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Case Report
An overlooked case of mandibular histiocytosis: Case report and review of the literature
1 Rabat Children’s hospital, Mohamed V University, Rabat, Morocco
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Kaoutar Imrani
Rabat Children’s hospital, Mohamed V University, Rabat,
Morocco
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Article ID: 100993Z01KI2019
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Imrani K, Ayouche O, Nazik A, Latifa C. An overlooked case of mandibular histiocytosis: Case report and review of the literature. Int J Case Rep Images 2019;10:100993Z01KI2019.ABSTRACT
Introduction: Langerhans histiocytosis is a rare disorder resulting from the proliferation and accumulation of a specific histiocytic cell called the Langerhans cell. Mandibular histiocytosis specifically can be asymptomatic or manifest as a jaw mass, Orthopanthogram as well as CT are required for the early diagnosis and prevention of total destruction of the osseous bone.
Case Report: We report the case of a 7-years-old patient, with a history of a non-resolving right jaw swelling treated as a dental abscess with antibiotics and analgesics for 14 days. The patient has no history of trauma, and was referred to our department for a right jaw swelling evolving for three months. Oral examination found an ulceroproliferative process of the posterior aspect of the dental part of the right mandible. Dental contrast enhanced CT showed a destruction of the periodontium, with an extensive geographical osteolytic homogeneous mass destroying the mandible. The diagnosis of histiocytosis was suggested histologically after biopsy. After a 6 weeks regimen of vinblastine and prednisolone with a clear regression, a surgical curettage of the bone lesion was performed. Local control was achieved with no complications in the six months follow up examination.
Conclusion: In conclusion, our case suggests that rapid diagnosis followed by minimally invasive surgery can be determinant in the prevention of possible deformation, especially in the pediatric population. Further investigation of the role of microbiopsy for screening should be discussed. Computed tomography is the first choice imaging technique in the diagnosis of mandibular histiocytosis.
Keywords: Computed tomography, Histiocytosis, Lytic bone, Mandible
SUPPORTING INFORMATION
Author Contributions
Othman Ayouche - Acquisition of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Allali Nazik - Acquisition of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Chat Latifa - Acquisition of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Kaoutar Imrani - Acquisition of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Guarantor of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementWritten informed consent was obtained from the patient for publication of this case report.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest.
Copyright© 2019 Kaoutar Imrani et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.
