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Internal Medicine (Tokyo, Japan) Jun 2018Objective In Japan, pleurodesis is often performed using OK-432. However, OK-432 may cause severe chest pain and fever. The risk factors for these complications are...
Objective In Japan, pleurodesis is often performed using OK-432. However, OK-432 may cause severe chest pain and fever. The risk factors for these complications are unclear. The aim of this study was to identify the risk factors for chest pain and fever caused by pleurodesis with OK-432. Methods The clinical data of 94 patients who underwent pleurodesis with OK-432 were retrospectively analyzed. Patients who developed chest pain (indicated by a record of rescue pain medication) and/or fever (a recorded temperature of >38°C) were identified. A logistic regression analysis was performed to determine the risk factors for these complications. Results Rescue medication for chest pain was required by 43.6% of the patients and 40.4% developed pyrexia after pleurodesis with OK-432. The univariate analysis showed that the likelihood of requiring rescue medication for chest pain was significantly increased in patients of <70 years of age (p=0.028) and in those who were not premedicated with a nonsteroidal anti-inflammatory drug (NSAID; p=0.003). Age <70 years (adjusted odds ratio 2.97, 95% confidence interval 1.10-8.00, p=0.031) and a lack of premedication with an NSAID (adjusted odds ratio 4.21, 95% confidence interval 1.47-12.04, p=0.007) remained significant factors in a multivariate analysis. The absence of NSAID premedication was the only statistically significant risk factor for fever in the univariate analysis (p=0.034). The multivariate analysis revealed no significant risk factors for fever. Conclusion The results of the present study suggest that premedication with an NSAID might be useful for preventing the chest pain caused by pleurodesis with OK-432. Furthermore, caution is advised when managing chest pain in adults of <70 years of age. Prospective studies should be performed to further investigate this issue.
Topics: Adult; Age Factors; Aged; Aged, 80 and over; Anti-Inflammatory Agents, Non-Steroidal; Chest Pain; Female; Fever; Humans; Japan; Male; Middle Aged; Odds Ratio; Picibanil; Pleurodesis; Retrospective Studies; Risk Factors
PubMed: 29434153
DOI: 10.2169/internalmedicine.9637-17 -
Medicine Nov 2017Linear nevus sebaceous syndrome (LNSS) is a rare neurocutaneous syndrome, characterized by nevus sebaceous,central nervous system (CNS), ocular and skeletal...
RATIONALE
Linear nevus sebaceous syndrome (LNSS) is a rare neurocutaneous syndrome, characterized by nevus sebaceous,central nervous system (CNS), ocular and skeletal abnormalities. The present study describes KRAS somatic mosaic mutation in a case of LNSS with lymphatic malformations (LMs).
PATIENT CONCERNS
A 4-month-old female with a clinical diagnosis of LNSS presented with infantile spasms, mental retardation, skull dysplasia, ocular abnormalities, congenital atrial septal defect, and LMs.
DIAGNOSIS
Cervical ultrasonography revealed a 4.6 × 4.6 × 2.2cm no echo packet with clear boundary in the subcutaneous tissues of the right neck. The neck MRI indicated a cyst in the subcutaneous tissues of the right neck. Whole-exome sequencing revealed a low-level heterozygous mutation of the KRAS gene (c.35C > T; p.G12D, 19%) in the skin lesion sample. This mutation was not present in the blood samples of the patient and her parents.
INTERVENTIONS
The patient received sclerotherapy with paicibanil (OK-432) injection for the cyst.
OUTCOMES
Following 1 year of treatment, the patient exhibited fewer seizures. The mental and motor development was significantly improved. The patient can currently walk with assistance and speak simple words.
LESSONS
LNSS is a rare, congenital neurocutaneous syndrome consisting of a spectrum of abnormalities involving the skin, central nervous system, eyes, LMs and other systems. LNSS can be caused by postzygotic somatic mutation in the RAS family of genes. Multidisciplinary evaluation and treatment is needed.
Topics: Antineoplastic Agents; Female; Humans; Infant; Nevus, Sebaceous of Jadassohn; Picibanil; Sclerotherapy
PubMed: 29381910
DOI: 10.1097/MD.0000000000008016 -
Respirology (Carlton, Vic.) Jan 2018Talc pleurodesis is commonly performed to manage refractory pleural effusion or pneumothorax. It is considered as a safe procedure as long as a limited amount of large...
BACKGROUND AND OBJECTIVE
Talc pleurodesis is commonly performed to manage refractory pleural effusion or pneumothorax. It is considered as a safe procedure as long as a limited amount of large particle size talc is used. However, acute respiratory distress syndrome (ARDS) is a rare but serious complication after talc pleurodesis. We sought to determine the risk factors for the development of ARDS after pleurodesis using a limited amount of large particle size talc.
METHODS
We retrospectively reviewed patients who underwent pleurodesis with talc or OK-432 at the University of Tokyo Hospital.
RESULTS
Twenty-seven and 35 patients underwent chemical pleurodesis using large particle size talc (4 g or less) or OK-432, respectively. Four of 27 (15%) patients developed ARDS after talc pleurodesis. Patients who developed ARDS were significantly older than those who did not (median 80 vs 66 years, P = 0.02) and had a higher prevalence of underlying interstitial abnormalities on chest computed tomography (CT; 2/4 vs 1/23, P < 0.05). No patient developed ARDS after pleurodesis with OK-432. This is the first case series of ARDS after pleurodesis using a limited amount of large particle size talc.
CONCLUSION
Older age and underlying interstitial abnormalities on chest CT seem to be risk factors for developing ARDS after talc pleurodesis.
Topics: Adult; Age Factors; Aged; Aged, 80 and over; Female; Humans; Lung Diseases, Interstitial; Male; Middle Aged; Particle Size; Picibanil; Pleural Effusion; Pleurodesis; Pneumothorax; Respiratory Distress Syndrome; Retrospective Studies; Risk Factors; Talc; Tomography, X-Ray Computed
PubMed: 28980363
DOI: 10.1111/resp.13192 -
The Journal of Craniofacial Surgery Oct 2017Herein, the authors aim to describe their findings of novel architectural types of lymphatic malformations (LM) and explain the relationship between these architectures...
PURPOSE
Herein, the authors aim to describe their findings of novel architectural types of lymphatic malformations (LM) and explain the relationship between these architectures and OK-432 treatment outcomes.
METHODS
A retrospective review was conducted of all patients diagnosed with a LM treated with OK-432 at the Vascular Anomalies Clinic at BC Children's Hospital from December 2002 to January 2012.
RESULTS
Twenty-seven patients were included in the study. Sixty percent of lesions were present by 2 years of age with the majority located in the head and neck (59%). The average number of sclerotherapy procedures was 1.4 per patient. Treatment under fluoroscopic guidance revealed 3 new LM architectures: open-cell microcystic, closed-cell microcystic, and lymphatic channel. Response to treatment was complete or good for 14/19 macrocystic and for 1/2 mixed lesions. Open-cell microcystic LMs gave a complete or good response for 3/3, which was attributed to OK-432 freely communicating between cysts. Closed-cell microcystic LM had localized cysts that did not allow OK-432 to freely communicate and were associated with partial responses, 2/2. The lymphatic channel had a partial response. There were 2 minor complications and 1 instance of recurrence.
CONCLUSIONS
The identification of 3 new LM architectures expands the current accepted classification to include: open-cell microcystic, closed-cell microcystic, and lymphatic channels. The majority of complete responses to OK-432 were found with macrocystic lesions. Open-cell microcystic lesions respond better to OK-432 than closed-cell microcystic lesions, and lymphatic channels may respond to OK-432. These key architecture-response relationships have direct clinical implications for treatment with OK-432 sclerotherapy.
Topics: Cysts; Fluoroscopy; Head; Humans; Lymphatic Abnormalities; Neck; Picibanil; Retrospective Studies; Sclerotherapy; Treatment Outcome
PubMed: 28834841
DOI: 10.1097/SCS.0000000000003789 -
Oral and Maxillofacial Surgery Sep 2017Lymphatic malformations are rare slow-flow vascular malformations, with high tendency to appear in the head and neck region. The treatment of these lesions ranges from...
Lymphatic malformations are rare slow-flow vascular malformations, with high tendency to appear in the head and neck region. The treatment of these lesions ranges from follow-up to sclerosing agent injection to surgical excision. The authors present a case of a new born with large extensive lingual and submandibular lymphatic malformation, for which the patient underwent tracheostomy and gastrostomy insertion. He was then treated successfully with sclerosing agent injections followed by surgical excision, with 7 years follow-up. The second case presented is a two and a half baby with large lingual lymphatic malformation, treated successfully with doxycycline injections followed by intraoral excision of the lesion. Pathology of the excised lesion is then demonstrated, which shows for the first time the different layers affected by the sclerosing agent.
Topics: Child; Child, Preschool; Combined Modality Therapy; Doxycycline; Follow-Up Studies; Humans; Imaging, Three-Dimensional; Infant; Infant, Newborn; Lymphatic Abnormalities; Lymphatic Vessels; Macroglossia; Magnetic Resonance Imaging; Male; Picibanil; Sclerotherapy; Tomography, X-Ray Computed; Tongue; Tracheostomy
PubMed: 28735345
DOI: 10.1007/s10006-017-0640-3 -
Internal Medicine (Tokyo, Japan) 2017Objective Pleurodesis is an effective therapy for malignant pleural effusion (MPE). While interstitial lung disease (ILD) has been regarded as a serious complication of...
Objective Pleurodesis is an effective therapy for malignant pleural effusion (MPE). While interstitial lung disease (ILD) has been regarded as a serious complication of pleurodesis, its clinicopathological characteristics have not been fully understood. This study was conducted to elucidate the incidence of ILD and the risk factors for ILD in patients who underwent pleurodesis to control MPE. Methods The medical records of patients who underwent pleurodesis in Aichi Medical University between March 2008 and February 2013, the period before the approval of talc in Japan, were retrospectively analyzed. Results A total of 84 patients underwent pleurodesis, all using OK-432. ILD occurred in 13 patients (15.5%). The development of ILD after pleurodesis was significantly associated with old age (odds ratio [OR]: 4.82, 95% confidence interval [CI]: 1.22-19.08) and epidermal growth factor receptor-tyrosine kinase inhibitor (EGFR-TKI) treatment (OR: 5.97, CI: 1.7-20.9). A multivariate analysis revealed that >67 years of age (p=0.01) and EGFR-TKI treatment (p=0.02) were significantly associated with the development of pleurodesis-related ILD. Among the patients who received both pleurodesis and EGFR-TKIs (n=23), 8 patients developed ILD. All of these patients were receiving EGFR-TKI therapy at the time of pleurodesis or within 30 days after pleurodesis. In contrast, no cases of ILD were observed among the patients who stopped EGFR-TKIs before pleurodesis or started EGFR-TKIs at more than 30 days after pleurodesis. Conclusion ILD seemed to be a frequent complication of pleurodesis in patients using OK-432, especially elderly patients and those who underwent pleurodesis while receiving EGFR-TKI therapy or who started EGFR-TKI therapy within 30 days after pleurodesis.
Topics: Adult; Aged; Aged, 80 and over; Antineoplastic Agents; Female; Humans; Japan; Lung Diseases, Interstitial; Male; Middle Aged; Odds Ratio; Picibanil; Pleural Effusion, Malignant; Pleurodesis; Retrospective Studies; Risk Factors
PubMed: 28717073
DOI: 10.2169/internalmedicine.56.7464 -
BMC Medical Imaging Jul 2017Congenital cystic lymphangiomas are benign malformations due to a developmental disorder of lymphatic vessels. Besides surgical excision, sclerosant therapy of these...
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.
Topics: Contrast Media; Humans; Infant; Lymphangioma; Male; Microbubbles; Picibanil; Sclerosing Solutions; Sclerotherapy; Treatment Outcome; Ultrasonography
PubMed: 28683719
DOI: 10.1186/s12880-017-0213-9 -
The Laryngoscope Oct 2017Evidence supporting any one treatment for plunging ranula is limited. Standard treatment-complete excision of the sublingual gland and ranula-is invasive and morbid...
Evidence supporting any one treatment for plunging ranula is limited. Standard treatment-complete excision of the sublingual gland and ranula-is invasive and morbid given the close operative proximity to the submandibular duct and lingual nerve. OK-432 (Picibanil; Chugai Pharmaceutical Co., Tokyo, Japan) sclerotherapy has been studied as a less invasive treatment but is inaccessible in the United States. This report illustrates the successful management of a plunging ranula using ultrasound-guided percutaneous ethanol injection. Within 2 months of the procedure, the patient had complete resolution of the plunging ranula, with no associated side effects. We propose that ultrasound-guided percutaneous ethanol injection be considered for the management of plunging ranula. Laryngoscope, 127:2239-2241, 2017.
Topics: Ablation Techniques; Aged, 80 and over; Ethanol; Humans; Injections, Subcutaneous; Male; Ranula; Sclerotherapy; Ultrasonography, Interventional
PubMed: 28407263
DOI: 10.1002/lary.26505 -
Cancer May 2017There are limited options for the curative treatment of refractory bone and soft tissue sarcomas. The purpose of this phase 1/2 study was to assess the immunological and...
BACKGROUND
There are limited options for the curative treatment of refractory bone and soft tissue sarcomas. The purpose of this phase 1/2 study was to assess the immunological and clinical effects of dendritic cells (DCs) pulsed with autologous tumor lysate (TL) in patients with advanced bone and soft tissue sarcomas.
METHODS
Thirty-seven patients with metastatic or recurrent sarcomas were enrolled in this study. Peripheral blood mononuclear cells obtained from the patients were suspended in media containing interleukin 4 (IL-4) and granulocyte-macrophage colony-stimulating factor. Subsequently, these cells were treated with TL, tumor necrosis factor α, and OK-432. The DCs were injected into the inguinal or axillary region. One treatment course comprised 6 weekly DC injections. The toxicity, clinical response (tumor volume, serum interferon-γ [IFN-γ], and serum IL-12), and oncological outcomes were observed.
RESULTS
In total, 47 courses of DC therapy were performed in 37 patients. No severe adverse events or deaths associated with the DC injections were observed in the study patients. Increased serum IFN-γ and IL-12 levels were observed 1 month after the DC injection. Among the 37 patients, 35 patients were assessed for clinical responses: 28 patients showed tumor progression, 6 patients had stable disease, and 1 patient showed a partial response 8 weeks after the DC injection. The 3-year overall and progression-free survival rates of the patients were 42.3% and 2.9%, respectively.
CONCLUSIONS
Although DC therapy appears safe and resulted in an immunological response in patients with refractory sarcoma, it resulted in an improvement of the clinical outcome in only a small number of patients. Cancer 2017;123:1576-1584. © 2017 American Cancer Society.
Topics: Adolescent; Adult; Aged; Antineoplastic Agents; Bone Neoplasms; Child; Chondrosarcoma; Dendritic Cells; Disease-Free Survival; Female; Granulocyte-Macrophage Colony-Stimulating Factor; Histiocytoma, Malignant Fibrous; Humans; Immunotherapy; Interferon-gamma; Interleukin-12; Interleukin-4; Leiomyosarcoma; Leukocytes, Mononuclear; Male; Middle Aged; Osteosarcoma; Picibanil; Sarcoma; Sarcoma, Clear Cell; Sarcoma, Synovial; Soft Tissue Neoplasms; Treatment Outcome; Tumor Necrosis Factor-alpha; Young Adult
PubMed: 28241093
DOI: 10.1002/cncr.30606 -
Journal of Vascular and Interventional... Mar 2017
Spontaneous Regression of Untreated Tumors with Immuno-Radiofrequency Ablation, RF Ablation in Combination with Local Injection of OK-432, in a Patient with Lung Metastases of Colon Cancer.
Topics: Aged; Antineoplastic Agents; Catheter Ablation; Colonic Neoplasms; Combined Modality Therapy; Female; Humans; Injections, Intralesional; Liver Neoplasms; Lung Neoplasms; Picibanil; Radio Waves; Remission, Spontaneous; Reoperation; Tomography, X-Ray Computed
PubMed: 28231930
DOI: 10.1016/j.jvir.2016.11.092