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

Ambre Bellot; Rémi Curien; Anaïs Derache; Bruno Delaître; Raffaele Longo; Yinka Zevering; Julie Guillet; Bérengère Phulpin

doi:10.1016/j.ijscr.2020.09.098

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

Int J Surg Case Rep. 2020:75:367-371. doi: 10.1016/j.ijscr.2020.09.098. Epub 2020 Sep 17.

Authors

Ambre Bellot¹, Rémi Curien², Anaïs Derache³, Bruno Delaître⁴, Raffaele Longo⁵, Yinka Zevering⁶, Julie Guillet⁴, Bérengère Phulpin⁴

Affiliations

¹ Department of Oral Surgery, CHR Metz-Thionville, 1 Allée du Château, 57085, Ars-Laquenexy, France; Department of Odontology, CHRU Nancy, Rue du Morvan, 54500, Vandoeuvre-lès-Nancy, France. Electronic address: ambre.bellot10@gmail.com.
² Department of Oral Surgery, CHR Metz-Thionville, 1 Allée du Château, 57085, Ars-Laquenexy, France.
³ Department of Odontology, CHRU Nancy, Rue du Morvan, 54500, Vandoeuvre-lès-Nancy, France.
⁴ Department of Odontology, CHRU Nancy, Rue du Morvan, 54500, Vandoeuvre-lès-Nancy, France; Odontology Faculty of Nancy, 96 Avenue du Maréchal de Lattre de Tassigny, 54004, Nancy, France.
⁵ Department of Oncology, CHR Metz-Thionville, 1 Allée du Château, 57085, Ars-Laquenexy, France; Clinical Research Support Unit, CHR Metz-Thionville, 1 Allée du Château, 57085, Ars-Laquenexy, France.
⁶ Clinical Research Support Unit, CHR Metz-Thionville, 1 Allée du Château, 57085, Ars-Laquenexy, France.

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.

Publication types

Case Reports