Pearls and Pitfalls of Intrathecal Amphotericin B Therapy for Refractory Coccidioidal Meningitis in Children: An Illustrative Pediatric Case Series.

Abstract

Introduction: Incidence of pediatric coccidioidomycosis has risen over the last 20 years, and coccidioidal meningitis is the most severe form of disseminated disease. Intrathecal amphotericin B deoxycholate (IT AmBd) has historically improved outcomes and remains a mainstay of therapy when response to azole therapy is inadequate. However, the dosing, route of administration and duration of therapy in young pediatric patients pose unique challenges due to their anatomy, inability to communicate and the need for hospital-based administration. We present 4 instructive cases over 30 years to provide guidance on the use of IT AmBd.

Methods: Case information was abstracted from the electronic health record at University of California Los Angeles. Literature review was conducted to look for additional cases on Pubmed with relevant search terms.

Results: We present 4 cases including 2 patients with successful outcomes and 2 who did not survive. Starting doses, administration routes, complications and neurosurgical challenges are described. Patients had prolonged hospitalizations as the ability to administer outpatient IT amphotericin B was minimal. Therefore, more rapid dose escalation and weaning, with a shorter overall duration of therapy, was needed than described for adults. IT treatment was considered an initial stabilizing measure while attempting to optimize azole monotherapy. Cisternal administration is preferred but remains a logistical challenge.

Conclusion: Intrathecal AmBd remains an important treatment option in pediatric patients with refractory coccidioidal meningitis; however, due to unique administration, challenges may be more of a temporizing measure rather than definitive prolonged therapy. Further experience and multidisciplinary care are needed to reduce mortality.