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Pediatrics Sep 2019Lymphatic malformation (LM) is a congenital disorder resulting from an abnormal development of lymphatic vessels. LM may result in problems of cosmesis and functional...
Lymphatic malformation (LM) is a congenital disorder resulting from an abnormal development of lymphatic vessels. LM may result in problems of cosmesis and functional impairment, including airway compression. An 11-year-old girl was referred to our department with increasing dysphagia caused by a large left cervical LM with a long history of treatment. Because of the LM location, surgical resection was not an option, and various therapies, including use of picibanil, had proven ineffective. Celecoxib treatment (100 mg/day) was initiated for local pain management. Softening of the lesion was observed 2 weeks after treatment initiation, and the dose was increased to 200 mg/day with additional shrinking of the LM over the next 2 weeks. With parental consent, celecoxib was continued, with a 65% reduction in volume achieved at 6 months. The patient discontinued treatment at 12 months, and the LM volume increased. Control over the LM was achieved with resumption of celecoxib treatment. After 2 years of treatment, the LM persists, but the size of the malformation is significantly smaller. No adverse effects of celecoxib treatment were observed. The anti-cyclooxygenase-2 effect of celecoxib prevented lymphatic vessel growth through an inhibition of cyclooxygenase-2 activity in the conversion of prostaglandin to prostaglandin E2. In conclusion, celecoxib may be a promising therapeutic agent for LM management.
Topics: Airway Obstruction; Celecoxib; Child; Cyclooxygenase 2 Inhibitors; Deglutition Disorders; Female; Humans; Lymphatic Abnormalities; Magnetic Resonance Imaging; Medication Adherence; Recurrence
PubMed: 31462447
DOI: 10.1542/peds.2019-0319 -
Otology & Neurotology : Official... Sep 2019The aim of this article was to investigate the effectiveness and underlying mechanisms of OK-432 therapy in patients with auricular hematomas.
OBJECTIVES
The aim of this article was to investigate the effectiveness and underlying mechanisms of OK-432 therapy in patients with auricular hematomas.
STUDY DESIGN AND SETTING
Case series with planned data collection.
SUBJECTS AND METHODS
We tried this therapy in 47 patients with auricular hematoma between April 2008 and August 2018. We aspirated as much of the fluid content of each lesion as possible with a 21-gage needle. We injected OK-432 solution into the lesion with the same needle that we used for aspiration. We performed this treatment at an outpatient basis without hospitalization.
RESULTS
Disappearance and marked reduction of the lesion were observed in all patients who had this therapy, and local scarring and deformity of the auricle did not occur in any patients. As adverse effects, local pain at the injection site and fever (37-38.5°C) were observed in some cases of the patients who had this therapy. The concentrations of various cytokines in each aspirate before and after OK-432 therapy were investigated. The production of tumor necrosis factor-α, interleukin-6, interleukin-8, interferon gamma, vascular endothelial growth factor, and periostin was significantly elevated in the aspirate fluid after OK-432 therapy.
CONCLUSION
OK-432 therapy is simple, easy, safe, effective, and can be used as a substitute for surgery in the treatment of auricular hematoma.
Topics: Adolescent; Adult; Aged; Aged, 80 and over; Ear Auricle; Ear Diseases; Female; Hematoma; Humans; Male; Middle Aged; Paracentesis; Picibanil; Retrospective Studies; Treatment Outcome; Young Adult
PubMed: 31348135
DOI: 10.1097/MAO.0000000000002336 -
Acta Oto-laryngologica Sep 2019Although there are studies regarding the efficacy of OK-432 sclerotherapy on thyroglossal duct cyst (TDC), its effects on surgical procedure following this therapy have...
Although there are studies regarding the efficacy of OK-432 sclerotherapy on thyroglossal duct cyst (TDC), its effects on surgical procedure following this therapy have not been properly described. The present study aimed to delineate the prognostic factors of OK-432 sclerotherapy in patients with TDC and investigate its influence on subsequent surgical procedure and the histological characteristics in patients with poor response to OK-432 sclerotherapy. We conducted a retrospective analysis of the medical records of 20 TDC patients treated with OK-432 sclerotherapy. Of the 20 patients, OK-432 sclerotherapy was effective in 5 patients (25.0%). OK-432 showed a lower effective rate in multilocular cysts (9.1%) than in unilocular cysts (44.4%), although not significantly. Five cases were treated with surgery following OK-432 sclerotherapy. There was no significant difference in the operating time and the amount of bleeding between patients with and without OK-432 sclerotherapy. From the results of the histological examination of the cyst wall, two cases had stratified squamous epithelium and two cases showed the absence of lymphocyte infiltration. OK-432 sclerotherapy is an acceptable initial treatment for TDC, especially in unilocular cysts, because of lack of influence on surgical procedure.
Topics: Adult; Cohort Studies; Combined Modality Therapy; Female; Humans; Male; Middle Aged; Picibanil; Prognosis; Retrospective Studies; Sclerotherapy; Severity of Illness Index; Thyroglossal Cyst; Tomography, X-Ray Computed; Treatment Outcome
PubMed: 31271329
DOI: 10.1080/00016489.2019.1633019 -
Current Opinion in Ophthalmology Sep 2019Currently, there is no ideal management for orbital lymphatic malformations. Significant advances have been made since the discovery of new agents in the treatment. The... (Review)
Review
PURPOSE OF REVIEW
Currently, there is no ideal management for orbital lymphatic malformations. Significant advances have been made since the discovery of new agents in the treatment. The purpose of this manuscript is to review the recent evidence on new sclerotherapy agents and systemic medications.
RECENT FINDINGS
Traditional sclerosants are OK-432, sodium tetradecyl sulphate and ethanol. More recent developments are the use of doxycycline, bleomycin, and pingyangmycin. Sirolimus as a systemic medication has revolutionized the medical management of lymphatic malformations. Other oral drugs such as propranolol and sildenafil are controversial. Future treatment involves targeting lymphangiogenic pathways including inhibition of vascular endothelial growth factors and the phosphatidylinositol 4,5-bisphosphate 3-kinase catalytic subunit.
SUMMARY
The development of new agents allows multimodal management either as monotherapy or combined therapy to achieve better outcomes in this difficult to manage disease.
Topics: Angiogenesis Inhibitors; Antineoplastic Agents; Bevacizumab; Bleomycin; Doxycycline; Humans; Immunosuppressive Agents; Lymphangioma; Orbital Neoplasms; Picibanil; Sclerosing Solutions; Sclerotherapy; Sirolimus
PubMed: 31232717
DOI: 10.1097/ICU.0000000000000585 -
Acta Paediatrica (Oslo, Norway : 1992) Aug 2019We assessed the long-term health-related quality of life (HRQoL) of children who received sclerotherapy for lymphatic malformations. This treatment involved injecting...
Long-term health-related quality of life in children with lymphatic malformations treated with sclerotherapy generally matched age-appropriate standardised population norms.
AIM
We assessed the long-term health-related quality of life (HRQoL) of children who received sclerotherapy for lymphatic malformations. This treatment involved injecting drugs into the blood vessels to make them shrink.
METHODS
Our cross-sectional study retrospectively reviewed patients who received OK-432 sclerotherapy injections at Karolinska University Hospital, Stockholm, Sweden, from 1998 to 2013. We studied 49 patients (63% female) aged 8-18 at least five years after their first injection. HRQoL was assessed with the KIDSCREEN-52 questionnaire and a study-specific questionnaire addressed disease consequences and patient satisfaction. We determined associations between HRQoL and disease and treatment and the patient's sex.
RESULTS
Overall HRQoL paralleled age-appropriate norms in the general population, but some subgroups had lower levels. Regression-based estimates showed that larger numbers of injections were negatively associated with HRQoL in the dimensions autonomy, parent relations and home life, financial resources and school environment (p = 0.01-0.03). Malformations in the head and neck area were negative predictors across dimensions and were strongest for psychological well-being (p = 0.009), parent relations and home life (p = 0.017) and school environment (p = 0.006).
CONCLUSION
Despite generally positive outcomes, multiple injections and malformations in the head and neck were associated with impaired HRQoL.
Topics: Adolescent; Antineoplastic Agents; Child; Cross-Sectional Studies; Female; Humans; Lymphatic Abnormalities; Male; Picibanil; Quality of Life; Retrospective Studies; Sclerotherapy
PubMed: 30556934
DOI: 10.1111/apa.14700 -
Brazilian Journal of Otorhinolaryngology 2020
Topics: Airway Obstruction; Head and Neck Neoplasms; Humans; Infant; Lymphangioma; Male; Picibanil; Sclerosing Solutions; Sclerotherapy; Tracheostomy; Treatment Outcome
PubMed: 27297957
DOI: 10.1016/j.bjorl.2016.04.016