Surgical treatment of a severe microstomia developed following leucocytoclastic vasculitis

Abstract

Narrowing of the oral opening, also known as microstomia, is a rarely seen condition. It can develop iatrogenically after wide excision of perioral tumors with subsequent reconstruction or can be a component of severe scleroderma. In the pediatric population, exposure to caustic agents and subsequent scarring is another cause. This situation can cause problems in feeding, articulation, and oral hygiene. Most of the cases reported have been moderately affected; these require non-surgical treatment or a small procedure, such as a comissuroplasty, to provide an adequate oral opening. A case of severe microstomia developed as a result of a rare condition known as leucocytoclastic vasculitis is presented. A boy aged 2 1/2 years was admitted complaining of severely restricted mouth opening. This resulted from a complicated vasculitic illness, the etiology of which was unknown. The condition subsequently caused necrosis and gangrene with autoamputation of the toes of the right foot, the distal two phalanges of the right index finger, and the distal phalanx of the left ring finger. In addition to the limb problems, he also developed a similar situation around the left side of his mouth. For 21 days, he was treated with antibiotics and antifungal medication. He also had serial skin biopsies, which showed only a leucocytoclastic vasculitis. The initial examination revealed significant scarring around the mouth with a severe microstomia. His mouth opening area was restricted to 1 cm. The surgical correction consisted of opening the mouth using full-thickness incisions on both oral commissures to slightly overcorrect the situation. A moderately extensive dissection was carried out to identify the orbicularis muscles and to allow reconstruction of the muscular element of the commissures. Once the functional area of the commissures had been reconstructed, mucosal flaps were developed and advanced to form the red margin. After 7 years of follow-up, his mouth opening remains satisfactory, and he had been able to have extensive orthodontic treatment.