Case report of osteomyelitis of the mandible in osteopetrosis and management considerations

Type Article
Date 2021
Language English
Author(s) Allal Sarah1, Rabuel Valentin1, Gengler Charline1, Douchet Catherine2, Allal FrancoisORCID3, Zwetyenga Narcisse1, 4
Affiliation(s) 1 : Department of maxillofacial and oral surgery, University Hospital of Dijon, 2 boulevard du Maréchal de Lattre de Tassigny, BP 77908, 21079, Dijon cedex, France
2 : Department of Pathology, University Hospital of Dijon, Dijon, France
3 : MARBEC, Univ. Montpellier, Ifremer, CNRS, IRD, 34250, Palavas-les-Flots, France
4 : CIC-EC 1432 (Centre d'Investigation Clinique - Epidémiologie clinique) - Pr QUANTIN Catherine, Chirurgie endocrinienne, Dr GOUDET Pierre, France
Source International Journal Of Surgery Case Reports (2210-2612) (Elsevier BV), 2021 , Vol. 81 , P. 105813 (5p.)
DOI 10.1016/j.ijscr.2021.105813
Keyword(s) Osteomyelitis, Mandible, Case report, Osteopetrosis, Management
Abstract

Introduction and importance

Osteopetrosis is a poorly known and probably underdiagnosed pathology. It is caused by various genetic abnormalities resulting in osteoclast dysfunction. Functional and aesthetic consequences have a major impact on the patient’s quality of life. Ten percent of osteopetrosis cases develop osteomyelitis that usually involves the mandible. Management of this complication remains complex and often unsatisfactory.

Case presentation

We report a case of a 62-year-old woman with osteopetrosis, complicated by mandibular osteomyelitis with intra-oral bone exposure and submental fistulas. Management was performed with antibiotic therapy and surgical necrotic resection. This cured the fistulas but the bone exposure persisted.

Discussion

This case report highlights the difficulty of achieving complete healing of osteomyelitis in osteopetrosis. Antibiotic therapy, surgical management, or even hyperbaric oxygen therapy are required, but must be adapted to the case. A free flap procedure is undesirable but, when it is necessary, a bone marrow transplant could be considered to restore osteoclast function.

Conclusion

The management of mandibular osteomyelitis in patients with osteopetrosis must adapt to the situation and severity. To avoid most cases of osteomyelitic complications in patients suffering from osteopetrosis, we propose that a preventive strategy of better dental care should be considered.

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