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Background. Lymphangiomas are benign tumors of abnormal lymphatic tissue. Approximately 6% of all lymphangiomas occur on the tongue. A lymphangioma of the tongue may present as a localized or a diffused growth, which may enlarge to cause macroglossia, impaired speech, and difficulty in mastication. This article reports a 21-year follow-up of a male infant who presented with a giant tongue lymphangioma. This long-term follow-up with multidisciplinary management including partial glossectomy, sclerotherapy, and orthodontic treatment to diminish complications of the disease in adulthood.

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OBJECTIVES: To review the effectiveness and safety of chemical ablation using ethanol or OK-432 for the treatment of TGDCs (thyroglossal duct cysts). METHODS: MEDLINE and EMBASE databases were searched up to May 29, 2020, to identify studies reporting the safety and efficacy of chemical ablation using ethanol or OK-432 for the treatment of TGDCs. The search query consisted of synonyms of thyroglossal duct cysts and ethanol or OK-432 ablation. The pooled success and complication rates were calculated using the inverse variance method to calculate weights, and pooled proportions were determined using the DerSimonian-Laird random-effects method. RESULTS: Seven original articles including a total of 129 patients were included. The efficacy of chemical ablation was acceptable, with a pooled success rate of 70% (95% CI, 47-86%). The pooled success rate of ethanol ablation was superior to that of OK-432 ablation, although with equivocal statistical significance (84% vs. 51%, p = 0.055). Repeat ethanol ablation achieved a pooled success rate of 47% (95% CI, 24-71%). The chemical ablation procedures were safe, with a pooled major complication rate of 0.9% (95% CI, 0.1-5.8%). CONCLUSIONS: Chemical ablation using ethanol or OK-432 for the treatment of TGDCs had acceptable success and low complication rates, and repeat treatment after initial failure was also feasible. In addition, it is an inexpensive and simple procedure and could therefore be considered a first-line treatment for TGDCs. KEY POINTS: • The efficacy of chemical ablation using ethanol or OK-432 was acceptable, with a pooled success rate of 70% (95% CI, 47-86%). The pooled success rate of ethanol ablation was superior to that of OK-432 ablation, although with equivocal statistical significance (84% vs. 51%, p = 0.055). • Repeat ethanol ablation was also feasible, with a pooled success rate of 47% (95% CI, 24-71%). • The chemical ablation procedures were safe, with a pooled major complication rate of 0.9% (95% CI, 0.1-5.8%).

Asunto(s)

Técnicas de Ablación , Quiste Tirogloso , Etanol , Humanos , Picibanil/uso terapéutico , Escleroterapia , Quiste Tirogloso/tratamiento farmacológico , Quiste Tirogloso/cirugía

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OBJECTIVE: The role of sclerotherapy for vascular lesions of the head and neck is well established. However, the efficacy of sclerotherapy for benign cystic lesions of the head and neck is less clear. The objective of this review is to determine the efficacy and safety of sclerotherapy for benign cystic lesions of the head and neck. DATA SOURCES: PubMed/MEDLINE, Cochrane Library, and Embase. REVIEW METHODS: The PRISMA guidelines (Preferred Reporting Systems for Systematic Reviews and Meta-analyses) were followed for this systematic review. Studies of patients with benign head and neck cystic masses treated primarily with sclerotherapy were included. Thirty-two studies met criteria for inclusion. RESULTS: A total of 474 cases of sclerotherapy were reviewed. Agents comprised OK-432, ethanol, doxycycline, tetracycline, and bleomycin. Lesions in the analysis were ranula, thyroglossal duct cyst, branchial cleft cyst, benign lymphoepithelial cyst, parotid cyst, thoracic duct cyst, and unspecified lateral neck cyst. A total of 287 patients (60.5%) had a complete response; 132 (27.9%) had a partial response; and 55 (11.6%) had no response. OK-432 was the most widely utilized agent, with a higher rate of complete response than that of ethanol (62.0% vs 39.4%, P = .015). Fifty-three cases (11.2%) required further surgical management. One case of laryngeal edema was reported and managed nonoperatively. CONCLUSION: Sclerotherapy appears to be a safe and efficacious option for benign cystic lesions if malignancy is reliably excluded. Efficacy rates are comparable to those of sclerotherapy for vascular malformations. The rate of serious complications is low, with 1 incident of airway edema reported in the literature.

Asunto(s)

Quistes/terapia , Escleroterapia , Malformaciones Vasculares/terapia , Branquioma/terapia , Etanol/administración & dosificación , Humanos , Linfocele/terapia , Cuello , Enfermedades de las Parótidas/terapia , Picibanil/administración & dosificación , Ránula/terapia , Quiste Tirogloso/terapia

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OBJECTIVE: In advanced ovarian cancer, traditional therapy included debulking surgery and postoperative chemotherapy. We proposed immunochemotherapy (IMCT) combined with picibanil (OK-432), interleukin-2 (IL-2), and traditional platinum- and taxol-based chemotherapy as a better treatment option for advanced ovarian cancer. METHODS: We retrospectively reviewed the medical records of 51 patients with advanced ovarian cancer between 2007 and 2015 at Chang Gung Memorial Hospital Linkou Medical Center, including 26 patients who were treated with OK-432, IL-2, and platinum- and taxol-based chemotherapy (IMCT group) after debulking surgery; another 25 were treated with traditional platinum- and taxol-based chemotherapy (traditional chemotherapy group) after debulking surgery. We analyzed the difference in age, follow-up period, recurrence rate, and diagnosis-to-recurrence period between the 2 groups. We also analyzed the difference in complete blood cell counts, differentiating counts, and cancer antigen 125 (CA-125) at 1 month after treatment. RESULTS: The recurrence rate between the IMCT and traditional chemotherapy groups showed a significant difference (53.8% vs 88%; P = .0128). The diagnosis-to-recurrence duration was longer in the IMCT than in the traditional chemotherapy groups (33.21 vs 25.63 months), although no statistical significance was found ( P = .4668). In laboratory analysis at 1 month after treatment, the white blood cell, absolute neutrophil, and absolute lymphocyte counts (ALCs) were significantly higher in the IMCT group. On the other hand, CA-125 was significantly lower, and ALC was significantly higher in the nonrecurrence group. CONCLUSIONS: Combined IMCT and chemotherapy have lower recurrence rate compared to traditional chemotherapy after debulking surgery for the treatment of advanced ovarian cancer.

Asunto(s)

Protocolos de Quimioterapia Combinada Antineoplásica/uso terapéutico , Inmunoterapia , Interleucina-2/uso terapéutico , Recurrencia Local de Neoplasia/prevención & control , Neoplasias Ováricas/tratamiento farmacológico , Picibanil/uso terapéutico , Anciano , Femenino , Humanos , Persona de Mediana Edad , Estadificación de Neoplasias , Estudios Retrospectivos , Resultado del Tratamiento

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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.

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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.

Asunto(s)

Humanos , Linfangioma , Picibanil , Escleroterapia , Adulto , Conducto Cístico

RESUMEN

PURPOSE: The management of lymphatic malformation (LM) in pediatric patients is challenging. Complete excision of LM is difficult to achieve in some cases. We reviewed our experience how to manage LM. METHODS: We retrospectively reviewed the patients who were treated for LM between 2010 and 2017. Medical records were reviewed about age of diagnosis, age of treatment age, gender, symptom, location of tumor, treatment modality, response and complication. RESULTS: Sixty-three patients (39 boys and 24 girls) were included. Mean age at diagnosis was 14.5±28.0 months (range, neonate-10 years). The involved lesion were head and neck in 27 patients (42.9%), abdominal cavity in 7 patients (11.1%), chest wall and abdominal wall in 11 patients (17.5%), buttock in 7 patients (11.1%), and extremities in 11 patients (17.5%). The treatment options were including surgical resection in 32 patients, sclerotherapy in 7 patients, surgical resection combined sclerotherapy in 19 patients, and close observation in 5 patients. We achieved complete remission in 39 patients. Fourteen patients showed partial remission and 6 showed recurrences. CONCLUSION: Despite surgical difficulty, meticulous excision with supportive treatment, and adjuvant sclerotherapy could get a favorable outcome without fatal complication. Decision should be based on surgeon's experience, location of LM, related symptoms, and consultation with patient's parents.

Asunto(s)

Humanos , Cavidad Abdominal , Pared Abdominal , Nalgas , Diagnóstico , Extremidades , Cabeza , Linfangioma , Registros Médicos , Cuello , Padres , Picibanil , Recurrencia , Estudios Retrospectivos , Escleroterapia , Pared Torácica

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BACKGROUND: Congenital cystic lymphangiomas are benign malformations due to a developmental disorder of lymphatic vessels. Besides surgical excision, sclerosant therapy of these lesions by intracavitary injection of OK-432 (Picibanil®), a lyophilized mixture of group A Streptococcus pyogenes, is a common therapeutical option. For an appropriate application of OK-432, a detailed knowledge about the structure and composition of the congenital cystic lymphangioma is essential. SonoVue® is a commercially available contrast agent commonly used in sonography by intravenous and intracavitary application. CASE PRESENTATION: Here we report the case of 2 month old male patient with a large thoracic congenital cystic lymphangioma. Preinterventional imaging of the malformation was performed by contrast-enhanced ultrasound after intracavitary application of SonoVue® immediately followed by a successful sclerotherapy with OK-432. CONCLUSIONS: Contrast agent-enhanced ultrasound imaging offers a valuable option to preinterventionally clarify the anatomic specifications of a congenital cystic lymphangioma in more detail than by single conventional sonography. By the exact knowledge about the composition and especially about the intercystic communications of the lymphangioma sclerosant therapy becomes safer and more efficient.

Asunto(s)

Linfangioma/diagnóstico por imagen , Linfangioma/terapia , Soluciones Esclerosantes/uso terapéutico , Medios de Contraste , Humanos , Lactante , Linfangioma/congénito , Masculino , Microburbujas , Picibanil/uso terapéutico , Escleroterapia , Resultado del Tratamiento , Ultrasonografía

RESUMEN

Background: Lymphangiomas are congenital malformations of the lymphatic system with characteristic dilated endothelium-lined spaces. It is vulnerability to infection or chemical irritants cause spontaneous reduction in size and in some cases complete resolution. Intralesional injection of OK-432 or Picibanil (lyophilized incubation mixture of Group A Streptococcus pyogenes of human origin) is slowly gaining recognition as its safety and efficacy standards have shown to avoid complications resulting from surgical interventions. The objective of this study was to evaluate the clinical outcomes of cystic hygroma patients who received OK-432 injections. Methods: In between 2011 and 2013, six patients with cystic hygroma received intralesional injection of OK-432. All the patients were assessed clinically and radiologically either via ultrasound, computer tomography (CT) or magnetic resonant imaging (MRI) prior to and after receiving the injections. Patients’ response towards treatment was classified as total shrinkage, marked shrinkage (greater than 50% reduction in size), slight shrinkage (less than 50% reduction in size) or non-responsive to treatment. Results: Mean duration of follow-up was 12 months. Total shrinkage was achieved in one patient, marked shrinkage in three patients and one patient experienced mild shrinkage. Only one out of the six patients showed no response to treatment. None of the patients in this study experienced serious complications or adverse effects post intralesional injection of OK-432. Conclusions: Intralesional OK-432 injection is an effective and safe alternative in treating cystic hygroma.

RESUMEN

INTRODUÇÃO: Conduziu-se revisão sistemática retrospectiva da literatura incluindo estudos relatando o uso de picibanil para tratar malformações linfáticas (ML). MÉTODOS: A pesquisa foi realizada com estudos publicados no PubMed de janeiro de 1990 a 14 de abril de 2013. Na estratégia de busca, usou-se os descritores "OK-432" ou "Picibanil" e "lymphatic malformation". Os seguintes elementos foram comparados aos de outras modalidades relatadas e, então, compilados: mecanismo de ação, indicações, contraindicações, eficácia, administração, efeitos colaterais, complicações, vantagens e desvantagens. RESULTADOS: Foram encontrados 44 estudos, 27 dos quais atenderam aos critérios de inclusão. O picibanil é uma preparação liofilizada de uma cepa de baixa virulência de Streptococcus pyogenes inativada pela penicilina G. Seu mecanismo de ação ainda não definido claramente, mas especula-se que provoque uma resposta inflamatória controlada com adesão das paredes dos cistos. O picibanil é indicado quase que unanimemente para o tratamento da ML macrocística, cuja resposta é mais efetiva do que em lesões microcísticas ou mistas. Em geral, o picibanil é administrado por meio de punção com visualização direta ou guiada por ultrassonografia, com o paciente sob anestesia geral. A preparação comumente utilizada consiste em 0,1 mg de picibanil em 10 ml de soro fisiológico. Os efeitos colaterais são, em geral, leves; sendo dor, inchaço e febre os mais frequentemente relatados. CONCLUSÃO: Os estudos apresentam pouca evidência científica. A revisão sistemática identificou que o picibanil é útil no tratamento da ML de qualquer tipo, mas tem resultados melhores em lesões macrocísticas. A eficácia foi comparável à de outras terapias. Não foi apresentada nenhuma contraindicação específica. Embora o mecanismo de ação ainda não tenha sido determinado, o picibanil trata-se de opção de tratamento.

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INTRODUCTION: We performed a retrospective systematic review of studies reporting the use of Picibanil for treatment of lymphatic malformations (LMs). METHODS: We searched the PubMed database for available studies, including those published between January 1990 and April 14, 2013. The search strategy involved the use of the keywords "OK-432" or "Picibanil" and "lymphatic malformation." Information was compiled regarding the reported mechanism of action, indications, contraindications, efficacy, administration, side effects, complications, and advantages and disadvantages compared to those of other modalities. RESULTS: Forty-four studies were found, of which 27 fulfilled the inclusion criteria. Picibanil is a lyophilized preparation of a low-virulence strain of Streptococcus pyogenes inactivated with penicillin G. Its mechanism of action is unclear, but it has been speculated that it causes a controlled inflammatory response with adhesion of cyst walls. Picibanil is almost unanimously indicated for the treatment of macrocystic LMs, which show a greater effectiveness response compared to that shown by microcystic or mixed LMs. Picibanil is usually administered by puncturing, either with direct visualization or guided by ultrasound, with the patient under general anesthesia. The most widely used preparation comprises 0.1 mg of Picibanil in 10 mL of saline. Side effects are mostly mild, with pain, swelling, and fever being the most frequently reported. CONCLUSION: The studies had low scientific evidence. A systematic review found that Picibanil is useful against any LM, with better results in macrocystic lesions. Efficacy was comparable to that of other therapies. No specific contraindication was presented. Although the mechanism of action has not been established, the inclusion of Picibanil as a treatment option is warranted.

Asunto(s)

Humanos , Historia del Siglo XXI , Picibanil , Streptococcus pyogenes , Terapéutica , Escleroterapia , Eficacia , Resultado del Tratamiento , Infusiones Intralesiones , Anomalías Linfáticas , Revisión Sistemática , Tejido Linfoide , Picibanil/efectos adversos , Picibanil/uso terapéutico , Picibanil/farmacología , Streptococcus pyogenes/efectos de los fármacos , Streptococcus pyogenes/patogenicidad , Terapéutica/efectos adversos , Terapéutica/métodos , Escleroterapia/efectos adversos , Escleroterapia/métodos , Eficacia/métodos , Infusiones Intralesiones/efectos adversos , Infusiones Intralesiones/métodos , Anomalías Linfáticas/complicaciones , Anomalías Linfáticas/patología , Anomalías Linfáticas/terapia , Tejido Linfoide/anomalías , Tejido Linfoide/crecimiento & desarrollo , Tejido Linfoide/patología

RESUMEN

OBJECTIVE: This study aimed to assess the histopathological effect of OK-432 (Picibanil) on rabbit nasal turbinates. METHODS: A total of 21 rabbits were divided into 3 treatment groups and various parts of both nasal turbinates were injected with 0.5 ml OK-432, 0.2 ml OK-432 or 0.6 ml saline (control). Bilateral nasal turbinates were later excised and studied under light microscopy to assess any histopathological changes. RESULTS: Animals in the 0.2 ml and 0.5 ml OK-432 groups exhibited mild ciliary loss, goblet cell loss and epithelial damage, and a marked increase in inflammatory cell infiltration, submucosal vascularisation and fibrosis. There was a significant difference in histopathological changes between the two OK-432 treated groups. In addition, each OK-432 treated group had significantly more inflammatory cell infiltration, increased submucosal vascularisation and fibrosis compared with controls. CONCLUSION: The marked fibrosis observed in OK-432-injected turbinates may be responsible for a reduction in turbinate size.

Asunto(s)

Obstrucción Nasal/tratamiento farmacológico , Picibanil/farmacología , Cornetes Nasales/efectos de los fármacos , Cornetes Nasales/patología , Animales , Biopsia con Aguja , Modelos Animales de Enfermedad , Relación Dosis-Respuesta a Droga , Esquema de Medicación , Inmunohistoquímica , Inyecciones Intralesiones , Masculino , Obstrucción Nasal/patología , Conejos , Distribución Aleatoria , Sensibilidad y Especificidad

RESUMEN

OBJECTIVE: Here, we describe our experience with different therapeutic modalities used to treat cystic lymphangiomas in children in our hospital, including single therapy with OK-432, bleomycin and surgery, and a combination of the three modalities. METHODS: We performed a retrospective, cross-sectional study including patients treated from 1998 to 2011. The effects on macrocystic lymphangiomas and adverse reactions were evaluated. Twenty-nine children with cystic lymphangiomas without any previous treatment were included. Under general anesthesia, patients given sclerosing agents underwent puncture of the lesion (guided by ultrasound when necessary) and complete aspiration of the intralesional liquid. The patients were evaluated with ultrasound and clinical examinations for a maximum follow-up time of 4 years. RESULTS: The proportions of patients considered cured after the first therapeutic approach were 44% in the surgery group, 29% in the bleomycin group and 31% in the OK-432 group. These proportions were not significantly different. Sequential treatment increased the rates of curative results to 71%, 74% and 44%, respectively, after the final treatment, which in our case was approximately 1.5 applications per patient. CONCLUSION: The results of this study indicate that most patients with cystic lymphangiomas do not show complete resolution after the initial therapy, regardless of whether the therapy is surgical or involves the use of sclerosing agents. To achieve complete resolution of the lesions, either multiple operations or a combination of surgery and sclerotherapy must be used and should be tailored to the characteristics of each patient. .

Asunto(s)

Preescolar , Femenino , Humanos , Lactante , Masculino , Bleomicina/uso terapéutico , Neoplasias de Cabeza y Cuello/terapia , Linfangioma Quístico/terapia , Picibanil/uso terapéutico , Punciones/métodos , Soluciones Esclerosantes/uso terapéutico , Brasil , Estudios Transversales , Terapia Combinada/métodos , Estudios de Seguimiento , Inyecciones Intralesiones , Inducción de Remisión , Estudios Retrospectivos , Soluciones Esclerosantes/administración & dosificación , Resultado del Tratamiento

RESUMEN

Surgical excision has traditionally been the treatment of choice for benign non-thyroid cystic neck masses, including lymphatic malformation, ranula, branchial cleft cyst, thyroglossal duct cyst, and parathyroid cyst. However, there is a tendency toward recurrence after surgery, and surgery may be accompanied by complications, including nerve injuries, vascular injuries, and scar formation. Ultrasound-guided sclerotherapy using various agents has been challenged and successfully applied as an alternative treatment for benign non-thyroid cystic neck masses. This report reviews the available sclerosing agents and describes the applications of sclerotherapy to the treatment of benign cystic masses in the neck.

RESUMEN

BACKGROUND/PURPOSE: Prolonged air leak is the most common complication after thoracoscopic operation for primary spontaneous pneumothorax (PSP), and the role of chemical pleurodesis in treating air leaks remains unclear. This study evaluated the safety and efficacy of chemical pleurodesis with a comparison between minocycline and OK-432. METHODS: Between 1994 and 2011, 1083 PSP patients were treated by thoracoscopic operation. After the operation, patients with persistent air leak for 3 days or more were managed by minocycline or OK-432 pleurodesis. The demographic and outcome data for these patients were collected by retrospective chart review. RESULTS: Seventy-nine patients (7.3%) with prolonged air leak after thoracoscopy underwent minocycline pleurodesis (60 patients) or OK-432 pleurodesis (19 patients) as the primary treatment. The primary success rate was 63% (38/60) for minocycline pleurodesis and 95% (18/19) for OK-432 pleurodesis (p = 0.009). Postpleurodesis pain was common and comparable between the two groups. No major complications were noted after a total of 121 treatments. Patients undergoing primary OK-432 pleurodesis had shorter durations of postpleurodesis chest drainage (mean 8.5 vs. 2.3 days; p < 0.001) and postoperative hospital stay (mean 11.9 vs. 6.8 days; p < 0.001) than those undergoing primary minocycline pleurodesis. After a median follow-up of 16 months, recurrence was noted in one patient in the OK-432 group and none in the minocycline group. Long-term pulmonary function in the two groups was comparable. CONCLUSION: Chemical pleurodesis using OK-432 or minocycline is safe and convenient for prolonged air leak after thoracoscopic treatment for PSP. Our experience suggested that OK-432 may be more effective than minocycline in reducing air leak.

Asunto(s)

Pleurodesia/métodos , Neumotórax/cirugía , Complicaciones Posoperatorias/terapia , Cirugía Torácica Asistida por Video/efectos adversos , Adulto , Femenino , Humanos , Masculino , Minociclina/administración & dosificación , Picibanil/administración & dosificación , Estudios Retrospectivos

RESUMEN

Surgical excision has traditionally been the treatment of choice for benign non-thyroid cystic neck masses, including lymphatic malformation, ranula, branchial cleft cyst, thyroglossal duct cyst, and parathyroid cyst. However, there is a tendency toward recurrence after surgery, and surgery may be accompanied by complications, including nerve injuries, vascular injuries, and scar formation. Ultrasound-guided sclerotherapy using various agents has been challenged and successfully applied as an alternative treatment for benign non-thyroid cystic neck masses. This report reviews the available sclerosing agents and describes the applications of sclerotherapy to the treatment of benign cystic masses in the neck.

Asunto(s)

Branquioma , Cicatriz , Cuello , Picibanil , Ránula , Recurrencia , Soluciones Esclerosantes , Escleroterapia , Quiste Tirogloso , Ultrasonografía , Lesiones del Sistema Vascular

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Toll-like receptors (TLRs) have long been known for their ability to initiate innate immune responses upon exposure to conserved microbial components such as lipopolysaccharide (LPS) and double-stranded RNA. More recently, this family of pattern recognition receptors has been attributed a critical role in the elicitation of anticancer immune responses, raising interest in the development of immunochemotherapeutic regimens based on natural or synthetic TLR agonists. In spite of such an intense wave of preclinical and clinical investigation, only three TLR agonists are currently licensed by FDA for use in cancer patients: bacillus Calmette-Guérin (BCG), an attenuated strain of Mycobacterium bovis that operates as a mixed TLR2/TLR4 agonist; monophosphoryl lipid A (MPL), a derivative of Salmonella minnesota that functions as a potent agonist of TLR4; and imiquimod, a synthetic imidazoquinoline that activates TLR7. One year ago, in the August and September issues of OncoImmunology, we described the main biological features of TLRs and discussed the progress of clinical studies evaluating the safety and therapeutic potential of TLR agonists in cancer patients. Here, we summarize the latest developments in this exciting area of research, focusing on preclinical studies that have been published during the last 13 mo and clinical trials launched in the same period to investigate the antineoplastic activity of TLR agonists.

RESUMEN

BACKGROUND: Picibanil (OK-432) is a lyophilized mixture of group A Streptococcus pyogenes with antineoplastic activity. Because of its capacity to produce a selective fibrosis of lymphangiomas (LMs), it has been approved by Japanese administration in 1995 for the treatment of LMs. MATERIALS AND METHODS: We treated 15 children (age range: 6-60 months) affected by head and neck macrocystic LMs with intracystic injections (single dose of 0.2 mL) of Picibanil (1-3 injections). RESULTS: Complete disappearance of the lesion was noticed in eight (53.33%) cases, a marked (>50%) reduction of LMs was found five (33.33%) cases, while a moderate (<50%) response was recorded in two (13.33%) cases. Picibanil side effects included fever, local inflammation, and transitory increase of blood platelets' concentration; a single case of anemia was resolved with concentrated red blood cells transfusion. CONCLUSIONS: Intracystic injection of Picibanil is an effective and safe treatment for macrocystic LMs in pediatric patients and may represent the treatment of choice in such cases, especially where surgical excision is associated with the risk of functional/cosmetic side effects.

RESUMEN

Mucocele y ránula, son términos clínicos aplicados para describir un pseudoquiste asociado con extravasación mucosa en los tejidos circundantes, donde se hallen glándulas salivales menores. Estas lesiones ocurren como resultado de un trauma del conducto excretor de laglándula salival o por la obstrucción de flujo salival por otra lesión asociada, como un sialolito o un tapón bacteriano. Estas patologías se originan en una glándula salival menor y son también conocidos como fenómeno de retenciónmucosa o reacción de escape mucoso.El manejo de estas lesiones es quirúrgico en todos los casos, sin embargo, presenta algunas dificultades propias del evento, complicacionespostquirúrgicas y secuelas permanentes en el paciente; en la actualidad se ha manejado este tipo de lesiones con otro tipo de terapéutica, como escleroterapia, uso de toxina Botulínicatipo A, LASER, o el OK-432 (Picibanil), que es una mezcla de Streptococo Pyogenes del grupo A mermado en su virulencia y penicilina G en polvo liofilizado que funciona como agente esclerosante para linfangiomas y agenteantineoplásico. En este trabajo se presenta una revisión bibliográficaacerca de esta patología y una nuevaopción de tratamiento con este medicamento, sus indicaciones y contraindicaciones, así como su administración, reacciones secundarias y complicaciones durante el manejo de lesiones reactivas asociadas a las glándulas salivales menores.

Asunto(s)

Humanos , Mucocele/tratamiento farmacológico , Picibanil/uso terapéutico , Ránula/tratamiento farmacológico , Soluciones Esclerosantes/uso terapéutico , Diagnóstico Diferencial , Complicaciones Posoperatorias

RESUMEN

Fetal chylothorax is a rare congenital manifestation that shows variable clinical outcome ranging from complete spontaneous resolution to progression into hydrops or lung hypoplasia. There is no consensus in the literature as to the optimal antenatal management despite several complications such as preterm delivery, pulmonary hypoplasia, and perinatal death. Pleuroamniotic shunting has been the treatment of choice in fetal chylothorax. Recently, new fetal therapy such as OK-432 (Picibanil) pleurodesis is being introduced. Herein, we present two cases of women referred at early 2nd trimester because of fetal hydrothorax by routine ultrasonography. Cytology obtained by thoracocentesis revealed abundant lymphocytes, suggesting chylothorax. Effusion was aspirated and OK-432 (Picibanil) was injected into the pleural space of fetus. On follow up ultrasonography, the pleural effusion was nearly resolved by adhesion of the intrathoracic space and resulted in the delivery of a healthy neonate. Intrapleural OK-432 injection may be feasible therapeutic option for selected cases in early 2nd trimester with persistent chylothorax for effective control of pleural effusion with no adverse effects.

Asunto(s)

Femenino , Humanos , Recién Nacido , Quilotórax , Consenso , Edema , Terapias Fetales , Feto , Estudios de Seguimiento , Hidrotórax , Pulmón , Linfocitos , Picibanil , Derrame Pleural , Pleurodesia

RESUMEN

undergo radical neck dissection. Subsequent chyle leakage can cause complications such as skin flap necrosis, orocutaneous fistula, electrolyte imbalance and protein loss. Chyle leakage is managed conservatively with total parenteral nutrition and mediumchain triglyceride diet or is treated surgically with leakage site ligation or thoracic duct ligation. Sclerotherapy can be one of the treatment options and tetracycline and povidone-iodine have been reported to be used as sclerosing agents. However, Picibanil sclerotheray for post-neck dissection chyle leakage has not been reported. This paper presents our experience in the management of a intractable chyle leakage which was irresponsive to conservative management and thoracic duct ligation, by successfully employing Picibanil.

Asunto(s)

Quilo , Dieta , Fístula , Ligadura , Cuello , Disección del Cuello , Necrosis , Nutrición Parenteral Total , Picibanil , Povidona Yodada , Soluciones Esclerosantes , Escleroterapia , Piel , Tetraciclina , Conducto Torácico

RESUMEN

BACKGROUND AND OBJECTIVES: Although surgical excision has been considered to be the treatment of choice by most of the surgeons, sclerotherapy of ranula has gained popularity during recent years. A prospective clinical trial was conducted to evaluate the efficacy of Picibanil(R) sclerotherapies for treating ranula children. SUBJECTS AND METHOD: Three patients were enrolled in the study conducted during 2002. All patients were not hospitalized. Picibanil(R) was injected into the lesion. One girl and two boys with ages ranging from 8 to 12 years were treated. RESULTS: Disappearance or marked reduction of the lesion and no recurrence of the ranula were observed in all children. Following each injection, local pain at the injection site was observed in 2 children and transient fever was observed in one child, but such problems resolved within a few days and no serious complication was developed. CONCLUSION: Our experience gives a suggestion that intralesional injection therapy using Picibanil(R) for intraoral ranula is relatively safe and can be used as a substitute for surgery in the treatment of ranula.

Asunto(s)

Niño , Femenino , Humanos , Fiebre , Inyecciones Intralesiones , Picibanil , Estudios Prospectivos , Ránula , Recurrencia , Escleroterapia