Evidence map›Paper›PMID 41367019›Full record

ArticleMedicine2025

A rare case of littoral cell angioma with paroxysmal nocturnal hemoglobinuria.

Zhenpeng Li, Sensen Zhang, Jun Wu, Zhonghai Du, Shuxian Niu

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In one paragraph

Article in Medicine, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

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2 · The registry

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3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

5 authors.

Zhenpeng LiFirst Clinical Medical College of Shandong University of Traditional Chinese Medicine, Jinan City, Shandong Province, China.
Sensen ZhangWeifang Municipal Hospital of Traditional Chinese Medicine, Weifang, Shandong Province, China.
Jun WuWeifang Municipal Hospital of Traditional Chinese Medicine, Weifang, Shandong Province, China.
Zhonghai DuWeifang Municipal Hospital of Traditional Chinese Medicine, Weifang, Shandong Province, China.
Shuxian NiuWeifang Municipal Hospital of Traditional Chinese Medicine, Weifang, Shandong Province, China.ORCID 0009-0006-2359-6638

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

rationaleWe 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.

interventionsThe 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.

Indexed as

HemangiomaHemoglobinuria, ParoxysmalSplenic NeoplasmsAbdominal PainAdultHumansMaleSplenectomycase reportlaparoscopic splenectomylittoral cell angiomaparoxysmal nocturnal hemoglobinuria

Identifiers

PMID41367019
PMCPMC12689099

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