Primary lymphangioma circumscriptum of the vulva presenting as warty plaques

Gude, Geethanjali; Gupta, Parikshaa; Sharma, Ramesh Kumar; Rajwanshi, Arvind

doi:10.1111/ajd.13014

Cited by 3 publications

(5 citation statements)

References 7 publications

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“…No additional articles were identified based on a search of article references. Ultimately, 78 articles, 1,3–79 comprising a total of 133 patients, met the eligibility criteria and were included in this qualitative analysis. Articles were published between 1979 and 2022.…”

Section: Resultsmentioning

confidence: 99%

Acquired vulvar lymphangioma: risk factors, disease associations, and management considerations: a systematic review

Duong

Balfour

Kraus

2023

International Journal of Women’s Dermatology

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Background: Acquired vulvar lymphangioma (AVL) is not well-characterized. Diagnosis is delayed and the condition is often refractory to therapy.Objective: The objective of this study was to provide a systematic review of AVL including risk factors, disease associations, and management options.Methods: A primary literature search was conducted using 3 databases: PubMed, CINAHL, and OVID, from all years to 2022.Results: In total, 78 publications with 133 patients (48 ± 17 years) were included. Most studies were case reports/series. The most common disease association was prior malignancy (70 patients, 53% of cases) and inflammatory bowel disease (6 patients, 5% of cases). The most common malignancy was cervical cancer (57 patients, 43% of cases). Most patients had prior radiation or surgery, with 36% (n = 48) treated with radiation, 30% (n = 40) with lymph node dissection, and 27% (n = 36) with surgical resection. Common presenting symptoms included discharge/oozing, pain, and pruritus. Most patients underwent surgical treatment for AVL with 39% treated with excision, 12% with laser therapy (the majority used CO 2 ), and 11% with medical therapies. Most patients had failed prior therapies and there was a diagnostic delay.Limitations: Retrospective nature. Most studies were limited to case reports and case series, with interstudy variability and result heterogeneity. Conclusion:AVL is an underrecognized entity and should be considered in patients with a history of malignancy or radiation to the urogenital area. Treatment should include multidisciplinary care and address underlying lymphatic changes, manage any existing inflammatory conditions, and utilize skin-directed therapies and barrier agents while addressing symptoms of pruritus and pain. Prospective studies are needed to further characterize AVL and develop treatment guidelines.

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Section: Resultsmentioning

confidence: 99%

Acquired vulvar lymphangioma: risk factors, disease associations, and management considerations: a systematic review

Duong

Balfour

Kraus

2023

International Journal of Women’s Dermatology

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“…Primary LC presents at birth or in early childhood, whereas secondary LC develops because of damage or impairment of lymphatic flow secondary to a variety of causes such as tuberculosis, filariasis, lymphogranuloma venereum, Crohn disease, neoplasms affecting the local lymphatics, and local surgeries such as vasectomy. [3][4] Because of the permeative growth of LC throughout all tissue layers, treatment is often challenging. Vesicles of LC often have associated deeper components.…”

Section: Discussionmentioning

confidence: 99%

Dermoscopic Assessment of Lymphangioma Circumscriptum Treated With Topical Sirolimus Ointment: A Case Report

Ankad¹,

Smitha²,

Nagur³

2022

International Journal of Dermatology and Venereology

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Introduction: Lymphangioma circumscriptum (LC) is characterized by malformation of superficial lymphatics. Dermoscopy reveals brownish-yellow lacunae and white septa. In scrotal LC, gray and red areas are also noted. Treatment of LC is challenging because of its high recurrence.We herein report a case of scrotal LC that was successfully treated with topical sirolimus with dermoscopic assessment of the therapeutic response.Case presentation: A male patient presented with fluid-filled lesions on the scrotum after partial penectomy. Examination revealed multiple skin-colored vesicles containing serous fluid on either side of the scrotum. Dermoscopy showed brownish-yellow lacunae with white septa as specific patterns. A diagnosis of secondary LC was made based on the clinical and dermoscopic features, and the patient was treated with 0.1% topical sirolimus ointment.Discussion: Management of LC includes close follow-up. Sirolimus, by inhibiting cellular proliferation and angiogenesis, helps in the regression of vesicular lesions of LC. Furthermore, dermoscopy supports an accurate evaluation of the therapeutic response. Conclusion:Sirolimus is a promising topical agent in the treatment of LC. Dermoscopy comes handy in the evaluation of treatment response. Any residual lesions could be noted and treated accordingly.

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“…Acquired causes of lymphangioma circumscriptum include filariasis, tuberculosis, scrofuloderma, lymphogranuloma venereum, hidradenitis suppurativa, lichen planus, scleroderma, recurrent cellulitis, severe herpes simplex infection, Crohn disease, local surgery, and radiotherapy. [2][3][4][5][6][7][8] Congenital lymphangioma circumscriptum usually occurs at birth or shortly thereafter, while the acquired type can occur at an age. 7 Lymphangioma circumscriptum is characterized by clusters of vesicles resembling frog spawn.…”

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confidence: 99%

“…4 Intermittent swelling, hemorrhage, or oozing of fluid from the superficial vesicles may occur. 6,10 With time, the vesicles may undergo verrucous changes and have a warty appearance. 1,6 The condition is usually asymptomatic but occasionally can cause pruritus or pain.…”

mentioning

confidence: 99%