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.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
Takatao T, Ohsone H, Tsukakoshi H (1989) Treatment of severe microstomia caused by swallowing of caustic soda. Oral Surg Oral Med Oral Pathol 67:20–24
La Trenta G, Grant RT, Haworth RD, Madden M, Hoffman LA (1992) Functional reconstruction for severe postburn microstomia. Ann Plast Surg 29:178–181
Maragakis GM, Tempone MG (1998) Microstomia following facial burns. J Clin Pediatr Dent 23:69–74
Sanders B, McKelvy B, Cruickshank G (1977) Correction of microstomia secondary to sclerodermatomyositis. J Oral Surg 35:57–59
Pizzo G, Scardina GA, Messina P (2003) Effects of a nonsurgical exercise program on the decreased mouth opening in patients with systemic scleroderma. Clin Oral Invest 7:175–178
Conine TA, Carlow DL, Moore PS (1987) Dynamic orthoses for the management of microstomia. J Rehabil Res 24:43–48
Ferreira LM, Minami E, Andrews JM (1994) Freeman–Sheldon syndrome: surgical correction of microstomia. Br J Plast Surg 47:201–202
Guyuron B, Winkler PA (1988) Craniocarpotarsal dysplasia: the whistling face syndrome. Ann Plast Surg 20:86–88
Freeman EA, Sheldon JH (1938) Cranio-carpo-tarsaldystrophy. An undescribed congenital malformation. Arch Dis Child 13:277–283
Burian F (1963) The ‘whistling face’ characteristic in a compound cranio-facio-corporal syndrome. Br J Plast Surg 16:140–163
Taylor LB, Walker J (1997) A review of selected microstomia prevention appliances. Pediatr Dent 19:413–418
Dougherty ME, Warden GD (2003) A thirty-year review of oral appliances used to manage microstomia, 1972 to 2002. J Burn Care Rehabil 24:418–431
Carlow DL, Conine TA, Moore PS (1987) Static orthoses for the management of microstomia. J Rehabil Res 24:35–42
Berlet AC, Ablaza VJ, Servidio P (1993) A refined technique for oral commissurotomy. J Oral Maxillofac Surg 51:1400–1403
Mehra P, Caiazzo A, Bestgen S (1998) Bilateral oral commissurotomy using buccal mucosa flaps for management of microstomia: report of a case. J Oral Maxillofac Surg 56:1200–1203
Sawada Y, Ara M, Yotsuyanagi T (1990) Expanding oral angle plasty using a subcutaneous pedicle flap to correct severe microstomia after extensive facial burns. Burns 16:230–232
Anderson R, Kurtay M (1971) Reconstruction of the corner of the mouth. Plast Reconstr Surg 47:463
Fernandez Villoria JM (1972) A new method of elongation of the corner of the mouth. Plast Reconstr Surg 49:52
Claudy A (1998) Pathogenesis of leukocytoclastic vasculitis. Eur J Dermatol 8:75–79
Koutkia P, Mylonakis E, Rounds S, Erickson A (2001) Leucocytoclastic vasculitis an update for the clinician. Scand J Rheumatol 30:315–322
Jorizzo JL (1993) Classification of vasculitis. J Invest Dermatol 100:106–110
Callen JP, Ekenstam E (1987) Cutaneous leucocytoclastic vasculitis: clinical experience in 44 patients. Southern Med J 80:848–851
Chang JS, Lin JS, Peng CT, Tsai CH (1999) Kawasaki disease complicated by peripheral gangrene. Pediatr Cardiol 20:139–142
Mordjikian E (2002) Severe microstomia due to burn by caustic soda. Burns 28:802–805
Converse JM (1959) Technique of elongation of the oral fissure and restoration of the angle of the mouth. The surgical treatment of facial injuries. Williams & Wilkins, Baltimore, MD
Kazanjian VH, Roopenian A (1954) The treatment of lip deformities resulting from electric burns. Am J Surg 88:884
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Tuncer, S., Bello-Rojas, G., Blake, M. et al. Surgical treatment of a severe microstomia developed following leucocytoclastic vasculitis. Eur J Plast Surg 30, 183–187 (2007). https://doi.org/10.1007/s00238-007-0170-6
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00238-007-0170-6