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Eplasty 2022. 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...
Long-Term Follow-up With Multispecialty Management of a Giant Lymphangioma of an Infant Tongue Contributed to Reduced Complications of the Disease: A Case Report of a 21-Year Follow-up.
. 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.
PubMed: 36545641
DOI: No ID Found -
Cureus Sep 2022Orbital and periorbital venolymphatic malformations (VLMs) are benign congenital vascular lesions and constitute 1%-3% of all orbital masses. Widespread facial venous...
Orbital and periorbital venolymphatic malformations (VLMs) are benign congenital vascular lesions and constitute 1%-3% of all orbital masses. Widespread facial venous malformations have a high incidence of associated intracranial developmental venous anomalies (DVAs). In such cases, there can be a sudden increase in proptosis following upper respiratory infection or minor trauma. Numerous percutaneous intralesional sclerosing agents like sodium tetradecyl sulfate (STS), bleomycin, doxycycline, ethanol, and OK-432 (Picibanil) have been used for treating VLMs. We hereby report a rare case of retro-orbital VLM treated successfully with STS injection and an isolated dural arterio-venous (AV) fistula in the same patient.
PubMed: 36259024
DOI: 10.7759/cureus.29173 -
Annals of Medicine and Surgery (2012) Sep 2022Congenital cystic lymphangiomas (CCL) or lymphatic malformations (LMs) are benign malformations due to a developmental disorder of lymphatic vessels. Besides surgical...
INTRODUCTION
Congenital cystic lymphangiomas (CCL) or lymphatic malformations (LMs) 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.
METHODS
In a single center retrospective study we analyzed 37 consecutive patients (30 children, 3 adolescents and 4 adults) who were diagnosed with lymphangioma and subsequently treated with OK-432 (Picibanil®) in a general hospital between October 2000 and November 2021.
RESULTS
The median follow-up period was 2.5 months (range 0.7-56.7 months). The lymphangiomas were localized in the head and neck region (n = 25), the thorax/abdomen (n = 6) and extremities (n = 6). The majority of patients had 1 injection with OK-432 (n = 28), five patients had 2 injections, three patients had 3 injections and one patient had more than 3 injections. The most common complications were swelling (89%), fever (81%), redness at the injection site (81%) and pain (73%). The response to therapy was excellent or good in 32 patients (86.4%), 2 patients had a medium response and 3 patients did not show any response. The clinical reaction after the instillation of OK-432 is not predictive for the quality of outcome.
CONCLUSION
The application of Picibanil is safe and without serious side effects. Parents and patients prefer local sclerotherapy versus surgery as it has less complications. We therefore suggest that Picibanil sclerotherapy should be the first-line treatment for macrocystic and mixed type lymphangiomas.
PubMed: 36147081
DOI: 10.1016/j.amsu.2022.104531 -
International Journal of Molecular... Jul 2022Dendritic cells (DC) and leukaemia derived DC (DC) are potent stimulators of anti-leukaemic activity in acute myeloid leukaemia (AML) and can be generated from...
Dendritic cells (DC) and leukaemia derived DC (DC) are potent stimulators of anti-leukaemic activity in acute myeloid leukaemia (AML) and can be generated from mononuclear cells in vitro following standard DC/DC-generating protocols. With respect to future clinical applications though, DC/DC-generating protocols specifically designed for application in a whole-blood-(WB)-environment must be established. Therefore, we developed ten new DC/DC-generating protocols (kits; Kit-A/-C/-D/-E/-F/-G/-H/-I/-K/-M) for the generation of DC/DC from leukaemic WB, containing calcium-ionophore, granulocyte-macrophage-colony-stimulating-factor (GM-CSF), tumour-necrosis-factor-alpha, prostaglandin-E (PGE), prostaglandin-E (PGE) and/or picibanil (OK-432). All protocols were evaluated regarding their performance in generating DC/DC using refined classification and/or ranking systems; DC/DC were evaluated regarding their performance in stimulating anti-leukaemic activity using a cytotoxicity fluorolysis assay. Overall, we found the new kits capable to generate (mature) DC/DC from leukaemic WB. Through refined classification and ranking systems, we were able to select Kit-I (GM-CSF + OK-432), -K (GM-CSF + PGE) and -M (GM-CSF + PGE) as the most efficient kits in generating (mature) DC/DC, which are further competent to stimulate immunoreactive cells to show an improved anti-leukaemic cytotoxicity as well. This great performance of Kit-I, -K and -M in mediating DC/DC-based anti-leukaemic immunity in a WB-environment in vitro constitutes an important and directive step for translating DC/DC-based immunotherapy of AML into clinical application.
Topics: Dendritic Cells; Granulocyte-Macrophage Colony-Stimulating Factor; Humans; Leukemia, Myeloid, Acute; Picibanil; Prostaglandins; Prostaglandins E
PubMed: 35955486
DOI: 10.3390/ijms23158333 -
Cancer Cell International Jun 2022Colorectal cancer (CRC) with pulmonary metastasis usually indicates a poor prognosis, whereas patients may benefit from adoptive cell therapy. Tumor-specific cytotoxic T...
BACKGROUND
Colorectal cancer (CRC) with pulmonary metastasis usually indicates a poor prognosis, whereas patients may benefit from adoptive cell therapy. Tumor-specific cytotoxic T lymphocytes (CTLs) have been reported as a promising treatment for CRC. However, the antitumor effect of CTLs remains limited partially due to insufficient production of effector cells via the activation by antigen-presenting dendritic cells (DCs).
METHOD
This study showed that a combination of CD40 mAb and Picibanil (OK-432) could significantly enhance the activation of CTLs by DCs, both in vitro and in vivo. Flow cytometry, colon cancer mouse model, and pathological staining were employed to demonstrate the specific functions.
RESULTS
This approach promoted the maturation of DCs, augmented the production of stimulatory cytokines, and suppressed the secretion of inhibitory cytokines. Additionally, it facilitated the killing efficiency of CTLs via stimulating their proliferation while restraining the number of Tregs, concomitantly with the positive regulation of corresponding cytokines. Furthermore, the combined unit could hurdle the expansion of tumor cells on metastatic lungs in the colon cancer mouse model.
CONCLUSION
Collectively, the combination of CD40-mAb and OK-432 facilitated the maturation of DCs and enhanced the cytotoxicity of T cells, promising therapeutic approach against CRC.
PubMed: 35715855
DOI: 10.1186/s12935-022-02630-x -
Transfusion Medicine and Hemotherapy :... Feb 2022Myeloid leukaemic blasts can be converted into leukaemia-derived dendritic cells (DC), characterised by the simultaneous expression of dendritic- and...
Interferon Gamma Secretion of Adaptive and Innate Immune Cells as a Parameter to Describe Leukaemia-Derived Dendritic-Cell-Mediated Immune Responses in Acute Myeloid Leukaemia in vitro.
INTRODUCTION
Myeloid leukaemic blasts can be converted into leukaemia-derived dendritic cells (DC), characterised by the simultaneous expression of dendritic- and leukaemia-associated antigens, which have the competence to prime and enhance (leukaemia-specific) immune responses with the whole leukaemic antigen repertoire. To display and further specify dendritic cell (DC)- and DC-mediated immune responses, we analysed the interferon gamma (IFNy) secretion of innate and adaptive immune cells.
METHODS
DC/DC were generated from leukaemic whole blood (WB) with (blast)modulatory Kit-I (granulocyte-macrophage colony-stimulating factor [GM-CSF] + Picibanil [OK-432]) and Kit-M (GM-CSF + prostaglandin E1) and were used to stimulate T cell-enriched immunoreactive cells. Initiated anti-leukaemic cytotoxicity was investigated with a cytotoxicity fluorolysis assay. Initiated IFNy secretion of T, NK, CIK, and iNKT cells was investigated with a cytokine secretion assay (CSA). IFNy positivity was additionally evaluated with an intracellular cytokine assay (ICA). Recent activation of leukaemia-specific cells was verified through addition of leukaemia-associated antigens (LAA; WT-1 and Prame).
RESULTS
We found Kit-I and Kit-M competent to generate mature DC and DC from leukaemic WB without induction of blast proliferation. Stimulation of immunoreactive cells with DC/DC regularly resulted in an increased anti-leukaemic cytotoxicity and increased IFNy secretion of T, NK, and CIK cells, pointing to the significant role of DC/DC in leukaemia-specific alongside anti-leukaemic reactions. Interestingly, an addition of LAA did not further increase IFNy secretion, suggesting an efficient activation of leukaemia-specific cells. Here, both the CSA and ICA yielded comparable frequencies of IFNy-positive cells. Remarkably, the anti-leukaemic cytotoxicity positively correlated with the IFNy secretion in T, T, T, and NK cells.
CONCLUSION
Ultimately, the IFNy secretion of innate and adaptive immune cells appeared to be a suitable parameter to assess and monitor the efficacy of in vitro and potentially in vivo acute myeloid leukaemia immunotherapy. The CSA in this regard proved to be a convenient and reproducible technique to detect and phenotypically characterise IFNy-secreting cells. In respect to our studies on DC-based immunomodulation, we were able to display the potential of DC/DC to induce or improve leukaemia-specific and anti-leukaemic activity.
PubMed: 35221867
DOI: 10.1159/000516886 -
Otolaryngologia Polska = the Polish... Jun 2021<b>Introduction:</b> Recurrent thyroglossal duct cyst after surgery is not a rare condition and first-line treatment has not been established...
<b>Introduction:</b> Recurrent thyroglossal duct cyst after surgery is not a rare condition and first-line treatment has not been established yet.<br/><br/> <b>Aim:</b> Evaluation of outcomes and complications of OK-432 treatment in patients with recurrent thyroglossal duct cyst after surgery. <br/><br/> <b>Material and methods:</b> This study is designed as a case series with planned data collection at Tohoku Medical and Pharmaceutical University and Fukase Clinic. Five patients with recurrent thyroglossal duct cyst after surgery received this therapy between January 2014 and February 2020 on an outpatient basis, without hospitalization. OK-432 solution was injected into the lesion using an 18- or 27-gauge needle, depending on the location and size of the lesion, as well as on possible complications.<br/> <br/> <b>Results:</b> Lesions showed marked reduction or total shrinkage in all patients, with no local scarring or deformity at the injection site. Side effects manifested as local pain at the site of injection and fever (37.5-38.5°C) observed in three patients, but the symptoms resolved within a few days.<br/> <br/> <b>Conclusions:</b> Since OK-432 therapy is simple, easy, safe and effective, it can be used as an alternative to surgery in the treatment of recurrent thyroglossal duct cyst after surgery.
Topics: Child; Humans; Picibanil; Thyroglossal Cyst
PubMed: 35175217
DOI: 10.5604/01.3001.0014.9073 -
Gynecology and Minimally Invasive... 2021This is a case report of a uterine cancer with the International Federation of Gynecology and Obstetrics staging 3c2 with the initial clinical presentation of...
This is a case report of a uterine cancer with the International Federation of Gynecology and Obstetrics staging 3c2 with the initial clinical presentation of postmenopausal vaginal bleeding in August 2015. Endometrium biopsy showed invasive nests of poorly differentiated grade 3 endometrioid adenocarcinoma. The patient received robotic surgery including total hysterectomy, bilateral salpingo-oophorectomy, bilateral pelvic lymph node dissection, para-aortic lymph node dissection, and washing cytology. The final pathology showed an endometrioid carcinoma with myometrium invasion up to 85% and para-aortic and pelvic lymph nodes invasion. The patient received six courses of adjuvant chemotherapy (paclitaxel and carboplatin) with concurrent chemoradiotherapy after the surgery. Later, immunotherapy with Picibanil (OK-432) and interleukin-2 (IL-2) was given, and cancer did not recur for 34 months until tumor recurrence at the liver dome and bilateral lung was noted by positron-emission tomography scan in July 2018. The patient received laparoscopic surgery for intra-abdominal tumor excision in December 2018, and the tumor found extended to the right diaphragm, liver surface, omentum, bilateral flank to pelvic peritoneum, Douglas pouch, and upper rectum. We continued the immunotherapy with OK-432, IL-2, Aldara cream (imiquimod), and later on, virotherapy (human papillomavirus vaccine). The immune risk profiles showed T-cells' proliferation and alteration of the Th1/Th2 activation after immunotherapy and virotherapy. Proctectomy with colon-anal anastomosis and cytoreduction surgery with hyperthermic intraperitoneal chemotherapy (HIPEC) (doxorubicin and paclitaxel) was performed in January 2019. After the surgery, the patient received chemotherapy (topotecan, paclitaxel, lipodox, and carboplatin) and continued the immunotherapy. The immune risk profiles showed CD4, CD4/CD8 increase after HIPEC and immunotherapy. The patient continued the therapy until May 2020.
PubMed: 34485069
DOI: 10.4103/GMIT.GMIT_153_20 -
Scientific Reports Jun 2020Treatment options for metastatic osteosarcoma are limited. The present study aimed to evaluate whether radiofrequency ablation (RFA) combined with intratumoural OK-432...
Treatment options for metastatic osteosarcoma are limited. The present study aimed to evaluate whether radiofrequency ablation (RFA) combined with intratumoural OK-432 injection induces systemic anti-tumour immunity in rat osteosarcoma model. Eighty of 145 rats were assigned to four groups to evaluate overall survival and tumour size: control (no treatment), RFA-only, OK-432, and RFA-OK-432. The remaining 65 were assigned for histological examination. Maximum diameters of tibial and lung tumours were determined. Tumour samples were histologically examined using haematoxylin-eosin and immunohistochemical staining. Overall survival was significantly prolonged in the RFA-OK-432 group compared to the RFA-only and OK-432 groups. Only rats in the RFA-OK-432 group exhibited significant decreases in maximum tumour diameter after treatment. Ki-67-positive tumour cells in the RFA-OK-432 group were significantly stained negative on immunohistochemical analysis as opposed to those in the RFA-only and OK-432 groups. The number of CD11c+, OX-62+, CD4+, and CD8 + cells significantly increased in the RFA-OK-432 group compared to the RFA-only group. RFA with intratumoural OK-432 injection resulted in distant tumour suppression, prolonged survival, and increased dendritic cells, cytotoxic T cells, IFN-γ, and TNF-α, whereas RFA or OK-432 alone did not produce this effect. This combination may induce an abscopal effect in human osteosarcoma.
Topics: Animals; Antineoplastic Agents; Bone Neoplasms; Cell Line, Tumor; Combined Modality Therapy; Osteosarcoma; Picibanil; Radiofrequency Ablation; Rats; Treatment Outcome; Tumor Burden; Xenograft Model Antitumor Assays
PubMed: 32541941
DOI: 10.1038/s41598-020-66934-6 -
Annals of Plastic Surgery Oct 2020This study aimed to determine the benefits of sclerotherapy with OK-432 for the treatment of postoperative chronic lymphocele.
OBJECTIVE
This study aimed to determine the benefits of sclerotherapy with OK-432 for the treatment of postoperative chronic lymphocele.
BACKGROUND
Postoperative chronic lymphocele formation is common and accounts for a high postoperative morbidity. Nonsurgical strategies comprise repetitive percutaneous fluid aspiration or percutaneous sclerotherapy. OK-432 has been used to treat congenital lymphatic malformations with several reports of promising results. We hypothesized that it is more beneficial than repetitive percutaneous fluid aspiration for the treatment of symptomatic lymphocele.
METHODS
Two cohorts of melanoma patients who developed recurrent lymphocele after lymph node dissection from January 2013 to August 2017 were compared. The first cohort was treated with repetitive percutaneous fluid aspiration (n = 20). The second cohort received OK-432 sclerotherapy (n = 20). Primary end points were overall treatment duration, number of treatment sessions, and the clinical success in both cohorts. Secondary end points were surgical site infection rate, need for additional antibiotic treatment, wound healing disorders, and the need for revision surgery.
RESULTS
Mean overall duration of treatment with sclerotherapy was significantly shorter than with repetitive aspiration (9.4 ± 7.2 vs 47.5 ± 31.9 days, P < 0.01). Mean number of sclerotherapy treatment sessions were 2.5 ± 1.2. Clinical success with OK-432 was 19 of 20, and that with repeated aspiration was 7 of 20 (χ = 15.82, P < 0.001). No surgical site infection occurred in the sclerotherapy cohort, which was significantly lower than those treated with repetitive aspiration (P < 0.03). Surgical revision was mandatory in 12 of 20 patients who were treated with repetitive aspiration, and only 1 of 20 patients in the sclerotherapy cohort.
CONCLUSION
Sclerotherapy with OK-432 for the treatment of postoperative lymphocele is highly beneficial with a significant reduction of morbidity and the overall treatment time compared with repetitive aspiration.
Topics: Cohort Studies; Humans; Lymph Node Excision; Lymphocele; Picibanil; Retrospective Studies; Sclerotherapy
PubMed: 32000251
DOI: 10.1097/SAP.0000000000002251