Hemangiolinfangioma mesentérico: reporte de un caso / Mesenteric hemangiolymphangioma: case report

El hemangiolinfangioma es un tipo muy raro de malformación del sistema vascular, caracterizado histológicamente por la presencia de vasos venosos y linfáticos dilatados quísticamente, cuyas células endoteliales de revestimiento son positivas para marcadores de inmunohistoquímica como CD31, CD34 y D2-40. El compromiso extenso retroperitoneal y del tracto gastrointestinal es infrecuente. Se presenta el caso de una paciente femenina de 24 años con antecedente de dolor pélvico crónico, con exacerbación de síntomas. El diagnóstico imagenológico mostró una masa retroperitoneal multiquística. Se hizo hemicolectomía derecha y resección de la masa, encontrándose que dicha lesión estaba íntimamente adherida al mesenterio con compromiso extenso del tracto gastrointestinal, y cuyo estudio histopatológico reveló un hemangiolinfangioma, con mejoría clínica posterior a la resección quirúrgica. Aportamos a la literatura mundial, la caracterización de los hallazgos clínicos, imagenológicos e histopatológicos de este tipo de malformaciones

Hemangiolymphangioma is a very rare type of malformation of the vascular system, characterized histologically by the presence of cystically dilated venous and lymphatic vessels, whose lining endothelial cells are positive for immunohistochemical markers such as CD31, CD34 and D2-40. Extensive retroperitoneal and gastrointestinal tract involvement is uncommon. We present the case of a 24-yearold female patient with a history of chronic pelvic pain with exacerbation of symptoms. The imaging diagnosis revealed a multicystic retroperitoneal mass. A right hemicolectomy and resection of the mass was performed, finding that the lesion was intimately adherent to the mesentery with extensive involvement of the gastrointestinal tract, and whose histopathological study revealed a hemangiolymphangioma, with clinical improvement after surgical resection. We contribute to the world literature with the characterization of the clinical, imaging and histopathological findings of this type of malformations

Uso del sildenafilo oral en el tratamiento del linfangioma orbitario / Use of Oral Sildenafil in the Treatment of Orbital Lymphangioma

Los linfangiomas orbitarios son malformaciones vasculares benignas, de crecimiento lento, abortivas y no funcionales, que se presentan principalmente en la primera década de la vida. Las opciones terapéuticas en estos casos presentan una resolutividad limitada, algunos tratamientos suelen ser agresivos y provocar daños del aparato visual. Se presenta un caso de una paciente femenina de 6 años de edad atendida por proptosis del ojo izquierdo a la que se le realizó el diagnóstico clínico-imagenológico de linfangioma de la órbita, con el objetivo de mostrar el resultado alcanzado en el manejo de la misma mediante el uso del sildenafilo por vía oral, modalidad terapéutica en estudio a nivel mundial en el tratamiento de estas afecciones. El tratamiento con sildenafilo en el linfangioma orbitario demostró ser eficaz en la mejoría del cuadro clínico y por imágenes. Durante el tratamiento no se reportaron reacciones adversas(AU)

Orbital lymphatic malformations are benign, slow-growing, abortive, nonfunctional vascular malformations that occur mainly in the first decade of life. Therapeutic options in these cases present limited resolution, some treatments are usually aggressive and cause damage to the visual apparatus. We present a case of a 6-year-old female patient treated for proptosis of the left eye. The clinical-imaging diagnosis of lymphangioma of the orbit was made to show the results achieved in its treatment through the use of oral sildenafil, a therapeutic modality under study worldwide in the treatment of these conditions. The treatment with sildenafil in orbital lymphangioma proved to be effective in the improvement of the clinical and imaging picture. No adverse reactions were reported during treatment(AU)

Malformaciones linfáticas abdominales en una población pediátrica: experiencia en un centro de referencia de Medellín, Colombia / Abdominal lymphatic malformations in a pediatric population: Experience in a referral center in Medellín, Colombia

Introducción. Los linfangiomas son anormalidades benignas del sistema linfático, que corresponden a dilataciones quísticas de estos vasos y se localizan especialmente en el cuello. Solo el 10 % de todas estas malformaciones se encuentran en el abdomen y presentan síntomas variables de acuerdo al tamaño y su ubicación especifica, siendo el dolor abdominal el principal síntoma. Métodos. Se presentan cinco pacientes pediátricos con malformaciones linfáticas abdominales. Se describen su cuadro clínico, localización, tratamiento y la experiencia en el manejo de dicha patología en un hospital de referencia. Resultados. Los métodos más apropiados para hacer una aproximación diagnóstica son la ecografía, la tomografía computarizada y la resonancia nuclear magnética. Dentro de las opciones descritas para el tratamiento están la farmacológica, la escleroterapia y la resección quirúrgica, tanto por vía abierta como por laparoscopia. Conclusión. Existe una variedad de métodos para realizar la resección de los linfangiomas abdominales, pero la cirugía sigue siendo la más efectiva, especialmente cuando se cuenta con la laparoscopia como una herramienta terapéutica.

Introduction. Lymphangiomas are benign abnormalities of the lymphatic system, which correspond to cystic dilations of these vessels and are located especially in the neck. Only 10% of all these malformations are found in the abdomen and present variable symptoms according to size and their specific location, with abdominal pain being the main symptom. Methods. Five pediatric patients with abdominal lymphatic malformations are presented. Their clinical presentation, location, treatment and experience in the management of this pathology in a referral hospital are described. Results. The most appropriate methods to make a diagnostic approach are ultrasound, computed tomography and magnetic resonance imaging. Among the options described for treatment are pharmacological, sclerotherapy and surgical resection, both open and laparoscopic. Conclusion. There are a variety of methods for resecting abdominal lymphangiomas, but surgery remains the most effective, especially when laparoscopy is used as a therapeutic tool.

Evaluación del tratamiento de linfangiomas con bleomicina en población pediátrica de 0 a 18 años / Evaluation of the treatment of lymphangiomas with bleomycin in a pediatric population aged 0 to 18 years

INTRODUCCIÓN. Los linfangiomas son una malformación infrecuente a escala mundial y constituye una preocupación para los padres del infante; está asociado a problemas estéticos y a posibles efectos deletéreos debido a la obstrucción o compresión de órganos vitales. Se ha descrito a la escleroterapia como la mejor opción de tratamiento. OBJETIVO. Determinar la eficiencia del tratamiento con bleomicina en linfangiomas en la población pediátrica de 0 a 18 años. MATERIALES Y MÉTODOS. Estudio transversal analítico retrospectivo. Población y muestra conocida de 20 datos de Historias Clínicas electrónicas de pacientes diagnosticados con linfangiomas y tratados con bleomicina en el Hospital de Especialidades Carlos Andrade Marín, desde enero 2015 a enero 2018. Criterios de inclusión: pacientes de 0 a 18 años de edad con diagnóstico de linfangioma mediante ecografía y angiotomografía computarizada. Criterios de exclusión: pacientes mayores de 18 años de edad o sospecha diagnóstica de linfangioma sin estudios de imagen, y/o que no acudieron a la cita de control, pacientes diagnosticados de linfangioma que no recibieron bleomicina para su tratamiento, niños con otras malformaciones vasculares. El análisis de datos se realizó en el programa estadístico International Business Machines Statistical Package for the Social Sciences. RESULTADOS. La mediana de edad en mujeres fue de 6,25 años y 3,8 años en hombres. 10 pacientes fueron hombres. El promedio de seguimiento fue de 26,86 +/- 16,78 meses. El tamaño promedio de los linfangiomas fue de 5,77 +/- 3,73 cm. La localización más frecuente fue cervical con un 52,38%. La mayoría fueron macronodulares con un 85,71%. La respuesta fue buena o excelente en el 81,00% de los casos con la primera infiltración y subió al 95,00% con la segunda y tercera, según requerimiento. CONCLUSIÓN. El tratamiento de los linfangiomas con bleomicina fue muy efectivo en la población estudiada.

INTRODUCTION. Lymphatic malformation is a rare malformation worldwide and is a concern for the parents of the infant; it is associated with aesthetic problems and possible deleterious effects due to obstruction or compression of vital organs. Sclerotherapy has been described as the best treatment option. OBJECTIVE. To determine the efficiency of bleomycin treatment in lymphangiomas in the pediatric population aged 0 to 18 years. MATERIALS AND METHODS. Retrospective analytical cross-sectional study. Population and known sample of 20 data from Electronic Medical Records of patients diagnosed with lymphangiomas and treated with bleomycin at the Carlos Andrade Marín Specialties Hospital, from January 2015 to January 2018. Inclusion criteria: patients aged 0 to 18 years with diagnosis of lymphangioma by ultrasound and computed angiotomography. Exclusion criteria: patients older than 18 years of age or diagnostic suspicion of lymphangioma without imaging studies, and/or who did not attend the control appointment, patients diagnosed with lymphangioma who did not receive bleomycin for treatment, children with other vascular malformations. Data analysis was performed in the statistical program International Business Machines Statistical Package for the Social Sciences. RESULTS. The median age in women was 6,25 years and 3,8 years in men. Ten patients were men. The average follow-up was 26,86 +/- 16,78 months. The average size of the lymphatic malformations was 5,77 +/- 3,73 cm. The most frequent location was cervical with 52,38%. Most were macronodular with 85,71%. The response was good or excellent in 81,00% of cases with the first infiltration and rose to 95,00% with the second and third, as required. CONCLUSION. The treatment of lymphangiomas with bleomycin was very effective in the population studied.

Clinical analysis of 40 newborns with head and neck occupying lesions / 中华耳鼻咽喉头颈外科杂志

Objective: To investigate the clinical features, diagnoses and treatments of head and neck occupying lesions in newborns. Methods: All newborns with head and neck occupying lesions admitted to Neonatel Intensive Care Unit of the First Affiliated Hospital of Zhengzhou University form January 2014 to November 2019 were included. There were 23 males and 17 females, admission age was from 2 d-28 d, and the clinical manifestations, examinations, treatments and outcomes were evaluated. Results: Among 40 newborns with head and neck occupying lesions, 22 cases were admitted with dyspnea, 15 cases with masses in oral cavity or head and neck, 2 cases with fever as the first symptom, and 1 case with hoarseness as the first symptom. There were 5 cases with local infection. All cases were examined with local ultrasound and CT or MRI. Nine cases with severe dyspnea were treated with invasive ventilationm, of them 6 cases underwent invasive ventilation for more than 48 hours, 4 cases received tracheal intubation and artificial nose. Diagnostic punctures were performed in 2 cases. Seven cases received conservative treatments. Surgeries were performed in 31 cases, and 25 cases obtained pathologic diagnoses, including 3 cases of soft palate mature teratomas, 1 case of hard palate teratoma, 1 case of granulosa cell tumor, 1 case of lobulated spindle cell tumor in tongue base, 1 case of polyp in right glottis, 1 case of polyp at esophageal entrance, 4 cases of lingual root cysts, 1 case of laryngeal cyst, 2 cases of thyroglossal duct cysts, 2 cases of lymphangiomas, 1 case of lymphangioma with hibernoma, 1 case of tracheal cyst, 1 case of esophageal cyst, 3 cases of left neck abscesses, 1 case of occipital hemangioma, and 1 case of left temporoparietal abscess. Conclusions: The head and neck occupying lesions in the newborn is prone to upper airway obstruction. Imaging examination can assist the diagnosis. Different treatments can be selected according to the natures of occupying lesions.

Thoracic duct lymphangioma and chylous ascites in a 24-year-old female

Lymphangiomas are rare and correspond to 0.7% to 4.0% of mediastinal tumors, and isolated mediastinal location occurs in 1% of cases. They are benign tumors that originate from a congenital malformation of the lymphatic vessels and are diagnosed more frequently in children less than 2 years of age. Chylous ascites is a clinical manifestation of thoracic duct lymphangioma and is composed of lymph accumulation caused by dilation of this lymphatic channel. It appears milky in the peritoneal cavity, containing triglyceride levels higher than 200 mg/dl. We report the case of a young patient with chylous ascites and lymphangioma of the thoracic duct, who was conservatively treated with octreotide and a low-fat diet with medium-chain triglycerides.

Linfangiectasia renal bilateral / Bilateral renal lymphangiectasia

Introducción: La linfangiectasia renal es una afección infrecuente en la que existe dilatación de los vasos linfáticos renales. Usualmente es bilateral y de buen pronóstico. Objetivos: Presentar un caso de linfangiectasia renal bilateral, sus características clínicas y aspectos del diagnóstico imaginológico. Caso clínico: Paciente femenina de 59 años de edad, hipertensa controlada, que asistió a la consulta externa de urología por dolor lumbar bilateral, de moderada intensidad y coloración rojiza de la orina, ambos de forma intermitente. El examen físico general y regional fue negativo. Se confirmó microhematuria en el examen general de la orina. Los estudios de la analítica sanguínea fueron normales. El ultrasonido renal reportó pielocaliectasia bilateral moderada y quistes parapiélicos, bilaterales, a predominio izquierdo. La tomografía axial computarizada renal contrastada, reveló la presencia de formaciones hipodensas renales bilaterales, de aspecto quístico parapiélicas y en el seno renal, compatibles con linfangiectasia bilateral. La paciente ha evolucionado favorablemente. Conclusiones: La linfangiectasia renal se debe tener en cuenta en el diagnóstico deferencial de la enfermedad quística renal. Para el diagnóstico imaginológico de certeza es esencial la tomografía axial computarizada renal contrastada(AU)

Introduction: The renal linfangiectasia is an uncommon, in which dilatation of the lymphatic renal vessels exists. It is usually bilateral and of good presage. Objectives: To present a case of renal bilateral lymphangiectasia, their clinical characteristics and aspects of the imaginologic diagnosis. Clinical case: Patient feminine of 59 years of age, with controlled hypertension that attended the external consultation of Urology for lumbar bilateral pain, of moderate intensity, and reddish coloration of the urine, both in an intermittent way. The general and regional physical exam was negative. Microhematuria was confirmed in the general exam of the urine. The sanguine studies of the analytic one were all normal ones. The renal ultrasound reported bilateral moderate pielocaliectasia and parapielic cysts, with left prevalence. The renal contrasted computed tomography revealed the presence of bilateral renal hipodenses formations, of aspect cystic parapielic and in the renal sinus, and compatible with bilateral linfangiectasia. The patient has evolved favorably. Conclusions: The renal linfangiectasia it should be kept in mind in the deferential diagnosis of the cystic renal illness. For the imaginologic diagnostic of certainty it is essential the renal contrasted computed tomography(AU)

Atypical lymphangioma and hyperkeratosis in a patient with morbid obesity

Abstract Lymphangioma is a rare and understudied pathology that is usually detected in the first decade of life, and its appearance in adults is rare. This report details a 51-year-old man with morbid obesity who presented, for the last eight months, multiple asymmetric tumor lesions with extension to the scrotal region. The diagnosis of circumscribed lymphangioma with associated infection was confirmed. This case report demonstrates an unusual presentation of the characteristics of the lymphangioma that are seldom described in the literature.

Diagnóstico y manejo de lesiones vasculares en cabeza y cuello de pacientes pediátricos de la Fundación Hospital de la Misericordia, período 2012­2019 / Diagnose and care of head and neck vascular lesions in pediatric patients from Fundación Hospital de la Misericordia, from 2012­2019

Objetivo: identificar el manejo y respuesta a tratamientos que presentaron los pa-cientes pediátricos diagnosticados con hemangiomas y malformaciones vasculares en cabeza y cuello, que acudieron al servicio de Cirugía Oral y Maxilofacial de la Fundación Hospital de la Misericordia (HOMI) durante el período 2012­2019. Métodos: 11 p a c i e n-tes fueron atendidos en el servicio de cirugía oral y maxilofacial de la Fundación HOMI, entre enero de 2012 y noviembre de 2019, con diagnóstico de hemangioma y/o malfor-maciones vasculares. De estos, 9 fueron tratados con doxiciclina como agente esclero-sante. Resultados: se atendieron 5 pacientes masculinos y 6 femeninos con rango de edad entre los 8 meses y 13 años, con un promedio de 86,8 meses. La escleroterapia se realizó con doxiciclina en dosis de 100 mg disuelta en una ampolla de bicarbonato de sodio de 10 ml, inyectada en 5 pacientes con ecografía y en 4 por aspiración directa. De los 11 pacientes, 9 fueron tratados con escleroterapia; de estos 5 recibieron una sesión de escleroterapia, mientras que a los otros 4 se les realizó entre 2 y 3 sesiones. En 5 pacientes fue posible hacer seguimiento, ya que la atención depende de la autorización de la EPS. Conclusiones: la doxiciclina como agente esclerosante utilizado en lesiones vasculares ha demostrado ser un medicamento seguro, de bajo costo y efectivo como tratamiento inicial de hemangiomas y malformaciones vasculares en la cara. El número de aplicaciones depende del tipo de lesión a tratar.

Objective: Identify the treatment provided to patients with hemangioma and vascular deformities diagnosis .in head and neck who resorted to Oral and Maxillofacial service in Fundación Hospital de la Misericordia (HOMI) from 2012-2019 and the outcome of this treatments. Methods: Eleven (11) patients were admitted and treated by the surgical and maxillofacial team in "Fundación Hospital la Misericordia HOMI", between January 2012 and November 2019 with the hemangioma and vascular deformities diagnosis. Nine (9) of them were treated using doxycycline as a sclerosing agent. Results: Out of the eleven (11) patients, five (5) were males and the other six (6) were females, with an age range going from 8 months, up to 13 years, for an average of 86,8 months. A sclerotherapy was performed using doxycycline, with a dosage of 100 mg dissolved in a syringe with 10 mL of sodium bicarbonate on five (5) patients with ultrasound, and four (4) with direct aspiration. Out of the eleven (11) patients, five (5) received one sclerotherapy session the remaining four (4) received 2 or 3 sessions. In five (5) of the patients it was possible to complete follow-ups since the attention depends on their healthcare provider. Conclusions: Doxycycline as a sclerosing agent used in vascular lesions has demonstrated to be a safe treatment, with low cost, and effective as an initial treatment for hemangiomas and vascular deformities on the face. The number of sessions depend on the lesion that is going to be treated.

Hemangiolinfangioma con extensión abdominal y pélvica. Reporte de caso / Hemangiolymphangioma with abdominal and pelvic extension. Case report

El hemangiolinfangioma es una malformación vascular de rara ocurrencia. Posee componentes mixtos, venosos y linfáticos, y su manejo es limitado en casos de gran extensión. Se presenta el caso de un hombre con alteración vascular de bajo flujo, de predominio linfático, con extensión intrabdominal, retroperitoneal, pélvica y de miembros inferiores.

Hemangiolymphangioma is a vascular malformation of rare occurrence. It has mixed venous and lymphatic components and its management is limited in cases of large extension. We present the case of a man with low-flow vascular alteration, predominantly lymphatic, with intra-abdominal, retroperitoneal, pelvic and lower limb extension.

Tratamiento esclerosante del linfangioma facial. Presentación de un caso / Sclerotherapic treatment of facial lymphangioma

This article details the treatment of lymphangioma of the face with intralesional bleomycin: with a case report and literature review. Surgical treatment of lymphangioma of the face is a difficult task to achieve, due to close vicinity of the lesion to the facial nerve and possibility of scar tissue formation. Inefficient surgical removals generally will give rise to high recurrence rates because of infiltrative and diffuse extension of the lesion. However, complete cure has been described by non-surgical methods with intralesional bleomycin injection under ultrasonographic guidance. Lymphangioma is a rare congenital malformation of the lymphatic system, frequently seen in the head and neck. Percutaneous sclerotherapy of lymphangioma involves the injection of sclerosing substances into the lymphangioma. This study aims to evaluate the effectiveness of intralesional bleomycin sclerotherapy in the treatment of lymphangioma, and to determine the incidence of complications in the treatment. Intralesional bleomycin therapy was very effective in the treatment of lymphangioma. Bleomycin administered as intralesional injection was found to be safe as there was no lesions complicating or side effects observed in the study.

Linfangioma mesentérico en la edad pediátrica / Mesenteric Lynphangioma in the pediatric ages

Resumen Introducción: Dentro del espectro de las malformaciones congénitas con las cuales debe lidiar un cirujano pediatra, los tumores y malformaciones vasculares representan un grupo especial de entidades que continúan actualmente en estudio. Objetivo: Determinar las características clínicoquirúrgicas de una serie de dos casos con malformaciones de origen linfático. Presentacion de casos: Dos pacientes con edades pediátricas, 12 y 4 años, respectivamente, ingresaron en servicio de urgencia con signos de oclusión intestinal y lesiones tumorales quísticas abdominales que requirieron tratamiento quirúrgico con modalidades de técnicas quirúrgicas diferentes. La evolución final fue satisfactoria. Conclusión: Los linfagiomas intestinales forman parte de la gama de presentaciones clínicas de las malformaciones de origen linfático, con una incidencia baja pero que, dada su localización y síntomas, pueden requerir tratamiento quirúrgico de urgencia. Su evolución y resolución es satisfactoria, debido al bajo índice de recidiva tras la exéresis completa(AU)

ABSTRACT Introduction: Within the range of congenital malfomations that surgeons have to deal with, vascular tumors and malformations represent an special group of entities that are still under study. Objective: To determine the clinical-surgical characteristics of a series of two cases with congenital malformations of lymphatic origin. Cases presentation: Two patients of 12 and 4 years old respectively that were admitted in the Emergencies services in the Clinical-Surgical Hospital of Guayaquil, Ecuador, with manifestations of intestinal obstruction and abdominal cystic tumoral lesions that required surgical treatments with different techniques. Final evolution was satisfactory. Conclusion: Intestinal lymphoangiomas are part of the kinds of clinical presentations of lymphatic origin´s malformations. These have a low incidence but due to their localization and symptoms can require emergency surgical treatment. Their evolution and resolution is satisfactory due to the low index of recidive after complete exeresis(AU)

Adult-onset cystic hygroma in the axilla in a 44-year old female: A case report

@#This is a case of a 44- year old female presenting with an 18cm x 17cm soft, movable, non-tender mass at the right axilla extending to the lateral aspect of the right breast. Computerized tomographic scan of the chest revealed a lobulated, multi-septated hypodense mass. The patient underwent excision of the right axillary mass and final histopathology revealed cystic lymphangioma. Adult-onset cystic hygroma of the axilla is a rare case, with less than 10 studies documented in PubMed. Total surgical excision remains to be its primary treatment.

A Case of Adult Orbital Lymphatic Malformation in the Lacrimal Gland

PURPOSE: When there is a mass in the superior temporal orbit area, a lacrimal gland tumor should be suspected. We report a rare case of orbital lymphatic malformation that was histologically diagnosed in a patient with typical clinical features of the lacrimal gland. CASE SUMMARY: A 55-year-old female with no underlying disease and no ophthalmic history visited our clinic with a right upper eyelid edema associated with an enlarged painless eyelid mass 1 month prior to her visit. The patient stated that she discovered the mass 1 year previously.The palpebral lobe of the lacrimal gland protruded slightly with congestion of the surrounding conjunctiva. Enhanced computed tomography showed a 3 cm well-defined heterogeneous mass in the right lacrimal gland area and several well-defined round calcifications within the mass. Orbital tissue or bone involvement was not observed. The pleomorphic adenoma of the lacrimal gland was the most clinically suspicious, so complete resection of the mass was performed using lateral orbitotomy. Histopathologically, lymphangioma (lymphatic malformation) originating from the lacrimal gland was diagnosed. CONCLUSIONS: Orbital lymphatic malformation can occur in the lacrimal gland. The present case showed that differential diagnosis can reveal the presence of an adult lacrimal gland tumor.

Incidentally found cystic lymphangioma of the adrenal gland in an elderly male cadaver / 영남의대학술지

Adrenal cystic lymphangiomas are extremely rare entities that are often identified incidentally, with less than 60 cases reported to date. We found a protruding ovoid mass consisting of a multiloculated cystic lesion within right adrenal gland in the cadaver of a 75-year-old Korean man. The epithelial cells lining the adrenal cyst were diffusely positive for cluster of differentiation 31 and podoplanin, and negative for pan-cytokeratin. The histopathological diagnosis confirmed a cystic lymphangioma arising from the adrenal gland. Post-mortem findings of the present case are discussed based on the clinicopathological features of adrenal cystic lymphangiomas.

Reduction glossectomy of congenital macroglossia due to lymphangioma

Macroglossia is a rare clinical condition defined as an enlarged tongue. Macroglossia can cause structural deformities like diastema and disproportionate mandibular growth and present functional disorders such as dysarthria, dysphonia, and respiratory problems. A 7-year-old boy who had lymphangiomatous macroglossia was treated with a reduction glossectomy by anchor-shaped combination of a U-shape and modified key-hole resection. Postoperatively, the reduced tongue was contained completely within the oral cavity, but open bite remained due to prognathism. Sensory and motor nerves to the tongue appeared to be intact, and circulation was adequate. This patient will be monitored for recurrence of tongue enlargement.

Cutaneous Lymphangioma Circumscriptum Occurred Seven Years after Surgery with Soft Tissue Cystic Lymphangioma

Lymphangiomas are rare benign congenital lymphatic malformations. They can be divided into three groups: cutaneous lymphangioma circumscriptum (CLC), cavernous, and cystic. CLC is the most common type, and rarely occurs with cavernous or cystic lymphangioma under the lesion. Here, we describe the case of a 9-year-old girl who presented with an asymptomatic vesicular lesion on her back. She was finally diagnosed with CLC by clinical manifestations, dermoscopic findings, and histologic findings. Seven years ago, there was a history of surgical operation in the department of general surgery, and the surgery was soft tissue cystic lymphangioma removal surgery. There was no skin lesion for seven years after surgery, but one occurred a month ago. Herein, we present a case of CLC that occurred after a long interval after surgery for soft tissue cystic lymphangioma.

Cervical lymphangioma in adults: case report and current treatment

ABSTRACT Introduction: Cystic tumors of the neck are rare in adults. Some of them include metastatic nodes, branchial cysts, thyroglossal cysts and lymphangiomas, among others. Clinically speaking, lymphangiomas are slow-growing soft masses located in different spaces of the neck. Case report: This paper reports the case of a 36-year-old woman presenting with lymph-angioma, who consulted due to a right lateral mass in the neck of 20 days of evolution without associated systemic symptoms. Although relapse is frequent, the patient was successfully treated with surgery, without evidence of recurrence at 12 months of follow-up. Discussion: When cystic tumors of the neck occur in children, surgical urgencies may arise due to obstruction of the airway. However, lymphangioma in adults only produce contour deformity and rarely require urgent intervention, which allows for conservative management such as observation, repeated drainage or sclerotherapy that can be done using OK-432 (Picibanil). Nevertheless, surgery remains a good treatment option, but some complications may occur. Conclusion: Different treatment options were reviewed, which led to conclude that surgical resection of lymphangiomas continues to be a good treatment for this complex neck lesion.

RESUMEN Introducción: Los tumores quísticos del cuello son inusuales en los adultos. Sin embargo, se pueden encontrar metástasis a ganglios, quistes branquiales, quistes tiroglosos, linfangiomas, entre otros. Clínicamente, estos últimos son masas blandas de crecimiento lento que se localizan en diferentes espacios del cuello. Reporte de caso: Se reporta un caso de linfangioma en una mujer de 36 años, quien consultó por masa lateral derecha del cuello con evolución de 20 días sin síntomas sistémicos asociados. A pesar de que la recaída es frecuente, la paciente fue tratada con cirugía exitosa sin evidencia de recidiva durante 12 meses de seguimiento. Discusión: Cuando se presentan en niños, los tumores quísticos del cuello se pueden convertir en urgencias quirúrgicas debido a obstrucción de la vía aérea; no obstante, en los adultos solo producen deformidad de contorno y rara vez requieren una intervención apremiante, lo que permite conductas conservadoras como la observación, el drenaje repetido o la escleroterapia. Esta última puede hacerse con el OK-432 (Picibanil); sin embargo, la cirugía es una buena opción de tratamiento sin estar exenta de complicaciones. Conclusión: Se realizó revisión de las diferentes opciones de tratamiento y se concluyó que la resección quirúrgica de los linfagiomas continúa siendo la opción más adecuada para el manejo de esta compleja lesión del cuello.

Experiencia con OK-432 en malformaciones vasculares linfáticas en un hospital del norte de México / Experience with OK-432 in lymphatic vascular malformations in a hospital from northern Mexico

Resumen Introducción: Las malformaciones vasculares linfáticas (MVL), anteriormente llamadas linfangiomas, son malformaciones congénitas que se presentan en uno de cada 6,000 a 16,000 nacimientos. El sistema de clasificación más útil para el manejo del linfangioma se basa en el tamaño de los quistes. La resolución espontánea es infrecuente, por lo que el tratamiento expectante no se recomienda. El tratamiento clásico es la cirugía de escisión, pero puede afectar a estructuras vecinas o haber recidivas, por lo que se empezaron a estudiar sustancias esclerosantes, como el OK-432. La mayoría de los estudios incluyen pocos pacientes; los más grandes realizados en México se enfocan a lesiones específicas (macroquísticas) o únicamente a una región anatómica. Hasta la fecha, no existen estudios del uso de este fármaco en la población del norte de México. Método: Se describe la experiencia con OK-432 mediante un estudio retrospectivo, descriptivo, en los pacientes con MVL, de 2011 a 2016, en un hospital de referencia del norte de México. Resultados: Veintiséis pacientes con MVL recibieron tratamiento con OK-432. La mayoría fueron macroquísticos (69%), microquísticos (19%) y mixtos (12%). Del total de pacientes, 11 presentaron curación total. El 72% de la población estudiada tuvo una reducción de > 50% del tamaño de las lesiones con solo dos aplicaciones de tratamiento; no se presentaron recidivas. Se reportaron complicaciones en dos pacientes (hiperpigmentación de la piel). Conclusiones: El manejo con OK-432 demostró ser efectivo para el tratamiento de las MVL en un hospital de referencia del norte de México.

Abstract Background: Lymphatic vascular malformations (LVM) or formerly called lymphangiomas are congenital malformations present in about 1 out of 6000 to -16000 births. The most relevant classification system for lymphangioma management is based on the size of the cysts. Spontaneous resolution is uncommon; thus, expectant management is not recommended. The classic treatment is excisional surgery, but it can affect adjacent structures or have relapses, so, sclerosing substances like OK-432 are being studied. The majority of the studies are small in number of patients and are from Japan; the largest studies in Mexico are focused on specific lesions (macrocystic) or a determined anatomical region. To date, there are no studies of the population of the north of Mexico. Methods: The experience with OK-432 was described through a retrospective, descriptive study in patients with LVM, from 2011 to 2016, in a reference hospital of northern Mexico. Results: A total of 26 patients with LVM were treated with OK-432. The majority of the lesions were macrocystic (69 %), microcystic (19 %) and mixed (12 %). From the total number of patients, 11 fully healed, and 72 % of the study population had >50 % reduction in lesion size, with only 2 applications. There were no recurrences. Complications were reported in 2 patients who had skin hyperpigmentation. Conclusions: OK-432 probed to be an effective treatment for LVM in a reference hospital in the north of Mexico.