Tacrolimus ointment for the treatment of superficial kaposiform hemangioendothelioma and tufted angioma

Zhang, Xuepeng; Yang, Kaiying; Chen, Siyuan; Ji, Yi

doi:10.1111/1346-8138.15031

Cited by 12 publications

(19 citation statements)

References 15 publications

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“…Several case reports and case series have reported success using topical sirolimus and tacrolimus ointment in superficial KHE/TA. The investigators have shown a good response of KHE/TA to twice daily topical application of these drugs [130,131]. Tacrolimus is an anti-T-cell immunosuppressive drug that is FDA-approved as a topical gel formulation (available in a concentration of 0.03 and 0.1%) for the treatment of cutaneous inflammatory/fibrotic diseases [132].…”

Section: Topical Treatmentsmentioning

confidence: 99%

Kaposiform hemangioendothelioma: current knowledge and future perspectives

Chen

Yang

et al. 2020

Orphanet J Rare Dis

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Kaposiform hemangioendothelioma (KHE) is a rare vascular neoplasm with high morbidity and mortality. The initiating mechanism during the pathogenesis of KHE has yet to be discovered. The main pathological features of KHE are abnormal angiogenesis and lymphangiogenesis. KHEs are clinically heterogeneous and may develop into a lifethreatening thrombocytopenia and consumptive coagulopathy, known as the Kasabach-Merritt phenomenon (KMP). The heterogeneity and the highly frequent occurrence of disease-related comorbidities make the management of KHE challenging. Currently, there are no medications approved by the FDA for the treatment of KHE. Multiple treatment regimens have been used with varying success, and new clinical trials are in progress. In severe patients, multiple agents with variable adjuvant therapies are given in sequence or in combination. Recent studies have demonstrated a satisfactory efficacy of sirolimus, an inhibitor of mammalian target of rapamycin, in the treatment of KHE. Novel targeted treatments based on a better understanding of the pathogenesis of KHE are needed to maximize patient outcomes and quality of life. This review summarizes the epidemiology, etiology, pathophysiology, clinical features, diagnosis and treatments of KHE. Recent new concepts and future perspectives for KHE will also be discussed.

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Section: Topical Treatmentsmentioning

confidence: 99%

Kaposiform hemangioendothelioma: current knowledge and future perspectives

Chen

Yang

et al. 2020

Orphanet J Rare Dis

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120

180

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“…Although surgical excision has been recommended for most cases as a specific treatment, medication therapy for TA with large lesion range and pain remains a clinical intricacy 1,5 . In a previous study, we have reported two patients with TA showing response after topical tacrolimus application 6 . Here, we further report a case of TA with severe pain been treated successfully by tacrolimus ointment.…”

Section: Figurementioning

confidence: 70%

“…1,5 In a previous study, we have reported two patients with TA showing response after topical tacrolimus application. 6 Here, we further report a case of TA with severe pain been treated successfully by tacrolimus ointment.…”

mentioning

confidence: 79%

“…1,5 Tacrolimus, on the other hand, has emerged as a promising treatment option. 6 In the present case, we described a TA case with pain treated successfully by tacrolimus ointment. Despite the slow color fading of TA, pain of the lesion was considerably F I G U R E 1 A, Skin lesion before treatment: scattered, irregular warm dark-red patches/plaques on the left thigh.…”

mentioning

confidence: 80%

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Topical tacrolimus reduces the severe pain of tufted angioma: Case report

Xiao

Hao

et al. 2020

Dermatologic Therapy

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“…In total, 137 patients were diagnosed with KHE during this 8-year interval. These KHE patients had previously been invited to participate in studies of epidemiologic factor, clinical feature, and/or treatment analyses [ 7 – 13 ].…”

Section: Methodsmentioning

confidence: 99%

Chronic lymphedema in patients with kaposiform hemangioendothelioma: incidence, clinical features, risk factors and management

Chen

Xia

et al. 2020

Orphanet J Rare Dis

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Objectives There are no cohort studies of chronic lymphedema in patients with kaposiform hemangioendothelioma (KHE). We sought to characterize the incidence, clinical features, risk factors and management of chronic lymphedema in patients with KHE. Methods We conducted a multicenter retrospective analysis of patients who had a minimum of 3 years of follow-up after the onset of KHE and/or Kasabach–Merritt phenomenon (KMP). Clinical features were reviewed to determine the possible cause of chronic lymphedema. The degree of lymphedema, risk factors and management strategies were analyzed. Results Among the 118 patients, chronic lymphedema was confirmed by lymphoscintigraphy 1 year after the onset of KHE and/or KMP in 13 patients. In 8 patients with lymphedema, extremity swelling was evident in the presence of KHE and/or KMP. In all patients with lymphedema, a unilateral extremity was affected, along with ipsilateral KHE. Most (84.6%) patients reported moderate lymphedema. Lymphedema was more common in patients with larger (≥ 10 cm) and mixed lesions involving the extremities (P < 0.01). A history of KMP and sirolimus treatment were not predictors of lymphedema (P > 0.05). Overall, 76.9% of patients received sirolimus treatment after referral, including 53.8% who presented extremity swelling before referral. Seven (53.8%) patients received compression therapy. Five (38.5%) patients reported lymphedema-associated decreased range of motion at the last follow-up. Conclusions Chronic lymphedema is a common sequela of KHE and can occur independently of KMP and sirolimus treatment. Patients with large and mixed KHE involving extremities should be closely monitored for this disabling complication.

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Tacrolimus ointment for the treatment of superficial kaposiform hemangioendothelioma and tufted angioma

Cited by 12 publications

References 15 publications

Kaposiform hemangioendothelioma: current knowledge and future perspectives

Kaposiform hemangioendothelioma: current knowledge and future perspectives

Topical tacrolimus reduces the severe pain of tufted angioma: Case report

Chronic lymphedema in patients with kaposiform hemangioendothelioma: incidence, clinical features, risk factors and management

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