Title: Neuropathic Facial Pain and Anesthesia Dolorosa in Face Transplant Rejection
Authors: Gene W. Lee, MD; Casey Wollenman, MD; Stephanie Jones, MD
Institution: Department of Anesthesiology and Pain Management, University of Texas Southwestern Medical School, Dallas, TX
Background: A face transplant is a rare and relatively new procedure. First three full facial transplants in the US were all performed in 2011, and fewer than 10 full face transplants have been performed in the nation. Its long-term course remains under investigation. No loss of a transplanted face on patients taking the immunosuppression drugs appropriately have been reported . Rejection nearly always appears first dermatologically, which facilitates its detection compared to internal organ transplants. Rejection can present as a blotchy or spotty rash, and it may appear anywhere on the transplant .
Abstract: A 32-year-old man who had undergone a full facial transplant ~7 years prior presented concerning for transplant rejection. He had several days of persistent lip ulcers and malaise. Dermatology service indicated the erosions were more typical of rejection than vesicles (HSV). He was hospitalized for 7 days receiving treatment for organ rejection along with empiric HSV treatment comprised of methylprednisolone, mycophenolate, tacrolimus, and valacyclovir.
Pain service was consulted on day 2 of admission for facial pain evaluation. Patient noticed pain on his face a few days prior to admission. Patient localized the pain to the bilateral forehead and the triangular area from the corners of the upper lips to the glabella (i.e., the danger triangle of the face), including anesthesia dolorosa of right forehead where he typically lacks sensation. He denied tenderness to palpation. Pain was described as burning without radiation and was constant. There were no known triggers or temporal changes. Patient reported experiencing the same pain in the past when he has had rejection episodes. Pain was rated at 5/10 with oral (PO) and intravenous (IV) narcotics and 10/10 without medication. Patient stated that he has tried gabapentin, pregabalin, and homeopathic medicine in the past without relief. He reported having baseline facial pain that was different in quality than his current pain. The baseline pain was dull and “in the bone,” and it was controlled by 1 tablet of hydrocodone-acetaminophen 10-325 mg every 6 hours as needed (PRN).
Pain service recommended increasing the frequency of hydrocodone administration to every 4 hours PRN with hydromorphone (IV) 0.5mg every 4 hours PRN for breakthrough pain since opioids have provided relief for the patient (patient requested nearly all the PO and half the IV doses during hospitalization). We also recommended topiramate 25 mg at bedtime since the quality of the pain appeared neuropathic. This achieved adequate pain control per patient.
Conclusion: Pain associated with face transplant rejection can be present in areas without tactile sensation (i.e., anesthesia dolorosa), suggesting there is a central representation of the area responsible for painful sensations, which is affected by the rejection process. The combination of opioid analgesics and neuropathic medications, in this case topiramate, was able to control an acute exacerbation of pain. Gabapentin and pregabalin could have been a sensible alternative (had patient not experienced the lack of efficacy in the past).
1. Lee, W. Facial Transplantation. Retrieved from URL <http://www.hopkinsmedicine.org/transplant/programs/reconstructive_transp....
2. Kanitakis J, Badet L, Petruzzo P, Béziat JL, Morelon E, Lefrançois N, Françès C, Claudy A, Martin X, Lengelé B, Testelin S, Devauchelle B, Dubernard, JM. Clinicopathologic Monitoring of the Skin and Oral Mucosa of the First Human Face Allograft: Report on the First Eight Months. Transplantation 2006; 82:1610.