Temporomandibular Joint Prosthesis in a Patient with Congenital Infiltrating Lipomatosis of the Face with Bony Ankylosis of the Temporomandibular Joint: A Case Report

Lauren C.M. Bulthuis*, Jean Pierre T.F. Ho, Petra C.M. Zuurbier, Michail Koutris, Jitske W. Nolte, Jan de Lange

*Corresponding author for this work

Research output: Contribution to JournalArticleAcademicpeer-review

Abstract

Hemifacial hyperplasia (HFH) is a rare congenital disorder characterized by marked unilateral overgrowth of the facial tissues. A subtype of HFH is congenital infiltrating lipomatosis of the face (CIL-F). This disease is characterized by unilateral diffuse infiltration of mature adipose cells in the facial soft tissue and is associated with skeletal hypertrophy. This work aims to report a case of a CIL-F patient with right facial asymmetry and progressive growth at adolescent age, causing mandibular asymmetry due to signs of concomitant unilateral condylar hyperplasia. At the age of seventeen, a condylectomy was performed to stop the progression of asymmetric mandibular growth. Five years later, the patient developed CIL-F-associated temporomandibular joint ankylosis, manifesting as progressive restricted mouth opening along with temporal facial pain. In this CIL-F patient, a TMJ reconstruction with an alloplastic total joint prosthesis was successfully performed with optimal maximal mouth opening, complete alleviation of temporal facial pain, and stable dental occlusion one year postoperatively. A TMJ reconstruction with a complete alloplastic total joint prosthesis proved to be a predictable, stable, and safe treatment option in a patient with CIL-F-associated TMJ ankylosis who was previously treated with condylectomy due to progressive mandibular asymmetry.

Original languageEnglish
Article number7723
Pages (from-to)1-10
Number of pages10
JournalJournal of Clinical Medicine
Volume12
Issue number24
DOIs
Publication statusPublished - 2 Dec 2023

Bibliographical note

This article belongs to the Special Issue Updates and Challenges in Maxillo-Facial Surgery.

Publisher Copyright:
© 2023 by the authors.

Keywords

  • case report
  • hemifacial hyperplasia
  • hemimandibular elongation
  • mandibular asymmetry
  • maxillofacial surgery

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