Lipschütz acute vulvar ulcer related to COVID-19 vaccination: First case report in South America / Úlcera vulvar aguda de Lipschütz relacionada con la vacunación por COVID-19: primer caso notificado en Sudamérica

Lipschütz ulcer is a non-sexually transmitted genital lesion of unknown etiology, which presents as a painful vulvar ulcer. Lipschütz ulcers have been described in most continents. This is the first case reported in Peru and South America. We present the case of a 33-year-old female patient with a Lipschütz ulcer after being vaccinated with the second dose of the AstraZeneca COVID-19 vaccine. She reported having had only one sexual partner in her lifetime. Laboratory results were negative for herpes simplex 2, Cytomegalovirus, Toxoplasma gondii, Epstein-Barr virus, and syphilis. The patient received symptomatic treatment. Ten days after the onset, the patient was significantly better during follow-up. This case report displays a potential adverse effect of the AstraZeneca COVID-19 vaccine as a Lipschütz ulcer triggered by the host humoral immune response. However, further research is needed to establish the causal relationship between these two.

La úlcera de Lipschütz es una lesión genital no transmitidas por vía sexual de etiología desconocida, se presenta como una úlcera vulvar muy dolorosa. Las úlceras de Lipschütz se han descrito en la mayoría de los continentes. Este es el primer caso que se reporta en Perú y Sudamérica. En este reporte de caso se presentauna paciente de 33 años con una úlcera de Lipschütz luego de haber sido vacunada con la segunda dosis de la vacuna AstraZeneca COVID-19. Refirió haber tenido una sola pareja sexual a lo largo de su vida. Los resultados de laboratorio fueron negativos para herpes simplex 2, Citomegalovirus, Toxoplasma gondii, virus de Epstein-Barr y sífilis. La paciente recibió tratamiento sintomático. Diez días después, durante el seguimiento, la paciente estaba significativamente mejor. Este reporte de caso expone un potencial efecto adverso de la vacuna AstraZeneca COVID-19, en forma de úlcera de Lipschütz, desencadenado por la respuesta inmune humoral del huésped. Sin embargo, es necesario realizar más investigación para establecer la relación causal entre ambos.

Lipschütz Ulcer: An Unusual Diagnosis that Should Not be Neglected

Abstract The diagnosis of genital ulcers remains a challenge in clinical practice. Lipschütz ulcer is a non-sexually transmitted rare and, probably, underdiagnosed condition, characterized by the sudden onset of vulvar edema along with painful necrotic ulcerations. Despite its unknown incidence, this seems to be an uncommon entity, with sparse cases reported in the literature. We report the case of an 11-year-old girl who presented at the emergency department with vulvar ulcers. She denied any sexual intercourse. The investigation excluded sexually transmitted infections, so, knowledge of different etiologies of non-venereal ulcers became essential. The differential diagnoses are extensive and include inflammatory processes, drug reactions, trauma, and malignant tumors. Lipschütz ulcer is a diagnosis of exclusion. With the presentation of this case report, the authors aim to describe the etiology, clinical course, and outcomes of this rare disease, to allow differential diagnosis of genital ulceration.

Úlceras vulvares agudas en adolescentes, reporte de una serie de casos clínicos / Severe vulvar ulcers in adolescents, a report of clinical cases series / Úlceras vulvares agudas em adolescentes, informe de uma série de casos clínicos

Introducción: las úlceras genitales se caracterizan por su gran heterogeneidad clínica y etiológica. Pueden corresponder a entidades patológicas cutáneas o sistémicas de naturaleza infecciosa o no infecciosa. Dentro de estas últimas, se destacan las úlceras de Lipschütz y la enfermedad de Behcet. Objetivo: describir cuatro casos clínicos de adolescentes con úlceras vulvares agudas de causa no infecciosa hospitalizadas en dos prestadores del Sistema Nacional Integrado de Salud de Montevideo con el objetivo de brindar herramientas a profesionales para el correcto abordaje diagnóstico y terapéutico. Discusión: la realización de una anamnesis detallada y un examen físico minucioso son esenciales para realizar un adecuado abordaje diagnóstico y terapéutico. En el análisis clínico es importante considerar la edad de la paciente, el inicio de relaciones sexuales, el número, topografía y características de las úlceras incluyendo la presencia o no de dolor, así como la asociación de manifestaciones sistémicas. Resulta necesario el conocimiento de esta patología poco frecuente en niñas y adolescentes cuyo abordaje diagnóstico y terapéutico depende de la orientación etiológica basada en criterios epidemiológicos y clínicos bien definidos. Ello contribuirá a mejorar la calidad de la asistencia y a minimizar las repercu¬siones biológicas y psicológicas.

Introduction: genital ulcers are characterized by great clinical and etiological heterogeneity. They may correspond to infectious or non-infectious skin or systemic pathologies. Lipschütz ulcer and Behcet disease stand out among non-infectious conditions. Objective: to describe four clinical cases of adolescents with non-infectious severe vulvar ulcers hospitalized in two institutions of the national integrated health system in Montevideo, with the purpose of providing professionals with tools for the adequate diagnostic and therapeutic approach. Discussion: a detailed anamnesis questionnaire and a thorough physical examination are of the essence for an adequate diagnostic and therapeutic approach. The clinical analysis should include considering the patient's age, initiation of sexual intercourse, the number, topography and characteristics of the ulcers including finding out whether there is pain or not, as well as the association of systemic manifestations. It is important to learn about this rather unusual condition in girls and adolescents, whose diagnostic and therapeutic approach depends on the etiological orientation based on well-defined epidemiological and clinical criteria. This will contribute to improving the quality of assistance and minimizing biological and psychological effects.

Introdução: as úlceras genitais se caracterizam por uma grande heterogeneidade clínica e etiológica. Podem ser causadas por patologias cutâneas ou sistémicas tanto infecciosas como não infecciosas. Entre as últimas, destacam-se as úlceras de Lipschütz e a doença de Behçet. Objetivo: descrever quatro casos clínicos de adolescentes com úlceras vulvares agudas de causa não infecciosa hospitalizadas em duas unidades de saúde do sistema nacional integrado de saúde de Montevidéu buscando oferecer ferramentas para uma correta abordagem diagnóstica e terapêutica. Discussão: é fundamental realizar uma anamnese detalhada e um exame físico minucioso para uma abordagem diagnóstica e terapêutica adequada. O exame clínico deve incluir a idade da paciente, o inicio das relações sexuais, o número, topografia e características das úlceras incluindo a presencia ou não de dor, bem como a associação com manifestações sistémicas. É necessário conhecer esta patologia pouco frequente em meninas e adolescentes cuja abordagem diagnóstica e terapêutica depende da orientação etiológica baseada em critérios epidemiológicos e clínicos bem definidos. Isso contribuirá para melhorar a qualidade da assistência e a minimizar as repercussões biológicas e psicológicas.

Úlcera vulvar en una paciente de 15 años: ¿se puede reconocer la alergia a fármacos? / Vulvar ulcer in a 15-year-old girl: can we recognize adverse drug reactions?

Introducción. Los pediatras consideran la úlcera vulvar como una patología poco común. Sin embargo, los médicos que se especializan en la atención de adolescentes y niñas con patología vulvar consideran las úlceras vulvares como un motivo de consulta frecuente. Caso clínico. Se trató de un paciente femenino de 15 años con cuadro de fiebre y dolor faríngeo diagnosticado como faringitis bacteriana. Recibió tratamiento con trimetoprima-sulfametoxazol. Dos días después cursó con aparición de placas hiperpigmentadas de centro ampolloso, que evolucionaron a una úlcera de aproximadamente 4 cm de diámetro. Inicialmente se consideró un origen infeccioso de la lesión y se manejó con fluconazol tópico; posteriormente, amoxicilina clavulanato. No presentó mejoría y, tras la valoración por el dermatólogo, se diagnosticó eritema pigmentado fijo con base en la morfología de lesión inicial. Recibió tratamiento con betametasona y loratadina oral, así como hidrocortisona tópica al 1%. Evolucionó con resolución de las lesiones. Conclusiones. En la práctica del pediatra es trascendental conservar en mente no solo la patología de origen infeccioso y venéreo en las pacientes adolescentes con úlcera vulvar, sino también las reacciones de alergia a fármacos.

Background. Pediatricians consider vulvar ulcer as an uncommon pathology. However, clinicians specialized in the care of young girls and teenagers with vulvar pathology actually consider vulvar ulcer not to be an infrequent primary complaint. Case report. We report the case of a 15-year-old girl who presented fever and sore throat for which she was treated with trimethoprim-sulfamethoxazol. Two days after taking the drug she presented hyperpigmented patches with a bullous center that developed into an ulcer of ~4 cm in diameter. First considered infectious in origin, she received treatment with topical fluconazol and afterwards with amoxicillin clavulanate without any improvement. She was then assessed by a dermatologist, and the diagnosis of fixed drug eruption was established on the basis of the initial morphology of the lesion. She was treated with betamethasone and loratadine as well as topical 1% hydrocortisone. The lesions evolved to complete resolution. Conclusions. In pediatric practice, it is important for the pediatrician to consider not only infectious and venereal causes in teenage girls with vulvar ulcer, but also allergic drug reactions.

Vulvar ulcer as a presentation of systemic langerhans cell histiocytosis.

We report a 38-year-old housewife with systemic Langerhans cell histiocytosis (LCH) presenting as a chronic vulvar and peri-anal ulcer. She had systemic involvement in the form of diabetes insipidus and bone "hot-spots". She responded favorably to etoposide, 6-mercaptopurine, and systemic steroids, and has been in remission since 10 years. Chronic vulvar ulcers not responding to routine therapy should not be neglected and need to be biopsied repeatedly to come to a specific diagnosis. The vulvar ulcer in our case provided a vital clue to a systemic LCH, with a successful outcome.