Case Report: Coombs-Negative Hemolytic Anemia as the Initial Manifestation of Wilson Disease in an Adolescent Female: A Case Report

Background Wilson disease is a rare autosomal recessive disorder of copper metabolism caused by mutations in the ATP7B gene. Although hepatic and neurological manifestations are well recognized, acute Coombs-negative hemolytic anemia as the initial presentation is uncommon and may delay diagnosis. Early recognition is essential because prompt initiation of anti-copper therapy can reverse both hematologic and hepatic abnormalities. Case Presentation A previously healthy 16-year-old female basketball player presented with progressive easy fatigability, exertional dyspnea, recurrent jaundice, and high-colored urine for three months. Laboratory evaluation revealed severe Coombs-negative hemolytic anemia with a hemoglobin of 6.3 g/dL, predominantly indirect hyperbilirubinemia, markedly reduced serum alkaline phosphatase (6 U/L), elevated aspartate aminotransferase with near-normal alanine aminotransferase, prolonged international normalized ratio, and reticulocytosis. During hospitalization, hemoglobin declined further to 5.4 g/dL, necessitating packed red blood cell transfusion. Viral hepatitis and autoimmune serology were negative. Slit-lamp examination demonstrated bilateral Kayser–Fleischer rings, serum ceruloplasmin was reduced (16.97 mg/dL), and 24-hour urinary copper excretion was markedly elevated (863.23 μg/24 h), confirming Wilson disease. Oral zinc therapy was initiated while awaiting urinary copper estimation, followed by gradual introduction of D-penicillamine with pyridoxine supplementation. Serial follow-up over three months demonstrated complete resolution of symptoms, improvement in hemoglobin to 11.1 g/dL, normalization of serum bilirubin, recovery of alkaline phosphatase, and reduction in urinary copper excretion to 343.68 μg/24 h. Conclusion Wilson disease should be considered in adolescents presenting with unexplained Coombs-negative hemolytic anemia and jaundice, particularly when accompanied by disproportionately low serum alkaline phosphatase and mild hepatic dysfunction. Early diagnosis and timely initiation of anti-copper therapy can result in excellent clinical and biochemical recovery.

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Journal
Wellcome Open Research
Published
2026-10-26
DOI
https://doi.org/10.12688/wellcomeopenres.27414.1
Primary Topic
Trace Elements in Health
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article
Field-Weighted Citation Impact
0.00

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article

Case Report: Coombs-Negative Hemolytic Anemia as the Initial Manifestation of Wilson Disease in an Adolescent Female: A Case Report

Aashis Poudel, Buddha Basnyat, Roshan Shrestha
Wellcome Open Research
Trace Elements in Health
article

Case Report: Coombs-Negative Hemolytic Anemia as the Initial Manifestation of Wilson Disease in an Adolescent Female: A Case Report

Aashis Poudel, Buddha Basnyat, Roshan Shrestha
article en

Abstract

Background Wilson disease is a rare autosomal recessive disorder of copper metabolism caused by mutations in the ATP7B gene. Although hepatic and neurological manifestations are well recognized, acute Coombs-negative hemolytic anemia as the initial presentation is uncommon and may delay diagnosis. Early recognition is essential because prompt initiation of anti-copper therapy can reverse both hematologic and hepatic abnormalities. Case Presentation A previously healthy 16-year-old female basketball player presented with progressive easy fatigability, exertional dyspnea, recurrent jaundice, and high-colored urine for three months. Laboratory evaluation revealed severe Coombs-negative hemolytic anemia with a hemoglobin of 6.3 g/dL, predominantly indirect hyperbilirubinemia, markedly reduced serum alkaline phosphatase (6 U/L), elevated aspartate aminotransferase with near-normal alanine aminotransferase, prolonged international normalized ratio, and reticulocytosis. During hospitalization, hemoglobin declined further to 5.4 g/dL, necessitating packed red blood cell transfusion. Viral hepatitis and autoimmune serology were negative. Slit-lamp examination demonstrated bilateral Kayser–Fleischer rings, serum ceruloplasmin was reduced (16.97 mg/dL), and 24-hour urinary copper excretion was markedly elevated (863.23 μg/24 h), confirming Wilson disease. Oral zinc therapy was initiated while awaiting urinary copper estimation, followed by gradual introduction of D-penicillamine with pyridoxine supplementation. Serial follow-up over three months demonstrated complete resolution of symptoms, improvement in hemoglobin to 11.1 g/dL, normalization of serum bilirubin, recovery of alkaline phosphatase, and reduction in urinary copper excretion to 343.68 μg/24 h. Conclusion Wilson disease should be considered in adolescents presenting with unexplained Coombs-negative hemolytic anemia and jaundice, particularly when accompanied by disproportionately low serum alkaline phosphatase and mild hepatic dysfunction. Early diagnosis and timely initiation of anti-copper therapy can result in excellent clinical and biochemical recovery.

Wellcome Open ResearchVol. 11
Patan Academy of Health Sciences (NP), Oxford University Clinical Research Unit (VN)
Wellcome Trust
Openalex Percentile: Top 13%
Trace Elements in Health
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