FSIP2

fibrous sheath interacting protein 2

Ensembl:
ENSG00000188738
UniProt:
Q5CZC0
OMIM:
615796
Synonyms:
FLJ34780

Cilia effects upon perturbation of FSIP2

Loss-of-function effect:
Shorter (sperm flagella, MMAF)

Ciliogenesis screen results (3 screens)

  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
  • Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-2.86) PMID:41160700

Subcellular localization

basal body, cilia

Functional category

  • Ciliary assembly/disassembly
  • Actin & cytoskeleton regulation
  • T cell biology
  • Reproduction & sperm
  • Cell migration & adhesion

Function

Major structural protein of the fibrous sheath (FS) shown to bind to AKAP4, required for FS assembly. Regulaties PKA anchoring to AKAP4. Involved in the control of sperm flagellum elasticity and sig ling pathways that regulate sperm motility. Mutations in FSIP2 are a recurrent cause of multiple morphological abnormalities of the sperm flagella. All spermatozoa from patients FSIP2_3 and FSIP2_4 have shorter flagella than controls. (30137358)

Model organism evidence

Danio rerio (1 reference)

(#)Contributed equally PURPOSE: This study identified novel variants of the FSIP2 and SPEF2 genes in multiple morphological abnormalities of the sperm flagella (MMAF) patients and to investigate the potential effect of variations on male infertility and assisted reproductive outcomes.

PMIDs: 39753944

Mus musculus (1 reference)

BACKGROUND: Loss-of-function mutations in FSIP2 result in multiple morphological abnormalities of the flagella in humans and mice.

PMIDs: 35654582