picibanil and Thoracic-Neoplasms

Lymphangiomas are rare congenital malformations of the lymphatic system. They typically appear within the first two years of life. They present as soft, indolent and benign tumors. As they have a tendency to grow in an infiltrating manner and rarely regress spontaneously, treatment may be necessary if the lymphangioma's location is functionally and cosmetically disturbing. Surgical therapy, which has been the treatment of choice so far, includes a risk of complications, depending on the location and extent of the tumor. Therefore alternative treatments such as sclerotherapy, especially with OK-432, have been developed. We present the case of a female baby with a huge macrocystic lymphangioma, extending over one half of the trunk, located intra- and extrathoracically, which was treated successfully by surgical resection and several intralesional injections of OK-432.

Topics: Female; Humans; Infant; Lymphangioma; Picibanil; Remission Induction; Sclerotherapy; Thoracic Neoplasms

The treatment of massive osteolysis with lymphangioma and/or hemangioma (Gorham-Stout syndrome) has been controversial. The authors report on a patient with multiple massive osteolyses and extensive lymph-hemangiomatosis whose lesions were reduced by interferon alfa therapy. A 2-year-old girl had complained of left chylothorax. Thoracoscopy showed an increase in small lymphatic vessels in the chest wall. The chylothorax was improved by coagulation of the lymphatic vessels. Later, multiple massive osteolyses appeared in the left 11th and 12th ribs, the TH10-L3 vertebrae, and the right femur. There were also hemangiomas in the liver and spleen, a tumor lesion in the left lower chest wall, and hemangiomatous change on the skin surface of the left back. The left lung had only a minimal air content. After OK-432 was injected into the femur and chest wall lesions, the femur lesion disappeared. Then, as right chylothorax appeared, OK-432 was injected into the right pulmonary cavity. The chylothorax disappeared, but pericardial effusion appeared. After steroid pulse therapy, pericardial effusion disappeared. During these treatments, the 7th to 10th ribs disappeared from the x-ray and scoliosis developed. One month later, a cloudy fluid collection in the right lung was found on computed tomography. Interferon alfa and steroid pulse therapy were started. Interferon alfa (1,500,000 units) was subcutaneously administered daily for 2 months and was gradually reduced and maintained at 1,500,000 unit/wk. Steroids were also reduced and maintained at 5 mg/d of predonine. Later, the progress of osteolysis and the extension of lymph-hemangiomatosis stopped. Ten months later, hemangioma in the back disappeared, and the 7th to 10th ribs, which had disappeared, reappeared. The interferon alfa therapy was stopped 14 months after it was administered. The patient's condition has been stable for 10 months since then. At this time, computed tomography shows regression of the hemangiomatous lesion in the back. The authors clinically diagnosed the patient as having Gorham-Stout syndrome with extension of lymph-hemangiomatosis. Interferon alfa with or without steroid therapy should be a choice for patients with extension lesions.

Topics: Adrenal Cortex Hormones; Angiogenesis Inhibitors; Antineoplastic Agents; Child, Preschool; Chylothorax; Drug Therapy, Combination; Female; Femur; Hemangioma; Humans; Interferon-alpha; Liver Neoplasms; Lymphangioma; Neoplasms, Multiple Primary; Osteolysis, Essential; Picibanil; Pleural Effusion; Pulmonary Atelectasis; Remission Induction; Ribs; Scoliosis; Skin Neoplasms; Spine; Splenic Neoplasms; Syndrome; Thoracic Neoplasms

Between April 1988 and July 1995, 11 children with lymphangioma were treated with intralesional OK-432 injection. In 7 patients it was the primary therapy and total shrinkage of the lesion was obtained in 5 of them. Two patients did not respond and the children underwent surgery. Following incomplete surgical removal or recurrence of the lymphangioma, intralesional OK-432 injection was used as secondary therapy in 4 patients. Total regression was observed in 2 cases and marked regression in the 2 others. No serious side-effects except fever lasting for 2-3 days and slight tenderness with swelling of the lymphangioma for 3-4 days after the injection was noted. Local inflammatory reaction did not cause any damage to the overlying skin and did not lead to scar formation. Depending on the size, location, and anatomical relationship of the airway, intralesional injections of the lymphangiomas were performed under general anaesthesia and the children were observed for 24 h. There was no recurrence after follow up periods ranging from 2 months to 7 years.. Intralesional injection OK-432 represents an alternative, safe and effective treatment for lymphangiomas. It can be used as the primary therapy, after partial surgical excision, or in recurrent lymphangiomas.

Topics: Antineoplastic Agents; Child; Child, Preschool; Facial Neoplasms; Female; Follow-Up Studies; Head and Neck Neoplasms; Humans; Infant; Lymphangioma; Neoplasm Recurrence, Local; Picibanil; Sclerosing Solutions; Thoracic Neoplasms

A 61-year-old female was admitted to our hospital because of a mass in right lateral chest wall and chest pain. Chest X-P and CT scans showed a right chest wall tumor and pleural effusion. Biopsy specimen from the chest wall tumor revealed an adenocarcinoma, not a mesothelioma, based on immunohistochemical study. Cancer cells were also detected in pleural effusion. Imaging diagnostic analysis could detect no primary tumor in lung field or other organs. High levels of CA125 were noticed: 9,610 U/ml in serum and 37,600 U/ml in pleural effusion, respectively. Finally, there was a possibility that the chest wall tumor might be a metastatic lesion from undetected ovarian cancers, so three cycles of combined chemotherapy (CDDP+ADM+CPA) were done. CDDP plus OK-432 was also injected two times intrapleurally. After chemotherapy, the chest wall tumor and effusion disappeared and the serum CA125 level decreased to the normal range.

Topics: Adenocarcinoma; Antineoplastic Combined Chemotherapy Protocols; CA-125 Antigen; Cisplatin; Cyclophosphamide; Doxorubicin; Drug Administration Schedule; Female; Humans; Middle Aged; Picibanil; Remission Induction; Thoracic Neoplasms

Giant cavernous haemangioma was treated successfully with a new therapy consisting of intralesional injection of OK-432 (group A Streptococcus pyogenes of human origin). Complete regression was observed within 3 months without serious side effects except for fever of 2-3 days' duration and local inflammatory reaction lasting for 3-4 days. Local inflammatory reaction did not cause any damage to the overlaying skin and did not lead to scar formation.

Topics: Biological Products; Dose-Response Relationship, Drug; Female; Hemangioma, Cavernous; Humans; Infant; Injections; Picibanil; Skin Neoplasms; Thoracic Neoplasms