Fibrodysplasia Ossificans Progressiva (FOP): A Case Report with Oral and Maxillofacial Manifestations and new radiographic feature

Mozafari, Pegah Mosannen; Malek, Abdolreza; Anbiaee, Najmeh; Mostafavi Tabatabaee, Rosa; Talafi Noghani, Mahsa

doi:10.22038/ijp.2022.62645.4790

Fibrodysplasia Ossificans Progressiva (FOP): A Case Report with Oral and Maxillofacial Manifestations and new radiographic feature

Document Type : case report

Authors

Pegah Mosannen Mozafari ¹

Abdolreza Malek ²

Najmeh Anbiaee ³

Rosa Mostafavi Tabatabaee ⁴

Mahsa Talafi Noghani ⁵

¹ Associate Professor of Oral and Maxillofacial Diseases, School of Dentistry, Mashhad University of Medical Sciences, Mashhad,Iran.

² Clinical Research Development Unit of Akbar Hospital, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.

³ Associate Professor of Oral & Maxillofacial Radiology, School of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran.

⁴ Department of Oral and Maxillofacial Medicine, Dental School of Lorestan University of Medical Science, Khorramabad, Iran.

⁵ Post-graduate student, Department of Pediatric Dentistry, School of dentistry, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran.

10.22038/ijp.2022.62645.4790

Abstract

Fibrodysplasia ossificans progressiva (FOP) is a rare form of genetic disorder categorized by progressive heterotopic ossification and congenital deformity of the big toes. Heterotopic ossification follows gradual inflammation of the soft tissues (flare-up) and results in limited movements in joints such as the Temporomandibular joint (TMJ). No effective medical treatment has been recognized for the treatment of FOP. FOP is commonly misdiagnosed, especially in the maxillofacial region. Patients with FOP often experience temporomandibular joint ankylosis. Therefore, dental professionals should be careful in planning treatment, including avoiding anesthesia injections, especially in the mandible. This study presents a case of FOP with temporomandibular joint ankylosis. An eight-year-old boy with the chief complaint of reduced mouth opening and clinical and radiological features of FOP. The patient was referred to Mashhad Dental School in January 2016. He had not previously been diagnosed with FOP.

Keywords

fibrodysplasia ossificans progressive

TMJ

trismus

Kleyenstüber T. Anesthetic considerations in a child with fibrodysplasia ossificans progressiva. Southern African Journal of Anesthesia and Analgesia. 2019; 25 (1).
Vashisht R, Prosser D. Anesthesia in a child with fibrodysplasia ossificans progressiva. Pediatric Anesthesia. 2006; 16(6):684-8.
Rikhotso RE, Pillay L. Fibrodysplasia Ossificans Progressiva: Report of two cases and review of the literature. Oral and Maxillofacial Surgery Cases. 2020:100198.
Geddis-Regan A. Severe trismus and contraindicated exodontia in a patient with fibrodysplasia ossificans progressiva: case report. British Journal of Oral and Maxillofacial Surgery. 2018; 56(5):427-9.
Wang Z, Wang X, Liu B, Hou Y. Fibrodysplasia ossificans progressiva in a young adult with genetic mutation: Case report. Medicine. 2021; 100(9).
Roberts T, Stephen L, Scott C, Urban M, Sudi S, Beighton P. Fibrodysplasia ossificans progressiva (FOP) in South Africa: dental implications in 5 cases. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 2011; 112(1):11-8.
Braga JM, Silva MFAM, Silva LCPd, Seraidarian PI, Cruz RdA. Improvement of mouth opening for a patient with fibrodysplasia ossificans progressiva: a case report. Special Care in Dentistry. 2011; 31(6):220-5.
Sleem HA. Fibrodysplasia Ossificans Progressiva: TMJ Involvement and Feeding Dilemma. Oral Health Dental Sci. 2019; 3(2):1-3.
Van der Meij E, Becking A, Van der Waal I. Fibrodysplasia ossificans progressiva. An unusual cause of restricted mandibular movement. Oral diseases. 2006; 12(2):204-7.
Frederick S. Kaplan MDTUoP, Philadelphia, PA, Robert J. Pignolo MD, Ph.D.; The Mayo Clinic, Rochester, MN, editors. The medical management of fibrodysplasia ossificans progressiva: current treatment considerations. Proc Intl Clin Council FOP; 2021.
Orhan K, Uyanık LO, Erkmen E, Kilinc Y. Unusually severe limitation of the jaw attributable to fibrodysplasia ossificans progressiva: a case report with cone-beam computed tomography findings. Oral surgery, oral medicine, oral pathology and oral radiology. 2012; 113(3):404-9.
Ibourk A, Bouzoubaa SM, Yahya IB. Characteristics of the odontological management of patients with progressive ossifying fibrodysplasia. Journal of Oral Medicine and Oral Surgery. 2019; 25(4):33.
Luchetti W, Cohen RB, Hahn GV, Rocke DM, Helpin M, Zasloff M, et al. Severe restriction in jaw movement after routine injection of local anesthetic in patients who have fibrodysplasia ossificans progressiva. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 1996; 81(1):21-5.
Pignolo RJ, Shore EM, Kaplan FS. Fibrodysplasia ossificans progressiva: diagnosis, management, and therapeutic horizons. Pediatric endocrinology reviews: PER. 2013; 10(0 2):437.
Phore S, Panchal RS. Beaten silver appearance: normal or pathognomic in children? International Journal of Oral Health Sciences. 2018; 8(1):51-4.
Anitha N, Babu NA, Christina LMDAS. Importance of Radiographic Patterns هn Dentistry. European Journal of Molecular & Clinical Medicine. 2020; 7(5):1505-17.