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D-Penicillamine Induced Myelotoxicity: A Unique Case
Aliya Abdulla1, Aryan Rezvani2, Christopher Nelsen1
1Department of Pharmacy, Houston Methodist Hospital, Houston, TX, USA.
Abstract:
Purpose: A case of D-penicillamine-related myelotoxicity in a patient with Wilson's disease is reported. Summary: There is a paucity of literature regarding D-penicillamine (DPA) induced myelotoxicity in the setting of Wilson's disease (WD). A 22-year-old male presented with a 1-week history of bleeding gums and dizziness. Four months prior, he had been diagnosed with Wilson's disease and started on a regimen of DPA. His blood counts demonstrated profound pancytopenia. Due to concern for suspected drug-induced myelotoxicity, DPA was discontinued. Parvovirus B19, Epstein-Barr virus, cytomegalovirus, and varicella zoster virus polymerase chain reaction studies were negative and there was no evidence of hematological malignancy. Bone marrow biopsy demonstrated hypocellularity and trilineage hypoplasia with corresponding aspirate flow cytometry confirming the absence of acute leukemia. The patient was started on subcutaneous granulocyte-colony stimulating factor, provided transfusion support with packed red blood cells and platelets. Despite these measures, his blood count failed to recover, and he was discharged on eltrombopag 150 mg daily with plans for outpatient transfusion support. DPA was permanently discontinued, and he was prescribed trientine 750 mg daily. Unfortunately, his myelotoxicity remained consistent, requiring regular transfusions. He is currently undergoing evaluation for bone marrow transplant. Conclusion: DPA-induced myelotoxicity is a rare clinical entity. Our case demonstrates a unique clinical presentation of this phenomenon. Guidelines to mitigate the risk of and treat this toxicity remain to be determined.
Insights
D-penicillamine (DPA) can cause rare myelotoxicity in Wilson
Area of Science:
- Hematology
- Pharmacology
- Genetics
Background:
- Wilson's disease (WD) is a genetic disorder of copper metabolism.
- D-penicillamine (DPA) is a chelating agent used to treat WD.
- Drug-induced myelotoxicity is a serious adverse effect of some medications.
Purpose of the Study:
- To report a rare case of D-penicillamine-induced myelotoxicity in a patient with Wilson's disease.
- To highlight the clinical presentation and management challenges of this adverse drug reaction.
Main Methods:
- Case report of a 22-year-old male diagnosed with Wilson's disease and treated with DPA.
- Clinical presentation, laboratory findings, bone marrow biopsy, and treatment interventions were documented.
- Exclusion of other causes of pancytopenia, including viral infections and hematological malignancies.
Main Results:
- The patient developed profound pancytopenia after starting DPA therapy.
- Bone marrow biopsy showed hypocellularity and trilineage hypoplasia.
- Despite supportive care and discontinuation of DPA, myelotoxicity persisted, necessitating transfusions and evaluation for bone marrow transplant.
Conclusions:
- D-penicillamine-induced myelotoxicity is a rare but severe complication in Wilson's disease patients.
- This case underscores the importance of vigilant monitoring for hematological toxicity during DPA treatment.
- Further research is needed to establish clear guidelines for managing and mitigating DPA-induced myelotoxicity.
