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Congenital infiltrating lipomatosis of the face with exophytic temporomandibular joint ankylosis: a case report and review of the literature.

ABSTRACT: Congenital infiltrating lipomatosis of the face (CIL-F) is a rare lipomatous lesion with diffuse fatty infiltration of tissues and hyperplasia of underlying bone. We report clinical and CT findings in an unusual case of CIL-F presenting with progressive hemifacial asymmetry, manifesting as severely restricted mouth opening owing to exophytic temporomandibular joint ankylosis. The role of imaging in diagnosis is presented with a review of the literature. Differential diagnosis of CIL-F and its exclusion as a cause of hemifacial hyperplasia are also discussed.

SUBMITTER: Sahai S

PROVIDER: S-EPMC3667539 | biostudies-other | 2013

REPOSITORIES: biostudies-other

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Project description:Temporomandibular dysfunction involving anterior disc derangement with or without reduction, secondary to posterior ligament insufficiency is typically managed conservatively with success in a majority of patients. When conservative management fails, the next step in the continuum of care is unclear. Platelet-rich plasma injection combined with a 3-week immobilization period may be effective in treating posterior ligament insufficiency following a period of physical therapy. The result of this case was exceptionally successful, with the patient reporting 100% improvement 6 months post-injection. Prior to this case, we predicted a 20% success rate based on her inability to maintain the effects of conservative management over the long term.A 33-year-old white woman presented with temporomandibular dysfunction, which responded to an initial course of physical therapy aimed at restoring the mechanics of her temporomandibular joint, exercise management, and education on self-management strategies. She returned 20 months later and responded well to another course of physical therapy. Despite improvement in pain, range of motion, and mechanics, she continued to present with a reduction click at the end range of opening. The crisp and loud nature of the reduction click indicated a viable posterior ligament and reduction of the anteriorly displaced disc. She opted for platelet-rich plasma injection, provided by a chronic pain specialist with the assistance of a physical therapist. She was immobilized for 3 weeks, followed by a weaning period with reduced posterior support for an additional 5 weeks. Follow-up appointments with a physical therapist occurred at 3 weeks, 8 weeks, and 6 months post-injection.At 6 months, she reported 100% improvement. Objectively, there was no indication that the disc condyle relationship was disrupted. At 6 months post-platelet-rich plasma injection, preceded by a period of conservative physical therapy management, and followed with appropriate physical therapy follow-up, this individual had complete resolution of her anterior disc derangement with reduction.

Dataset Information

Congenital infiltrating lipomatosis of the face with exophytic temporomandibular joint ankylosis: a case report and review of the literature.

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