Medicine · 2025
A rare case of littoral cell angioma with paroxysmal nocturnal hemoglobinuria.
СтатьяКлинический случайНазвание и аннотация: Перевод в очереди
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Li Zhenpeng, Zhang Sensen, Wu Jun, Du Zhonghai, Niu Shuxian
Аннотация
RATIONALE: We report a rare case of littoral cell angioma (LCA) in a patient with paroxysmal nocturnal hemoglobinuria (PNH). To our knowledge, this is the first report of these 2 rare conditions occurring together, which may help clarify their relationship and its importance in clinical practice.
PATIENT CONCERNS: A 38-year-old man had recurrent anemia and dark brown urine, accompanied by abdominal pain that worsened over time, which required several hospital visits and long-term follow-up.
DIAGNOSES: Follow-up scans showed progressive splenomegaly with multiple nodules. In combination with histopathology and immunohistochemistry results (CD31, CD34, and ERG positive; Ki-67 around 5%), these findings confirmed a final diagnosis of LCA in a patient with previously diagnosed PNH.
INTERVENTIONS: The patient received treatment with oral prednisone, sodium bicarbonate, and vitamin E for PNH. As the splenic lesions enlarged and blood counts worsened, he subsequently underwent laparoscopic splenectomy in February 2025.
OUTCOMES: After surgery, both LCA and anemia concurrently resolved, with normalization of hematologic parameters and resolution of abdominal symptoms.
LESSONS: This case suggests a potential association between LCA and PNH. Splenectomy provided dual benefits by treating the splenic tumor and improving hemolysis. Given the possible risk of malignant transformation, long-term follow-up is recommended.
PATIENT CONCERNS: A 38-year-old man had recurrent anemia and dark brown urine, accompanied by abdominal pain that worsened over time, which required several hospital visits and long-term follow-up.
DIAGNOSES: Follow-up scans showed progressive splenomegaly with multiple nodules. In combination with histopathology and immunohistochemistry results (CD31, CD34, and ERG positive; Ki-67 around 5%), these findings confirmed a final diagnosis of LCA in a patient with previously diagnosed PNH.
INTERVENTIONS: The patient received treatment with oral prednisone, sodium bicarbonate, and vitamin E for PNH. As the splenic lesions enlarged and blood counts worsened, he subsequently underwent laparoscopic splenectomy in February 2025.
OUTCOMES: After surgery, both LCA and anemia concurrently resolved, with normalization of hematologic parameters and resolution of abdominal symptoms.
LESSONS: This case suggests a potential association between LCA and PNH. Splenectomy provided dual benefits by treating the splenic tumor and improving hemolysis. Given the possible risk of malignant transformation, long-term follow-up is recommended.