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Niemann-Pick disease type B

NPD type B is a rare inborn error of metabolism of autosomal recessive inheritance.

Patients with Niemann-Pick disease type B (NPDB) have a benign course and prognosis, while Niemann-Pick disease type A (NPDA) and Niemann-Pick disease type C (NPDC) are always associated with severe neurological involvement. They are known to be complicated with varying degrees of prognosis-determining liver dysfunction.

Synopsis

- short stature (less common)
- cherry-red maculae (less common)
- dyspnea
- frequent respiratory infections
- decreased pulmonary diffusion secondary to alveolar infiltration
- diffuse reticular or finely nodular infiltrations
- hepatomegaly
- progressive liver failure (7655746)
- splenomegaly
- absence of neurologic manifestations

Pathology

- Multiple visceral organs (lung, liver, spleen, kidney) contain foamy resident cells and histiocytes
- large vacuolated foam cells (?NP cells?) on bone marrow biopsy
- sea blue histiocytes
- decreased platelets

Associations

- generalized AL amyloidosis of kappa type (7655746)

Ultrastructure

- foam cells with lamellar inclusions

LABORATORY ABNORMALITIES

- Decreased acid sphingomyelinase activity
- Increased LDL cholesterol
- Increased triglycerides
- Decreased HDL cholesterol

MISCELLANEOUS

- Onset in infancy or childhood
- Variable phenotype
- More common in Ashkenazi Jews
- Allelic disorder to Niemann-Pick disease type A (257200)

Etiology

- Caused by mutations in the acid lysosomal sphingomyelin phosphodiesterase-1 gene (SMPD1) (MIM.607608)

See also

- lysosomal storage diseases

- Niemann-Pick diseases (NPDs)

References

- Takahashi T, Akiyama K, Tomihara M, Tokudome T, Nishinomiya F, Tazawa Y, Horinouchi K, Sakiyama T, Takada G. Heterogeneity of liver disorder in type B Niemann-Pick disease. Hum Pathol. 1997 Mar;28(3):385-8. PMID: 9042807