A possible case of complex regional pain syndrome in the orofacial region

Pain Medicine 11 (2), 274-80 (Feb 2010)

 Sakamoto E, Shiiba S, Noma N, Okada-Ogawa A, Shinozaki T, Kobayashi A, Kamo H, Koike K, Imamura Y;

Objective. To present a case of complex regional pain syndrome (CRPS) type II with sympathetic dysfunction and trophic changes in the orofacial region, which was partially responsive to intravenous ketamine.

Patient. The patient was a 68-year-old man who suffered from inveterate pain with trophic changes of the right face and tongue and vasomotor dysfunction on the right side of the face after ipsilateral trigeminal nerve block. Allodynia and hyperalgesia were observed on the affected side of the face. Pain initially improved after sympathetic nerve block, but similar pain returned that was unresponsive to the same procedure. Repeated intravenous administration of low-dose ketamine preceded by intravenous midazolam alleviated the pain, but trophic changes of the tongue persisted.

Discussion. CRPS in the orofacial region has not been clearly defined and has been infrequently documented. Clinical findings in this patient met the criteria of the International Association for the Study of Pain's and Harden's diagnostic criteria for CRPS. The reason for gradual pain relief after induction of intravenous ketamine therapy was unclear, but the fact that only ketamine and not other various pain medicines or procedures alleviated the pain is important to note.

Conclusion. Distinct cases of CRPS involving the orofacial region are rare. Thorough observations and documentation of signs and symptoms may lead to future standardization of diagnostic criteria and treatment strategies for this disorder.

A Severe Case of Complex Regional Pain Syndrome I (Reflex Sympathetic Dystrophy) Managed with Spinal Cord Stimulation

Pain Practice
Volume 10 Issue 1, Pages 78 - 83
Bernard Canlas, MD*; Thomas Drake, PA-C*; Eric Gabriel, MD †
*Institute of Pain Management, P.A., Jacksonville, Florida; † St. Vincent's Brain and Spine Institute, Jacksonville, Florida, U.S.A.
Correspondence to Bernard R. Canlas, MD, Institute of Pain Management, P.A.––4243 Sunbeam Road Ste.6 Jacksonville, FL 32257, U.S.A. E-mail: drcanlas@comcast.net.
Disclosure: Dr.Canlas and Dr. Gabriel are guest consultants of St. Jude Neuromodulation division. Dr. Canlas' has stock ownership with St. Jude Medical.

ABSTRACT
Complex regional pain syndrome is a condition that usually affects the upper or lower extremities. The cause is not clearly understood. We report a case of a severe form of a rapidly progressive complex regional pain syndrome type I developing after a right shoulder injury managed with spinal cord stimulation (SCS). After failed conservative treatments, a rechargeable SCS system was implanted in the cervical spine. Allodynia and dystonia improved but the patient subsequently developed similar symptoms in lower right extremity followed by her lower left extremity. The patient became wheelchair bound. A second rechargeable SCS with a paddle electrode was implanted for the lower extremity coverage. The patient's allodynia and skin lesions improved significantly. However, over time, her initial symptoms reappeared which included skin breakdown. Due to the need for frequent recharging, the system was removed. During explantation of the surgical paddle lead, it was noted by the neurosurgeon that the contacts of the paddle lead were detached from the lead. After successful implantation of another SCS system, the patient was able to reduce her medications and is now able to ambulate with the use of a left elbow crutch.

Postdural Puncture Headache in Complex Regional Pain Syndrome: A Retrospective Observational Study

Pain Medicine

Early View (Articles online in advance of print)

Published Online: 26 Oct 2009

© 2009 American Academy of Pain Medicine

Alexander G. Munts, MD,* Joan H. C. Voormolen, MD, ^† Johan Marinus, PhD,* Elmar M. Delhaas, MD,* and Jacobus J. van Hilten, MD, PhD*

Departments of *Neurology and ^† Neurosurgery, Leiden University Medical Center, Leiden, The Netherlands

Correspondence to Jacobus J. van Hilten, MD, PhD, Department of Neurology, Leiden University Medical Center, PO Box 9600, 2300 RC Leiden, The Netherlands. Tel: 31-71-5262134; Fax: 31-71-5248253; E-mail: j.j.van_hilten@lumc.nl.

ABSTRACT

Objective. To describe the unusual course of postdural puncture headache (PDPH) after pump implantation for intrathecal baclofen (ITB) administration in patients with complex regional pain syndrome (CRPS)-related dystonia.

Design. Case series based on data collected from 1996 to 2005.

Setting. Movement disorders clinic, university hospital.

Patients. A total of 54 patients with CRPS-related dystonia who were treated with ITB.

Results. A high incidence (76%) and prolonged course (median 18 days, range 2 days to 36 months) of PDPH was found. Radionuclide studies performed in two patients with long-lasting symptoms (12–16 months) did not reveal cerebrospinal fluid (CSF) leakage. In patients without signs of CSF leakage (N = 38), epidural blood patches administered in 24 patients were effective in 54%, while ketamine infusions administered in six patients were effective in 67%.

Conclusions. Our observations may suggest that other mechanisms besides intracranial hypotension play a role in the initiation and maintenance of PDPH in CRPS and stimulate new directions of research on this topic.

Reduction of allodynia in patients with complex regional pain syndrome: A double-blind placebo-controlled trial of topical ketamine

Reduction of allodynia in patients with complex regional pain syndrome: A double-blind placebo-controlled trial of topical ketamine
(http://www.painjournalonline.com/article/PIIS0304395909003042/abstract?)
Philip M. Finchab, Lone Knudsenb, Peter D. Drummondb
Received 4 February 2009; received in revised form 22 April 2009; accepted 19 May 2009. published online 25 August 2009. Corrected Proof
Abstract
A double-blind placebo-controlled crossover trial was used to determine the effects of topical ketamine, an N-methyl-d-aspartate (NMDA) receptor antagonist, on the sensory disturbances in 20 patients with complex regional pain syndrome (CRPS). On two occasions separated by at least one week, sensory tests to light touch, pressure, punctate stimulation, light brushing and thermal stimuli were performed in the symptomatic and contralateral limb and on each side of the forehead before and 30min after 10% ketamine cream was applied to the symptomatic or healthy limb. Venous blood for the plasma estimations of ketamine and norketamine was obtained 1h after application of the creams. Ketamine applied to the symptomatic limb inhibited allodynia to light brushing and hyperalgesia to punctate stimulation. Systemic effects of the ketamine are unlikely to account for this as the plasma levels were below detectable limits. As touch thresholds were unchanged, NMDA receptors may contribute to the sensory disturbances in CRPS via actions at cutaneous nociceptors. Allodynia and hyperalgesia were detected in the ipsilateral forehead to a range of stimuli (brushing, pressure, punctate stimulation, cold, heat, and warmth). In several patients, ketamine treatment of the symptomatic limb inhibited allodynia to brushing the ipsilateral forehead, suggesting that the mechanism that mediates allodynia in the symptomatic limb contributed to allodynia at more remote sites. The present study shows promise for the use of topical ketamine as opposed to parenteral and oral forms which often result in undesirable side effects.