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      Penile Livedoid Vasculopathy: First Reported Case


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          Livedoid vasculopathy is a thromboocclusive cutaneous vasculopathy manifested by livedoid changes, atrophie blanche, and ulceration. The pathogenesis is speculated to involve increasing coagulation or impaired thrombolysis leading to the occlusion of dermal blood vessels with fibrin thrombi. Livedoid vasculopathy is known to primarily affect the lower extremities. We report the first case of livedoid vasculopathy affecting the penis. A 60-year-old male was evaluated for a split urine stream with associated irritation and peeling of the skin of the glans penis. His penile ulcer continued to enlarge despite steroids and antibiotics. Due to diagnostic uncertainty, a biopsy was performed which revealed hyaline thrombi within the lumens of small vessels within the upper to mid dermis, fibrinoid material in the walls of these blood vessels and within the perivascular stroma with overlying and adjacent epidermal spongiosis, and mild perivascular lymphocytic infiltrate with a few scattered neutrophils most consistent with livedoid vasculitis. He was started on aspirin and pentoxifylline with limited improvement and was later started on apixaban with near-complete resolution in 6 months. Penile livedoid vasculopathy has not been previously reported in the English literature. Early diagnosis and treatment are imperative to limit morbidity.

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          Livedoid vasculopathy: further evidence for procoagulant pathogenesis.

          To further characterize the clinical and pathologic features, disease associations, and laboratory abnormalities of livedoid vasculopathy. Retrospective study of patients identified from our institutional database from January 1, 1990, to December 31, 2000. Tertiary care institution. Patients Forty-five patients with biopsy-proved livedoid vasculopathy. Clinical presentation, histopathologic diagnosis, results of testing for coagulation abnormalities, and assessment of vascular status. Thirty-two patients (71.1%) were female (mean age, 45 years; age range, 10-85 years). Bilateral lower extremity disease occurred in 36 patients (80.0%), ulceration in 31 (68.9%), and atrophie blanche in 32 (71.1%). In patients tested, transcutaneous oximetry measurements were decreased in 20 (74.1%) of 27, and factor V Leiden mutation (heterozygous) was noted in 2 (22.2%) of 9, decreased activity for protein C or protein S in 2 (13.3%) of 15, prothrombin G20210A gene mutation in 1 (8.3%) of 12, and lupus anticoagulant in 5 (17.9%) of 28. Anticardiolipin antibodies were present in 8 (28.6%) of 28 patients, and elevated homocysteine levels in 3 (14.3%) of 21. Intraluminal thrombosis was observed in 44 (97.8%) of 45 skin biopsy specimens. Direct immunofluorescence disclosed multiple vascular conjugates in 31 (86.1%) of 36 biopsy specimens. Livedoid vasculopathy was predominantly bilateral, affected the lower extremities, and was associated with ulceration and atrophie blanche. Histologic evidence of intraluminal thrombosis was observed in almost all biopsy specimens reviewed. Laboratory testing revealed numerous heterogeneous coagulation abnormalities, providing further evidence of procoagulant mechanisms.
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            Treatment for Livedoid Vasculopathy: A Systematic Review.

            Livedoid vasculopathy is a painful, ulcerative condition of the lower extremities for which no established treatment exists. Current treatment paradigms rely on low levels of evidence, primarily case reports and case series.
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              Livedoid vasculopathy: An in-depth analysis using a modified Delphi approach

              Livedoid vasculopathy (LV) is a noninflammatory thrombotic condition presenting in a primary idiopathic or secondary subtype associated with abnormal coagulation factors. Different from atrophie blanche (AB), which is a clinical manifestation of certain scars, LV may have AB in combination with recurrent livedo reticularis with chronic and painful skin ulcers particularly around the ankle region, and at the back of the feet. Histology is characterized by segmental hyalinizing changes at the subintimal region of small dermal vessels with thrombotic occlusions. LV skin ulcers resolve with stellate, porcelain-white scars that need to be distinguished from similar changes seen with venous insufficiency. "Atrophie blanche" was originally used synonymously with "livedoid vasculopathy." AB describes spontaneously occurring porcelain-white skin areas with red dots that typically occur in the context of skin changes attributed to chronic venous insufficiency. The 2 forms of AB--(1) the LV-AB complex and (2) AB in the context of chronic venous insufficiency--are unrelated and require separate diagnostic and therapeutic approaches. Using a modified Delphi method, we have developed an international consensus document on the diagnosis and management of LV. Individual sections of this document provide advice on diagnosis and management of LV.

                Author and article information

                Case Rep Vasc Med
                Case Rep Vasc Med
                Case Reports in Vascular Medicine
                4 July 2023
                : 2023
                : 6920383
                1Ochsner Medical Center, New Orleans, Louisiana, USA
                2Texas Tech University Health Sciences Center, USA
                Author notes

                Academic Editor: Muzaffer Sindel

                Author information
                Copyright © 2023 Ahmad Hallak et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                : 31 May 2022
                : 11 April 2023
                : 9 June 2023
                Case Report


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