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A CASE WITH WOLFRAM (DIDMOAD) SYNDROME AND ORAL FINDINGS (A CASE REPORT)

Year 2007, Issue: 014, 137 - 140, 17.12.2007

Abstract

Wolfram Syndrome is an unusual, congenital, multi system disorder. Diabetes mellitus, Diabetes insipidus, optic

atrophy and deafness are most observable symptoms. Neurological, urinary tract and psychiatric disorders are the

other common symptoms. Diabetes mellitus and optic atrophy are usually the first findings.

In this case report we present 20 year old-male patient, that was have Wolfram syndrome. Oral clinical and

radiological findings were presented.

References

  • [1] Internet: OMIM, In: Online Mendelian Inheritance in Man.http://www.ncbi.nlm.nih.gov/entrez/dispomim.cgi?cmd=entry&id=222300 (2007)
  • [2] Swift, R.G., Sadler, D.B., Swift, M.,” Psychiatric findings in Wolfram syndrome homozygotes”, Lancet,336: 667-669 (1990).
  • [3] Barrett, T.G., Bundey, S.E., Macleod, A.F., “Neurodegeneration and diabetes: UK nationwide study of Wolfram (DIDMOAD) syndrome”, Lancet, 2;346 (8988): 1458-63 (1995).
  • [4] Hardy, C., Khanim, F., Torres, R., Scott-Brown M., Seller A, Poulton J., Collier D., Kirk J.,Polymeropoulos M., Latif F., Barrett T., “Clinical and molecular genetic analysis of 19 Wolfram syndrome kindreds demonstrating a wide spectrum of mutations in WFS1”, Am J Hum Genet.,65(5):1279-90 (1999).
  • [5] Bu, X., Rotter, J.I., ”Wolfram syndrome: a mitochondrial-mediated disorder ?”, Lancet, 4;342 (8871):598-600 (1993).
  • [6] Sugano, N., Kawamoto, K., Numazaki, H., Murai, S., Ito, K., “Detection of mitochondrial DNA mutations in human gingival tissues”, J. Oral Sci., 42 (4):221-223 (2000).
  • [7] Zen, P.R., Pinto, L.L., Schwartz, I.V., Barrett, T.G., Paskulin, G., “Report of a Brazilian patient with Wolfram Syndrome”, J. Pediatr (Rio J)., 78(6): 529-32 (2002).
  • [8] Rotig, A., Cormier, V., Chatelain, P., Francois, R., Saudubray, J.M., Rustin, P., Munnich, A., ”Deletion of mitochondrial DNA in a case of early-onset diabetes mellitus, optic atrophy, and deafness (Wolfram syndrome, MIM 222300)”, J. Clin. Invest., 91(3): 1095-8 (1993).
  • [9] Langlais, R.P., Miller, C.S., ”Color Atlas of Common Oral Diseases”, Lea & Febiger, Philadelphia/London, 46-47 (1992).
  • [10] Mealey, B.,” Periodontal Medicine”, Rose, F.L., Mealey B.L., Genco, R..J., Cohen, W.D., B.C. Decker Inc., Toronto, 121-150 (1999).
  • [11] Terezhalmy, G. T., “Internal Medicine for Dentistry, 2 nd edition”, Kaye, D., Rose, L.F., The C.V.Mosby Company, St Louis, 1153-1154 (1990).

WOLFRAM (DIDMOAD) SENDROMU BULUNAN BİR OLGUDAKİ ORAL BULGULAR (OLGU BİLDİRİMİ)

Year 2007, Issue: 014, 137 - 140, 17.12.2007

Abstract

Wolfram Sendromu, nadir görülen konjenital, birçok sistemi etkileyebilen sendromdur. Diabetes mellitus,

diabetes insipidus, optik atrofi ve sağırlık ile karakterizedir. Hastalarda nörolojik rahatsızlıklar ile üriner sistem

rahatsızlıkları ve psikolojik rahatsızlıklar da izlenir. Diabetes mellitus ve optik atrofi ilk ortaya çıkan bulgulardır.

Bu olgu raporunda 20 yaşındaki Wolfram sendromlu erkek hastanın klinik ve radyografik ağız bulguları

sunulmaktadır.

References

  • [1] Internet: OMIM, In: Online Mendelian Inheritance in Man.http://www.ncbi.nlm.nih.gov/entrez/dispomim.cgi?cmd=entry&id=222300 (2007)
  • [2] Swift, R.G., Sadler, D.B., Swift, M.,” Psychiatric findings in Wolfram syndrome homozygotes”, Lancet,336: 667-669 (1990).
  • [3] Barrett, T.G., Bundey, S.E., Macleod, A.F., “Neurodegeneration and diabetes: UK nationwide study of Wolfram (DIDMOAD) syndrome”, Lancet, 2;346 (8988): 1458-63 (1995).
  • [4] Hardy, C., Khanim, F., Torres, R., Scott-Brown M., Seller A, Poulton J., Collier D., Kirk J.,Polymeropoulos M., Latif F., Barrett T., “Clinical and molecular genetic analysis of 19 Wolfram syndrome kindreds demonstrating a wide spectrum of mutations in WFS1”, Am J Hum Genet.,65(5):1279-90 (1999).
  • [5] Bu, X., Rotter, J.I., ”Wolfram syndrome: a mitochondrial-mediated disorder ?”, Lancet, 4;342 (8871):598-600 (1993).
  • [6] Sugano, N., Kawamoto, K., Numazaki, H., Murai, S., Ito, K., “Detection of mitochondrial DNA mutations in human gingival tissues”, J. Oral Sci., 42 (4):221-223 (2000).
  • [7] Zen, P.R., Pinto, L.L., Schwartz, I.V., Barrett, T.G., Paskulin, G., “Report of a Brazilian patient with Wolfram Syndrome”, J. Pediatr (Rio J)., 78(6): 529-32 (2002).
  • [8] Rotig, A., Cormier, V., Chatelain, P., Francois, R., Saudubray, J.M., Rustin, P., Munnich, A., ”Deletion of mitochondrial DNA in a case of early-onset diabetes mellitus, optic atrophy, and deafness (Wolfram syndrome, MIM 222300)”, J. Clin. Invest., 91(3): 1095-8 (1993).
  • [9] Langlais, R.P., Miller, C.S., ”Color Atlas of Common Oral Diseases”, Lea & Febiger, Philadelphia/London, 46-47 (1992).
  • [10] Mealey, B.,” Periodontal Medicine”, Rose, F.L., Mealey B.L., Genco, R..J., Cohen, W.D., B.C. Decker Inc., Toronto, 121-150 (1999).
  • [11] Terezhalmy, G. T., “Internal Medicine for Dentistry, 2 nd edition”, Kaye, D., Rose, L.F., The C.V.Mosby Company, St Louis, 1153-1154 (1990).
There are 11 citations in total.

Details

Primary Language English
Subjects Engineering
Journal Section Articles
Authors

Hasan Hatipoğlu This is me

Müjgan Güngör Hatipoğlu This is me

Özden Kansu This is me

Publication Date December 17, 2007
Published in Issue Year 2007 Issue: 014

Cite

APA Hatipoğlu, H., Hatipoğlu, M. G., & Kansu, Ö. (2007). A CASE WITH WOLFRAM (DIDMOAD) SYNDROME AND ORAL FINDINGS (A CASE REPORT). Journal of Science and Technology of Dumlupınar University(014), 137-140.

HAZİRAN 2020'den itibaren Journal of Scientific Reports-A adı altında ingilizce olarak yayın hayatına devam edecektir.