A Refractory Livedoid Vasculopathy Accompanied by Methylene Tetrahydrofolate Reductase Gene Polymorphism Successfully Treated with Hyperbaric Oxygen Therapy
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Livedoid vasculopathy (LV) is a chronic coagulation disorder characterized by recurrent, painful ulcers on the lower extremities. Methylene tetrahydrofolate reductase (MTHFR) gene polymorphism is associated with coagulopathy. Therapeutic options usually include anti-inf lammatory or immunosuppressive agents. However, the condition is still highly challenging to manage and no consensus over the first-line treatment for LV exists. Further- more, when LV is accompanied with MTHFR gene polymorphism, clinical presentations could be more severe and resistant to treatment. We report a case of refractory LV accom- panied by MTHFR gene polymorphism, which was successfully treated with hyperbaric oxygen therapy (HBOT). A 63-year-old female patient presented with multiple painful ul- cers, atrophie blanches, and retiform purpura on both lower legs and feet. Histopathologic findings were compatible with LV. LV was diagnosed based on these clinicopathological findings. Following the diagnosis, we treated the patient with pentoxifylline, aspirin, sys- temic corticosteroid, antihistamine, and antibiotics. In spite of six-month treatment, the skin lesions did not improve; hence, HBOT was performed. It was performed at 2.0 absolute atmosphere for 120 minutes each time, three times a week. After 4 sessions, the ulcers began to heal and after 13 sessions, the skin lesions almost healed. During the eight-month follow- up period, the skin ulcers did not recur and the symptoms remained stable. Additionally, it was confirmed that she had MTHFR gene polymorphism after a genetic test. In conclusion, we wish to provide evidence regarding the effectiveness of HBOT and suggest that HBOT might be a considerable treatment option in refractory LV.
Abstract
Livedoid vasculopathy (LV) is a chronic coagulation disorder characterized by recurrent, painful ulcers on the lower extremities. Methylene tetrahydrofolate reductase (MTHFR) gene polymorphism is associated with coagulopathy. Therapeutic options usually include anti-inf lammatory or immunosuppressive agents. However, the condition is still highly challenging to manage and no consensus over the first-line treatment for LV exists. Further- more, when LV is accompanied with MTHFR gene polymorphism, clinical presentations could be more severe and resistant to treatment. We report a case of refractory LV accom- panied by MTHFR gene polymorphism, which was successfully treated with hyperbaric oxygen therapy (HBOT). A 63-year-old female patient presented with multiple painful ul- cers, atrophie blanches, and retiform purpura on both lower legs and feet. Histopathologic findings were compatible with LV. LV was diagnosed based on these clinicopathological findings. Following the diagnosis, we treated the patient with pentoxifylline, aspirin, sys- temic corticosteroid, antihistamine, and antibiotics. In spite of six-month treatment, the skin lesions did not improve; hence, HBOT was performed. It was performed at 2.0 absolute atmosphere for 120 minutes each time, three times a week. After 4 sessions, the ulcers began to heal and after 13 sessions, the skin lesions almost healed. During the eight-month follow- up period, the skin ulcers did not recur and the symptoms remained stable. Additionally, it was confirmed that she had MTHFR gene polymorphism after a genetic test. In conclusion, we wish to provide evidence regarding the effectiveness of HBOT and suggest that HBOT might be a considerable treatment option in refractory LV.
참고문헌 (17)
- Fritsch P, "Livedo vasculitis", Hautarzt, 46, pp.215~224 (1995)
- Jorizzo JL, "Livedoid vasculopathy : what is it?", Arch Dermatol, 134, pp.491~493 (1998)
- Vasudevan B, "Livedoid vasculopathy : a review of pathogenesis and principles of management", Indian J Dermatol Venereol Leprol, 82, pp.478~488 (2016)
- Abou Rahal J, "Livedoid vasculopathy in a patient with lupus anticoagulant and MTHFR mutation : treatment with low-molecular-weight heparin", J Thromb Thrombolysis, 34, pp.541~544 (2012)
- 전인경, "MTHFR 유전자 돌연변이로 인한 고호모시스테인혈증 환자에게서 발생한 울혈반모양 혈관병증", 대한피부과학회지, 51(4), pp.276~279 (2013), 대한피부과학회
- Yang CH, "Intractable livedoid vasculopathy successfully treated with hyperbaric oxygen", Br J Dermatol, 149, pp.647~652 (2003)
- Polo Gascón MR, "Quality-of-life impairment in patients with livedoid vasculopathy", J Am Acad Dermatol, 71, pp.1024~1026 (2014)
- Micieli R, "Treatment for livedoid vasculopathy : a systematic review", JAMA Dermatol, 154, pp.193~202 (2018)
- Lee JS, "Livedoid vasculopathy in Koreans : clinical features and response to rivaroxaban treatment", J Eur Acad Dermatol Venereol, 34, pp.e176~e178 (2020)
- Key NS, "Hyperhomocyst(e)inemia and thrombophilia", Arch Pathol Lab Med, 126, pp.1367~1375 (2002)
- Dauwe PB, "Does hyperbaric oxygen therapy work in facilitating acute wound healing : a systematic review", Plast Reconstr Surg, 133, pp.208~ (2014)
- Oztürk F, "Hyperbaric oxygen therapy for the management of chronic wounds", Cutan Ocul Toxicol, 32, pp.72~77 (2013)
- Juan WH, "Livedoid vasculopathy : long-term follow-up results following hyperbaric oxygen therapy", Br J Dermatol, 154, pp.251~255 (2006)
- Bhutani S, "Livedoid vasculopathy managed with hyperbaric oxygen therapy", Med J Armed Forces India, 68, pp.389~391 (2012)
- Ray R, "Livedoid vasculopathy with hyperhomocysteinemia responding to hyperbaric oxygen therapy", Indian J Dermatol, 60, pp.524~ (2015)
- Irani-Hakime NA, "Livedoid vasculopathy associated with combined prothrombin G20210A and factor V(Leiden)heterozygosity and MTHFR C677T homozygosity", J Thromb Thrombolysis, 26, pp.31~34 (2008)
- Lee JS, "Methylene tetrahydrofolate reductase C677T polymorphism in Korean livedoid vasculopathy patients", J Am Acad Dermatol, 84, pp.1068~1069 (2021)