Case
Report
Reflex Sympathetic Dystrophy Syndrome
Complicating the Management of TMJ Symptoms. A Case Report
M. Jeffrey Morton,
D.M.D., Mark L. Pitel, D.M.D.
Clinical Report
A case report and a review of a rare
facial pain syndrome is presented. Reflex sympathetic dystrophy syndrome (RSDS) is a
condition that is commonly seen and treated by other health practitioners, but which is
rarely seen by dental practitioners. Because many of the signs and symptoms of RSDS are
similar to TMJ and other facial pain syndromes, it should bean important consideration in
the differential diagnosis and selection of proper treatment.
More widely known as "causalgia,'' RSDS is a complicated. poorly understood condition
that has been known for over 100 years. The goals of this paper are to (1) familiarize the
dental profession with the symptoms and the currently accepted terminology to describe the
condition, (2) present the current theory of the pathogenesis. and (3) describe a case
where post-traumatic TMJ symptoms and their treatment were complicated by an associated
case of RSDS involving the face. It is important to state at the outset that the reporting
of a case Where the patient responded poorly to therapy is highly significant because it
points out the need for the differentiation of RSDS from other conditions more readily
managed by the dentist.
Definition of the Condition
Reflex sympathetic dystrophy syndrome is an extremely interesting condition that is well
known to the medical and podiatric professions, but has only rarely been reported in the
dental literature It is a poorly understood neurogenic pain syndrome that appears to
involve a disturbance of the reflex pathway of the sympathetic nervous system following
some local in-jury-. The syndrome is characterized by persistent pain, discoloration.
increased perspiration and elevated cutaneous temperature of the affected part. In severe
cases there may be edema. deossification of the underlying bone (osteoporosis), and
secondary trophic changes such as atrophy of skin and muscle.
and grooving and fissuring of nails. The pain of RSDS usually spreads far beyond the area
of the injury. persists long after the area heals, and is not confined to the distribution
of a particular sensory nerve. This has been interpreted as involving a change in the
internuncial neuron pool.
The uncertain pathogenesis of RSDS, as well as the variability of the symptoms has led
to a large number of descriptions and terms that all refer to the same syndrome. The
syndrome was first described by Mitchell et al,
1 in 1864 who called the
syndrome causalgia. which means ''burning pain.'' Dentists are probably most familiar with
this term. It has also been widely known as Sudeck's Atrophy: since about 1900. The
currently accepted term Reflex sympathetic dystrophy originated with Evans
3
in 1947, causalgia, and Sudeck's atrophy are the three most commonly used descriptors.
Causalgia has been frequently described in the dental literature however, the reports
of RSDS specifically affecting the trigeminal system are very limited. The majority of
case reports center around ''causalgic-like'' symptoms following tooth extraction, pulp
extirpation. or toothache of nondental origin. Patients describe the pain as persistent.
unremitting and burning in quality. giving it a neuritic quality uncharacteristic of true
odontogenic pain. Because the pain may often involve several teeth. attempts at treatment
frequently lead to multiple dental extractions or endodontic interventions all to no
avail. The RSDS may follow trauma, minor or major oral surgery. or infection so the exact
dental relationship may be obscure to both dentist and patient. It is likely that some
cases diagnosed as craniomandibular dysfunction. TMJ or myofacial pain dysfunction
syndromes, which were refractory to conventional therapy, were in fact RSDS or
combinations of diseases.
Pathogenesis
RSDS has been found to be caused most commonly by trauma, infections, and neoplasms.4
Though the actual mechanism of RSDS is still poorly understood. the best current theory is
as follows:
1. Peripheral tissue injury causes damage to Sensory neurons. As a result of nerve
damage. a barrage of afferent impulses is generated by the dorsal ganglion and propagated
both orthodromically and antidromically.5
2. Damage to the sensory neuron disrupts the myelin sheath and induces neural sprout
formation. The neural sprouts become sensitized to norepinephrine creating an artificial
synapse or 'short circuit'' to nearby efferent sympathetic fibers. Autonomic impulses
being conducted along vasomotor, pilomotor. and sudomotor pathways cross over to the
damaged afferent nerve and induce a nociceptive response.6,7
This would explain why causalgic pain is effected by temperature changes and emotional
stimulation. It also explains why the most successful therapies to date have been stellate
ganglion blockade adenergic blockade, guanethedine blockade. sympathetic blocks and
sympathectomy.
The increased flow of afferent impulses orthodromically induces an abnormal state of
activity in the internuncial neuron center of the spinal cord, This has been confirmed by
the work of Erlanger and Gasser.8 Augmentation of afferent impulses by the
internuncial pool explains why the pain of RSDS is rarely confined to the distribution of
a particular sensory nerve.
Case Discussion
The patient. a 20-year-old white female. was referred for TMJ evaluation and treatment
approximately one month following trauma received in an automobile accident. During the
accident. the patient was thrown forward and apparently struck the right side of her face
on the front windshield. The chief complaint was severe right-sided facial pain,
limitation of opening, jaw sounds. moderate trismus. and pain during mastication. The pain
was described as continuous and throbbing. It was reported to have begun almost
immediately following the trauma, was always localized to the right facial region, and was
getting worse. In the morning her face felt tight. She noted having her teeth often
clenched. She related having the sensation of her face feeling swollen and looking swollen
on the right side. The patient also reported unusual cutaneous thermal sensations ranging
from cold to hot.
A review of the patient's medical history revealed no significant systemic diseases or
allergies. She was hospitalized three limes during the year for surgery, including
tonsillectomy, cholecystectomy. and a sympathectomy for persistent left-sided rib pain. In
the case of the latter surgery, a diagnosis of reflex sympathetic dystrophy had been made
and the sympathectomy apparently proved to be successful in eliminating the rib pain. This
pain was the result of injury from a previous car accident. The patient was taking the
following medications: Tolectin, Flexoril. and Talwin.
Clinical examination revealed a well-developed 20-year-old female in acute distress
from facial pain. She had a distinctive asymmetry to the face. The right side appeared
slightly swollen from the zygoma to the inferior border of the mandible. The area had a
blotchy mottling with irregular zones of pink. white. and red skin coloration. The parotid
gland was palpated and felt larger than its contralateral counterpart. The patient had a
maximum interincisal distance of 15 mm when opening her mouth. Palpation of the
muscles of mastication. facial, and neck muscles. TMJ, and skin on the right side of the
face produced immediate severe pain to the patient. Early clicking of the right TMJ was
detected during mandibular depression. Translation of the right condyle was diminished
when compared to the left during mandibular opening.
Radiographic studies demonstrated no fractures. dislocations, or other significant
findings.
Dental examination showed generalized marginal and papillary- gingivitis. Considerable
plaque, calculus, and materia alba were present on visible tooth surfaces. Numerous
carious teeth were noted. A parafunctional habit of clenching was observed.
Our differential diagnosis included post-traumatic arthropathy of the right TMJ. bur
clearly this would not account for all of the unusual symptoms seen in this patient. RSDS
was also suspected because of her previous history of this condition. Confirmation of the
diagnosis of RSDS in the face has been documented by successful relief of pain following
sympathectomy or sympathetic blockade (usually of the stellate ganglion) )9,12
A referral was made to a neurologist who confirmed the RSDS problem in our patient by
relieving the pain with sympathectomy. Since the pain relief proved only transient. we
began conventional supportive TMJ treatment in hopes that minimal disarticulation of the
dentition would provide some bruxism relief and patient comfort while the neurologists'
comprehensive therapy would be successful in man-aging the patient's pain.
Treatment
A mandibual acrylic bruxism splint was initially used to disarticulate the
tooth-to-tooth contracts and to control the parafunctional clenching habit. Over a
five-week period. the mandibular position was gradually shifted from habitual centric
position to a protrusive relation. This did apparently reduce some of the TMJ component of
the pain, but the overall relief was minimal.
We next initiated a series or needling visits both for localization of any pain trigger
zones and for pain control. Initially we used sterile saline. and 3% Mepivicaine (plain)
to first localize trigger point locations. Some success was achieved in mapping three
locations as shown in Figure 1. After providing some pain relief to the
patient. the decision was made to try to give some hours of pain relief using the
nonvasoconstriction anesthetic and later 2% Xylocaine with 1:100,000 epinephrine and
Marcaine with the hope of prolonging the pain relief. It was discussed with the other
therapists and the patient. properly noted and of some concern that the later solutions
had greater myotoxicity. Hot compresses to increase blood flow, vapocoolant cryotherapy,
and conventional TENS treatments were all used with only marginal effect.
Restoration of the patient's carious teeth and periodontal management were completed in a
single visit under general anesthesia. This had no effect on the pain. We also tried a
variant modality of TENS called the ''pain suppressor.''13 which is reported to
be somewhat different from the conventional TENS units.
Finally. having reduced the bruxism component of the pain and achieved all that we could
within the scope of dental practice. the patient was referred back to the neurologist for
a more definitive management of the RSDS problem. Future follow-up indicated poor pain
control was achieved through neurosurgical attempts and medical treatment was going toward
a more chronic pain management direction. Discussions of medication use for pain
management was being discussed at the time of submission of this paper.
Conclusion
A case is presented where an unusual pain syndrome reflex sympathetic dystrophy
complicated the diagnosis and management of post-traumatic TMJ symptoms. Diagnosis was
based on the history, clinical examination. signs. and symptoms. Confirmation of RSDS was
made by relief of pain following sympathectomy. Numerous other therapies were attempted to
comprehensively manage the patient's pain. These included bruxism splint. needling of pain
trigger zones, moist heat, vapocoolant cryotherapy. TENS. ''pain suppressor-TENS''
therapy, and restoration of the carious teeth. None could be considered completely
successful in eliminating or controlling significant aspects of the pain. The needling
visits were useful in demonstrating and outlining specific trigger zones for the pain.
These are outlined in Figure 1. Initially, the patient reported high
levels of pain relief from the local anesthetic injections for periods of up to three
hours; however, she quickly became refractory to these. Some pain relief has come from use
of the ''pain suppressor.'' As has been pointed out. the relief of pain by sympathetic
blockade or sympathectomy is diagnostic for RSDS. In many cases these are also therapeutic
as they were when this patient suffered from RSDS of the back. In this case, however. they
produced only transient relief of the facial pain. This may be due to the greater
abundance of nerve fibers found in the face and head than other locations often reported
for RSDS such as arms and legs. In any event, this case identifies a rare condition that
dentists should be aware of and consider in the differential diagnosis of pain syndromes
that affect the face and oral cavity.
Further information about RSDS can be obtained by contacting the RSDS Association. P.O.
Box 821. Haddonfield. New Jersey 08033.
Reprint request to:
M. Jeffrey Morton. D.M.D.
520 Stokes Rd. Rt. 541
Medford. New Jersey 08055
References
1. Mitchell SW, Moorehouse GR , Keen WW, Gunshot Wounds and Other Injuries of Nerves.
Philadelphia. JB Lippincott,Co 1864.
2. Sudeck P: Uber die akute entzundlicke knockentrophie. Arch F Klin Chin 1900 62: 147
3. Evans JA, Reflex sympathetic dystrophy. Report on 57 cases Ann Intern Med 1947; 26:
417
4. DeLorimer AA. Minear WL. Boyd HR: Reflex Lyperemic deossification regional to joints
of the extremities. Radiology 1946 40: 227
5. Watt PD. Devor NI. Sensory afferent impulse. originate from dorsal root ganglia as
well as from the periphery in normal and nerve injured rats. Pain 1983 17:321-339
6. Bell WE: Orofacial Pain;. Classification Diagnosis. Management. P 303. Third
Edition. Chicago. Yearbook Medical Publishers. 1985
7. Melament lB. Click JR: Sudecks Atrophy. the Clinical Syndrome. J. Am Pad Assoc 1983
73 (7): 362-367
8. Erlanger I, Gasser HS: Electrical signs of nervous activity. Philadelphia University
of Pennsylvania Press. 1937
9. Jaeger B. Singer F. Kroening R: Reflex sympathetic dystrophy or the face. Report of
two cases and a review literature. Arch Neurol l986 43 (17): 693-695
10. Khoury R. Kennedy SF. MacNamara TE. Facial causalgia. Report of cases. J Oral Surg
1980 38:782-783
11. Hanowell ST. Kennedy SF: Phantom tongue pain and causalgia: Case presentation and
treatment. Anesth Analg 1979 58: 436-438
12. Bingham JAW: Causalgia of the face: Two cases successfully treated by
sympathectomy. Br Med J 1947 1:
804-805
13. Pain Suppression Labs. Inc. 550 River Dr. Elmwood Park. New Jersey 07407-0441
Mark L. Pitel, D.M.D.
Dr. Mark L. Pitel graduated from the Temple University School of Dentistry in 1982. He is
currently in private general practice in Swedesboro. New Jersey. Dr. Pitel serves as vice
president of the New Jersey Academy of General Dentistry and chairman of the Speaker's
Bureau of the Southern Dental Society. He is a member of the American Dental Association
and Academy of General Dentistry-
Jeffrey Norton, D.M.D.
Dr. Jeffrey Morton received his D.M.D. from the University of Pennsylvania School of
Dentistry. He is presently in private practice in Medford. New Jersey. Dr. Morton's
professional memberships include the American Association of Periodontology, the
Philadelphia Society of Periodontology, the Southern Dental Association, and the American
Dental Association.
July 1989. Vol. 7. NO. 3: 239-242 THE JOURNAL OF CRANIOMANDIBULAR PRACTICE
