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Thrombocytopenia 13, syndromic

MedGen UID:
1052978
Concept ID:
CN377271
Disease or Syndrome
Synonym: THROMBOCYTOPENIA, AUTOSOMAL RECESSIVE, 13
 
Gene (location): GALE (1p36.11)
 
Monarch Initiative: MONDO:0958333
OMIM®: 620776

Definition

Syndromic thrombocytopenia-13 (THC13) is an autosomal recessive disorder characterized mainly by congenital thrombocytopenia resulting in increased bleeding. Platelets tend to be enlarged (macrothrombocytopenia) and/or gray and show functional defects. Some patients have infection-induced leukopenia or anemia and pancytopenia. Additional more variable features have also been reported, including mitral valve malformations, pyloric stenosis, and impaired intellectual development (Seo et al., 2019; Febres-Aldana et al., 2020; Marin-Quilez et al., 2023). For a discussion of genetic heterogeneity of thrombocytopenia, see THC1 (313900). [from OMIM]

Professional guidelines

PubMed

Fattizzo B, Marchetti M, Michel M, Cantoni S, Frederiksen H, Giordano G, Glenthøj A, González-López TJ, Murakhovskaya I, Napolitano M, Mingot ME, Arguello M, Patriarca A, Raso S, Vianelli N, Barcellini W
Lancet Haematol 2024 Aug;11(8):e617-e628. Epub 2024 Jul 2 doi: 10.1016/S2352-3026(24)00144-3. PMID: 38968944
Sciascia S, Amigo MC, Roccatello D, Khamashta M
Nat Rev Rheumatol 2017 Sep;13(9):548-560. Epub 2017 Aug 3 doi: 10.1038/nrrheum.2017.124. PMID: 28769114
Carson JL, Guyatt G, Heddle NM, Grossman BJ, Cohn CS, Fung MK, Gernsheimer T, Holcomb JB, Kaplan LJ, Katz LM, Peterson N, Ramsey G, Rao SV, Roback JD, Shander A, Tobian AA
JAMA 2016 Nov 15;316(19):2025-2035. doi: 10.1001/jama.2016.9185. PMID: 27732721

Recent clinical studies

Etiology

Platzbecker U, Santini V, Fenaux P, Sekeres MA, Savona MR, Madanat YF, Díez-Campelo M, Valcárcel D, Illmer T, Jonášová A, Bělohlávková P, Sherman LJ, Berry T, Dougherty S, Shah S, Xia Q, Sun L, Wan Y, Huang F, Ikin A, Navada S, Feller F, Komrokji RS, Zeidan AM
Lancet 2024 Jan 20;403(10423):249-260. Epub 2023 Dec 1 doi: 10.1016/S0140-6736(23)01724-5. PMID: 38048786
Sehgal A, Hoda D, Riedell PA, Ghosh N, Hamadani M, Hildebrandt GC, Godwin JE, Reagan PM, Wagner-Johnston N, Essell J, Nath R, Solomon SR, Champion R, Licitra E, Fanning S, Gupta N, Dubowy R, D'Andrea A, Wang L, Ogasawara K, Thorpe J, Gordon LI
Lancet Oncol 2022 Aug;23(8):1066-1077. Epub 2022 Jul 12 doi: 10.1016/S1470-2045(22)00339-4. PMID: 35839786
Kamdar M, Solomon SR, Arnason J, Johnston PB, Glass B, Bachanova V, Ibrahimi S, Mielke S, Mutsaers P, Hernandez-Ilizaliturri F, Izutsu K, Morschhauser F, Lunning M, Maloney DG, Crotta A, Montheard S, Previtali A, Stepan L, Ogasawara K, Mack T, Abramson JS; TRANSFORM Investigators
Lancet 2022 Jun 18;399(10343):2294-2308. doi: 10.1016/S0140-6736(22)00662-6. PMID: 35717989
Brocklebank V, Wood KM, Kavanagh D
Clin J Am Soc Nephrol 2018 Feb 7;13(2):300-317. Epub 2017 Oct 17 doi: 10.2215/CJN.00620117. PMID: 29042465Free PMC Article
Kremer Hovinga JA, Coppo P, Lämmle B, Moake JL, Miyata T, Vanhoorelbeke K
Nat Rev Dis Primers 2017 Apr 6;3:17020. doi: 10.1038/nrdp.2017.20. PMID: 28382967

Diagnosis

Fattizzo B, Marchetti M, Michel M, Cantoni S, Frederiksen H, Giordano G, Glenthøj A, González-López TJ, Murakhovskaya I, Napolitano M, Mingot ME, Arguello M, Patriarca A, Raso S, Vianelli N, Barcellini W
Lancet Haematol 2024 Aug;11(8):e617-e628. Epub 2024 Jul 2 doi: 10.1016/S2352-3026(24)00144-3. PMID: 38968944
Cavero T, Auñón P, Caravaca-Fontán F, Trujillo H, Arjona E, Morales E, Guillén E, Blasco M, Rabasco C, Espinosa M, Blanco M, Rodríguez-Magariños C, Cao M, Ávila A, Huerta A, Rubio E, Cabello V, Barros X, Goicoechea de Jorge E, Rodríguez de Córdoba S, Praga M
Nephrol Dial Transplant 2023 May 4;38(5):1217-1226. doi: 10.1093/ndt/gfac248. PMID: 36002030
Palma LMP, Vaisbich-Guimarães MH, Sridharan M, Tran CL, Sethi S
Pediatr Nephrol 2022 Sep;37(9):1967-1980. Epub 2022 Jan 18 doi: 10.1007/s00467-021-05370-8. PMID: 35041041Free PMC Article
Brocklebank V, Wood KM, Kavanagh D
Clin J Am Soc Nephrol 2018 Feb 7;13(2):300-317. Epub 2017 Oct 17 doi: 10.2215/CJN.00620117. PMID: 29042465Free PMC Article
Kremer Hovinga JA, Coppo P, Lämmle B, Moake JL, Miyata T, Vanhoorelbeke K
Nat Rev Dis Primers 2017 Apr 6;3:17020. doi: 10.1038/nrdp.2017.20. PMID: 28382967

Therapy

Platzbecker U, Santini V, Fenaux P, Sekeres MA, Savona MR, Madanat YF, Díez-Campelo M, Valcárcel D, Illmer T, Jonášová A, Bělohlávková P, Sherman LJ, Berry T, Dougherty S, Shah S, Xia Q, Sun L, Wan Y, Huang F, Ikin A, Navada S, Feller F, Komrokji RS, Zeidan AM
Lancet 2024 Jan 20;403(10423):249-260. Epub 2023 Dec 1 doi: 10.1016/S0140-6736(23)01724-5. PMID: 38048786
Kamdar M, Solomon SR, Arnason J, Johnston PB, Glass B, Bachanova V, Ibrahimi S, Mielke S, Mutsaers P, Hernandez-Ilizaliturri F, Izutsu K, Morschhauser F, Lunning M, Maloney DG, Crotta A, Montheard S, Previtali A, Stepan L, Ogasawara K, Mack T, Abramson JS; TRANSFORM Investigators
Lancet 2022 Jun 18;399(10343):2294-2308. doi: 10.1016/S0140-6736(22)00662-6. PMID: 35717989
Asakura H, Ogawa H
Int J Hematol 2021 Jan;113(1):45-57. Epub 2020 Nov 7 doi: 10.1007/s12185-020-03029-y. PMID: 33161508Free PMC Article
Brocklebank V, Wood KM, Kavanagh D
Clin J Am Soc Nephrol 2018 Feb 7;13(2):300-317. Epub 2017 Oct 17 doi: 10.2215/CJN.00620117. PMID: 29042465Free PMC Article
Kremer Hovinga JA, Coppo P, Lämmle B, Moake JL, Miyata T, Vanhoorelbeke K
Nat Rev Dis Primers 2017 Apr 6;3:17020. doi: 10.1038/nrdp.2017.20. PMID: 28382967

Prognosis

Fattizzo B, Marchetti M, Michel M, Cantoni S, Frederiksen H, Giordano G, Glenthøj A, González-López TJ, Murakhovskaya I, Napolitano M, Mingot ME, Arguello M, Patriarca A, Raso S, Vianelli N, Barcellini W
Lancet Haematol 2024 Aug;11(8):e617-e628. Epub 2024 Jul 2 doi: 10.1016/S2352-3026(24)00144-3. PMID: 38968944
Platzbecker U, Santini V, Fenaux P, Sekeres MA, Savona MR, Madanat YF, Díez-Campelo M, Valcárcel D, Illmer T, Jonášová A, Bělohlávková P, Sherman LJ, Berry T, Dougherty S, Shah S, Xia Q, Sun L, Wan Y, Huang F, Ikin A, Navada S, Feller F, Komrokji RS, Zeidan AM
Lancet 2024 Jan 20;403(10423):249-260. Epub 2023 Dec 1 doi: 10.1016/S0140-6736(23)01724-5. PMID: 38048786
Fakhouri F, Fila M, Hummel A, Ribes D, Sellier-Leclerc AL, Ville S, Pouteil-Noble C, Coindre JP, Le Quintrec M, Rondeau E, Boyer O, Provôt F, Djeddi D, Hanf W, Delmas Y, Louillet F, Lahoche A, Favre G, Châtelet V, Launay EA, Presne C, Zaloszyc A, Caillard S, Bally S, Raimbourg Q, Tricot L, Mousson C, Le Thuaut A, Loirat C, Frémeaux-Bacchi V
Blood 2021 May 6;137(18):2438-2449. doi: 10.1182/blood.2020009280. PMID: 33270832
Asakura H, Ogawa H
Int J Hematol 2021 Jan;113(1):45-57. Epub 2020 Nov 7 doi: 10.1007/s12185-020-03029-y. PMID: 33161508Free PMC Article
Brocklebank V, Wood KM, Kavanagh D
Clin J Am Soc Nephrol 2018 Feb 7;13(2):300-317. Epub 2017 Oct 17 doi: 10.2215/CJN.00620117. PMID: 29042465Free PMC Article

Clinical prediction guides

Platzbecker U, Santini V, Fenaux P, Sekeres MA, Savona MR, Madanat YF, Díez-Campelo M, Valcárcel D, Illmer T, Jonášová A, Bělohlávková P, Sherman LJ, Berry T, Dougherty S, Shah S, Xia Q, Sun L, Wan Y, Huang F, Ikin A, Navada S, Feller F, Komrokji RS, Zeidan AM
Lancet 2024 Jan 20;403(10423):249-260. Epub 2023 Dec 1 doi: 10.1016/S0140-6736(23)01724-5. PMID: 38048786
Beck DB, Bodian DL, Shah V, Mirshahi UL, Kim J, Ding Y, Magaziner SJ, Strande NT, Cantor A, Haley JS, Cook A, Hill W, Schwartz AL, Grayson PC, Ferrada MA, Kastner DL, Carey DJ, Stewart DR
JAMA 2023 Jan 24;329(4):318-324. doi: 10.1001/jama.2022.24836. PMID: 36692560Free PMC Article
Kamdar M, Solomon SR, Arnason J, Johnston PB, Glass B, Bachanova V, Ibrahimi S, Mielke S, Mutsaers P, Hernandez-Ilizaliturri F, Izutsu K, Morschhauser F, Lunning M, Maloney DG, Crotta A, Montheard S, Previtali A, Stepan L, Ogasawara K, Mack T, Abramson JS; TRANSFORM Investigators
Lancet 2022 Jun 18;399(10343):2294-2308. doi: 10.1016/S0140-6736(22)00662-6. PMID: 35717989
Dai ZN, Peng XF, Li JC, Zhao J, Wu YX, Yang X, Yang T, Zhang SF, Dai K, Guan XG, Yuan C, Yang ZD, Cui N, Lu QB, Huang Y, Fan H, Zhang XA, Xiao GF, Peng K, Zhang LK, Liu W, Li H
Emerg Microbes Infect 2022 Dec;11(1):1672-1682. doi: 10.1080/22221751.2022.2081617. PMID: 35603493Free PMC Article
Ferreri AJ, Cwynarski K, Pulczynski E, Ponzoni M, Deckert M, Politi LS, Torri V, Fox CP, Rosée PL, Schorb E, Ambrosetti A, Roth A, Hemmaway C, Ferrari A, Linton KM, Rudà R, Binder M, Pukrop T, Balzarotti M, Fabbri A, Johnson P, Gørløv JS, Hess G, Panse J, Pisani F, Tucci A, Stilgenbauer S, Hertenstein B, Keller U, Krause SW, Levis A, Schmoll HJ, Cavalli F, Finke J, Reni M, Zucca E, Illerhaus G; International Extranodal Lymphoma Study Group (IELSG)
Lancet Haematol 2016 May;3(5):e217-27. Epub 2016 Apr 6 doi: 10.1016/S2352-3026(16)00036-3. PMID: 27132696

Recent systematic reviews

Golub D, McBriar JD, Donaldson H, Wong T, Unadkat P, White TG, Quach ET, Haddock S, Chitti B, Ziemba Y, Goenka A, Singer S, Schulder M, Dehdashti AR
J Neurooncol 2023 Nov;165(2):229-239. Epub 2023 Nov 13 doi: 10.1007/s11060-023-04499-w. PMID: 37955760
Mossayebi MH, Iyer NS, McLaren RA Jr, Moussa HN, Sibai BM, Al-Kouatly HB
Am J Obstet Gynecol 2023 Nov;229(5):502-515.e10. Epub 2023 May 5 doi: 10.1016/j.ajog.2023.04.046. PMID: 37150281
Wang Y, Song Z, Wei X, Yuan H, Xu X, Liang H, Wen H
PLoS Negl Trop Dis 2022 Jun;16(6):e0010489. Epub 2022 Jun 17 doi: 10.1371/journal.pntd.0010489. PMID: 35714138Free PMC Article
Raina R, Sethi SK, Dragon-Durey MA, Khooblall A, Sharma D, Khandelwal P, Shapiro R, Boyer O, Yap HK, Bagga A, Licht C
Pediatr Nephrol 2022 Jul;37(7):1479-1493. Epub 2022 Feb 3 doi: 10.1007/s00467-022-05451-2. PMID: 35118546
Yue J, Dong BR, Yang M, Chen X, Wu T, Liu GJ
Cochrane Database Syst Rev 2016 Jan 7;2016(1):CD008056. doi: 10.1002/14651858.CD008056.pub3. PMID: 26758498Free PMC Article

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