Autoria: Ray R; Sharma A; Vasudevan B; Sridhar J; Deo R; Mohanty CS
Publicado em: 01/09/2015
A 30-year-old male presented to the dermatology department with complaints of multiple ulcers over both legs of 6 years duration. The ulcers had a waxing and waning course with present exacerbation of lesions since 1 month. Dermatological examination revealed multiple ulcers distributed in a reticular pattern over medial and lateral aspects of both lower legs, extensor aspect of both ankles and dorsum of both feet. Multiple interspersed atrophic porcelain white scars were also present. Investigations revealed raised serum homocysteine levels. A skin biopsy from the ulcers showed features of livedoid vasculopathy. Following recurrence of lesions after oral corticosteroid therapy, the patient was given a course of hyperbaric oxygen therapy for the ulcers to which he responded very well. This case is being presented for the novel option of hyperbaric oxygen therapy in livedoid vasculopathy, which by itself is rarely reported in this part of the world.
INTRODUCTION: Livedoid vasculopathy (LV) is a rare cutaneous disease which manifests mainly as recurrent leg ulcers. These ulcers heal with atrophic, porcelain white scars called atrophie blanche, which is an alternative named used for the disease. Inflammation, autoimmunity and hypercoagulability are the main pathogenetic factors with the latter being the most predominant among them. The treatment is controversial with antithrombotic measures taking precedence over the others. The recurrent nature and controversial pathogenesis makes this disorder a very challenging one to treat. We herein report a case of LV associated with hyperhomocysteinemia which responded well to hyperbaric oxygen therapy (HBOT), thus providing a novel, efficient and safe option for management.
CASE REPORT: This 30-year-old male presented with complaints of multiple ulcers over both legs of 6 years duration. The patient gave history of initially developing few ulcers on the left foot after prolonged wearing of boots for duration of 7 days (18 hours/day). He progressively developed similar multiple ulcers over both lower legs and dorsum of feet over a duration of 2 weeks. They were associated with moderate intensity pain and would increase on walking or running and reduce with rest. The ulcers had a waxing and waning course and would generally heal with course of antibiotics and rest. Presently he had exacerbation of lesions since 1 month. There were no other associated complaints or history of any other risk factors.
DISCUSSION: LV is presently described as a condition manifesting as recurrent leg ulcers with thrombosis of the microcirculation as the main pathogenesis. The important procoagulant causes include hyperhomocysteinemia, proteins C and S deficiency, factor V Leiden mutation, prothrombin gene mutation, antithrombin III deficiency and antiphospholipid antibody syndrome.
Hyperhomocystenemia can either be genetic or acquired. The genetic causes of homocystenemia are mainly due to enzyme defects such as cystathione-β-synthase, methylenetetrahydrofolate reductase and methionine synthase deficiencies. Acquired causes include nutritional deficiencies, renal failure, use of folic acid and vitamin B6 antagonists, cardiovascular diseases and peripheral arterial occlusive disease. Folic acid, vitamin B6/B12 are the most common nutritional deficiencies associated with homocystenemia. Serum homocysteine levels higher than 5–15 μmol/L have been found to correlate more with LV.
