Reduced Cancer Incidence in Huntington’s Disease: Analysis in the Registry Study.
J Huntingtons Dis.. 2018; 7(3): 209-222
DOI: 10.3233/jhd-170263
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1. J Huntingtons Dis. 2018;7(3):209-222. doi: 10.3233/JHD-170263.
Reduced Cancer Incidence in Huntington’s Disease: Analysis in the Registry Study.
McNulty P, Pilcher R, Ramesh R, Necuiniate R, Hughes A, Farewell D, Holmans P,
Jones L; REGISTRY Investigators of the European Huntington’s Disease Network.
Collaborators: Bonelli RM, Hecht K, Herranhof B, Holl A, Kapfhammer HP, Koppitz
M, Lilek S, Magnet M, Müller N, Otti D, Painold A, Reisinger K, Scheibl M,
Schöggl H, Ullah J, Braunwarth EM, Brugger F, Buratti L, Hametner EM, Hepperger
C, Holas C, Hotter A, Hussl A, Larcher B, Mahlknecht P, Müller C, Pinter B, Poewe
W, Reiter EM, Seppi K, Sprenger F, Wenning G, Ladurner G, Lilek S, Sinadinosa D,
Staffen W, Walleczek AM, Constant E, Gillardin AF, Léonard MC, Verellen-Dumoulin
C, van de Wyngaerde F, Dupuis M, Minet C, Ribaï P, Paemel DV, Boogaerts A,
Vandenberghe W, Reijen DV, Kaiserova M, Šenkárová Z, Klempíř J, Majerová V, Roth
J, Hasselstrøm Madsen L, Møller AT, Hjermind L, Jacobsen O, Lindquist S, Nielsen
J, Regeur L, Stockholm J, Larsen IU, Vangsted-Hansen C, Vinther-Jensen T, Lolk A,
Lundsgaard M, Wermuth L, Andersson C, Nyberg C, Sundblom J, Peippo M, Sipponen M, Hartikainen P, Ollokainen M, Åman J, Ignatius J, Kärppä M, Mustonen A, Kajula O, Jääskalainen O, Moilanen J, Santala M, Eklund P, Hiivola H, Hyppönen H,
Martikainen K, Tuuha K, Allain P, Bonneau D, Bost M, Gohier B, Guérid MA, Olivier
A, Prouzet J, Prundean A, Scherer-Gagou C, Verny C, Babiloni B, Debruxelles S,
Duché C, Goizet C, Jameau L, Lafoucrière D, Spampinato U, Couttier J, Debilly B,
Delaigue C, Durif F, Legendre P, Loiseau S, Ulla M, Vidal T, Bachoud-Lévi AC,
Badei F, Boissé MF, Boudali L, de Langavant LC, Lemoine L, Morgado G, Youssov K,
Annic A, Barthélémy R, De Bruycker C, Cabaret M, Carette AS, Carrière N, Decorte
E, Defebvre L, Delliaux M, Delval A, Depelchin A, Destee A, Dewulf-Pasz N,
Dondaine T, Dugauquier F, Dujardin K, Hopes L, Krystkowiak P, Lemaire MH,
Manouvrier S, Mutez E, Peter M, Plomhause L, Sablonnière B, Simonin C, Tard C,
Thibault-Tanchou S, Vuillaume I, Bellonet M, Benoit A, Berrisoul H, Blin S,
Courtin F, Duru C, Fasquel V, Flament M, Godefroy O, Mantaux B, Playe A, Roussel
M, Tir M, Schüler B, Wannepain S, Azulay JP, Chabot C, Delfini M, Eusebio A,
Fluchere F, Grosjean H, Mundler L, Nowak M, Raseta R, Benaich S, Brice A, Charles
P, Durr A, Ewenczyk C, Francisque H, Jauffret C, Justo D, Kassar A, Klebe S,
Lesne F, Milani P, Monin ML, Roze E, Tataru A, Tchikviladzé M, Bioux S, Bliaux E,
Girard C, Guyant-Maréchal L, Hannequin D, Hannier V, Jourdain S, Maltête D,
Pouliquen D, Lagha-Boukbiza O, Longato N, Marcel C, Phillipps C, Rudolf G,
Steinmetz G, Tranchant C, Wagner C, Zimmermann MA, Blondeau L, Calvas F, Cheriet
S, Delabaere H, Demonet JF, Pariente J, Pierre M, Rolland S, Kosinski CM,
Milkereit E, Probst D, Reetz K, Sass C, Schiefer J, Schlangen C, Werner CJ, Beuth
M, Gelderblom H, Priller J, Prüß H, Spruth E, Thiel S, Andrich J, Ellrichmann G,
Herrmann L, Hoffmann R, Kaminski B, Kraus P, Saft C, Stamm C, Lange H, Maiwald R,
Bosredon C, Hunger U, Löhle M, Maass A, Ossig C, Schmidt S, Storch A, Wolz A,
Wolz M, Kohl Z, Kozay C, Ullah J, Winkler J, Bergmann U, Böringer R, Capetian P,
Kammel G, Lambeck J, Mächtel M, Meier S, Rijntjes M, Zucker B, Boelmans K, Ganos
C, Goerendt I, Heinicke W, Hidding U, Lewerenz J, Münchau A, Orth M, Schmalfeld
J, Stubbe L, Zittel S, Diercks G, Dressler D, Francis F, Gayde-Stephan S,
Gorzolla H, Kramer B, Minschke R, Schrader C, Tacik P, Ribbat M, Longinus B,
Lüsebrink A, Mühlau M, Peinemann A, Städtler M, Weindl A, Winkelmann J, Ziegler
C, Bechtel N, Beckmann H, Bohlen S, Göpfert N, Hölzner E, Reilmann R, Rohm S,
Rumpf S, Sass C, Schepers S, Weber N, Bachmeier M, Dose M, Hofstetter N, Marquard
R, Mühlbäck A, Barth K, Buck A, Connemann J, Ecker D, Geitner C, Held C, Kesse A,
Landwehrmeyer B, Lezius F, Nepper S, Niess A, Schneider A, Schwenk D, Süssmuth S,
Trautmann S, Vogel M, Weydt P, Klebe S, Musacchio T, Leypold C, Nöth K, Cormio C,
Difruscolo O, Franco G, Sciruicchio V, Serpino C, Tommaso M, Calandra-Buonaura G,
Capellari S, Cortelli P, Gallassi R, Poda R, Scaglione C, Bertini E, Ghelli E,
Ginestroni A, Mechi C, Paganini M, Piacentini S, Pradella S, Romoli AM, Sorbi S,
Abbruzzese G, Bandettini di Poggio M, Ferrandes G, Mandich P, Marchese R, Di
Maria E, Albanese A, Castagliuolo S, Castaldo A, Di Donato S, Di Bella D, Gellera
C, Genitrini S, Mariotti C, Monza D, Nanetti L, Panzeri M, Paridi D, Soliveri P,
Spagnolo F, Taroni F, Tomasello C, Michele G, Di Maio L, Rinaldi C, Massarelli M,
Peluso S, Roca A, Russo CV, Salvatore E, Sorrentino P, Tucci T, Cannella M,
Codella V, De Gregorio F, De Nicola A, Elifani F, Martino T, Mazzante I,
Petrollini M, Simonelli M, Vezza M, Squitieri F, Lovo F, Bentivoglio AR, Bove F,
Catalli C, Di Giacopo R, Fasano A, Frontali M, Guidubaldi A, Ialongo T, Jacopini
G, Loria G, Modoni A, Petracca M, Piano C, Piccininni C, Quaranta D, Romano S,
Soleti F, Solito M, Spadaro M, Torlizzi F, Zinzi P, Coarelli G, Ferraldeschi M,
Ristori G, Romano S, van Hout MSE, van Vugt JPP, de Weert AM, Verhoeven M, Dekker
M, Leenders N, Oostrom JV, Klooster J, Kremer B, Baake V, van den Bogaard SJA,
Bos R, Dumas EM, ‘t Hart EP, Kampstra A, Roos RAC, Schoonderbeek A, Duits A,
Oosterloo M, Waber M, Verstappen C, Blinkenberg EØ, Hauge E, Tyvoll H, Aaserud O,
Bjørgo K, Borgeød N, Dramstad E, Fannemel M, Frich JC, Gørvell PF, Heiberg A,
Retterstøl L, Røsby O, Sikiric A, Stokke B, Walsem MV, Wehus R, Bjørnevoll I,
Sando SB, Haug MG, Størseth HH, Arntsen V, Dziadkiewicz A, Nowak M, Robowski P,
Sitek E, Slawek J, Soltan W, Szinwelski M, Arkuszewski M, Błaszczyk M,
Boczarska-Jedynak M, Ciach-Wysocka E, Gorzkowska A, Jasińska-Myga B, Kaczmarczyk
A, Kłodowska-Duda G, Opala G, Stompel D, Banaszkiewicz K, Boćwińska D,
Bojakowska-Jaremek K, Dec M, Grabska N, Krawczyk M, Kubowicz E, Malec-Litwinowicz
M, Rudzińska M, Stenwak A, Szczudlik A, Szczygieł E, Wójcik M, Wasielewska A,
Anioła J, Bryl A, Ciesielska A, Klimberg A, Marcinkowski J, Samara H, Sempołowicz
J, Wiśniewski B, Zielonka D, Gogol A, Janik P, Jamrozik Z, Kaminska A, Kwiecinski
H, Antczak J, Jachinska K, Krysa W, Rakowicz M, Richter P, Rola R, Ryglewicz D,
Sienkiewicz-Jarosz H, Stępniak I, Sułek A, Witkowski G, Zaremba J, Zdzienicka E,
Ziora-Jakutowicz K, Januário C, Júlio F, Almeida M, Calado A, Dias M, Morgado J,
Semedo C, Guedes LC, Coelho M, Ferreira JJ, Mestre T, Mendes T, Valadas A, Costa
C, Cardoso H, Andrade C, Costa A, Garrett C, Gago M, Guimarães J, Massano J,
Meireles J, Monteiro A, Herrera CD, Moreno PG, Bas J, Busquets N, Calopa M,
Classen SJ, Dedichá NR, Teresa Buongiorno M, de la Cerda Santa María A, Muñoz E,
Santacruz P, Barbera MA, Sebastián AR, Pardo SA, Guia DB, Calzado N, Hernanz LC,
Tartari Díaz-Zorita JP, Catena JL, Ferrer PQ, Carruesco GT, Robert MF, Viladrich
CM, Roca E, Ruiz Idiago JM, Riballo AV, Campolongo A, de Bobadilla RF, Bojarsky
JK, Martinez-Horta S, Pagonabarraga J, Perez JP, Ribosa R, Villa C, Angeles M,
Gil A, Corrales KB, Gomez Esteban JC, González A, Merino BT, Cubo E, Polo CG,
Mariscal N, Romero SG, Matías Arbelo J, de Molina RM, Martín I, Periañez JM,
Udaeta B, Alonso-Frech F, Del Valle Loarte M, Barrero F, Morales B, Frades B,
Villanueva MÁ, Zea Sevilla MA, Frech FA, Del Mar Fenollar M, García-Ramos García
R, Villanueva C, Bascuñana Garde M, Ventura MF, Caldentey JG, García Ribas G, de
Yébenes JG, López-Sendón Moreno JL, Mañanes Barral V, Cubillo PT, García Ruíz PJ,
García A, López RG, Bárcenas AH, Martínez-Descals A, Martin VP, Martínez NR,
Sainz Artiga MJ, Sánchez V, Martínez Pueyo A, Dolores Alarcón MM, Antúnez Almagro
C, Diéguez E, Fortuna L, Manzanares S, Muñoz JM, Antequera Torres MM, Perea FN,
Vivancos L, González S, Guisasola LM, Prieto MP, Ribacoba R, Salvador C, Lozano
PS, Ramirez IL, Arques PN, Lopera MR, Pastor BV, Gaston I, Garcia-Amigot F,
Martinez-Jaurrieta MD, Ramos-Arroyo MA, Carrillo F, Cáceres Redondo MT, Mir P,
González LV, Damas Hermoso F, García Moreno JM, Lucena CM, Pacheco Cortegana EM,
Chacón Peña J, Redondo L, Sánchez VS, Fernandez CM, Romero Lemos MD, Mata MP,
Villagrán Casado R, Bosca M, Burguera JA, Brugada FC, Vilaplana CP, Solís P,
Jeweinat Figuerola B, Palanca PM, Wahlström J, Høsterey-Ugander U, Fredlund G,
Constantinescu R, Neleborn-Lingefjärd L, Berglund M, Berglund P, Linnsand P,
Björnsson E, Paucar M, Pålhagen S, Svenningsson P, Wallden T, Loutfi G, Olofsson
C, Stattin EL, Westman L, Wikström B, Ekwall C, Göller ML, Sundblom J, Burgunder
JM, Stebler Y, Kaelin A, Romero I, Schüpbach M, Zaugg SW, Esposito F, Good JM,
Paus K, Vingerhoets F, Wider C, Jung HH, Petersen JA, Ligon-Auer M, Mihaylova V,
Downie L, Jack R, Matheson K, Miedzybrodzka Z, Rae D, Simpson SA, Summers F, Ure
A, Vaughan V, Harrower T, Vernon N, Akhtar S, Crooks J, Curtis A, de Souza J,
Rickards H, Wright J, Coulthard E, Hayward B, Sieradzan K, Wright A, Barker RA,
O’Keefe D, Gerrtiz A, Fisher K, Goodman A, Hill S, Mason S, Swain R, Guzman NV,
Busse M, Butcher C, Dunnett S, Clenaghan C, Fullam R, Hunt S, Jones U, Khalil H,
Minster S, Owen M, Price K, Townhill J, Rosser A, Goudie D, Buchanan L, McFadyen
P, Tonner A, Taylor AM, Edwards M, Ho C, McGill M, Porteous M, Pearson P, Irvine
S, Brockie P, Foster J, Johns N, McKenzie S, Rothery J, Thomas G, Yates S, Deith
C, Ireland J, Ritchie S, Burrows L, Fletcher A, Harding A, Laver F, Silva M,
Thomson A, Chu C, Evans C, Gallentree D, Hamer S, Kraus A, Markova I, Raman A,
Andrew A, Frost J, Noad R, Hobson E, Jamieson S, Longthorpe M, Markova I,
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C, Dipple H, Freire-Patino D, Hallam C, Middleton J, Alusi S, Davies R, Foy K,
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Peterson M, Rose S, Andrews T, Dougherty A, Golding C, Kavalier F, Laing H,
Lashwood A, Robertson D, Ruddy D, Santhouse A, Whaite A, Bruno S, Chu E, Doherty
K, Golding C, Haider S, Hensman D, Lewis M, Novak M, Patel A, Robertson N, Rosser
E, Tabrizi S, Taylor R, Warner T, Wild E, Arran N, Bek J, Callaghan J, Craufurd
D, Fullam R, Hare M, Howard L, Huson S, Johnson L, Jones M, Krishnamoorthy A,
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C, Haig-Brown D, Simpson S, Hare M, Majeed T, Bandmann O, Bradbury A, Fairtlough
H, Fillingham K, Foustanos I, Gill P, Kazoka M, O’Donovan K, Nevitt L, Peppa N,
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da Silva WV, Di Renzo M, Finisterra AM, Genoves C, Gilling M, Hvalstedt C,
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E, Zielonka E.
BACKGROUND: People with Huntington’s disease (HD) have been observed to have
lower rates of cancers.
OBJECTIVE: To investigate the relationship between age of onset of HD, CAG repeat
length, and cancer diagnosis.
METHODS: Data were obtained from the European Huntington’s disease network
REGISTRY study for 6540 subjects. Population cancer incidence was ascertained
from the GLOBOCAN database to obtain standardised incidence ratios of cancers in
the REGISTRY subjects.
RESULTS: 173/6528 HD REGISTRY subjects had had a cancer diagnosis. The
age-standardised incidence rate of all cancers in the REGISTRY HD population was
0.26 (CI 0.22-0.30). Individual cancers showed a lower age-standardised incidence
rate compared with the control population with prostate and colorectal cancers
showing the lowest rates. There was no effect of CAG length on the likelihood of
cancer, but a cancer diagnosis within the last year was associated with a greatly
increased rate of HD onset (Hazard Ratio 18.94, p