Case and Research Letter
Vulval Lymphangioma Circumscriptum as Painful Papulovesicles Mimicking Genital Warts
Linfangioma circunscrito vulvar manifestado como papulovesículas dolorosas, simulando verrugas genitales
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Note the improvement of the lesions after 6 months of treatment with apremilast 30<span class="elsevierStyleHsp" style=""></span>mg twice a day (C and D).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M. Mansilla-Polo, C. Abril-Pérez, M.Á. Navarro-Mira, R. Botella-Estrada" "autores" => array:4 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Mansilla-Polo" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Abril-Pérez" ] 2 => array:2 [ "nombre" => "M.Á." "apellidos" => "Navarro-Mira" ] 3 => array:2 [ "nombre" => "R." "apellidos" => "Botella-Estrada" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731023004908?idApp=UINPBA000044" "url" => "/00017310/unassign/S0001731023004908/v1_202307180913/en/main.assets" ] "asociados" => array:1 [ 0 => array:17 [ "pii" => "S0001731024008020" "issn" => "00017310" "doi" => "10.1016/j.ad.2024.10.028" "estado" => "S200" "fechaPublicacion" => "2024-11-04" "aid" => "4119" "copyright" => "AEDV" "documento" => "simple-article" "crossmark" => 0 "subdocumento" => "crp" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "es" => array:10 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Carta científico-clínica</span>" "titulo" => " Linfangioma circunscrito vulvar manifestado como papulovesículas dolorosas, simulando verrugas genitales" "tienePdf" => "es" "tieneTextoCompleto" => "es" "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Vulval Lymphangioma Circumscriptum as Painful Papulovesicles Mimicking Genital Warts" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figura 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 879 "Ancho" => 1409 "Tamanyo" => 238359 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Hallazgos microscópicos de linfangioma circunscrito. (A y B) Examen histopatológico que revela la presencia de una lesión polipoide con proliferación vascular cutánea y vasos dilatados compuestos por una capa única de células endoteliales (ver flechas). (C) Positividad parcheada para D2-40. (D) Receptores de progesterona expresados por las células estromales.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "L. Corbella-Bagot, A. García-Herrera, P. Fusté-Brull, I. Fuertes de Vega" "autores" => array:4 [ 0 => array:2 [ "nombre" => "L." "apellidos" => "Corbella-Bagot" ] 1 => array:2 [ "nombre" => "A." "apellidos" => "García-Herrera" ] 2 => array:2 [ "nombre" => "P." "apellidos" => "Fusté-Brull" ] 3 => array:2 [ "nombre" => "I." "apellidos" => "Fuertes de Vega" ] ] ] ] ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731024008020?idApp=UINPBA000044" "url" => "/00017310/unassign/S0001731024008020/v1_202411040359/es/main.assets" ] ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letter</span>" "titulo" => "Vulval Lymphangioma Circumscriptum as Painful Papulovesicles Mimicking Genital Warts" "tieneTextoCompleto" => true "saludo" => "<span class="elsevierStyleItalic">To the Editor,</span>" "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "L. Corbella-Bagot, A. García-Herrera, P. Fusté-Brull, I. Fuertes de Vega" "autores" => array:4 [ 0 => array:4 [ "nombre" => "L." "apellidos" => "Corbella-Bagot" "email" => array:1 [ 0 => "lluis.corbella96@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "García-Herrera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "P." "apellidos" => "Fusté-Brull" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "I." "apellidos" => "Fuertes de Vega" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Department of Dermatology, Clínic Barcelona, University of Barcelona, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Pathology, Clínic Barcelona, University of Barcelona, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of Gynecology, Clínic Barcelona, University of Barcelona, Barcelona, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Linfangioma circunscrito vulvar manifestado como papulovesículas dolorosas, simulando verrugas genitales" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1042 "Ancho" => 1674 "Tamanyo" => 436502 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Vulval lymphangioma circumscriptum. Non-inflammatory, coalescent, translucent, and millimeter-sized papulovesicles on the upper external wall of the two labia majora forming two papillomatous plaques. A vesicle with intracystic bleeding can also be identified (A).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Lymphangioma circumscriptum (LC) is a malformation-derived pseudotumoral lesion consisting of several cysts on the skin and subcutaneous tissue, occasionally after an acquired insult or stasis affecting the lymphatic system. It is often found on the head, neck, trunk and axillae; vulval lymphangiomas, though, are a rare finding.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> Vulval LC can be asymptomatic or cause discomfort or pruritus.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">2</span></a> LC requires histopathological confirmation. Misdiagnosis as condylomatosis is not rare, particularly in verrucous forms. Recurrences are frequent among all treatment modalities.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">2</span></a> We present two cases of very painful vulval LC initially diagnosed as condylomas, which resolved completely after CO<span class="elsevierStyleInf">2</span> laser therapy.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Case #1 was a woman in her 40s who presented with a 2-year history of periodically occurring vulval lesions. She reported the presence of pruritus and intense vulval pain. The physical examination revealed the presence of several translucent, coalescing papulovesicles on the two labia minora (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Condyloma acuminata was initially suspected, but acetowhite negativity signaled the need for a biopsy. The histopathological examination led to the diagnosis of LC (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Pelvic magnetic resonance imaging showed superficial bilateral tubulocystic structures on the vulva. No space occupying lesions on the draining lymphatics were identified. A single session of continuous-wave CO<span class="elsevierStyleInf">2</span> laser vaporization was performed, until all papulovesicles were gone. The patient will be followed-up at our center.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Case #2 was another woman in her 40s with a past medical history of chronic lymphedema on her lower limbs who came to our office due to spreading of the edema to the genitalia, and the onset of very painful vulval lesions. The gynecological examination revealed the presence of edematous vulval papulovesicles on both sides of labia majora and minora. Initially diagnosed as vulval condylomatosis, the patient received imiquimod, without a satisfactory response. The histopathological examination revealed the presence of enlarged lymphatic vessels in the papillary dermis, which led to a diagnosis of acquired lymphangioma circumscriptum. A diathermy loop was used to treat it that led to temporary remission. Five years later, the patient showed a recurrent vulval LC, which was successfully treated with a single session of continuous-wave CO<span class="elsevierStyleInf">2</span> laser vaporization and no further episodes have been reported to this date.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Congenital lymphangioma circumscriptum (LC) is a developmental defect of the skin lymphatic vessels, though most cases do not become apparent until adulthood. Non-inherited activating mutations in PIK3CA affecting the lymphatic endothelial cells have been identified<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">3</span></a> causing lymphatic hyperplasia and leading to cyst formation due to fluid stasis. In contrast, acquired LC is triggered by lymphatic disruption<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> following local processes such as pelvic surgery, radiation therapy, tuberculosis, fistulous Crohn's disease, or an obstructing neoplasm.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a> Cases #1 and #2 describe a late-onset congenital and an acquired LC due to chronic lymphedema, respectively. Pathology and treatment options are the same for both these subtypes.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Vulval LC may occur at almost any age.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">2</span></a> It presents with diffuse or clustered millimeter-sized, thin-walled translucent or serous vesicles. Sometimes LC can have a verrucous appearance due to hyperkeratosis. Lesions may present with no symptoms or pruritus, a burning sensation, tenderness, discharge, or dyspareunia.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a> Histopathology results show hyperplasia of the epidermis and dilatation of distorted, irregular lymphatic vessels in the papillary dermis, forming non-communicated cysts. Lymphatic channels can be identified by their flat endothelium expressing podoplanin (D2-40), PROX1, and VEGFR-3.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Neoplasm-related acquired vulval LCs are often due to pelvic and anogenital malignancies following radiation therapy or lymphadenectomy.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">6</span></a> The occurrence of lymphangiosarcoma in a case of long-standing LC has been reported.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">7</span></a> In the presence of a vulval LC without a previous history of lymphatic damage, an obstructive process in the draining lymphatics should be considered.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">8</span></a> We used magnetic resonance imaging over computerized tomography for a better study of the lymphatic system and soft tissues proximal to sites of a possible obstruction.</p><p id="par0035" class="elsevierStylePara elsevierViewall">LC can simulate relapsing, smooth and hyperkeratotic condylomatosis. Herpes, impetigo, contact dermatitis, erythema multiforme, fixed drug eruption and some autoimmune bullous diseases and tumors can also mimic LC. Dermoscopy of LC shows the combination of lacunae and vascular structures in 71% of the cases. Other common features include the presence of white lines and the hypopyon sign.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">9</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Indications for treatment of LC include cosmetic reasons, presence of symptoms, recurrent infections, or psychosexual dysfunction. Surgery,<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">10</span></a> sclerotherapy, and several destructive treatments have often been used. Local recurrences are frequent, and non-surgical approaches have twice the recurrence rate of surgery,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">2</span></a> since the latter can remove the deep communicating lymphatic cisterns considered to be the main cause of recurrences. The second case shows how the persistence of the underlying cause in acquired LC probably also has a significant impact on recurrences. We used ablative CO<span class="elsevierStyleInf">2</span> laser therapy as a less invasive, well-tolerated, and safe alternative to surgery. CO<span class="elsevierStyleInf">2</span> laser induces vaporization of intracellular water, can penetrate up to the reticular dermis, seal lymph vessels, and induce sclerosis of vascular channels in LC.<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">11</span></a> Laser settings are not standardized and vary widely between practitioners.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Vulval lymphangioma circumscriptum should be suspected in the presence of condylomatosis unresponsive to treatment. In vulval LC without a previous history of lymphatic damage, an obstructive process in the draining lymphatics should be ruled out. CO<span class="elsevierStyleInf">2</span> laser ablation is minimally invasive, effective, and safe against LC. However, more studies are needed to determine the most effective long-term treatment of vulval LC.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1042 "Ancho" => 1674 "Tamanyo" => 436502 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Vulval lymphangioma circumscriptum. Non-inflammatory, coalescent, translucent, and millimeter-sized papulovesicles on the upper external wall of the two labia majora forming two papillomatous plaques. A vesicle with intracystic bleeding can also be identified (A).</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1170 "Ancho" => 1874 "Tamanyo" => 439588 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Microscopic findings of lymphangioma circumscriptum. (A and B) Histopathology examination revealing the presence of a polypoid lesion with dermal vascular proliferation and dilated vessels outlined by a single layer of endothelial cells (see arrows). (C) Patched positivity for D2-40. (D) Progesterone receptors expressed by stromal cells.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0060" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Report of 4 cases of vulval lymphangioma: an update" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "C.E. Marchitelli" 1 => "M.C. Sluga" 2 => "M.M. Domenech" 3 => "M.S. Peremateu" 4 => "M.L. Absi" 5 => "A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 16 | 14 | 30 |
| 2024 October | 159 | 52 | 211 |
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| 2024 April | 139 | 46 | 185 |
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| 2023 December | 88 | 65 | 153 |
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