Revisiting Intra-arterial Chemotherapy—Palliative Utility in a Case of Recurrent Esthesioneuroblastoma and Literature Review

Background: Esthesioneuroblastomas (ENB) are rare, aggressive, and locally destructive neoplasms that occur in the superior nasal tracts originating from the olfactory neuroepithelium. Standard treatment commonly entails a combination of surgery and radiation with case-by-case adjunctive administration of chemotherapy. The role of intra-arterial (IA) therapeutics is not well-defined or described for ENBs.

Methods: We report an elderly recurrent ENB treated with IA carboplatin as a palliative therapy. A systematic review of PubMed and EmBase was also conducted, using search terms synonymous with “Esthesioneuroblastoma,” “Intra-arterial,” and “Chemotherapy” ([Fig. 1a]). Data on indications for IA chemotherapy, clinical course of treatment and outcomes were collected.

Results: A 77-year-old male presented with epistaxis and was biopsy-diagnosed with ENB. He underwent surgical resection 30 years ago and shortly thereafter received fractionated external beam radiation to the face and nasopharynx (total dose of 60 Gray over 30 fractions). Patient had been surveilled for several years and discharged from the clinic. Two years ago, patient returned with right eye visual loss and headaches in which there was found to be significant recurrence. Patient subsequently received 5 fractions of Gamma Knife radiosurgery totaling 25 Gy[JV1] . The mass did not respond with treatment progressively growing over time ([Fig. 2]) and palliative therapeutic options were discussed given patient had recurrent epistaxis, right sided vision loss secondary to mass effect on the optic nerve, confusion, and ataxic gait. Patient was found to have normal pressure hydrocephalus and was trialed with a large volume lumbar puncture with significant improvement. Patient subsequently underwent ventriculoperitoneal shunting with significant improvement in cognition and gait. For his recurrent ENB, one palliative IA carboplatin was discussed. After discussions with family regarding risks and rationale, the patient underwent an angiogram identifying bilateral internal maxillary arteries (IMA) as the predominant feeders of the recurrence ([Fig. 3]). Carboplatin was injected intra-arterially to the bilateral IMAs (total dose of 250 mg/m² suspended in 150 mL of sterile saline injected at rate of 4 mL/minute). Patient tolerated the procedure well and was discharged on post-procedure day 2. Patient is doing well 2 weeks post operatively with no new neurological sequelae and pending his 8 week posttreatment imaging.

Literature review yielded two primary and two recurrent ENBs mainly involving the ethmoid sinus with varying extension into the anterior and middle cranial fossa and cervical lymph nodes ([Fig. 1b], [Table 1]). No metastases were observed. IA cisplatin, cyclophosphamide and carmustine were administered through the maxillary or carotid arteries as adjuvant to surgical resection, radiotherapy or systemic chemotherapy. Patients did not experience any post-operative complications. All biopsied or resected specimens demonstrated ENB histology with one demonstrating admixed craniopharyngioma. Patients were followed up for 20 (12.5–30) months with two deaths, one each for primary and recurrent ENB.

Conclusion: We preliminarily report safe administration of IA carboplatin for a patient with recurrent ENB as a palliative therapy. There may be a role and need for further discussion on utility of IA therapy for recurrent ENBs with current improved selective microcatheter access.

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Artikel online veröffentlicht:
05. Februar 2024