Abnormal Pyramidal Decussation and Bilateral Projection of the Corticospinal Tract Axons in Mice Lacking the Heparan Sulfate Endosulfatases, Sulf1 and Sulf2

Aizawa, Satoshi; Okada, Takuya; Keino‐Masu, Kazuko; Doan, Tri Huu; Koganezawa, Tadachika; Akiyama, Masahiro; Tamaoka, Akira; Masu, Masayuki

doi:10.3389/fnmol.2019.00333

Cited by 9 publications

(9 citation statements)

References 66 publications

(116 reference statements)

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“…In rodents, Kuang and Kalil (1990) were the first to report a variety of fiber distributions from CS neurons in the lumbar cord gray mat- (Aizawa et al, 2020;Steward et al, 2021). The present study using AAV vectors confirms those observations and extends our knowledge of the anatomical organization of CS neurons by identifying the cortical area projecting to the lateral portion of the lumbar dorsal horn.…”

Section: Projection From the Cerebral Cortex To The Lumbar Cordsupporting

confidence: 83%

Parcellation of the murine cortical hindlimb area is demonstrated by its subcortical connectivity and cytoarchitecture

Kubo

Murabe

Mizukami

et al. 2022

J of Comparative Neurology

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Although corticospinal neurons are known to be distributed in both the primary motor and somatosensory cortices (S1), details of the projection pattern of their fibers to the lumbar cord gray matter remain largely uncharacterized, especially in rodents. We previously investigated the cortical area projecting to the gray matter of the fourth lumbar cord segment (L4) (L4 Cx) in mice. In the present study, we injected an anterograde tracer into multiple sites to cover the entire L4 Cx. We found that (1) the rostromedial part of the L4 Cx projects to the intermediate and ventral zones of the lumbar cord gray matter, (2) the lateral part projects to the medial dorsal horn, and (3) the caudal part projects to the lateral dorsal horn. We also found that the border between the rostromedial and caudolateral parts corresponds to the border between the agranular and granular cortex. Analysis of the somatotopic patterns formed by the cortical projection cells and the primary sensory neurons innervating the skin of the hindlimb and its related area suggests that the lateral part corresponds to the S1 hindlimb area and the caudal part to the S1 trunk area. Examination of thalamic innervation by the L4 Cx revealed that the caudolateral L4 Cx focally projects to the ventrobasal complex (VB) and the posterior complex (PO), while the medial L4 Cx widely projects to the PO but little to the VB. These findings suggest that the L4 Cx is parceled into subregions defined by the cytoarchitecture and subcortical projection.

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Section: Projection From the Cerebral Cortex To The Lumbar Cordsupporting

confidence: 83%

Parcellation of the murine cortical hindlimb area is demonstrated by its subcortical connectivity and cytoarchitecture

Kubo

Murabe

Mizukami

et al. 2022

J of Comparative Neurology

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“…Lastly, it is often observed that the phenotype of disease models may be altered depending on the genetic background. This is also the case in these sulfatases; namely, Sulf1(−/−);Sulf2(−/−) double-deficient mice on a C57BL/6 genetic background are lethal while those on a C57BL/6 and ICR mixed genetic background are not [129,140].…”

Section: Sulfatase Reactionmentioning

confidence: 89%

“…One report describes impaired axon guidance in the corticospinal tract [128]. A more recent study demonstrated impaired motor functions in these mice [129], with Sulf1(−/−);Sulf2(−/−) double-deficient mice exhibiting the most severe phenotype involving embryonic development that is similarly found in Ext1/Ext2and Ndst-deficient mice. Furthermore, the chondrogenic phenotype was also reported in Sulf1(−/−);Sulf2(−/−) double-deficient mice.…”

Section: Sulfatase Reactionmentioning

confidence: 99%

Physiology and Pathophysiology of Heparan Sulfate in Animal Models: Its Biosynthesis and Degradation

Mashima¹,

Okuyama²,

Ohira³

2022

IJMS

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Heparan sulfate (HS) is a type of glycosaminoglycan that plays a key role in a variety of biological functions in neurology, skeletal development, immunology, and tumor metastasis. Biosynthesis of HS is initiated by a link of xylose to Ser residue of HS proteoglycans, followed by the formation of a linker tetrasaccharide. Then, an extension reaction of HS disaccharide occurs through polymerization of many repetitive units consisting of iduronic acid and N-acetylglucosamine. Subsequently, several modification reactions take place to complete the maturation of HS. The sulfation positions of N-, 2-O-, 6-O-, and 3-O- are all mediated by specific enzymes that may have multiple isozymes. C5-epimerization is facilitated by the epimerase enzyme that converts glucuronic acid to iduronic acid. Once these enzymatic reactions have been completed, the desulfation reaction further modifies HS. Apart from HS biosynthesis, the degradation of HS is largely mediated by the lysosome, an intracellular organelle with acidic pH. Mucopolysaccharidosis is a genetic disorder characterized by an accumulation of glycosaminoglycans in the body associated with neuronal, skeletal, and visceral disorders. Genetically modified animal models have significantly contributed to the understanding of the in vivo role of these enzymes. Their role and potential link to diseases are also discussed.

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“…Sulfatases (Sulf1 and Sulf2) are enzymes that further process HS after its exposure to the extracellular space, removing O-sulfate groups, and their expression is important for pyramidal decussation and bilateral projections of corticospinal axons 42 .…”

Section: Developmental Dynamicsmentioning

confidence: 99%

“…40 Sulfatases (Sulf1 and Sulf2) are enzymes that further process HS after its exposure to the extracellular space, removing O-sulfate groups, and their expression is important for pyramidal decussation and bilateral projections of corticospinal axons. 41 Reduced HS sulfation levels correlates with tumor growth inhibition. 42,43 On the other hand, Sulf2 knockout resulted in less marked effects of nonalcoholic fatty liver disease.…”

Section: Heparan Sulfate: Biosynthesis and Biologymentioning

confidence: 99%

Heparan sulfate fine‐tunes stromal‐epithelial communication in the prostate gland

Barbosa

Biancardi

Carvalho

2020

Developmental Dynamics

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Several studies reported the concerted and mutual communication between the prostate epithelium and stroma, which determines the final organ architecture and function, but gets awry in cancer. Deciphering the mechanisms involved in this communication is crucial to find new therapeutic strategies. HS sequesters a number of secreted growth factors and cytokines, controlling their bioavailability to the target cells, suggesting that HS is an important regulator of the extracellular matrix (ECM) and a key player in the cell-cell and cell-microenvironment communication during prostate morphogenesis and physiology. We propose that by controlling HS biosynthesis and sulfation pattern, as well as the cleavage of the HS chain and/or the shedding of proteoglycans, epithelial and stromal cells are able to precisely tune the availability of signaling molecules and modulate ligand-receptor interaction and intracellular signal transduction.

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Abnormal Pyramidal Decussation and Bilateral Projection of the Corticospinal Tract Axons in Mice Lacking the Heparan Sulfate Endosulfatases, Sulf1 and Sulf2

Cited by 9 publications

References 66 publications

Parcellation of the murine cortical hindlimb area is demonstrated by its subcortical connectivity and cytoarchitecture

Parcellation of the murine cortical hindlimb area is demonstrated by its subcortical connectivity and cytoarchitecture

Physiology and Pathophysiology of Heparan Sulfate in Animal Models: Its Biosynthesis and Degradation

Heparan sulfate fine‐tunes stromal‐epithelial communication in the prostate gland

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