Evidence map›Paper›PMID 35109795›Full record

ReviewBMC pediatrics2022

A novel case of prolonged Ifosfamide encephalopathy and long-term treatment with methylene blue: a case report and review of literature.

Gabriel Chain, Mudit Kalia, Karen Kestenbaum, Lara Pappas, Anna Sechser-Perl, Gadi Abebe Campino, Nibal Zaghloul

Open access · goldAbstract readCase ReportsReview
In one paragraph

Review in BMC pediatrics, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
1.5field-weighted citation impact, top 19% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed, 10 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors at 2 institutions in 2 countries.

Gabriel ChainDepartment of Pediatrics, The Children's Hospital at Saint Peter's University Hospital, 254 Easton Avenue, New Brunswick, NJ, 08901, USA. gabrielschain@gmail.com.ORCID 0000-0001-7823-9036
Mudit KaliaDepartment of Pediatrics, The Children's Hospital at Saint Peter's University Hospital, 254 Easton Avenue, New Brunswick, NJ, 08901, USA.
Karen KestenbaumDivision of Pediatric Hematology/Oncology, The Children's Hospital at Saint Peter's University Hospital, New Brunswick, NJ, 08901, USA.
Lara PappasDivision of Pediatric Hematology/Oncology, The Children's Hospital at Saint Peter's University Hospital, New Brunswick, NJ, 08901, USA.
Anna Sechser-PerlDivision of Pediatric Hematology/Oncology, The Children's Hospital at Saint Peter's University Hospital, New Brunswick, NJ, 08901, USA.
Gadi Abebe CampinoPediatric Hemato-Oncology division, Safra Children's Hospital, Sheba Medical Center, Ramat Gan, Israel.
Nibal ZaghloulDepartment of Pediatrics, The Children's Hospital at Saint Peter's University Hospital, 254 Easton Avenue, New Brunswick, NJ, 08901, USA.
Saint Peter's University Hospital · USSheba Medical Center · IL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundEncephalopathy following Ifosfamide treatment is a well-described phenomenon that is typically treated with Methylene Blue (MB). Chloroacetaldehyde, a potentially neurotoxic metabolite of Ifosfamide is hypothesized to cause this encephalopathy. Current guidelines for treatment is to stop Ifosfamide and provide supportive care. MB acts to inhibit Chloroacetaldehyde formation and has been described as a therapy and prophylaxis for Ifosfamide-encephalopathy. MB is effective within 30 min and lasts up to 3 days. Prolonged encephalopathy and MB therapy has not been described in the literature as lasting longer than 30 days following treatment. CASE PRESENTATION: We present the case of an 11-year-old female with autistic spectrum disorder and recurrent episodes of severe somnolence for 7 months following Ifosfamide therapy for her Non-Germinomatous Germ Cell Tumor (GCT). Periods of somnolence occurred prior to receiving cranial RT. Administration of MB gave immediate but limited response, with resolution of somnolence lasting 1-2 days between administrations. The somnolence could not be explained by neuroimaging or laboratory evaluation, but EEG indicated persistent encephalopathy.

conclusionA literature review determines that neurotoxicity is a side effect of Ifosfamide, but this effect has not been described persisting longer than 30 days. Our case continued to require treatment with MB for 7 months following cessation of therapy. We report these novel clinical findings, and hypothesize that there could be a genetic/metabolic component linking this reaction to Ifosfamide with the case patient's pre-existing autism. This possible association may also correlate to the already-established link between autism and the development of GCTs. This hypothesis leads to further discussion on the suitable usage of Ifosfamide in children with co-morbidities and the necessity of screening prior to its usage.

Indexed as

Brain DiseasesNeurotoxicity SyndromesChildFemaleHumansIfosfamideMethylene BlueSleepinessIfosfamideMethylene BlueEncephalopathyIfosfamideMethylene blueSomnolence

Identifiers

PMID35109795
PMCPMC8807677
OpenAlexW4210368749

What OpenQuestion holds

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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.