Niemann-pick disease with visceral and pulmonary involvement in a resource limited setting: A rare case report

Niemann-Pick disease (NPD) is an autosomal recessive disease caused by deficient lysosomal enzyme or faulty cholesterol transport. A 9-year-old male patient presented with 6 years of abdominal swelling, previously treated as tuberculosis. He exhibited hepatosplenomegaly, delayed growth, and pancytopenia. Imaging revealed hepatosplenomegaly, a focal splenic infarct, diffuse interstitial septal thickening, and ground glass opacities in the lungs, raising suspicion of a storage disease. Further biopsies, of the bone marrow and liver, revealed the presence of foam cells with abundant multivacuolated cytoplasm and central round nuclei, suggesting NPD. Genetic testing and specific enzyme activity tests are unavailable in our setting. This case highlights the diagnostic challenges of rare disease in resource-limited settings, often mistaken for more common conditions like tuberculosis and lymphoma.

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Publication Details

Journal
Radiology Case Reports
Published
2024-12-21
DOI
https://doi.org/10.1016/j.radcr.2024.11.021
Citations
1
Primary Topic
Lysosomal Storage Disorders Research
Type
article
Field-Weighted Citation Impact
0.33
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article

Niemann-pick disease with visceral and pulmonary involvement in a resource limited setting: A rare case report

Daniel Zewdneh Solomon, Samuel Sisay Hailu, Eyasu Wakjira Korsa, Hewan Asfaw et al.
1 citations
Radiology Case Reports
Lysosomal Storage Disorders Research
0.33
article

Niemann-pick disease with visceral and pulmonary involvement in a resource limited setting: A rare case report

Daniel Zewdneh Solomon, Samuel Sisay Hailu, Eyasu Wakjira Korsa, Hewan Asfaw, Hanna Damtew
article en
1 citations

Abstract

Niemann-Pick disease (NPD) is an autosomal recessive disease caused by deficient lysosomal enzyme or faulty cholesterol transport. A 9-year-old male patient presented with 6 years of abdominal swelling, previously treated as tuberculosis. He exhibited hepatosplenomegaly, delayed growth, and pancytopenia. Imaging revealed hepatosplenomegaly, a focal splenic infarct, diffuse interstitial septal thickening, and ground glass opacities in the lungs, raising suspicion of a storage disease. Further biopsies, of the bone marrow and liver, revealed the presence of foam cells with abundant multivacuolated cytoplasm and central round nuclei, suggesting NPD. Genetic testing and specific enzyme activity tests are unavailable in our setting. This case highlights the diagnostic challenges of rare disease in resource-limited settings, often mistaken for more common conditions like tuberculosis and lymphoma.

Radiology Case ReportsVol. 20(3)
Children's Hospital of Philadelphia (US), Addis Ababa University (ET)
Openalex Percentile: Top 35%
Lysosomal Storage Disorders Research
0.33
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Niemann-pick disease with visceral and pulmonary involvement in a resource limited setting: A rare case report — Daniel Zewdneh Solomon, Samuel Sisay Hailu, et al. · Radiology Case Reports (2024) | TGRS Research Map | TGRS