KEY WORDS
Eczematous dermatitis - replantation - revascularisation - skin lesions - sympathetic
dysfunction
INTRODUCTION
Frequently reported complications after replantation and revascularisation of upper
extremity are vascular thrombosis leading to complete or partial failure, non-union
or malunion of bones, joint stiffness and incomplete or abnormal neurological recovery
including paraesthesia, dysaesthesia and hypoesthesia.[[1]]
Although abnormal skin eruptions and inflammatory skin lesions have been previously
mentioned in patients with limb trauma and complex regional pain syndrome (CRPS),[[2]
[3]
[4]
[5]
[6]] the development of skin lesions and abnormal sudomotor function following replantation
or revascularisation of the limb in such injuries has not been reported. We present
the case of a young adult who developed eczematous dermatitis over the revascularised
limb and discuss the role of sympathetic dysfunction in the development of skin lesions.
CASE REPORT
A 24-year-old right-handed male reported to our emergency department with the history
of trauma to his left forearm and hand while working on a wood-cutting machine. There
was no associated injuries or comorbidities. There was near-total amputation of the
left upper limb at two levels: at the junction of middle and lower one-third of forearm
and at the level of metacarpal shafts. All the forearm structures were transected
except preserved skin tag of 2 cm width on the ulnar side of the forearm [[Figure 1]]. The left thumb was also amputated at the level of mid distal phalynx. After the
bony fixation, revascularisation of the limb was undertaken by repair of ulnar and
radial arteries. Venous flow was established by repair of cephalic and basilic veins.
Perineural repair of the ulnar nerve, the median nerve, superficial branch of radial
nerve. and the lateral cutaneous nerve of forearm was carried out in an end-to-end
fashion. Similarly, the first, second and third common digital arteries and nerves
were repaired after the fixation of metacarpals. Thumb stump was closed primarily. Injury to reperfusion time was 7 h and total operating time was 13 h.
Figure 1: Pre-operative photograph and plain radiographs of the patient showing near-total
amputation of left forearm and hand
In the post-operative period, the patient developed vascular insufficiency of the
distal part of the hand which could not be salvaged. We performed trans-metacarpal
amputation of the left hand and covered the stump with a pedicled groin flap. Small
raw area on the dorsum of the forearm was skin grafted. Complete healing of the wounds
occurred after 1 month, at which time the patient was discharged from the hospital
[[Figure 2]]. The patient was referred to the physiotherapy unit for rehabilitation and was followed
up in our outpatient department monthly for sensory and motor evaluation.
Figure 2: Post-operative photographs showing complete healing after 4 weeks. Healed skin graft
on the dorsum of forearm and healed groin flap at the trans-metacarpal amputation
stump are seen
Four months after the discharge, the patient reported with acute-onset skin lesions
over the revascularised part of the forearm and hand. The physical examination revealed
weeping and crusting exudative lesions suggestive of of eczematous dermatitis. The
revascularised part of an extremity developed irregular erythematous plaques covered
with yellowish thick crust. There were multiple erosions at places along with serous
discharge. These lesions were strictly limited to the revascularised part of the limb;
rest of the limb was normal [[Figure 3]]. The patient did not have any allergy and other reactive skin lesions in the past.
Negative Patch test with standard series ruled out contact dermatitis. Skin biopsy
revealed the findings of acute eczematous dermatitis.
Figure 3: Photograph showing erythematous, exudating and crusting lesions of eczematous dermatitis
3 months postoperatively
Several studies in the past have reported the association between autonomic dysfunction
and skin lesions like atopic dermatitis, psoriasis and CRPS.[[2]
[3]
[4]] As the patient had suffered traumatic neuropathy, investigations were done to rule
out autonomic dysfunction in the revascularised part. We performed Ninhydrin sweat
test and Sympathetic Skin Response (SSR) test. Negative Ninhydrin sweat test indicated
disturbance of sweating function.
SSR test was performed using a four-channel electromyography machine as described
by Cicek et al.[[2]] The affected hand did not show any waveform indicating sudomotor dysfunction.
Sympathetic dysfunction in the revascularised part of the limb was confirmed by above
investigations.
Sympathetic dysfunction led to abnormal sudomotor and vasomotor functions and eczematous
dermatitis in the revascularised part of the limb. Therefore, we coined the term sympathetic dysfunction dermatitis to describe the skin lesions developing in the revascularised or replanted part of
an extremity.
The patient responded well to a topical cream of 2% fusidic acid and 0.1% betamethasone
along with oral prednisolone for 1 week. All the skin lesions subsided with appearance
of a normal skin texture [[Figure 4]]. However, there was one episode of recurrence 3 weeks after the resolution of the
skin lesions which was treated as before. There was no further episode of dermatitis.
The patient is presently on emollients to prevent recurrence.
Figure 4: All the lesions had completely healed after a short course of oral and topical steroids
To evaluate the sensory recovery, Semmes–Weinstein monofilament test and 2-point discrimination
test (2PD) were performed at the 4th, 5th, 6th and 8th months postoperatively. On evaluation at the 6th month, the patient was free of skin lesions and his sensory recovery improved.
DISCUSSION
Dermatitis at a surgical site was first reported by Carr and Rau in 1981[[5]] and later on by Bart in 1983[[6]] following vein graft harvest for cardiac surgery. The aetiology of this remained
largely unknown until the mid-nineties when injury to the neural plexuses and localised
venous congestion were implicated for dermatitis in these patients.[[4]]
A Japanese study cited saphenous neuropathy as the cause for occurrence of skin lesions.
Injury to the nerve plexus leads to sympathetic dystrophy leading to skin atrophy
and a state of hyper- or hypo-hidrosis.[[7]] Local venous congestion after trauma can also lead to oedema of the skin, a pigmented
purpura-like inflammatory dermatitis, and a non-immune bullous eruption.[[7]
[8]]
The association between autonomic dysfunction and development of skin lesions was
emphasised by some studies. These studies reported the presence of sympathetic dysfunction
in patients with atopic dermatitis, psoriasis and CRPS.[[2]
[3]
[4]] The unmyelinated C fibres are responsible for sudomotor activity which cause activation
of sweat glands. Involvement of these fibres leads to dryness and itchiness of the
skin, making it susceptible for the development of dermatitis.[[2]] Several interacting pathways are involved in causing skin inflammation, with autonomic
and sensory nerves modulating the response.[[9]] Loss of modulatory effect of autonomic and sensory nerves after amputation may
lead to skin inflammation in such patients.
More evidence into the relationship between sudomotor and sympathetic dysfunction
and development of inflammatory skin lesions can be found in patients with hand surgery
who develop CRPS.[[10]] CRPS is characterised by abnormal pain and sudomotor, vasomotor and trophic changes.
After traumatic denervation, there is distal degeneration of small-diameter axons
subserving nociception and sympathetic function.[[11]] There are reports of vascular and trophic changes along with increased interleukin-6
(IL-6), tumor necrosis factor-α and mast cell marker tryptase after trauma to the
limb.[[12]
[13]]
It can be safely assumed that similar derangements take place in patients with traumatic
amputations undergoing replantation or revascularisation. Along with sensory and motor
deficiencies, denervation of the distal part of the limb leads to trophic, vasomotor
and sudomotor changes which lead to the increased likelihood of developing dermatitis
in the amputated part.
Hruza and Hruza[[14]] found that this type of dermatitis typically occurs between 1 and 9 months and
is associated with traumatic neurosensory deficits. Dermatitis improves with the improvement
in sensory deficit.[[13]] This finding was observed in our patient where dermatitis was active between the
4th and 5th post-operative months and thereafter it subsided with the improved sensory recovery
of the affected limb. There may be variable clinical presentations. Cutaneous manifestations
can be seen in the form of cellulitis, dermatitis or xerosis.[[8]]
Zakrzewska-Pniewska and Jedras observed decreased sweating, hypohidrosis and skin
xerosis as markers of autonomic dysfunction and that autonomic dysfunction can be
successfully diagnosed with SSR test.[[15]] SSR test can be effectively used to evaluate the presence or return of autonomic
function in patients with replantation or revascularisation. It can be a useful addendum
to nerve conduction study and electromyography in monitoring return of neurological
function.
Dermatitis due to sympathetic dysfunction after trauma responds well to treatment
with short course of oral and topical steroids. However, there is a chance of recurrence
of dermatitis until adequate sensory recovery has occurred.[[7]
[8]]
After replantation or revascularisation of the extremity, the primary focus remains
on the survival of the limb and its functional rehabilitation. Skin changes might
receive less attention and subsequently remain unreported. This report of sympathetic
dysfunction dermatitis as a new complication after revascularisation or replantation
of limb which should be kept in mind. Further studies are required to assess the prevalence
and incidence of this entity.
CONCLUSION
Sympathetic dysfunction dermatitis is a rare form of dermatitis seen in patients with
traumatic denervation of the limb. It should be regarded as a potential complication
of denervation of the extremity, which is expected to subside after reinnervation.
Appropriate measures to keep the distal replanted part well hydrated may prevent this
complication. Sympathetic dysfunction dermatitis responds well to oral and topical
steroids.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms.
In the form, the patient has given his consent for his images and other clinical information
to be reported in the journal. The patient understands that name and initial will
not be published and due efforts will be made to conceal identity, but anonymity cannot
be guaranteed.
Financial support and sponsorship
None.
