Reactions 1249 - 25 Apr 2009

★ SAntineoplastics

Oesophageal stricture (first report withmercaptopurine and tretinoin) in a patient withDown syndrome: case report

A 16-year-old boy with Down syndrome developedoesophageal stricture while receiving maintenanceantineoplastic therapy [dosages and durations of treatmentsnot stated] for acute promyelocytic leukaemia; he had alsoexperienced severe mucositis during induction therapy.

The boy initially received induction therapy withcytarabine, daunorubicin and tretinoin [all-trans retinoicacid], but this phase of treatment was complicated bysevere mucositis, febrile neutropenia, respiratory distressassociated with tretinoin syndrome, and tumour lysissyndrome with acute renal failure. Once he had recovered,he received consolidation therapy with daunorubicin andreduced-dose tretinoin, followed by maintenance therapywith mercaptopurine, oral methotrexate and tretinoin.While receiving maintenance therapy, he developed nauseaand vomiting. He was treated with maximal antiemetictherapy with no improvement.

The boy’s chemotherapy doses were reduced, but hisvomiting continued and maintenance therapy wasdiscontinued 9 weeks early. Routine echocardiography afew weeks later revealed a posterior mediastinal mass. Achest CT scan revealed moderate upper thoracicoesophageal dilatation, and lower oesophageal narrowingand thickening. A barium swallow confirmed significantnarrowing of his distal oesophagus, and upper GIendoscopy revealed a severe inflammatory strictureresulting in a luminal diameter of approximately 5mm. Anoesophagectomy with gastric conduit reconstruction wasperformed, and a pathological examination of the stricturerevealed fibrosis secondary to chronic inflammation. Hewas able to tolerate oral feeding following surgery and heregained bodyweight; however, he experienced leukaemiarelapse 26 weeks after the end of his treatment, and hedied.

Author comment: "Our patient was possibly moresensitive to esophageal toxicity due to a combination offactors."Kassam A, et al. Chemotherapy induced esophageal stricture. Pediatric Blood andCancer 52: 551, No. 4, Apr 2009 - Canada 801139514

» Editorial comment: A search of AdisBase, Medline andEmbase did not reveal any previous case reports ofoesophageal stricture associated with mercaptopurine ortretinoin. The WHO ADR database contained one report ofoesophageal stricture (WHO-ART) associated withmercaptopurine and no reports of oesophageal strictureassociated with tretinoin.

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Reactions 25 Apr 2009 No. 12490114-9954/10/1249-0001/$14.95 © 2010 Adis Data Information BV. All rights reserved