Optimizing care for patients with maxillomandibular Gorham-Stout disease

Area of Science:

• Oral and Maxillofacial Surgery
• Vascular Anomalies
• Bone Metabolism

Background:

• Gorham Stout disease (GSD) is a rare condition characterized by progressive osteolysis.
• Maxillary and/or mandibular involvement occurs in approximately 20% of GSD cases.
• The etiology of GSD remains incompletely understood, necessitating effective management strategies.

Purpose of the Study:

• To propose a management algorithm for successful surgical reconstruction in maxillo-mandibular GSD.
• To identify factors influencing treatment outcomes in GSD affecting the jaws.
• To emphasize the importance of disease control prior to reconstructive procedures.

Main Methods:

• Systematic literature review of maxillo-mandibular GSD cases.
• Inclusion of two additional patient cases to the reviewed series.
• Logistic regression analysis to determine factors associated with treatment response.

Main Results:

• A total of 100 patients with maxillo-mandibular GSD were analyzed.
• Radiotherapy demonstrated a significant association with disease remission (OR = 7.363, P = .044).
• Reconstructive surgery without prior osteolysis stabilization led to disease recurrence and flap resorption.

Conclusions:

• Strict control of Gorham Stout disease is crucial before undertaking major reconstructive surgery.
• Radiotherapy emerged as the sole significant factor for achieving disease control in the studied cohort.
• This study underscores the necessity of stabilizing GSD to prevent surgical complications.