LETTERS AND COMMUNICATIONS Angiolymphoid Hyperplasia With Eosinophilia Responsive to Sclerotherapy Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon benign but potentially disfiguring vascular proliferation. It is most common in women during the third to fourth decade. The cause of ALHE is unknown, although trauma and reactive hyperplastic processes have been proposed.1,2 Angiolymphoid hyperplasia with eosinophilia is often associated with spontaneous bleeding, pain, growth, pulsation, and pruritus. Multiple treatment options have been reported for ALHE, and we report a case of ALHE responsive to sclerotherapy.
sent home with a pressure dressing, which was kept on for 12 hours. After the first treatment, spontaneous bleeding and pruritus had significant improvement. The lesions gradually decreased and completely disappeared after the seventh treatment (Figure 1). Sclerotherapy was stopped when lesions completely disappeared. The patient attended our clinic for follow-up visits every month after the remission. At 2year follow-up, there was no evidence of recurrence.
A 30-year-old woman was first seen in November 2010 for a 1-year history of subcutaneous bleeding and pruritic nodules of her occiput during pregnancy. Histology confirmed a diagnosis of ALHE. There is proliferation of large endothelial cells lining vascular spaces and lymphocytic and eosinophilic inflammatory infiltrations in the dermis. Plump endothelial cells show cytoplasmic vacuoles and hobnail changes.1 Blood eosinophilia was slightly raised. Immunologic investigations were normal. Pruritus and nodules were refractory to antihistamine and serial injections of diprospan.
Angiolymphoid hyperplasia with eosinophilia is an uncommon dermal angioproliferating tumor, characterized by red-to-brown papules or nodules on the head or neck, although it can also occur in the mouth, trunk, extremities, and inguinal area. A distinct pathological entity, ALHE, is marked by a proliferation of blood vessels with distinctive large endothelial cells. These blood vessels are accompanied by a characteristic inflammatory infiltrate that includes eosinophils. The lesion is benign but may be persistent and is difficult to eradicate. Multiple treatment modalities have been reported with varying rates of success.2 Surgical excision is the treatment of choice, but local recurrences may occur in 33% to 50% of the cases after standard surgical excision. Drawbacks of surgery are that it can be disfiguring and difficult, especially in the case of periauricular lesions. Alternative treatments include topical or intralesional corticosteroid, lasers, electrodessication, radiation, and cryotherapy, although recurrences have also been reported with these, particularly after incomplete removal.
We decided to treat the patient with intralesional polidocanol injections (Shanxi Tianyu Pharmaceutical, Xian, China) every 10 days. No additional topical treatment was given. Injection of 1% polidocanol solution was injected using a 25-G needle. The amount of solution injected into each lesion was 0.05 to 0.1 mL, and the total with each session was 1 to 2 mL. Care was taken to avoid any spillage or direct injection into the surrounding skin or into any artery. The anesthetic effects of polidocanol make the injection a little painful. Bleeding after the injection was controlled by applying pressure over the area. After the patient was observed for half an hour, the patient was
© 2014 by the American Society for Dermatologic Surgery, Inc. ISSN: 1076-0512 Dermatol Surg 2014;40:1042–1060
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·
·
Discussion
Polidocanol is a sclerosing agent commonly used for the treatment of varicose veins. Since 1967, the local anesthetic polidocanol has been used for sclerotherapy because its injection works by damaging the cell lining
Published by Lippincott Williams & Wilkins
·
1042
Copyright © American Society for Dermatologic Surgery. Unauthorized reproduction of this article is prohibited.
LETTERS AND COMMUNICATIONS
Figure 1. Clinical appearance before (left) and after (right) 7 sclerotherapy treatments.
of blood vessels, causing them to close and eventually be replaced by other types of tissue.3 Paravenous injection can make aseptic inflammations, fibrous tissue growing, veins oppression, and vascular occlusion around the venous blood vessels and surrounding tissues. We have found that this method represents a useful therapeutic option in the treatment of hemangiomas and vascular malformations in our outpatient department. Because of the vascular component of ALHE and reports of ALHEs associated with arteriovenous shunting,4,5 sclerotherapy has been considered. In this case, our patient has demonstrated continued improvement with each consecutive treatment. Spontaneous bleeding and pruritus has completely resolved after the second treatment. There was no recurrence after 24 months of follow-up. Sclerotherapy offers several advantages, such as the low risk of systemic toxicity or allergic reactions, and has a local anesthetic effect, hence causing less pain during injection. It seems to offer a good symptomatic and cosmetic result. Conclusion Although different methods for the treatment of ALHE are available, there is no specific treatment at present. Partial treatment and recurrences have usually been found through traditional therapy. Treating this rare dermatosis is still a challenge. In this case, a patient with ALHE responded exceptionally well to polidocanol sclerotherapy without any side effect. No
recurrence was found 2 years after follow-up. To the best of our knowledge, this is the first report of ALHE responsiveness to sclerotherapy. Given the success of our patient, we suggest further clinical research to examine whether polidocanol sclerotherapy is efficacious in addressing both the symptoms and lesions of ALHE.
References 1. Chong WS, Thomas A, Goh CL. Kimura’s disease and angiolymphoid hyperplasia with eosinophilia: two disease entities in the same patient: case report and review of the literature. Int J Dermatol 2006; 45:139–45. 2. Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 1969;81:1–14. 3. Rabe E, Pannier F. Sclerotherapy of varicose veins with polidocanol based on the guidelines of the German Society of Phlebology. Dermatol Surg 2010;36(Suppl 2):968–75. 4. James WD, Berger TG, Elston DM. Andrews’ diseases of the skin (10th ed). Elsevier/Saunders: Philadelphia, 2006. 5. Olsen TG, Helwig EB. Angiolymphoid hyperplasia with eosinophilia: a clinicopathologic study of 116 patients. J Am Acad Dermatol 1985; 12:781–96.
Yujue Wang, MD Yating Tu, MD Juan Tao, MD Yan Li, MD Department of Dermatology Affiliated Union Hospital Tongji Medical College University of Science and Technology Wuhan, China Y. Wang and Y. Tu have contributed equally to this work.
40:9:SEPTEMBER 2014
1043
Copyright © American Society for Dermatologic Surgery. Unauthorized reproduction of this article is prohibited.
sent home with a pressure dressing, which was kept on for 12 hours. After the first treatment, spontaneous bleeding and pruritus had significant improvement. The lesions gradually decreased and completely disappeared after the seventh treatment (Figure 1). Sclerotherapy was stopped when lesions completely disappeared. The patient attended our clinic for follow-up visits every month after the remission. At 2year follow-up, there was no evidence of recurrence.
A 30-year-old woman was first seen in November 2010 for a 1-year history of subcutaneous bleeding and pruritic nodules of her occiput during pregnancy. Histology confirmed a diagnosis of ALHE. There is proliferation of large endothelial cells lining vascular spaces and lymphocytic and eosinophilic inflammatory infiltrations in the dermis. Plump endothelial cells show cytoplasmic vacuoles and hobnail changes.1 Blood eosinophilia was slightly raised. Immunologic investigations were normal. Pruritus and nodules were refractory to antihistamine and serial injections of diprospan.
Angiolymphoid hyperplasia with eosinophilia is an uncommon dermal angioproliferating tumor, characterized by red-to-brown papules or nodules on the head or neck, although it can also occur in the mouth, trunk, extremities, and inguinal area. A distinct pathological entity, ALHE, is marked by a proliferation of blood vessels with distinctive large endothelial cells. These blood vessels are accompanied by a characteristic inflammatory infiltrate that includes eosinophils. The lesion is benign but may be persistent and is difficult to eradicate. Multiple treatment modalities have been reported with varying rates of success.2 Surgical excision is the treatment of choice, but local recurrences may occur in 33% to 50% of the cases after standard surgical excision. Drawbacks of surgery are that it can be disfiguring and difficult, especially in the case of periauricular lesions. Alternative treatments include topical or intralesional corticosteroid, lasers, electrodessication, radiation, and cryotherapy, although recurrences have also been reported with these, particularly after incomplete removal.
We decided to treat the patient with intralesional polidocanol injections (Shanxi Tianyu Pharmaceutical, Xian, China) every 10 days. No additional topical treatment was given. Injection of 1% polidocanol solution was injected using a 25-G needle. The amount of solution injected into each lesion was 0.05 to 0.1 mL, and the total with each session was 1 to 2 mL. Care was taken to avoid any spillage or direct injection into the surrounding skin or into any artery. The anesthetic effects of polidocanol make the injection a little painful. Bleeding after the injection was controlled by applying pressure over the area. After the patient was observed for half an hour, the patient was
© 2014 by the American Society for Dermatologic Surgery, Inc. ISSN: 1076-0512 Dermatol Surg 2014;40:1042–1060
·
·
·
Discussion
Polidocanol is a sclerosing agent commonly used for the treatment of varicose veins. Since 1967, the local anesthetic polidocanol has been used for sclerotherapy because its injection works by damaging the cell lining
Published by Lippincott Williams & Wilkins
·
1042
Copyright © American Society for Dermatologic Surgery. Unauthorized reproduction of this article is prohibited.
LETTERS AND COMMUNICATIONS
Figure 1. Clinical appearance before (left) and after (right) 7 sclerotherapy treatments.
of blood vessels, causing them to close and eventually be replaced by other types of tissue.3 Paravenous injection can make aseptic inflammations, fibrous tissue growing, veins oppression, and vascular occlusion around the venous blood vessels and surrounding tissues. We have found that this method represents a useful therapeutic option in the treatment of hemangiomas and vascular malformations in our outpatient department. Because of the vascular component of ALHE and reports of ALHEs associated with arteriovenous shunting,4,5 sclerotherapy has been considered. In this case, our patient has demonstrated continued improvement with each consecutive treatment. Spontaneous bleeding and pruritus has completely resolved after the second treatment. There was no recurrence after 24 months of follow-up. Sclerotherapy offers several advantages, such as the low risk of systemic toxicity or allergic reactions, and has a local anesthetic effect, hence causing less pain during injection. It seems to offer a good symptomatic and cosmetic result. Conclusion Although different methods for the treatment of ALHE are available, there is no specific treatment at present. Partial treatment and recurrences have usually been found through traditional therapy. Treating this rare dermatosis is still a challenge. In this case, a patient with ALHE responded exceptionally well to polidocanol sclerotherapy without any side effect. No
recurrence was found 2 years after follow-up. To the best of our knowledge, this is the first report of ALHE responsiveness to sclerotherapy. Given the success of our patient, we suggest further clinical research to examine whether polidocanol sclerotherapy is efficacious in addressing both the symptoms and lesions of ALHE.
References 1. Chong WS, Thomas A, Goh CL. Kimura’s disease and angiolymphoid hyperplasia with eosinophilia: two disease entities in the same patient: case report and review of the literature. Int J Dermatol 2006; 45:139–45. 2. Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 1969;81:1–14. 3. Rabe E, Pannier F. Sclerotherapy of varicose veins with polidocanol based on the guidelines of the German Society of Phlebology. Dermatol Surg 2010;36(Suppl 2):968–75. 4. James WD, Berger TG, Elston DM. Andrews’ diseases of the skin (10th ed). Elsevier/Saunders: Philadelphia, 2006. 5. Olsen TG, Helwig EB. Angiolymphoid hyperplasia with eosinophilia: a clinicopathologic study of 116 patients. J Am Acad Dermatol 1985; 12:781–96.
Yujue Wang, MD Yating Tu, MD Juan Tao, MD Yan Li, MD Department of Dermatology Affiliated Union Hospital Tongji Medical College University of Science and Technology Wuhan, China Y. Wang and Y. Tu have contributed equally to this work.
40:9:SEPTEMBER 2014
1043
Copyright © American Society for Dermatologic Surgery. Unauthorized reproduction of this article is prohibited.
