title: Aberrant facial flushing following monobloc fronto-facial distraction.
abstract: BACKGROUND
  Patients with syndromic forms of craniosynostosis may experience functional problems such as raised intracranial pressure, proptosis, obstructive sleep apnoea and failure to thrive. The monobloc fronto-facial advancement with osteogenic distraction is increasingly used to correct these functional problems in one procedure as well as improve appearance. The authors report the phenomenon of post operative aberrant facial flushing - an unusual and previously unreported complication of the procedure.
  
  
  METHODS
  The case notes of 80 consecutive patients undergoing fronto-facial advancement by distraction using the rigid external distraction device (RED) were reviewed for features of aberrant facial flushing.
  
  
  RESULTS
  Four out of eighty individuals developed facial flushing after monobloc fronto-facial distraction using the rigid external distractor (RED) frame. All were female with Crouzon or Pfeiffer syndromes causing the severe functional problems for which they underwent the surgery. They were aged 6-8 years. Following removal of the frame, they developed intermittent but severe facial flushing. The flushing spontaneously settled in three patients after up to four years but persists in the other child seven years after her surgery.
  
  
  CONCLUSIONS
  Aberrant facial flushing is a rare but significant complication of monobloc fronto-facial surgery. It occurred in 4 of our 80 (5%) patients. The skull base osteotomies essential for the procedure are made anterior to the pterygopalatine ganglion and it is our contention that damage from these was responsible for a neuropraxia of its efferent nerve branches. A review of the autonomic control of the facial vascular system suggests that the phenomenon is due to an unequal process of recovery that leaves the cutaneous vasodilating parasympathetic or beta-adrenergic innervation relatively unopposed - a situation that persists until with time a normal balance of autonomic input is achieved.
authors: Department of Oral & Maxillofacial Surgery, United Hospitals Bristol NHS Trust, Bristol, UK. Electronic address: Alistair.cobb@uhbristol.nhs.uk.
authors: Alistair R M
authors: AR
authors: Cobb
authors: Craniofacial Service, Great Ormond Street Hospital for Children, London, UK.
authors: Michael
authors: M
authors: Vourvachis
authors: Department of Otolaryngology, Royal London Hospital, London, UK.
authors: Jahangir
authors: J
authors: Ahmed
authors: Department of Paediatric Otolaryngology, Great Ormond Street Hospital for Children, London, UK.
authors: Michelle
authors: M
authors: Wyatt
authors: Craniofacial Service, Great Ormond Street Hospital for Children, London, UK.
authors: David
authors: D
authors: Dunaway
authors: Department of Paediatric Neurosurgery, Great Ormond Street Hospital for Children, London, UK.
authors: Richard
authors: R
authors: Hayward
mesh_terms: D000168:Acrocephalosyndactylia / Q000503:physiopathology / Q000601:surgery; D000402:Airway Obstruction / Q000601:surgery; D002648:Child; D003394:Craniofacial Dysostosis / Q000503:physiopathology / Q000601:surgery; D005094:Exophthalmos / Q000601:surgery; D016267:External Fixators; D005147:Facial Bones / Q000601:surgery*; D005260:Female; D005483:Flushing / Q000209:etiology*; D005500:Follow-Up Studies; D005726:Ganglia, Parasympathetic / Q000293:injuries; D006801:Humans; D019586:Intracranial Hypertension / Q000601:surgery; D019857:Osteogenesis, Distraction / Q000009:adverse effects / Q000295:instrumentation / Q000379:methods*; D010027:Osteotomy / Q000009:adverse effects / Q000379:methods; D011183:Postoperative Complications; D012189:Retrospective Studies
keywords: Autonomic dysfunction; Craniosynostosis; Facial flushing; Fronto-facial; Monobloc