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Current Opinion in Pediatrics Jun 2015To review the literature on lymphatic malformations and to provide current opinion about the management of these lesions. (Review)
Review
PURPOSE OF REVIEW
To review the literature on lymphatic malformations and to provide current opinion about the management of these lesions.
RECENT FINDINGS
Current treatment options include nonoperative management, surgery, sclerotherapy, radiofrequency ablation, and laser therapy. New therapies are emerging, including sildenafil, propranolol, sirolimus, and vascularized lymph node transfer. The primary focus of management centers on the patient's quality of life.
SUMMARY
Multimodal treatment of lymphatic malformations continues to expand as new information about the biology and genetics of these lesions is discovered, in addition to knowledge gained from clinical practice. A patient-centered approach should guide timing and modality of treatment. Continued study of lymphatic malformations will increase and solidify a treatment algorithm for these complicated lesions.
Topics: Antineoplastic Agents; Child; Humans; Laser Therapy; Lymphangioma; Lymphatic Abnormalities; Picibanil; Practice Guidelines as Topic; Quality of Life; Sclerotherapy; Treatment Outcome
PubMed: 25888145
DOI: 10.1097/MOP.0000000000000209 -
Otolaryngology--head and Neck Surgery :... Oct 1999Picibanil (OK-432) is a sclerosing agent derived from a low-virulence strain of Streptococcus pyogenes that induces regression of macrocystic lymphangiomas. This report... (Clinical Trial)
Clinical Trial
Picibanil (OK-432) is a sclerosing agent derived from a low-virulence strain of Streptococcus pyogenes that induces regression of macrocystic lymphangiomas. This report describes a prospective, nonrandomized trial to evaluate the efficacy of Picibanil in the treatment of 13 affected children ranging in age from 1 to 94 months. On average, 4.1 fluoroscopically guided intracystic injections were performed per child, with an average total dose of 0.56 mg of Picibanil. As judged by physical examination and radiographic studies, 5 children (42%) showed a complete or substantial response, and 2 children (16%) showed an intermediate response. No response was seen in 5 children (42%), 2 of whom had massive craniofacial lymphangioma. Factors that contribute to failure with Picibanil sclerotherapy are the presence of a significant microcystic component to the lesion, massive craniofacial involvement, and previous surgical resection. Macrocystic lymphangiomas of the infratemporal fossa or cervical area have the best response to therapy.
Topics: Adolescent; Antineoplastic Agents; Child; Child, Preschool; Facial Neoplasms; Female; Follow-Up Studies; Humans; Infant; Injections, Intralesional; Lymphangioma; Magnetic Resonance Imaging; Male; Otorhinolaryngologic Neoplasms; Picibanil; Prospective Studies; Sclerotherapy; Treatment Outcome
PubMed: 10504592
DOI: 10.1016/S0194-5998(99)70225-1 -
Ultrasound in Obstetrics & Gynecology :... Dec 2005
Review
Topics: Chylothorax; Fetal Diseases; Humans; Hydrops Fetalis; Picibanil; Pleurodesis
PubMed: 16273598
DOI: 10.1002/uog.2634 -
Archives of Otolaryngology--head & Neck... Oct 2002To describe and to determine the robustness of our study evaluating the efficacy of OK-432 (Picibanil) as a therapeutic modality for lymphangiomas. (Clinical Trial)
Clinical Trial Randomized Controlled Trial
OBJECTIVE
To describe and to determine the robustness of our study evaluating the efficacy of OK-432 (Picibanil) as a therapeutic modality for lymphangiomas.
DESIGN AND SETTING
Prospective, randomized trial and parallel-case series at 13 US tertiary care referral centers.
SUBJECTS
Thirty patients diagnosed as having lymphangioma. Ages in 25 ranged from 6 months to 18 years. Twenty-nine had lesions located in the head-and-neck area.
INTERVENTION
Every patient received a 4-dose injection series of OK-432 scheduled 6 to 8 weeks apart unless a contraindication existed or a complete response was observed before completion of all injections. A control group was observed for 6 months.
OUTCOME MEASURES
Successful outcome of therapy was defined as a complete or a substantial (>60%) reduction in lymphangioma size as determined by calculated lesion volumes on computed tomographic or magnetic resonance imaging scans.
RESULTS
Overall, 19 (86%) of the 22 patients with predominantly macrocystic lymphangiomas had a successful outcome.
CONCLUSIONS
OK-432 should be efficacious in the treatment of lymphangiomas. Our study design is well structured to clearly define the role of this treatment agent.
Topics: Adolescent; Adult; Aged; Aged, 80 and over; Antineoplastic Agents; Child; Child, Preschool; Female; Head and Neck Neoplasms; Humans; Infant; Lymphangioma; Magnetic Resonance Imaging; Male; Middle Aged; Picibanil; Prospective Studies; Sclerotherapy; Tomography, X-Ray Computed; Treatment Outcome
PubMed: 12365884
DOI: 10.1001/archotol.128.10.1137 -
International Immunopharmacology Jan 2008OK-432 (Picibanil), a Streptococcal immunotherapeutic agent, has been used for immunotherapy of various cancers as a biological response modifier (BRM). However, OK-432...
OK-432 (Picibanil), a Streptococcal immunotherapeutic agent, has been used for immunotherapy of various cancers as a biological response modifier (BRM). However, OK-432 contains multiple components consisting of immunotherapeutic ones and contaminants which may weaken the effects or exert side-effects. In this study, we investigated extraction of contaminants from OK-432 using Triton X-114 (TX-114)-water phase partitioning and examined an antitumor effect of the resulting preparation. OK-432 was subjected to TX-114 partitioning to give residual precipitate designated as OK-TX-ppt. OK-TX-ppt exerted no TLR2-mediated activity, but induced interleukin (IL)-6 in human PBMC. OK-TX-ppt also induced tumor necrosis factor (TNF)-alpha, IL-10, IL-12, and interferon (IFN)-gamma in PBMC. Moreover, IFN-gamma-inducing activity of OK-TX-ppt was significantly higher and IL-10 production was lower than that of OK-432. In tumor-bearing mice model, administration of OK-TX-ppt i.p. extended the survival time of Meth-A-bearing mice compared to OK-432. OK-TX-ppt also increased the levels of IL-12 and IFN-gamma in mouse spleen cells in vitro. These results indicated that TX-114 partitioning removed some contaminants, which attenuates the antitumor effect, from OK-432 and increase the immunotherapeutic effects of OK-432.
Topics: Adjuvants, Pharmaceutic; Animals; Antineoplastic Agents; Cell Line, Tumor; Cells, Cultured; Humans; Male; Mice; Mice, Inbred BALB C; Neoplasms, Experimental; Octoxynol; Picibanil; Polyethylene Glycols
PubMed: 18068095
DOI: 10.1016/j.intimp.2007.09.021 -
Klinische Padiatrie 2008The therapeutic gold standard of cystic hygroma is its complete resection. Because of its growth pattern and its main location in the head and neck region complete...
BACKGROUND
The therapeutic gold standard of cystic hygroma is its complete resection. Because of its growth pattern and its main location in the head and neck region complete resection is not always possible. An alternative is the local injection of Picibanil, but only few cases have been published about its use in infants.
PATIENTS/METHOD
We retrospectively analyzed the data of 8 infants (age: 2 weeks-12 months) who got Picibanil therapy because of cystic hygroma in the time period 2002 until 2006. Follow up ranged from 3 months up to 3 years.
RESULTS
During the postoperative period all patients had local swelling, in 4 cases accompanied with local inflammation and fever. Tumor reduction of >50% was obtained in 7 of 8 patients.
CONCLUSIONS
Local injection of Picibanil in infants with cystic hygroma seems to be a safe alternative to surgical therapy, especially when complete tumor resection means damage of important neighbouring structures. Prospective trials are necessary to confirm the better outcome after therapy with Picibanil compare to primary surgery.
Topics: Antineoplastic Agents; Chemotherapy, Adjuvant; Chylothorax; Combined Modality Therapy; Female; Head and Neck Neoplasms; Humans; Infant; Infant, Newborn; Injections, Intralesional; Lymphangioma, Cystic; Male; Neoplasm Recurrence, Local; Picibanil; Postoperative Complications; Retrospective Studies
PubMed: 18654945
DOI: 10.1055/s-2008-1073158 -
The British Journal of Ophthalmology Jan 2000
Topics: Adult; Female; Humans; Lymphangioma; Orbital Neoplasms; Picibanil; Sclerosing Solutions; Sclerotherapy; Treatment Failure
PubMed: 10611113
DOI: 10.1136/bjo.84.1.124b -
Oncology Reports Feb 2005We investigated the potent inhibitory effects of OK-432 (Picibanil) on both cellular adhesion and cell proliferation of estrogen-dependent (MCF-7) or...
We investigated the potent inhibitory effects of OK-432 (Picibanil) on both cellular adhesion and cell proliferation of estrogen-dependent (MCF-7) or estrogen-independent (MDA-MB-231) breast carcinoma cells. Cellular proliferation of both MCF-7 and MDA-MB-231 cells was markedly inhibited in a dose-dependent manner, when the carcinoma cells were exposed to OK-432. Cell attachment assay demonstrated that incubation with OK-432 for 24 h reduced integrin-mediated cellular adhesion of both cell types. However, fluorescence activated cell sorter (FACS) analysis revealed that incubation with OK-432 for 24 h did not decrease the cell surface expressions of any integrins. These results suggest that the binding avidity of integrins is reduced by OK-432 without alteration of the integrin expression. We conclude that OK-432 inhibits integrin-mediated cellular adhesion as well as cell proliferation of breast carcinoma cells regardless of estrogen-dependence, and that these actions of OK-432 contribute to prevention or inhibition of breast carcinoma invasion and metastasis.
Topics: Antineoplastic Agents; Breast Neoplasms; Cell Adhesion; Cell Proliferation; Estrogens; Female; Humans; Neoplasms, Hormone-Dependent; Picibanil; Tumor Cells, Cultured
PubMed: 15643513
DOI: No ID Found -
Medicina Oral, Patologia Oral Y Cirugia... 2005Cervical cystic lymphangioma (CCL) is a rare and benign tumour involving congenital and cystic abnormalities derived from lymphatic vessels. The most accepted treatment...
Cervical cystic lymphangioma (CCL) is a rare and benign tumour involving congenital and cystic abnormalities derived from lymphatic vessels. The most accepted treatment continues to be surgical excision. However, when this infiltrates vital neurovascular neck structures, complete excision is difficult and if only partial, the recurrence rate is very high. The most frequently used alternative treatment is to inject sclerosants into the lesion. The use of these techniques has reported good results in children; however, there are few references thereof with regard to adults. We are reporting on a cervical cystic lymphangioma in a male aged 22, treated with an intra-lesion injection of 20 cc with 0.01 mg/cc dilution of OK-432 (picibanil) in physiological serum. Sole complications were fever and local reaction where the solution was injected. One month after treatment the lymphangioma had totally remitted and sixteen months later continues in remittance.
Topics: Adult; Antineoplastic Agents; Head and Neck Neoplasms; Humans; Lymphangioma, Cystic; Male; Picibanil; Remission Induction; Sclerosing Solutions
PubMed: 16056191
DOI: No ID Found -
Otolaryngology--head and Neck Surgery :... Apr 2008Systematically review the published literature regarding the efficacy of nonsurgical therapies in the treatment of head and neck (H&N) lymphatic malformations (LM) in... (Review)
Review
OBJECTIVE
Systematically review the published literature regarding the efficacy of nonsurgical therapies in the treatment of head and neck (H&N) lymphatic malformations (LM) in children.
DATA SOURCE
MEDLINE.
REVIEW METHODS
MEDLINE was searched for literature relating to nonsurgical treatments for H&N LM.
RESULTS
The initial search returned 1876 articles, with 22 meeting criteria. The majority (20) were case series. All therapies were percutaneous, with OK-432 or bleomycin sclerotherapy being most common. Random-effects modeling revealed 43% (CI = 28.9%-57%) of patients undergoing OK-432 for LM achieved a complete/excellent response, 23.5% (CI = 5.8%-41.3%) achieved a good response, 16.9% (CI = 10.3%-23.4%) achieved a fair/poor response, and 15.4% (CI = 8.6%-22.2%) observed no response. In the bleomycin group, the results were: 35.2% (CI = 15.7%-54.6%) excellent, 37.1% (CI = 22%-52.3%) good, 18.4% (CI = 2.7%-34.2%) fair/poor, and 11.6% (CI = 3.5%-19.6%) no response. Seven major complications were noted out of the 289 patients in the series, including two mortalities.
CONCLUSIONS
The literature indicates that sclerotherapy for H&N LM achieves excellent/good clinical response in a majority of patients, with few complications, and anecdotally does not complicate future surgery.
Topics: Antibiotics, Antineoplastic; Bleomycin; Humans; Lymphangioma; Picibanil; Sclerotherapy; Treatment Outcome
PubMed: 18359347
DOI: 10.1016/j.otohns.2007.11.018