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Letter to the Editor
Vasculitic mononeuritis multiplex associated with livedoid vasculopathy
Mononeuritis múltiple vasculítica asociada a vasculopatía livedoide
Í. Corrala,
Corresponding author
inigo.corral@salud.madrid.org

Corresponding author.
, M.M. Kawiorskib, C. Morenob, H. Pianb
a Servicio de Neurología, Hospital Universitario Ramón y Cajal, Madrid, Spain
b Servicio de Anatomía Patológica, Hospital UniversitarioRamón yCajal, Madrid, Spain
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progression of pain and progressive loss of strength and sensitivity in both feet&#44; associated with erythematous lesions on the tops of the feet&#46; He also reported loss of sensitivity in the fourth and fifth fingers of the right hand&#46; He presented no constitutional symptoms and had no history of systemic disease&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The examination detected confluent erythematous macules in both feet&#44; compatible with livedo racemosa&#44; and no ulcers or atrophie blanche&#46; Muscle strength was 3&#47;5 in plantar flexion and 2&#47;5 in dorsiflexion&#46; Patellar and Achilles reflexes were absent in the left limb and hypoactive in the right&#46; The patient also displayed reduced tactile and pain sensitivity below the knees and in the fourth and fifth fingers of the right hand&#46; The remaining results of the neurological examination were normal&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Laboratory analyses yielded normal results&#44; including normal levels of homocysteine&#44; cryoglobulins&#44; lupus anticoagulant&#44; and rheumatoid factor&#46; Tests for antinuclear&#44; antineutrophil cytoplasmic&#44; anti-&#174;2 microglobulin&#44; anticardiolipin&#44; antiganglioside&#44; and antineuronal antibodies returned negative results&#59; results from serology tests for syphilis&#44; hepatitis B and C viruses&#44; and HIV were also negative&#46; CSF analysis yielded normal results&#44; with no oligoclonal bands&#46; Screening tests for hereditary thrombophilia and occult cancer yielded negative results&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">A neurophysiological study detected increased distal latencies and low-amplitude motor evoked potentials in the peroneal and tibial nerves&#46; Somatosensory evoked potentials were not detected in the sural nerves&#46; Low-amplitude motor evoked potentials were detected in the right ulnar nerve&#46; Electrical activity was normal in both median nerves and the left ulnar nerve&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">A skin biopsy revealed occlusive vasculopathy in the superficial and deep dermal arteries&#44; with areas of mural hyalinisation&#44; no inflammatory infiltrate&#44; and preserved epidermis&#59; these findings are consistent with LV &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; A sural nerve biopsy revealed intense loss of myelinated fibres&#46; An epineurial artery showed CD45&#43; inflammatory infiltrate&#44; with arterial wall infiltration and destruction &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; No thrombosis was observed in other arteries&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">The patient was treated with acetylsalicylic acid at 100&#8239;mg&#47;day&#44; prednisone at 1&#8239;mg&#47;kg&#47;day&#44; and amitriptyline and tramadol as rescue medications&#59; treatment relieved pain and strength and sensitivity progressively improved&#46; Skin lesions improved progressively&#44; leaving no atrophie blanche&#46; A year after the first consultation&#44; the patient presented deep vein thrombosis in the right leg&#44; which was attributed to immobilisation and treated with acenocoumarol for 6 months&#46; Again&#44; tests for thrombophilia and occult cancer yielded negative results&#46; The dose of prednisone was gradually reduced&#44; and the drug was suspended at 19 months&#46; The patient remains asymptomatic after 6 years of follow-up&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">LV was diagnosed based on the characteristic skin biopsy findings&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The condition is not always associated with atrophie blanche&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> The pathogenesis of LV is unclear&#46; In many cases&#44; the condition is associated with acquired or hereditary thrombophilia&#44; neoplasia&#44; or systemic diseases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> However&#44; it may also appear in isolation&#44; as in our patient&#59; in this case&#44; coagulopathy and systemic disease could not be confirmed&#44; despite extensive testing and a long follow-up period&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The literature includes cases of peripheral neuropathy as an extracutaneous manifestation of LV&#44; usually with a clinical and electrophysiological pattern of mononeuritis multiplex&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;9</span></a> In a retrospective series of 70 patients with LV&#44; 9&#37; presented peripheral neuropathy&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> In some patients&#44; biopsy studies reveal ischaemic neuropathy secondary to thrombosis of the vasa nervorum&#44; with no signs of vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;6&#44;7&#44;9</span></a> To our knowledge&#44; this is the first reported case of vasculitic MM associated with LV&#46; We should consider this diagnostic possibility and perform a nerve biopsy in these patients&#44; as immunosuppressive treatment may resolve the symptoms&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Our patient met the diagnostic criteria for non-systemic vasculitic neuropathy &#40;NSVN&#41;&#44; as the nerve biopsy revealed inflammatory infiltrate in the vessel wall and signs of vascular damage&#44; with no evidence of systemic vasculitis&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Copresence of NSVN and LV&#44; and the simultaneous improvement of both conditions after initiation of corticosteroid treatment&#44; suggest a pathogenic association between the 2&#44; although the nature of this association is unclear&#46; LV may be associated with other inflammatory diseases&#44; such as connective tissue disorders&#44; and may therefore be associated with NSVN&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0055" class="elsevierStylePara elsevierViewall">This study has received no specific funding from any public&#44; commercial&#44; or non-profit organisation&#46;</p></span></span>"
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