography showed enlarged masseter muscles with high-density areas around the anterior and lateral fascia. Magnetic resonance imaging also showed thickened tendons and aponeuroses on the anterior surface and inside bilateral masseter muscles. Finally, the patient was diagnosed with MMTAH. Bilateral aponeurectomy of the masseter muscles with coronoidectomy and masseter muscle myotomy was performed under general anesthesia. The maximum opening during surgery was 48 mm. Mouth opening training was started on day 3 after surgery. Histopathological examination of the surgical specimen showed that the muscle fibers were enlarged to 60 μm. Immunohistochemistry testing for calcineurin, which was associated with muscle hypertrophy due to overload in some case reports, showed positive results. Twelve months after surgery, the mouth self-opening and forced opening were over 35 mm and 44 mm, respectively. Conclusions Herein, we report a case of MMATH. Lower limb pain due to prolonged standing at work and overload due to clenching were considered risk factors for symptoms onset of MMATH.https://doi.org/10.1186/s40902-023-00386-6AponeurosisCalcineurinHyperplasiaMasticatory musclesSquare-shaped mandibleFascia
spellingShingle Wataru Katagiri
Daisuke Saito
Satoshi Maruyama
Makiko Ike
Hideyoshi Nisiyama
Takafumi Hayashi
Jun-ichi Tanuma
Tadaharu Kobayashi
Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature
Maxillofacial Plastic and Reconstructive Surgery
Aponeurosis
Calcineurin
Hyperplasia
Masticatory muscles
Square-shaped mandible
Fascia
title Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature
title_full Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature
title_fullStr Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature
title_full_unstemmed Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature
title_short Masticatory muscle tendon-aponeurosis hyperplasia that was initially misdiagnosed for polymyositis: a case report and review of the literature
title_sort masticatory muscle tendon aponeurosis hyperplasia that was initially misdiagnosed for polymyositis a case report and review of the literature
topic Aponeurosis
Calcineurin
Hyperplasia
Masticatory muscles
Square-shaped mandible
Fascia
url https://doi.org/10.1186/s40902-023-00386-6
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