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Hydroxyurea and colonic ulcers: a case report.

Boonyawat K, Wongwaisayawan S, Nitiyanant P, Atichartakarn V - BMC Gastroenterol (2014)

Bottom Line: However, a potentially fatal gastrointestinal ulceration was recently found and herein reported.Subsequently, ulcerative lesions also developed at the pharynx, histo-pathologic findings of which were not different from those in the colon.These ulcerative lesions resolved within a month after discontinuing hydroxyurea in April 2010 and have not recurred since.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Pathology, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand. sww563@hotmail.com.

ABSTRACT

Background: Hydroxyurea at a relatively low dose is frequently prescribed to induce hemoglobin F production in patients with sickle cell and β-thalassemia diseases because of its good efficacy and safety profiles. However, a potentially fatal gastrointestinal ulceration was recently found and herein reported.

Case presentation: A thirty-seven-year-old man with transfusion dependent hemoglobin E/β-thalassemia disease was treated with hydroxyurea to induce hemoglobin F production since 2007 without incident. From 2008 to April 2010, episodes of hematochezia, mucous diarrhea and epigastric pain intermittently manifested. Four colonoscopies done during the period repeatedly showed ulcerative lesions from the terminal ileum to the ascending colon with a non-specific histo-pathologic finding. Subsequently, ulcerative lesions also developed at the pharynx, histo-pathologic findings of which were not different from those in the colon. These ulcerative lesions resolved within a month after discontinuing hydroxyurea in April 2010 and have not recurred since.

Conclusion: The findings suggested role of hydroxyurea in the pathogenesis of these ulcers, and that it must be immediately discontinued to prevent further damage to the digestive mucosa.

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Related in: MedlinePlus

Histo-pathologic finding of the ascending colon (H&E × 200). The mucosa shows acute and chronic inflammatory cells infiltration with ulceration.
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Figure 2: Histo-pathologic finding of the ascending colon (H&E × 200). The mucosa shows acute and chronic inflammatory cells infiltration with ulceration.

Mentions: In December 2009, he presented with fever and mucous diarrhea. CT scan of the whole abdomen showed a circumferential thickened wall involving the ascending colon and the terminal ileum. Esophago-gastro-duodenoscopy revealed multiple erosions on the gastric mucosa, biopsy of which showed chronic gastritis. Colonoscopy revealed a markedly swollen and ulcerated IC valve and circumferentially thickened wall from the terminal ileum to the ascending colon. Biopsies of cecal ulcers showed acute ulcers with acute and chronic inflammatory cells infiltration to the muscularis mucosae. There was no feature of malignancy, vasculitis, viral inclusion or granuloma. GMS, Periodic Acid Schiff (PAS), and AFB stains were non-revealing. Various serologic tests for systemic vasculitis, such as antinuclear antibody, perinuclear and cytoplasmic antineutrophil cytoplasmic autoantibody were all negative. Empiric treatment with intravenous ceftriaxone was given.In February 2010, there was another episode of massive hematochezia, resulting in a partial right half colectomy. Multiple scattered ulcerative lesions involving the terminal ileum, the cecum and the ascending colon were found on gross examination (Figure 1). The histo-pathologic findings were acute mucosal ulcers with granulation tissue and tissue eosinophilia. The submucosa was thickened and edematous. The acute and chronic inflammatory cells infiltrated through the submucosa to the inner muscular layer, not unlike the previous ones (Figure 2). Leukocytoclastic vasculitis was not found. Special stains with GMS, PAS, and AFB were again non-revealing.


Hydroxyurea and colonic ulcers: a case report.

Boonyawat K, Wongwaisayawan S, Nitiyanant P, Atichartakarn V - BMC Gastroenterol (2014)

Histo-pathologic finding of the ascending colon (H&E × 200). The mucosa shows acute and chronic inflammatory cells infiltration with ulceration.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4127553&req=5

Figure 2: Histo-pathologic finding of the ascending colon (H&E × 200). The mucosa shows acute and chronic inflammatory cells infiltration with ulceration.
Mentions: In December 2009, he presented with fever and mucous diarrhea. CT scan of the whole abdomen showed a circumferential thickened wall involving the ascending colon and the terminal ileum. Esophago-gastro-duodenoscopy revealed multiple erosions on the gastric mucosa, biopsy of which showed chronic gastritis. Colonoscopy revealed a markedly swollen and ulcerated IC valve and circumferentially thickened wall from the terminal ileum to the ascending colon. Biopsies of cecal ulcers showed acute ulcers with acute and chronic inflammatory cells infiltration to the muscularis mucosae. There was no feature of malignancy, vasculitis, viral inclusion or granuloma. GMS, Periodic Acid Schiff (PAS), and AFB stains were non-revealing. Various serologic tests for systemic vasculitis, such as antinuclear antibody, perinuclear and cytoplasmic antineutrophil cytoplasmic autoantibody were all negative. Empiric treatment with intravenous ceftriaxone was given.In February 2010, there was another episode of massive hematochezia, resulting in a partial right half colectomy. Multiple scattered ulcerative lesions involving the terminal ileum, the cecum and the ascending colon were found on gross examination (Figure 1). The histo-pathologic findings were acute mucosal ulcers with granulation tissue and tissue eosinophilia. The submucosa was thickened and edematous. The acute and chronic inflammatory cells infiltrated through the submucosa to the inner muscular layer, not unlike the previous ones (Figure 2). Leukocytoclastic vasculitis was not found. Special stains with GMS, PAS, and AFB were again non-revealing.

Bottom Line: However, a potentially fatal gastrointestinal ulceration was recently found and herein reported.Subsequently, ulcerative lesions also developed at the pharynx, histo-pathologic findings of which were not different from those in the colon.These ulcerative lesions resolved within a month after discontinuing hydroxyurea in April 2010 and have not recurred since.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Pathology, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand. sww563@hotmail.com.

ABSTRACT

Background: Hydroxyurea at a relatively low dose is frequently prescribed to induce hemoglobin F production in patients with sickle cell and β-thalassemia diseases because of its good efficacy and safety profiles. However, a potentially fatal gastrointestinal ulceration was recently found and herein reported.

Case presentation: A thirty-seven-year-old man with transfusion dependent hemoglobin E/β-thalassemia disease was treated with hydroxyurea to induce hemoglobin F production since 2007 without incident. From 2008 to April 2010, episodes of hematochezia, mucous diarrhea and epigastric pain intermittently manifested. Four colonoscopies done during the period repeatedly showed ulcerative lesions from the terminal ileum to the ascending colon with a non-specific histo-pathologic finding. Subsequently, ulcerative lesions also developed at the pharynx, histo-pathologic findings of which were not different from those in the colon. These ulcerative lesions resolved within a month after discontinuing hydroxyurea in April 2010 and have not recurred since.

Conclusion: The findings suggested role of hydroxyurea in the pathogenesis of these ulcers, and that it must be immediately discontinued to prevent further damage to the digestive mucosa.

Show MeSH
Related in: MedlinePlus