Livedoid vasculopathy associated with peripheral neuropathy: a report of two cases

Tubone, Mariana Quirino; Escobar, Gabriela Fortes; Peruzzo, Juliano; Schestatsky, Pedro; Maldonado, Gabriela

doi:10.1590/abd1806-4841.20132363

Cited by 8 publications

(13 citation statements)

References 9 publications

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“…Livedoid vasculopathy may or may not be associated with underlying thrombotic risk factors, such as a paraproteinemia or antiphospholipid syndrome, or a systemic rheumatologic illness, such as systemic lupus erythematosus among others 10 . In rare cases, livedoid vasculopathy has been known to cause peripheral neuropathy, as was the case in our patient 11 …”

Section: Discussionsupporting

confidence: 71%

Cutaneous involvement by T‐cell prolymphocytic leukemia presenting as livedoid vasculopathy

et al. 2021

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T‐cell prolymphocytic leukemia (T‐PLL) is a rare, aggressive neoplasm derived from post‐thymic T‐cells. Patients are typically middle aged with a slight male predominance who present with a high white blood cell count, hepatosplenomegaly, lymphadenopathy, and other symptoms typically associated with leukemia. Although cutaneous involvement has been reported in up to 30% of cases of T‐PLL, to our knowledge, none have presented with a presentation resembling livedoid vasculopathy. In the correct clinical context, an underlying hematolymphoid neoplasm should be included in the differential diagnosis of a patient presenting with livedoid vasculopathy.

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Section: Discussionsupporting

confidence: 71%

Cutaneous involvement by T‐cell prolymphocytic leukemia presenting as livedoid vasculopathy

et al. 2021

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“…www.medicaljournals.se/acta (29). Peripheral neuropathy has been rarely described with a total of 9 detailed cases in the literature: one sensory ganglionopathy, one axonal neuropathy and 7 cases of mononeuritis multiplex (8,(10)(11)(12)(13)(14)(15). Among them, 5 nerve biopsies revealed the same images of axonal loss with a lymphocytic infiltrate, without signs of vasculitis.…”

Section: Discussionmentioning

confidence: 99%

“…Recently, a few cases have reported peripheral neuropathy in association with LV, most often mononeuritis multiplex (8)(9)(10)(11)(12)(13)(14)(15).…”

mentioning

confidence: 99%

Livedoid Vasculopathy: A French Observational Study Including Therapeutic Options

Gardette¹,

Moguelet²,

Bouaziz³

et al. 2018

Acta Derm Venerol

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Livedoid vasculopathy is a rare thrombotic cutaneous disease. This observational study aimed to assess the clinical and biological features of livedoid vasculopathy and the efficacy of treatments. Patients enrolled had typical livedoid vasculopathy both clinically and histologically. Investigation of thrombophilia was performed. Electromyography was undertaken in the presence of symptoms suggesting peripheral neuropathy. Eighteen women and 8 men were included, with a mean age of 35.5 years at onset. Twenty patients had at least one thrombophilia factor. Ten patients had a peripheral neuropathy with 2 of these patients demonstrating a specific thrombo-occlusive vasculopathy on muscle biopsy. Anticoagulation with low molecular weight heparin was the most prescribed therapy and was associated with the best outcome (effective in 14 patients). Eight patients had severe disease refractory to anticoagulation and required intravenous immunoglobulins, producing a good response in 6 patients.

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“…There have been 6 previously reported LV cases with multiple mononeuropathy in which nerve biopsies were performed (Table 4) [2][3][4][6][7][8]. Nerve biopsies in 3 cases (Pai and Pai [6], Toth et al [7], and Tubone et al [8]) showed chronic histologic findings; a nerve biopsy performed by Malaguti et al [4] showed pleomorphic mononuclear cell infiltration in the endoneurium, perineurium, and epineurium. Four histologic findings from nerve biopsies could not distinguish whether multiple mononeuropathy in LV was derived from inflammation or infarction.…”

Section: B Amentioning

confidence: 98%

Multiple Mononeuropathy Associated with Livedoid Vasculopathy: A Case Report and Review

Kang¹,

Lee²,

Kim³

et al. 2021

J Electrodiagn Neuromuscul Dis

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Livedoid vasculopathy (LV) is a chronic, painful, thrombo-occlusive cutaneous vasculopathy. It affects the lower extremities, and clinical symptoms are primarily confined to the skin [1]. In rare cases, there is involvement of the peripheral nervous system, which the most common form is multiple mononeuropathy [2].The exact pathophysiologic mechanism of the nerve involvement in LV and treatment for neurologic deficits remain unclear.This report describes a rare case of livedoid vasculopathy (LV) with sensory and motor neurologic deficits. A 46-year-old woman who had been diagnosed with LV developed acute left-foot drop. An electrodiagnostic study showed multiple mononeuropathy involving both the common peroneal and sural nerves, primarily on the left side. Nerve biopsy findings showed intravascular thrombosis in the perineurium and extensive infarct of the peripheral nerve. The patient was prescribed continuous anticoagulants, which resulted in a good clinical outcome. LV associated with multiple mononeuropathy is a rare condition, and the exact pathophysiologic mechanism of nerve involvement and treatment for neurologic deficits remain unclear. The histologic findings in the present case support the possibility that the peripheral neuropathy related to LV was primarily derived from an ischemic injury rather than inflammation. Continuous anticoagulant use could be helpful in the neurologic recovery of LV patients with multiple mononeuropathy.

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Livedoid vasculopathy associated with peripheral neuropathy: a report of two cases

Cited by 8 publications

References 9 publications

Cutaneous involvement by T‐cell prolymphocytic leukemia presenting as livedoid vasculopathy

Cutaneous involvement by T‐cell prolymphocytic leukemia presenting as livedoid vasculopathy

Livedoid Vasculopathy: A French Observational Study Including Therapeutic Options

Multiple Mononeuropathy Associated with Livedoid Vasculopathy: A Case Report and Review

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