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Case Reports
. 2023 Feb 21:11:1145941.
doi: 10.3389/fped.2023.1145941. eCollection 2023.

Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report

Elisabetta Bigagli  1 , Sara Agostiniani  1 , Alessandra Pugi  2 , Barbara Rombi  3 , Elena Eve Tornaboni  2 , Maria Luigia Censullo  4 , Carlotta Gemma Gori  4 , Rossana Pavone  4 , Iacopo Sardi  4
Affiliations
Case Reports

Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report

Elisabetta Bigagli et al. Front Pediatr. .

Abstract

In immunocompetent individuals, cytomegalovirus (CMV) infection is usually mild but may cause severe complications such as retinitis, pneumonitis, and encephalitis in immunocompromised individuals. So far, cases of CMV retinitis in patients with medulloblastoma undergoing chemotherapy and radiotherapy, have not been reported. We herein report the case of a pediatric patient with high-risk medulloblastoma who experienced an unexpected CMV retinopathy and leukoencephalopathy following high dose thiotepa and proton irradiation. The patient underwent a four-course induction therapy (1st cycle: methotrexate and vinorelbine; 2nd cycle: etoposide and hematopoietic stem cells apheresis; 3rd cycle: cyclophosphamide and vinorelbine; 4th cycle: carboplatin and vinorelbine) and then a consolidation phase consisting in high dose thiotepa followed by autologous HSC transplant and proton cranio-spinal irradiation plus boost to the primary tumor site and pituitary site with concomitant vinorelbine. After two months of maintenance treatment with lomustine and vinorelbine, the patient showed complete blindness and leukoencephalopathy. A diagnosis of CMV retinopathy was made and oral valganciclovir was administered. CMV retinopathy was judged to be possibly related to the use of high dose thiotepa worsened by radiotherapy. This case report suggests that in pediatric patients undergoing immunosuppressive chemo-radiotherapy, CMV reactivation should be carefully monitored to prevent serious complications such as retinopathy and visual loss.

Keywords: cytomegalovirus; drug safety—clinical pharmacology; leukoencephalopathy; medulloblastoma; pediatric; proton irradiation; retinopathy; thiotepa.

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Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

Figure 1
Figure 1
Pre-operative T2 (A) and post-gadolinum (B) sagittal and axial (C) MRI scans of our patient showing the extensive metastasis to hypothalamus. (D) post-gadolinium-enhanced T1-weighted sagittal spine MRI image showing leptomeningeal dissemination of medulloblastoma.
Figure 2
Figure 2
T2 (A) and FLAIR (B) axial MRI scans showing multifocal leukoencephalopathy after two months of maintenance treatment. Gadolinium-enhanced T1-weighted MRI (C, coronal section; D, axial section) shows a ring enhancement of the hypothalamic lesion compressing the chiasm and the optic tracts.
Figure 3
Figure 3
Clinical course of the patient.
See this image and copyright information in PMC

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