Oral management in a patient with Gardner-Diamond Syndrome: A case report

Abstract

Introduction: Gardner-Diamond Syndrome (GDS) is rare. It is characterized by the spontaneous formation of painful erythematous skin lesions that develop into ecchymoses within 24 h and then disappear progressively over days to weeks. The complications can be serious.

Presentation of case: A 35-year-old man with GDS was admitted to the oral surgery department for dental infectious focus eradication. Clinical and radiological examinations indicated extraction of tooth 17. It was performed with local anaesthesia, cardiac monitoring, and verbal reassurance therapy. After delivering anaesthesia, two intra-oral hematomas and a bruise quickly developed. Cardiovascular manifestations and a spontaneous painful right temporal erythematous skin lesion appeared in the next 24 h. The patient was briefly hospitalized in the cardiovascular medicine department. Over the next 21 days, some haematomas regressed, another expanded, and a new cervico-thoracic ecchymosis developed.

Discussion: The surgical and post-surgical complications in this clinical case raise several points concerning the oral management of patients with GDS. We propose to apply: verbal reassurance therapy during surgery; presurgical haematological evaluation and postsurgical daily haematological follow-up; and atraumatic surgery with intra- and post-surgical haemostatic precautions. Systematic antibiotic prophylaxis and non-steroidal anti-inflammatory drugs may also be useful. Other possibilities include desmopressin acetate, corticosteroids, antihistamines, plasmapheresis, and immunosuppressive agents; however, few cases have been treated with these strategies.

Conclusion: This case highlights the difficult management of patients with GDS who require oral surgery. Further studies are needed to improve the oral surgical procedures in these patients and to establish a systematic management algorithm.

Keywords: Bruising; Dental extraction; Ecchymosis; Gardner-Diamond Syndrome; Haematoma; Oral management.

Fig. 1

Cone-beam computerized tomography showing a peri-apical cyst on the upper-right second molar. A. Coronal section. B. Sagittal section.

Fig. 2

Immediately after anesthesia, two intra-oral hematomas of the right anterior (1) and posterior cheek (2) where the retractor was positioned, and a peri-apical bruising at the injection site (3).

Fig. 3

At 24 h, persistence of medium-sized hematoma of the inside right posterior cheek and right soft palate (A). Appearance of spontaneous and painful right temporal erythematous skin lesion (B).

Fig. 4

The haematoma on the inner right-posterior cheek had almost disappeared at postoperative day 14 (A) and was unchanged at 21 days (B). Lateralization of the right soft-palate haematoma was observed at postoperative day 14 (A) and its expansion to the anterior and posterior tonsillar pillar was noted at postoperative day 21 (B).

Fig. 5

On postoperative day 21, the patient also had a left cervico-thoracic ecchymosis that was resembled a bead necklace.