B-complex vitamins for patients with TANGO2-deficiency disorder

doi:10.1002/jimd.12585

. 2023 Mar;46(2):161-162.

doi: 10.1002/jimd.12585. Epub 2022 Dec 30.

B-complex vitamins for patients with TANGO2-deficiency disorder

Sarah E Sandkuhler¹, Lilei Zhang^{2

3}, Joshua K Meisner^{2

4}, Lina Ghaloul-Gonzalez^{2

5}, Cheyenne M Beach^{2

6}, David Harris^{2

7}, Pascale de Lonlay^{2

8}, Seema R Lalani^{2

3}, Christina Y Miyake^{2

9}, Samuel J Mackenzie^{2

10}

Affiliations

¹ Department of Pathology, University of Rochester Medical Center, Rochester, New York, USA.
² TANGO2 Research Foundation, Clinical Advisory Board, Middletown, Connecticut, USA.
³ Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
⁴ Department of Pediatrics, Michigan Medicine, Ann Arbor, Michigan, USA.
⁵ Department of Pediatrics, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, USA.
⁶ Department of Pediatrics, Yale School of Medicine, New Haven, Connecticut, USA.
⁷ Department of Neurology, Michigan Medicine, Ann Arbor, Michigan, USA.
⁸ Department of Pediatrics, Hôpital Necker-Enfants Malades, Paris, France.
⁹ Department of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.
¹⁰ Department of Neurology, University of Rochester Medical Center, Rochester, New York, USA.

PMID: 36550018
PMCID: PMC10204720
DOI: 10.1002/jimd.12585

B-complex vitamins for patients with TANGO2-deficiency disorder

Sarah E Sandkuhler et al. J Inherit Metab Dis. 2023 Mar.

. 2023 Mar;46(2):161-162.

doi: 10.1002/jimd.12585. Epub 2022 Dec 30.

Authors

Affiliations

¹ Department of Pathology, University of Rochester Medical Center, Rochester, New York, USA.
² TANGO2 Research Foundation, Clinical Advisory Board, Middletown, Connecticut, USA.
³ Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
⁴ Department of Pediatrics, Michigan Medicine, Ann Arbor, Michigan, USA.
⁵ Department of Pediatrics, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, USA.
⁶ Department of Pediatrics, Yale School of Medicine, New Haven, Connecticut, USA.
⁷ Department of Neurology, Michigan Medicine, Ann Arbor, Michigan, USA.
⁸ Department of Pediatrics, Hôpital Necker-Enfants Malades, Paris, France.
⁹ Department of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.
¹⁰ Department of Neurology, University of Rochester Medical Center, Rochester, New York, USA.

PMID: 36550018
PMCID: PMC10204720
DOI: 10.1002/jimd.12585

No abstract available

PubMed Disclaimer

Conflict of interest statement

CONFLICT OF INTEREST

The authors have no relevant financial or non-financial interests to disclose.

Cited by

TANGO2: A Rare but Important Mutation.
Walters B, McConkey N, Imundo JR. Walters B, et al. J Innov Card Rhythm Manag. 2024 May 15;15(5):5871-5875. doi: 10.19102/icrm.2024.15054. eCollection 2024 May. J Innov Card Rhythm Manag. 2024. PMID: 38808169 Free PMC article.
Defects in lipid homeostasis reflect the function of TANGO2 in phospholipid and neutral lipid metabolism.
Lujan AL, Foresti O, Sugden C, Brouwers N, Farre AM, Vignoli A, Azamian M, Turner A, Wojnacki J, Malhotra V. Lujan AL, et al. Elife. 2023 Mar 24;12:e85345. doi: 10.7554/eLife.85345. Elife. 2023. PMID: 36961129 Free PMC article.
Clinical case report of intractable paroxysmal sympathetic hyperactivity in TANGO2 deficiency disorder.
Morrison K, Koshiya H, Safier R, Brown A, May C, Vockley J, Ghaloul-Gonzalez L. Morrison K, et al. Am J Med Genet A. 2024 Sep;194(9):e63633. doi: 10.1002/ajmg.a.63633. Epub 2024 Apr 18. Am J Med Genet A. 2024. PMID: 38634641
Folate as a potential treatment for lethal ventricular arrhythmias in TANGO2-deficiency disorder.
Xu W, Cao Y, Stephens SB, Arredondo MJ, Chen Y, Perez W, Sun L, Yu AC, Kim JJ, Lalani SR, Li N, Horrigan FT, Altamirano F, Wehrens XH, Miyake CY, Zhang L. Xu W, et al. JCI Insight. 2024 Jun 10;9(11):e171005. doi: 10.1172/jci.insight.171005. JCI Insight. 2024. PMID: 38855866 Free PMC article.
TANGO2 deficiency disease is predominantly caused by a lipid imbalance.
Sacher M, DeLoriea J, Mehranfar M, Casey C, Naaz A, Gamberi C. Sacher M, et al. Dis Model Mech. 2024 Jun 1;17(6):dmm050662. doi: 10.1242/dmm.050662. Epub 2024 Jun 5. Dis Model Mech. 2024. PMID: 38836374 Free PMC article. Review.

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References

1. Kremer LS, Distelmaier F, Alhaddad B, et al. Bi-allelic truncating mutations in TANGO2 cause infancy-onset recurrent metabolic crises with encephalocardiomyopathy. Am J Hum Genet. 2016;98:358–362. - PMC - PubMed
1. Lalani SR, Liu P, Rosenfeld JA, et al. Recurrent muscle weakness with rhabdomyolysis, metabolic crises, and cardiac arrhythmia due to Bi-allelic TANGO2 mutations. Am J Hum Genet. 2016;98:347–357. - PMC - PubMed
1. Schymick J, Leahy P, Cowan T, et al. Variable clinical severity in TANGO2 deficiency: case series and literature review. Am J Med Genet A. 2022;188:473–487. - PubMed
1. Sun F, Zhao Z, Willoughby MM, et al. HRG-9 homologues regulate haem trafficking from haem-enriched compartments. Nature. 2022;610:768–774. - PMC - PubMed
1. Miyake CY, Lay EJ, Soler-Alfonso C, et al. Natural history of TANGO2 deficiency disorder: baseline assessment of 73 patients. Genet Med. 2022:S1098-3600(22)01057-7. [Epub ahead of print]. - PMC - PubMed

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[1] Kremer LS, Distelmaier F, Alhaddad B, et al. Bi-allelic truncating mutations in TANGO2 cause infancy-onset recurrent metabolic crises with encephalocardiomyopathy. Am J Hum Genet. 2016;98:358–362. - PMC - PubMed

[2] Kremer LS, Distelmaier F, Alhaddad B, et al. Bi-allelic truncating mutations in TANGO2 cause infancy-onset recurrent metabolic crises with encephalocardiomyopathy. Am J Hum Genet. 2016;98:358–362. - PMC - PubMed

[3] Lalani SR, Liu P, Rosenfeld JA, et al. Recurrent muscle weakness with rhabdomyolysis, metabolic crises, and cardiac arrhythmia due to Bi-allelic TANGO2 mutations. Am J Hum Genet. 2016;98:347–357. - PMC - PubMed

[4] Lalani SR, Liu P, Rosenfeld JA, et al. Recurrent muscle weakness with rhabdomyolysis, metabolic crises, and cardiac arrhythmia due to Bi-allelic TANGO2 mutations. Am J Hum Genet. 2016;98:347–357. - PMC - PubMed

[5] Schymick J, Leahy P, Cowan T, et al. Variable clinical severity in TANGO2 deficiency: case series and literature review. Am J Med Genet A. 2022;188:473–487. - PubMed

[6] Schymick J, Leahy P, Cowan T, et al. Variable clinical severity in TANGO2 deficiency: case series and literature review. Am J Med Genet A. 2022;188:473–487. - PubMed

[7] Sun F, Zhao Z, Willoughby MM, et al. HRG-9 homologues regulate haem trafficking from haem-enriched compartments. Nature. 2022;610:768–774. - PMC - PubMed

[8] Sun F, Zhao Z, Willoughby MM, et al. HRG-9 homologues regulate haem trafficking from haem-enriched compartments. Nature. 2022;610:768–774. - PMC - PubMed

[9] Miyake CY, Lay EJ, Soler-Alfonso C, et al. Natural history of TANGO2 deficiency disorder: baseline assessment of 73 patients. Genet Med. 2022:S1098-3600(22)01057-7. [Epub ahead of print]. - PMC - PubMed

[10] Miyake CY, Lay EJ, Soler-Alfonso C, et al. Natural history of TANGO2 deficiency disorder: baseline assessment of 73 patients. Genet Med. 2022:S1098-3600(22)01057-7. [Epub ahead of print]. - PMC - PubMed

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B-complex vitamins for patients with TANGO2-deficiency disorder

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B-complex vitamins for patients with TANGO2-deficiency disorder

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