The DOI matches its Crossref record; 54 of the 54 references it cites were found in a registry or an index; 8 of the 45 sources its claims cite were checked for support.
SH3TC2, a protein mutant in Charcot–Marie–Tooth neuropathy, links peripheral nerve myelination to endosomal recycling
Open accessAbstract
Patients with Charcot-Marie-Tooth neuropathy and gene targeting in mice revealed an essential role for the SH3TC2 gene in peripheral nerve myelination. SH3TC2 expression is restricted to Schwann cells in the peripheral nervous system, and the gene product, SH3TC2, localizes to the perinuclear recycling compartment. Here, we show that SH3TC2 interacts with the small guanosine triphosphatase Rab11, which is known to regulate the recycling of internalized membranes and receptors back to the cell surface. Results of protein binding studies and transferrin receptor trafficking are in line with a role of SH3TC2 as a Rab11 effector molecule. Consistent with a function of Rab11 in Schwann cell myelination, SH3TC2 mutations that cause neuropathy disrupt the SH3TC2/Rab11 interaction, and forced expression of dominant negative Rab11 strongly impairs myelin formation in vitro. Our data indicate that the SH3TC2/Rab11 interaction is relevant for peripheral nerve pathophysiology and place endosomal recycling on the list of cellular mechanisms involved in Schwann cell myelination.
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Written from the paper’s full text.
TL;DR
SH3TC2, the protein mutated in Charcot–Marie–Tooth disease type 4C (CMT4C), is a direct Rab11 effector localized to the endosomal recycling compartment, and CMT4C missense mutations disrupt this interaction. Rab11 activity regulates Schwann cell myelination in vitro, linking endosomal recycling to peripheral nerve myelination.
Key findings
- SH3TC2 was identified as a direct Rab11 interactor by tandem affinity purification/mass spectrometry, coimmunoprecipitation, GST pulldown, and yeast two-hybrid assays; binding was specific to Rab11 and occurred with active GTP-bound Rab11_Q70L but not dominant-negative Rab11_S25N or the effector-domain mutant Rab11a_Q70L_I44E.
- All tested CMT4C-associated missense substitutions (five in Fig. 4A) disrupted SH3TC2/Rab11a binding, while rodent Sh3tc2 still bound human Rab11a despite ~20% nonconserved residues.
- Wild-type SH3TC2 slowed transferrin receptor recycling and condensed Rab11a-positive perinuclear recycling endosomes; the N881S mutant had only a slight effect. In Sh3tc2 knockout sciatic nerves, Rab11a protein levels were reduced while Rab11a mRNA was unchanged.
- In in vitro myelination, dominant-negative Rab11a_S25N strongly impaired myelin basic protein-positive segments, whereas constitutively active Rab11a_Q70L increased myelination in dorsal root ganglion explants and Schwann cell/neuron co-cultures.
Methods
- Protein interactions were assayed by tandem affinity purification from inducible SH3TC2-C-TAP Flp-In T-REx 293 cells followed by mass spectrometry, coimmunoprecipitation in HEK293 cells, GST pulldown with in vitro translated SH3TC2/Sh3tc2 and Rab11 mutants, and yeast two-hybrid.
- Cell biology included immunofluorescence in HEK293, COS7, and RT4-D6P2T cells; subcellular fractionation on 5–25% Optiprep gradients; transferrin recycling by FACS (2000 GFP-positive cells, three repeats); and Rab11a surface reconstruction in 100 transfected COS7 cells per condition.
- Myelination was assessed in mouse embryonic day 13.5 dorsal root ganglion explants and rat Schwann cell/mouse dorsal root ganglion neuron co-cultures transduced with lentiviral GFP, Rab11a_Q70L-GFP, or Rab11a_S25N-GFP; myelin basic protein staining was quantified from 10 fields per coverslip in triplicate experiments with at least three coverslips per condition, using mean ± SEM and two-tailed Student’s t-test.
Limitations
- Stated: the authors could not identify the specific SH3TC2 protein region or a shorter linear motif mediating Rab11 binding, and CMT4C substitutions disrupting the complex are spread throughout the protein.
- Stated: SH3TC2 may also influence myelination in a Rab11-independent manner, and other proteins in the SH3TC2 complex were not investigated for PNS/Schwann cell roles.
- Stated/evident: no antibody against endogenous Sh3tc2 was available, so the Schwann cell line had to be transfected with a FLAG construct; the findings rely heavily on overexpression and in vitro myelination systems, with no in vivo rescue of the SH3TC2/Rab11 interaction.
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Claims the paper makes
26 Own result · 14 Not checked yet · 13 No full text of the sources · 13 Own interpretation · 4 Partly supported · 3 Sources not found · 2 Supported · 1 No basis given · accuracy not yet measured
The cited sources support only part of this claim — read each source’s verdict below.
In the paper
“We have recently shown that both overexpressed proteins, SH3TC2 and Rab11a, colocalize with -tubulin and internalized transferrin in the perinuclear recycling endosome (Arnaud et al., 2009).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Partly supportedcites Arnaud et al., 2009 sourceExperimental — accuracy not yet measured
The localization results (p. 2) and Fig. 3 legend (p. 4) show tagged SH3TC2 colocalizing with Rab11 in the endocytic recycling compartment, and tagged SH3TC2 and Rab11 colocalizing with internalized transferrin. They use γ-tubulin to locate SH3TC2 near the centrosome, but do not explicitly show both proteins colocalizing with γ-tubulin or identify the Rab11 construct as Rab11a. (LLM, conf=0.82)
plaincorrelational- Asserted: passes — The authors put it forward as true.
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- Partly supported
Every source the paper cites for this claim was checked and supports it.
In the paper
“Very recently, we found that a mouse model without a functional copy of the Sh3tc2 gene developed a peripheral neuropathy that largely reproduced the human phenotype (Arnaud et al., 2009).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Introduction
What the sources say
- Supportedcites Arnaud et al., 2009 source
The paper reports that mice lacking Sh3tc2 exon 1 develop progressive peripheral neuropathy, and the Discussion (p. 4) says their reduced nerve conduction velocities are comparable to those in CMT4C patients. It calls the mice a valid model of CMT4C neuropathy (Discussion, p. 5). The mice do not reproduce every feature: unlike many patients, they did not develop scoliosis (p. 2), which is consistent with the claim’s qualification “largely.” (LLM, conf=0.92)
plaindirect- Asserted: passes — The authors put it forward as true.
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- Supported
The cited sources support only part of this claim — read each source’s verdict below.
In the paper
“Obviously, the presence of this motif is not a prerequisite to function as a Rab11 effector, as it does not occur in all Rab11-interactors (Wu et al., 2005;Westlake et al., 2007).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not matchedcites Wu et al., 2005
Couldn’t be matched to a paper in Crossref, so it can’t be checked.
- Partly supportedcites Westlake et al., 2007 sourceExperimental — accuracy not yet measured
The Discussion (page unavailable) says Evi5 binds Rab11 despite having no other identified conserved domains, such as Rab-binding domains, in its binding fragment. The Results show a functional Rab11–Evi5 interaction, but the paper describes Evi5 as a Rab11-binding protein rather than establishing it as a Rab11 effector or specifically testing the motif named in the claim. (LLM, conf=0.82)
boosted- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: unclear — It is unclear whether the paper’s conclusions depend on it.
- Based: passes — The paper gives a reason why it holds.
- Not matched
A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“On the other hand, infection with a lentivirus containing constitutively active Rab11a resulted in an increased number of myelinated segments (Supplementary Fig. 5A and B).”
- Also stated in: § Results · plain
“expression of the constitutively active Rab11a resulted in moderately increased myelination (Fig. 6A and B).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
plaindirect- Asserted: passes — The authors put it forward as true.
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- Load-bearing: passes — The paper’s conclusions depend on it.
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- Also stated in: § Results · plain
A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“Conversely, we observed no colocalization of SH3TC2 with marker proteins for other perinuclear organelles (Supplementary Fig. 3).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
plain- Asserted: passes — The authors put it forward as true.
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- Load-bearing: unclear — It is unclear whether the paper’s conclusions depend on it.
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A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“The localization of both proteins overlapped in the perinuclear region in all three cell lines (Fig. 2A).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
plaincorrelationaln = 3- Asserted: passes — The authors put it forward as true.
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A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“Overexpression of wild-type SH3TC2 resulted in moderate slowing of transferrin recycling as compared with transferrin retention in cells transfected with CMT4C mutants or a GFP control vector (Fig. 5B and Supplementary Table 1).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
plaindirect- Asserted: passes — The authors put it forward as true.
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- Load-bearing: passes — The paper’s conclusions depend on it.
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A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“Here, we show that SH3TC2 interacts with the small guanosine triphosphatase Rab11, which is known to regulate the recycling of internalized membranes and receptors back to the cell surface.”
- Also stated in: § Results · plain
“In addition to the co-precipitation of Rab11a along with SH3TC2, immunoprecipitation in the other direction showed that SH3TC2 could be coprecipitated with GFP-Rab11a as well (Fig. 1C).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Abstract
plaincorrelational- Asserted: passes — The authors put it forward as true.
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- Also stated in: § Results · plain
A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“Results from GST pulldown and yeast two-hybrid experiments provided further evidence for the interaction between SH3TC2 and both Rab11 isoforms and showed that SH3TC2 binds directly to Rab11 (Fig. 1D and E; Supplementary Fig. 1B).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
boostedcorrelational- Asserted: passes — The authors put it forward as true.
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A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“SH3TC2 mutations that cause neuropathy disrupt the SH3TC2/Rab11 interaction”
- Also stated in: § Results · plain
“We found that all tested amino acid substitutions related to missense mutations observed in patients with CMT4C disrupted the SH3TC2/Rab11a interaction (Fig. 4A).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Abstract
plaindirect- Asserted: passes — The authors put it forward as true.
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- Also stated in: § Results · plain
- Probably not a substantial claim. 20
- It may state two separate claims as one. 11
The cited sources support only part of this claim — read each source’s verdict below.
In the paper
“During myelination, Schwann cells establish specialized microtubule networks, suggesting increased rates of vesicle transport (Trapp et al., 1995;Kidd et al., 1996).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Partly supportedcites Trapp et al., 1995 sourceExperimental — accuracy not yet measured
The Discussion (page unavailable) reports that vesicles travel along microtubules in myelinating Schwann cells, and describes microtubules in channels extending along the myelin internode. It does not show that the cells establish those networks during myelination or that vesicle transport rates increase. (LLM, conf=0.84)
- Partly supportedcites Kidd et al., 1996 sourceExperimental — accuracy not yet measured
The paper reports that myelinating Schwann cells reorganize their microtubules and, in the Discussion, suggests that this arrangement establishes transport pathways along the myelin internode. It does not say that vesicle transport occurs at increased rates; its suggestion concerns the location of transport pathways, not transport speed. (LLM, conf=0.85)
hedgedconditional- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it. It may state two separate claims as one.
- Partly supported
The cited sources support only part of this claim — read each source’s verdict below.
In the paper
“The outer (abaxonal) Schwann cell plasma membrane is specialized for extracellular interactions, while the inner (adaxonal) Schwann cell plasma membrane contains cell adhesion molecules (Previtali et al., 2001;Maurel et al., 2007;Spiegel et al, 2007).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked — no full text of the sourcecites Previtali et al., 2001 source
- Partly supportedcites Maurel et al., 2007 sourceExperimental — accuracy not yet measured
The Results section, “Necl proteins are localized at the internode of myelinated axons” (page not available), places the adhesion protein Necl-4 on the periaxonal Schwann cell membrane adjacent to the axon, supporting the adaxonal part of the claim. Our copy does not establish that the outer, abaxonal membrane is specialized for extracellular interactions. (LLM, conf=0.87)
- Partly supportedcites Spiegel et al, 2007 sourceExperimental — accuracy not yet measured
The Results (page not provided) locate the adhesion molecule Necl4 at the adaxonal Schwann cell membrane, supporting the claim about the inner membrane. The paper identifies β-dystroglycan as an abaxonal membrane marker but does not establish that the outer membrane is specialized for extracellular interactions. (LLM, conf=0.87)
plain- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim. The paper’s conclusions may not depend on it. It may state two separate claims as one.
Every source the paper cites for this claim was checked and supports it.
In the paper
“An extended interaction domain consisting of several tetratricopeptide repeat repeats (which are contained in SH3TC2 as well) has been reported to enable p67phox to interact with Rac1, a small GTPase of the Rho family (Lapouge et al., 2000).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Supportedcites Lapouge et al., 2000 source
The Results and Discussion report that p67phox’s N-terminal domain contains four predicted TPR motifs and binds activated Rac1. The Abstract identifies the Rac–p67phox complex as a Rho-family GTPase interaction. The SH3TC2 parenthetical is contextual framing, not a finding attributed to this source. (LLM, conf=0.91)
plaindirect- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim. The paper’s conclusions may not depend on it.
- Supported
In the paper
“while there is no binding to the dominant negative guanosine diphosphate-bound Rab11_S25N mutants or to Rab11a_Q70L_I44E, a constitutively active mutant which has a second mutation in its effector domain abrogating effector binding (Wallace et al., 2002) (Fig. 5A and Supplementary Fig. 4B).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Not checked — no full text of the sourcecites Wallace et al., 2002 source
plain- Asserted: passes — The authors put it forward as true.
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It may state two separate claims as one.
A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“No association was observed with Rab4a and Rab5a (associated with early endosomes) and Rab7a (associated with late endosomes) (Supplementary Fig. 4A).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
plain- Asserted: passes — The authors put it forward as true.
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It may state two separate claims as one.
A result the paper reports from its own data — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“We found that Rab11a was prominently expressed in cultured Schwann cells and in the PNS of adult animals.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
plain- Asserted: passes — The authors put it forward as true.
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It may state two separate claims as one.
In the paper
“the gene product, SH3TC2, localizes to the perinuclear recycling compartment.”
- Also stated in: § Introduction · plain
“localizes to the plasma membrane and a perinuclear membranous structure (Arnaud et al., 2009;Lupo et al., 2009) that corresponds to the endocytic recycling compartment (Arnaud et al., 2009).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Abstract
What the sources say
- Not matchedcites Arnaud et al., 2009;Lupo et al., 2009
Couldn’t be matched to a paper in Crossref, so it can’t be checked.
plain- Asserted: passes — The authors put it forward as true.
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It may state two separate claims as one.
- Also stated in: § Introduction · plain
In the paper
“Finally, myelin proteins undergo endocytic recycling in oligodendrocytes (Winterstein et al., 2008), which are the myelinating glia cells in the CNS.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked — no full text of the sourcecites Winterstein et al., 2008 source
plain- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim. The paper’s conclusions may not depend on it.
A claim is checked only against the cited paper’s full text, never its abstract, and none is available for its sources.
In the paper
“One crucial mechanism underlying establishment and maintenance of cellular polarity is differential sorting of proteins to distinct plasma membrane domains along the secretory and endosomal pathways (Keller et al., 2001;Kreitzer et al., 2003).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
plaindirect- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim. The paper’s conclusions may not depend on it.
In the paper
“Finally, the membranes of Schwann cell microvilli contain ligands for axonal cell adhesion molecules and contribute to sodium channel clustering at the nodes of Ranvier (Eshed et al., 2005).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked — no full text of the sourcecites Eshed et al., 2005 source
plaindirect- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim. The paper’s conclusions may not depend on it. It may state two separate claims as one.
None of the sources cited for this claim have been checked yet.
In the paper
“Membranes of the paranodal loops are enriched for junctional proteins that permit intra-Schwann cell junctions and Schwann cell-axon junctions (Fannon et al., 1995;Boyle et al., 2001).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
plaincorrelational- Asserted: passes — The authors put it forward as true.
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- Load-bearing: fails — The paper’s conclusions may not depend on it.
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Probably not a substantial claim. The paper’s conclusions may not depend on it.
A conclusion the paper draws from its own results — it cites no other source for it, so there is none to check. The quote shows it is in the paper.
In the paper
“Therefore, a more likely explanation would be that the mutated residues are all found on the surface of the protein, forming an extended Rab11-interacting domain.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
hedgedcorrelational- Asserted: passes — The authors put it forward as true.
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- Based: unclear — It is unclear why the paper says it holds.
It may state two separate claims as one.
None of the sources cited for this claim have been checked yet.
In the paper
“Rab11 is a key regulator of recycling pathways from endosomes to the plasma membrane (Ullrich et al., 1996) and consists of two isoforms, Rab11a and Rab11b, which are encoded by different genes and mainly differ in their C-termini.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Not checked yetcites Ullrich et al., 1996 source
Checked before — its earlier verdict is held back until a new check runs.
plaindirect- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim. The paper’s conclusions may not depend on it.
- Not checked yet
In the paper
“Rab11, a master regulator of recycling endosome functions (Maxfield and McGraw, 2004)”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked — no full text of the sourcecites Maxfield and McGraw, 2004 source
plaindirect- Asserted: passes — The authors put it forward as true.
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- Load-bearing: fails — The paper’s conclusions may not depend on it.
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Probably not a substantial claim. The paper’s conclusions may not depend on it.
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In the paper
“as it has been shown that Rab effectors can stabilize Rab-GTPases (Ganley et al., 2004).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Not checked yetcites Ganley et al., 2004 source
Checked before — its earlier verdict is held back until a new check runs.
plaindirect- Asserted: passes — The authors put it forward as true.
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Probably not a substantial claim.
- Not checked yet
None of the sources cited for this claim have been checked yet.
In the paper
“Several lines of evidence indicate that protein transport and targeting play an important role in myelin membrane assembly.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
hedgedcorrelational- Asserted: passes — The authors put it forward as true.
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- Load-bearing: passes — The paper’s conclusions depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim.
In the paper
“Assays for transferrin receptor recycling are commonly used as a paradigm to test recycling endosome functions (Sager et al., 1984).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Not checked — no full text of the sourcecites Sager et al., 1984 source
plain- Asserted: passes — The authors put it forward as true.
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- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it.
In the paper
“There is at least one known Rab11 effector, Rab11-FIP4, which causes a dramatic condensation of the recycling endosome (Wallace et al., 2002).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Not checked — no full text of the sourcecites Wallace et al., 2002 source
plaindirect- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it.
None of the sources cited for this claim have been checked yet.
In the paper
“During the preparation our manuscript, another article reporting the SH3TC2/Rab11 interaction and a potential effect of SH3TC2 on recycling endosome function was published (Roberts et al., 2010).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked yetcites Roberts et al., 2010 source
Checked before — its earlier verdict is held back until a new check runs.
plaincorrelational- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
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Probably not a substantial claim. The paper’s conclusions may not depend on it. It may state two separate claims as one.
- Not checked yet
A claim is checked only against the cited paper’s full text, never its abstract, and none is available for its sources.
In the paper
“Similarly, in polarized epithelial cells, transfected Schwann cell proteins are distinctly transported, potentially reflecting the segregation of compact and non-compact myelin components (Minuk and Braun, 1996;Kroepfl and Gardinier, 2001;Maier et al., 2006).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
hedgedcorrelational- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it.
In the paper
“Myelination requires dramatic changes in the cellular architecture of differentiating glia and a high degree of cell polarization that partitions the plasma membrane into distinct domains (Pfeiffer et al., 1993).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked — no full text of the sourcecites Pfeiffer et al., 1993 source
plaindirect- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it. It may state two separate claims as one.
In the paper
“Compact myelin in the PNS is largely composed of lipids (mainly cholesterol and sphingolipids) and exhibits narrow lamellar spacings, mediated by homophilic adhesion molecules (Filbin et al., 1990).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked — no full text of the sourcecites Filbin et al., 1990 source
plaindirect- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it. It may state two separate claims as one.
In the paper
“As there is increasing evidence that the development and maintenance of Schwann cells and the myelin sheath require endosomal sorting (Trapp et al., 2004;Simons and Trotter, 2007), we decided to follow the SH3TC2/Rab11 interaction further.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Results
What the sources say
- Not matchedcites Trapp et al., 2004;Simons and Trotter, 2007
Couldn’t be matched to a paper in Crossref, so it can’t be checked.
hedgeddirect- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: passes — The paper’s conclusions depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim.
- Not matched
A claim is checked only against the cited paper’s full text, never its abstract, and none is available for its sources.
In the paper
“Myelinating Schwann cells have at least five distinct membrane domains (Arroyo and Scherer, 2000;Simons and Trotter, 2007) that differ in their membrane lipid and protein composition.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
plain- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it.
None of the sources cited for this claim have been checked yet.
In the paper
“Sorting in the trans-Golgi network partitions myelin proteins into separate transport vesicles (Trapp et al., 1995) that are targeted to distinct Schwann cell compartments.”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
What the sources say
- Not checked yetcites Trapp et al., 1995 source
Checked before — its earlier verdict is held back until a new check runs.
plaindirect- Asserted: passes — The authors put it forward as true.
- Checkable: passes — It can be checked against evidence.
- Load-bearing: fails — The paper’s conclusions may not depend on it.
- Based: passes — The paper gives a reason why it holds.
Probably not a substantial claim. The paper’s conclusions may not depend on it.
- Not checked yet
The paper relies on this without support.
No proteins interacting with SH3TC2 had been identified before this study.
“However, no protein binding to SH3TC2 has been identified so far.”
The paper relies on this without support.
The physiological function of SH3TC2 had not yet been identified, and the pathomechanism causing impaired PNS myelination in CMT4C patients and Sh3tc2 knockout mice remained unclear.
“However, as the physiological function of the gene product, SH3TC2, has not yet been identified, the pathomechanism causing impaired PNS myelination in CMT4C patients and Sh3tc2 knockout mice has so far remained unclear.”
The paper relies on this without support.
There were no published data on Rab11 expression in the PNS.
“As there were no published data on Rab11 expression in the PNS, we studied Rab11a protein levels in Schwann cell lines, primary rat Schwann cell cultures and sciatic nerves of rat and mouse by western blotting (Fig. 3A).”
The paper relies on this without support.
The putative protein-binding motifs in SH3TC2 did not allow prediction of a bona fide interaction partner.
“as the putative protein binding motifs in SH3TC2 did not allow prediction of a bona fide interaction partner, we decided to perform an unbiased screen for interacting proteins.”
The paper relies on this without support.
Rab proteins cycle between inactive GDP-bound and active GTP-bound conformations.
“Like all GTPases, Rab proteins cycle between an inactive (guanosine diphosphatebound) and an active (GTP-bound) conformation.”
The paper relies on this without support.
The GTP-bound activated form of a Rab GTPase associates with effector molecules through which it carries out its functions.
“The GTP-bound, activated form of the GTPase associates with effector molecules through which it carries out its functions.”
The authors state this as an assumption.
Endocytic recycling may assist morphogenesis of the myelin sheath by sorting and redirecting myelin components.
“Assuming that endocytic recycling may assist morphogenesis of the myelin sheath by sorting and redirecting myelin components”
The paper relies on this without support.
Molecular pathways regulating vesicular transport during myelination have remained largely unknown.
“However, molecular pathways regulating vesicular transport during myelination have remained largely unknown.”
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
Each contradiction between two passages listed here was confirmed by an independent check.
Rated minor: it would not touch a result — a value no result rests on, a p-value that stays on the same side of the significance level, or a conflict that is clerical, resolved elsewhere in the paper, or outside its own findings.
In the paper
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > In vitro myelination
“Myelination was induced over 10 consecutive days with C-medium supplemented with 50 mg/ml ascorbic acid (Sigma).”
§ SH3TC2, a protein mutant in Charcot-Marie-Tooth neuropathy, links peripheral nerve myelination to endosomal recycling > Discussion
“(A) Immunofluorescence studies of primary rat Schwann cells/mouse dorsal root ganglion neuron co-cultures after 12 days of myelination.”
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Metadata
- Authors
- Stendel, C., Roos, A., Kleine, H., Arnaud, E., Özçelik, M., Sidiropoulos, P. N. M., Zenker, J., Schüpfer, F., Lehmann, U., Sobota, R. M., Litchfield, D. W., Lüscher, B., Chrast, R., Suter, U., & Senderek, J.
- Affiliations
- ETH Zurich (CH)
- RWTH Aachen University (DE)
- Centre Hospitalier Universitaire Vaudois (CH)
- University of Southern Denmark (DK)
- Western University (CA)
- ORCID
- Venue
- Brain
- Publisher
- Oxford University Press (OUP)
- Published
- 2010-07-24
- Volume
- 133
- Issue
- 8
- Pages
- 2462-2474
- ISSN
- 0006-8950 (ISSN-L)
- 1460-2156
- OpenAlex
- W2150495344
- Semantic Scholar
- 70bc7cade3b8bdee6e5412858bfe471eb390536c
- Cited by
- 102
- References
- 53
- Citations by year
- 2026: 1 · 2025: 6 · 2024: 8 · 2023: 5 · 2022: 6 · 2021: 5 · 2020: 6 · 2019: 3 · 2018: 8 · 2017: 6 · 2016: 8 · 2015: 3 · 2014: 10 · 2013: 8 · 2012: 14
- FWCI
- 3.01
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Brain (publishedVersion)
- PubMed (publishedVersion)
- SERVAL (Université de Lausanne) (publishedVersion, other-oa)
- reroDoc Digital Library (submittedVersion)
- IRIS (submittedVersion)
- Scholarship@Western (Western University) (submittedVersion)
- RWTH Publications (RWTH Aachen) (submittedVersion)
- Repository for Publications and Research Data (ETH Zurich) (submittedVersion, other-oa)
- ETH Zürich Research Collection
- Funders
- National Science Foundation
- Vanderbilt University
- Vanderbilt University Medical Center
- University College Cork
- Deutsche Forschungsgemeinschaft
- Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung
- RWTH Aachen University
- Topics
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Hereditary Neurological Disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Cellular transport and secretion
- Health Sciences › Medicine › Clinical Neurology › Peripheral Neuropathies and Disorders
- Keywords
- SH3TC2, Charcot-Marie-Tooth disease, peripheral nerve myelination, Schwann cells, Rab11, endosomal recycling, membrane trafficking, myelination
- MeSH terms
- rab11 GTP-Binding Proteins, Animals, Carrier Proteins, Cell Line, Chlorocebus aethiops, Charcot-Marie-Tooth Disease, Ganglia, Spinal, Humans, Mutation, Myelin Sheath, Peripheral Nerves, Endosomes, Schwann Cells, Sciatic Nerve, Mice, Knockout, COS Cells, rab GTP-Binding Proteins, Intracellular Signaling Peptides and Proteins, Mice, Rats
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
A language model drew the claims from the paper on 2026-11-10, under version 3 of the claim definition (model deepseek-v4.1-flash, prompt extract-v3).
A second model labelled each claim on 2026-11-10 (model jev-1.13-20260917, definition revision gold-guideline-v0.2).
Cited sources are checked with a language model’s help (gpt-6-sol).
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
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