ABSTRACT
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.
ABSTRACT
INTRODUCTION: Testicular neoplasms that are derived from connective tissue, blood vessels and musculature are uncommon and intra-testicular tumors of vascular origin are extremely rare; both are benign in nature. Testicular hemangioma is exceedingly rare and typically occurs in patients younger than 20 years, the age in which a primary germ cell tumor of the testis may present, necessitating a radical approach to management with orchidectomy, although potential conservative focal partial surgical excision is desirable. Hemangiomas of the testis have a similar sonographic and magnetic resonance imaging appearance to that of malignant tumors of the testis, especially seminoma. The work has been reported in line with the SCARE criteria. PRESENTATION OF CASE: We present a case of testicular torsion in a 15-year-old male patient who had a painful left testis for 6 days, no vomiting, no fever or dysuria. With clinical suspicion of an old testicular torsion the patient was examined by ultrasound which confirmed the clinical diagnosis. The patient underwent emergency surgical exploration. The left testis was found to be necrotic after a 360° testicular torsion and an orchiectomy was performed. RESULTS: The postoperative course was uneventful. The patient was discharged on day 4 after surgery. Histology showed a complete ischemic infarction of the testicular parenchyma as part of a ruptured intratesticular cavernous hemangioma. DISCUSSION AND CONCLUSION: Cavernous hemangioma is a rare tumor of the testicle in either childhood or adult period. The particularity of the presented case is the possible association of a cavernous intratesticular hemangioma with the torsion of the testis in a teenager. Clinicians and pathologists must be aware of the rare entity of testicular hemangiomas, as clinical examination and imaging studies do not often suffice to arrive at a correct diagnosis.
