Diane K. Burke

Spastic diplegia and other motor disturbances in infants receiving interferon-alpha.

Objective: To determine how frequently the use of ‐interferon (‐IFN) is associated with the development of spastic diplegia.

Efficacy and safety of OK‐432 immunotherapy of lymphatic malformations

To determine the efficacy and safety of the immunostimulant OK‐432 (Picibanil) as a treatment option in the management of children with cervicofacial lymphatic malformations.

Treatment of lymphangiomas in children: an update of Picibanil (OK-432) sclerotherapy.

Picibanil (OK-432) is a sclerosing agent derived from a low-virulence strain of Streptococcus pyogenes that induces regression of macrocystic lymphangiomas. This report describes a prospective, nonrandomized trial to evaluate the efficacy of Picibanil in the treatment of 13 affected children ranging in age from 1 to 94 months. On average, 4.1 fluoroscopically guided intracystic injections were performed per child, with an average total dose of 0.56 mg of Picibanil. As judged by physical examination and radiographic studies, 5 children (42%) showed a complete or substantial response, and 2 children (16%) showed an intermediate response. No response was seen in 5 children (42%), 2 of whom had massive craniofacial lymphangioma. Factors that contribute to failure with Picibanil sclerotherapy are the presence of a significant micro-cystic component to the lesion, massive craniofacial involvement, and previous surgical resection. Macrocystic lymphangiomas of the infratemporal fossa or cervical area have the best response to therapy.

Treatment of massive or life-threatening hemangiomas with recombinant α2a-interferon

OBJECTIVE: To assess the response of massive, life-threatening, or function-impairing hemangiomas in pediatric patients receiving daily α2a-interferon subcutaneously. METHODS: The effect of 3 or more months of subcutaneous α2a-interferon (3 mU/m2) was prospectively evaluated in 10 patients with hemangiomas necessitating medical intervention. Hemangioma characteristics and extent were initially assessed by radiographic imaging in all but one patient. α2a-Interferon tolerance was monitored, and reduction in hemangioma size was recorded as marked (>50%), moderate (25% to 50%), or minimal (<25%). RESULTS: Hemangiomas were apparent at birth in 8 of 10 patients, and α2a-interferon was initiated at a median age of 4.5 months. Symptoms necessitating therapeutic intervention included congestive heart failure, airway obstruction, dysphagia, infection, failure to thrive, external auditory canal occlusion, visual axis impairment, and severe facial deformity. Four patients received treatment before referral that included systemic steroids (n = 2), intralesional steroids (n = l), or surgical/laser excision (n = 2). α2a-Interferon therapy was well tolerated. Most patients had a temporary elevation in body temperature during the first month of therapy. One patient with anorexia required nasogastric feedings and a temporary reduction in her α2a-interferon dose. An additional patient with irritability was withdrawn from the study at his parents’ request even though this symptom persisted after drug cessation. Hemangioma response to α2a-interferon was marked in six patients, moderate in two, and minimal in one whose lesion had features suggestive of a vascular malformation. Early signs of involution were usually evident within 6 weeks and often heralded by cutaneous blanching. α2a-Interferon therapy was concluded in four patients after a mean duration of 20 months. CONCLUSIONS: Daily subcutaneous α2a-interferon is well tolerated in pediatric patients and appears effective in hastening involution of symptomatic hemangiomas. A significant response is unlikely in lesions with features suggestive of a vascular malformation. (Otolaryngol Head Neck Surg 1997;117:99-110.)

Acyclovir in the treatment of recurrent respiratory papillomatosis. A pilot study.

Six patients with severe, recalcitrant, juvenile-onset recurrent respiratory papillomatosis were treated with 7 independent trials of acyclovir. In 2 trials, patients received acyclovir in place of interferon-α; the remaining 5 trials were in patients not otherwise receiving chemotherapy. Quantitative analysis of overall disease extent, laryngeal involvement, and degree of glottic obstruction for the 6 months prior to acyclovir administration and during acyclovir administration demonstrated a statistically significant decrease in all parameters evaluated in those patients who were otherwise unmedicated. The 2 patients who discontinued interferon-α immediately prior to beginning acyclovir demonstrated worsening disease, consistent with the well-recognized rebound phenomenon associated with stopping interferon. This study suggests that acyclovir decreases the extent of respiratory papillomatosis in patients with recalcitrant disease. The beneficial effect of acyclovir appears to be insufficient to counteract the rebound of disease when interferon is stopped abruptly.

Interferon alfa 2a for treatment of severe hemangiomas

Acute and serous otitis media are the most common diseases encountered in the pediatric age group. These diseases can be treated medically and/or with surgery--myringotomy with or without ventilation tube (VT) insertion. VT insertion is the most common surgical procedure in modern otology. Even though the procedure is simple and has considerable benefits, it also results in complications, the most common of which is posttympanostomy otorrhea. The purpose of this study was to compare various prophylactic treatments aimed at preventing the immediate onset of postoperative otorrhea. The study reviewed the records of 1252 children who underwent bilateral VT insertion for persistent serous otitis media. The children were divided into four groups: