Pharmacological chaperones stabilize retromer to limit APP processing

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SUPPLEMENTARY INFORMATION Pharmacological chaperones stabilize retromer to limit APP processing Vincent J. Mecozzi 1,3, Diego E. Berman 2,3, Sabrina Simoes 2, Chris Vetanovetz 2, Mehraj R. Awal 1, Vivek M. Patel 2, Remy T. Schneider 2, Gregory A. Petsko 1,4, Dagmar Ringe 1,4, Scott A. Small 2,4 1 Department of Biochemistry and Chemistry, Rosenstiel Basic Medical Sciences Research Center, Brandeis University, Waltham, Massachusetts, 02454, 2 The Taub Institute for Research on Alzheimer s Disease and the Aging Brain, Department of Neurology and Pathology, Columbia University College of Physicians and Surgeons, New York NY 10032 5 Helen and Robert Appel Alzheimer s Disease Research Institute, Weill Cornell Medical College, New York NY 10065 3 These authors contributed equally to this work 4 Correspondence should be addressed to Greg Petsko (gpetsko@med.cornell.edu), Scott A. Small (sas68@columbia.edu), or Dagmar Ringe (ringe@brandeis.edu)

SUPPLEMENTARY RESULTS

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Aβ42 Aβ40 fg/μg fg/μg VEHICLE 5uM R53 VEHICLE 5uM R53 sappα sappβ α-secretase β-secretase Aβ FL-APP βctf

Vps35 Vps26 actin

sappα in media FL-APP βctf

VEHICLE-Δ VEHICLE-CRE NT R55-Δ R55-CRE NT Vps35 actin Vps26

R55-Compound1 Certificate of Analysis Product Name: MedKoo Cat#: TPT-260 (Thiophene thiourea derivative) 504210 Chemical name Thiophene-2,5-diylbis(methylene) dicarbamimidothioate dihydrochloride N/A Synonyms Chemical structure CAS# and Theoretical analysis Name: TPT-260 2HCl MedKoo Code# 504210 CAS# 2076-91-7 Lot#ZY20120915 Chemical Formula: C 8 H 12 N 4 S 3 2HCl Molecular Weight: 333.32 R55-Compound1 TPT-260 (free base): Chemical Formula: C8H12N4S3 Exact Mass: 260.02241 Molecular Weight: 260.40 Batch No.: Mfg. Date: Test Date: Retest date: ZY20120915 September 15, 2012 September 15, 2012 September 14, 2014 Analysis item Specification Results Appearance White solid powder Complies Structure H-NMR and MS spectrum of the sample Complies correspond to the data reported in the literature Purity (HPLC) 97% 98.0% Solubility Soluble in DMF,DMSO Complies Additional information: N/A Conclusion This product conforms with MedKoo s quality s standards Shipping condition Ambient temperature Storage condition Short term storage (weeks): 0 4 o C under dry condition Long term storage (months): -20 o C under dry condition Self life At least 2 years if properly stored. CAUTION: NOT FULLY TESTED. FOR RESEARCH ONLY, NOT FOR HUMAN USE MedKoo Biosciences http://www.medkoo.com sales@medkoo.com P O Box 16222, Chapel Hill, NC 27516-6222, USA Tel: 919-279-0682 Fax: 704-967-0446

R55-Compound1 TPT-260.2HCl ----- C13-NMR analysis solvent: DMSO-d6 Normalized Intensity 1.0 0.9 0.8 0.7 0.6 0.5 0.4 0.3 0.2 0.1 ThipheneBisuraC 168.68 138.83 127.76 39.52 40.35 40.07 39.78 38.67 38.95 39.23 MedKoo Code#: 504210 Name: TPT-260: CAS#: 2076-91-7 (TPT-260, HCl salt) Chemical Formula: C8H14Cl2N4S3 Exact Mass: 331.97576 Molecular Weight: 333.32456 29.31 0 200 180 160 140 120 100 80 60 40 20 0 Chemical Shift (ppm) MedKoo Biosciences www.medkoo.com sales@medkoo.com

R55-Compound1 thiophene-bisuh 1.0 0.9 0.8 TPT-260.2HCl ----- H1-NMR analysis Solvent: DMSO-d6 7.00 4.80 MedKoo Code#: 504210 Name: TPT-260: CAS#: 2076-91-7 (TPT-260, HCl salt) Chemical Formula: C8H14Cl2N4S3 Exact Mass: 331.97576 Molecular Weight: 333.32456 0.7 Normalized Intensity 0.6 0.5 0.4 9.36 9.45 2.49 2.49 0.3 0.2 0.1 0 0.26 0.07 0.15 10 9 8 7 6 5 4 3 2 1 0 Chemical Shift (ppm) MedKoo Biosciences www.medkoo.com sales@medkoo.com

D:\DATA\2012\September2012\n120914\Pan\20120914_EJ_Pan_ZY-2_CHC_RP2465_ Sample 20120914_EJ_Pan_ZY-2_CHC_RP2465_suppOFF\0_C3\1 Comment 1 suppoff\0_c3\1 Pan Comment 2 ZY-2_CHC RP2465_suppressionOFF Intens. [a.u.] R55-Compound1 x10 4 3.0 2.5 185.041 TPT-260 ---- MS analysis 2.0 187.030 (M + K)+ = 299.00 1.5 172.041 190.050 TPT-260 (free base) Chemical Formula: C8H12N4S3 Exact Mass: 260.02241 Molecular Weight: 260.40 298.050 1.0 0.5 0.0 164.070 167.985 173.882 192.056 197.027 202.108 207.053 212.029 215.025 219.029 224.054 228.003 234.018 237.040 160 180 200 220 240 260 280 300 320 m/z m/z SN Quality Fac. Res. Intens. Area 38.971 76.1 1373 910 308.48 14 45.051 23.9 262 1079 96.92 5 62.064 39.1 831 1021 157.50 11 71.093 45.3 808 1208 181.43 13 243.274 248.027 251.007 254.055 259.019 264.024 270.279 273.036 Acquisition method name D:\Methods\flexControlMethods\Specification\RP_2465.par Number of shots 100 Voltage polarity POS Laser beam attenuation 85.185 Date of acquisition 2012-09-14T10:18:53.687-04:00 printed: 9/14/2012 277.119 281.018 10:38:16 AM 293.030 295.038 300.052 305.565 311.033 MedKoo Biosciences www.medkoo.com sales@medkoo.com

R33-Compound3 Certificate of Analysis Product Name: MedKoo Cat#: TPT-172 HCl (Thiophene thiourea derivative) 504211 Chemical name thiophen-2-ylmethyl carbamimidothioate hydrochloride N/A Synonyms Chemical structure R33-Compound3 CAS# and Theoretical analysis Name: TPT-172 HCl MedKoo Code# 504211 CAS# 2032415-42-2 Lot#ZY20120719 Chemical Formula: C 6 H 8 N 2 S 2 HCl Molecular Weight: 208.73 TPT-172 (free base): Chemical Formula: C6H8N2S2 Exact Mass: 172.01289 Molecular Weight: 172.27 Batch No.: Mfg. Date: Test Date: Retest date: ZY20120917 Sepetember 17, 2012 Sepetember 17, 2012 September 16, 2014 Analysis item Specification Results Appearance White solid powder Complies Structure H-NMR and MS spectrum of the sample Complies correspond to the data reported in the literature Purity (HPLC) 97% 98.0% Solubility Soluble in DMF,DMSO Complies Additional information: N/A Conclusion This product conforms with MedKoo s quality s standards Shipping condition Ambient temperature Storage condition Short term storage (weeks): 0 4 o C under dry condition Long term storage (months): -20 o C under dry condition Self life At least 2 years if properly stored. CAUTION: NOT FULLY TESTED. FOR RESEARCH ONLY, NOT FOR HUMAN USE MedKoo Biosciences http://www.medkoo.com sales@medkoo.com P O Box 16222, Chapel Hill, NC 27516-6222, USA Tel: 919-279-0682 Fax: 704-967-0446

R33-Compound3 TPT-172.HCl ----- H1-NMR analysis solvent: DMSO-d6 thiophene-monothiou 4.86 1.0 0.9 0.8 0.7 MedKoo Code#: 504211 Name: TPT-172.HCl CAS#: 32415-42-2 (TPT-172 HCl salt) Lot#ZY20120917 Chemical Formula: C6H9ClN2S2 Molecular Weight: 208.73206 Normalized Intensity 0.6 0.5 0.4 0.3 0.2 9.49 9.38 7.50 7.49 7.16 7.15 6.97 6.96 6.95 0.1 0 0.23 0.05 0.06 0.06 0.13 10 9 8 7 6 5 4 3 2 1 0 Chemical Shift (ppm) MedKoo Biosciences www.medkoo.com sales@medkoo.com

R33-Compound3 TPT-172.HCl ---- C13-NMR analysis solvent: DMSO-d6 Normalized Intensity 1.0 0.9 0.8 0.7 0.6 0.5 0.4 Thiophene-monoUC 168.78 137.91 128.08 127.10 126.84 40.07 39.78 39.50 38.95 39.23 29.26 MedKoo Code#: 504211 Name: TPT-172.HCl CAS#: 32415-42-2 (TPT-172 HCl salt) Lot#ZY20120917 Chemical Formula: C6H9ClN2S2 Molecular Weight: 208.73206 0.3 0.2 0.1 0 200 180 160 140 120 100 80 60 40 20 0 Chemical Shift (ppm) MedKoo Biosciences www.medkoo.com sales@medkoo.com

D:\DATA\2012\September2012\n120914\Pan\20120914_EJ_Pan_ZY-1_CHC_RP2465_ Sample 20120914_EJ_Pan_ZY-1_CHC_RP2465_suppOFF\0_C2\1 Comment 1 suppoff\0_c2\1 Pan Comment 2 ZY-1_CHC RP2465_suppressionOFF Intens. [a.u.] x10 4 2.5 R33-Compound3 173.036 TPT-172 ---- MS analysis 2.0 Name: TPT-172 Chemical Formula: C6H8N2S2 Exact Mass: 172.01289 Molecular Weight: 172.27 1.5 97.029 1.0 0.5 77.041 107.361 127.081 146.072 164.065 190.050 212.010 268.998 294.040 335.081 362.846 379.092 421.192 443.516 475.121 573.681 0.0 100 200 300 400 500 600 700 800 900 1000 m/z m/z SN Quality Fac. Res. Intens. Area 62.065 16.0 158 1078 16.02 1 66.290 13.0 2277 8754 16.25 0 72.288 10.1 123 4493 10.53 0 77.041 189.3 3549 1093 189.86 15 Acquisition method name D:\Methods\flexControlMethods\Specification\RP_2465.par Number of shots 100 Voltage polarity POS Laser beam attenuation 85.185 Date of acquisition 2012-09-14T10:14:55.171-04:00 printed: 9/14/2012 10:32:19 AM MedKoo Biosciences www.medkoo.com sales@medkoo.com