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[Gorlin-Goltz syndrome].

P Roth1, K Grätz, H Sailer

  • 1Clinique et Policlinique de Chirurgie Maxillo-Faciale, Université de Zurich.

Swiss Dent
|January 1, 1991
PubMed
Summary
This summary is machine-generated.

This case study details a 22-year patient journey with Gorlin-Goltz Syndrome, highlighting aggressive basal cell carcinomas and odontogenic keratocysts. It emphasizes the complex surgical interventions and management required for this rare genetic disorder.

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Area of Science:

  • Oncology
  • Genetics
  • Oral and Maxillofacial Surgery

Background:

  • Gorlin-Goltz Syndrome, also known as nevoid basal cell carcinoma syndrome, is a rare autosomal dominant disorder.
  • It is characterized by multiple basal cell carcinomas, keratocystic odontogenic tumors, skeletal abnormalities, and intracranial calcifications.

Observation:

  • A patient with Gorlin-Goltz Syndrome was followed for over 22 years, experiencing severe disease progression.
  • This included multiple resections for basal cell carcinomas on the face and trunk, and numerous maxillary keratocysts.
  • One cyst showed histologic features of ameloblastic fibroma, and a squamous cell carcinoma necessitated mandibular resection and radiotherapy.

Findings:

  • The patient underwent extensive surgical management, including mandibular resection, reconstruction with an AO plate, and radiotherapy.

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  • A recurrence required further resection and secondary reconstruction with a myocutaneous flap, despite the presence of lung metastases.
  • A literature review of similar cases and the authors' therapeutic approach are discussed.

    • Implications:

    • This case underscores the aggressive nature of Gorlin-Goltz Syndrome and the need for long-term, multidisciplinary management.
    • It highlights the challenges in treating recurrent malignancies and odontogenic tumors associated with the syndrome.
    • The study contributes to understanding the natural history and therapeutic strategies for Gorlin-Goltz Syndrome.