Pure White Cell Aplasia and Immune Thrombocytopenia after Thymoma Resection: A Case Report and Review of the Literature

Youssef, Michael; Stratton, Tyler W.; Gallant, Reid C.; Young, Christine; Li, Daniel Y.; Piran, Siavash

doi:10.1155/2022/8271069

Cited by 3 publications

(2 citation statements)

References 26 publications

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“…Seven of these cases were effectively treated with CSA (Table 3), and the patient in this study benefited from CSA treatment both after surgery and after recurrence. These results [20] 33 [21] 74/M Granulocytic hypoplasia indicate that low-dose CSA after PCWA therapy was sufficient to maintain long-term remission after thymectomy. [15] Accordingly, patients with thymoma-associated PCWA benefit more firstline immunosuppressive therapy.…”

Section: Concomitantmentioning

confidence: 84%

Pure white cell aplasia before and after thymectomy in the rare conundrum of thymoma: A case report and review of the literature

Yang,

Chen,

Zheng

et al. 2024

Medicine

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Rationale: Pure white cell aplasia (PWCA) is a rare paraneoplastic syndrome that occurs in patients with thymomas. Currently, the pathogenesis and treatment of this disease remain in the exploratory stage. Patient concerns: We report a 68-year-old woman with thymoma experienced PWCA involvement as her first presentation. The patient had high fever and agranulocytosis at the onset of the disease. The white blood cell count in the complete blood count was 1.9 × 109/L with a neutrophil of 0.1 × 109/L. The bone marrow aspirates showed decreased granulocyte proliferation. Computed tomography showed a large mass in the anterior mediastinum. Diagnoses: The final diagnosis of our patient was PWCA and thymoma. Interventions: She underwent a thymectomy and cyclosporine A administration during first remission. Outcomes: Long-term remission was achieved following the readministration of cyclosporine A after the disease recurrence. Lessons: PWCA or agranulocytosis with thymoma has been confirmed to be an extremely rare disease. Thymomas with PWCA correlate with autoimmunity. From this case study and the literature review, we concluded that the pathogenesis of thymomas in PWCA is mainly related to the activation of autoreactive T cells. Thymectomy and the immunosuppressive drug, cyclosporine A, were chosen for treatment. The patient’s granulocyte levels were unable to recover after surgery because of the inability to promptly clear activated T cells. After surgery, cyclosporine A continued to take for a long time. Thymectomy combined with prolonged cyclosporine A administration may be an effective method for treating this rare disease.

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Section: Concomitantmentioning

confidence: 84%

Pure white cell aplasia before and after thymectomy in the rare conundrum of thymoma: A case report and review of the literature

Yang,

Chen,

Zheng

et al. 2024

Medicine

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“…In addition to concurrent paraneoplastic anemias, the patient also developed neutropenia and thrombocytopenia. Hypothesized etiologies for the neutropenia and thrombocytopenia included an occult presentation of T-LGLL causing cytopenias versus thymoma-associated neutropenia and thrombocytopenia, though the latter is extremely rare [ 4 , 27 , 28 ]. In contrast, the relationship between T-LGLL and immune-mediated cytopenias is well described in the literature [ 29 ].…”

Section: Discussionmentioning

confidence: 99%

Thymoma With Triple Threat: Pure Red Cell Aplasia, Autoimmune Hemolytic Anemia, and T-Cell Large Granular Lymphocytic Leukemia

2022

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Thymomas are a rare neoplasm of the anterior mediastinum and often associated with paraneoplastic syndromes. Though myasthenia gravis is the most common and well-known, the list of reported paraneoplastic syndromes occurring with thymoma is extensive and ever-growing. Paraneoplastic syndromes can involve nearly every organ system, including hematologic abnormalities affecting any or all cell lines. This can present challenges to the clinician in terms of diagnosis, prognostic impact, and management. We present the case of a previously healthy 41-year-old female who was diagnosed with thymoma and three rare hematologic paraneoplastic syndromes: pure red cell aplasia (PRCA), autoimmune hemolytic anemia (AIHA), and T-cell large granular lymphocytic leukemia (T-LGLL). To the best of our knowledge, there have been only four other reported cases of PRCA and AIHA in a single patient with thymoma, all of which were treated with thymectomy. Upfront surgical resection was not possible in the present case and thus the patient was alternatively treated with corticosteroids and octreotide, which proved successful in resolving the anemia. The authors present this case to share these findings of an alternative treatment strategy for thymoma-associated PRCA and AIHA and to highlight the importance of careful monitoring with routine blood work for these complex patients.

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Anti-inflammatories/danazol/granulocyte-colony-stimulating-factors

2022

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Pure White Cell Aplasia and Immune Thrombocytopenia after Thymoma Resection: A Case Report and Review of the Literature

Cited by 3 publications

References 26 publications

Pure white cell aplasia before and after thymectomy in the rare conundrum of thymoma: A case report and review of the literature

Pure white cell aplasia before and after thymectomy in the rare conundrum of thymoma: A case report and review of the literature

Thymoma With Triple Threat: Pure Red Cell Aplasia, Autoimmune Hemolytic Anemia, and T-Cell Large Granular Lymphocytic Leukemia

Anti-inflammatories/danazol/granulocyte-colony-stimulating-factors

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