Multidisciplinary oral rehabilitation of an adolescent suffering from juvenile Gorlin-Goltz syndrome - a case report.


Journal

Head & face medicine
ISSN: 1746-160X
Titre abrégé: Head Face Med
Pays: England
ID NLM: 101245792

Informations de publication

Date de publication:
08 Feb 2019
Historique:
received: 07 02 2018
accepted: 30 01 2019
entrez: 10 2 2019
pubmed: 10 2 2019
medline: 18 12 2019
Statut: epublish

Résumé

The Gorlin-Goltz syndrome is an autosomal dominant disorder characterized by keratocystic odontogenic tumors in the jaws, multiple basal cell carcinomas and skeletal abnormities. Frequently, the manifestation of the syndrome occurs in the adolescent years. An 11-year-old boy was referred to our clinic due to the persistence of the lower deciduous molars. The further diagnosis revealed bilateral keratocystic odontogenic tumors in the region of teeth 33 and 45 representing a symptom of a Gorlin-Goltz syndrome. This case of the oral rehabilitation of an adolescent with bilateral keratocystic odontogenic tumors shows the approach of a multidisciplinary treatment concept including the following elements: Enucleation and bone defect augmentation using a prefabricated bone graft; distraction osteogenesis to extend the graft-block vertically after cessation of growth; accompanying orthodontic treatment, guided implant placement and prosthetic rehabilitation. Six months after implant insertion, a new keratocystic odontogenic tumor in the basal part of the left sinus maxillaris had to be removed combined with the closure of the oroantral fistula. During the follow-up period of 18 months in semi-annual intervals, the patient showed no sign of pathology. In the presented case could be shown that distraction osteogenesis of prefabricated bone blocks is possible. With a multidisciplinary approach in a long-term treatment a sufficient oral rehabilitation of the patient suffering from extended keratocystic odontogenic tumors was possible.

Sections du résumé

BACKGROUND BACKGROUND
The Gorlin-Goltz syndrome is an autosomal dominant disorder characterized by keratocystic odontogenic tumors in the jaws, multiple basal cell carcinomas and skeletal abnormities. Frequently, the manifestation of the syndrome occurs in the adolescent years.
CASE PRESENTATION METHODS
An 11-year-old boy was referred to our clinic due to the persistence of the lower deciduous molars. The further diagnosis revealed bilateral keratocystic odontogenic tumors in the region of teeth 33 and 45 representing a symptom of a Gorlin-Goltz syndrome. This case of the oral rehabilitation of an adolescent with bilateral keratocystic odontogenic tumors shows the approach of a multidisciplinary treatment concept including the following elements: Enucleation and bone defect augmentation using a prefabricated bone graft; distraction osteogenesis to extend the graft-block vertically after cessation of growth; accompanying orthodontic treatment, guided implant placement and prosthetic rehabilitation. Six months after implant insertion, a new keratocystic odontogenic tumor in the basal part of the left sinus maxillaris had to be removed combined with the closure of the oroantral fistula. During the follow-up period of 18 months in semi-annual intervals, the patient showed no sign of pathology.
CONCLUSION CONCLUSIONS
In the presented case could be shown that distraction osteogenesis of prefabricated bone blocks is possible. With a multidisciplinary approach in a long-term treatment a sufficient oral rehabilitation of the patient suffering from extended keratocystic odontogenic tumors was possible.

Identifiants

pubmed: 30736811
doi: 10.1186/s13005-019-0189-5
pii: 10.1186/s13005-019-0189-5
pmc: PMC6367745
doi:

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

5

Références

J Dent Res. 1999 Jul;78(7):1345-53
pubmed: 10403462
Quintessence Int. 2001 May;32(5):381-3
pubmed: 11444071
Mod Pathol. 2002 Mar;15(3):331-41
pubmed: 11904346
J Prosthet Dent. 2003 May;89(5):436-42
pubmed: 12806318
N Engl J Med. 1960 May 5;262:908-12
pubmed: 13851319
J Ir Dent Assoc. 2003;49(3):83-8
pubmed: 14603664
J Am Acad Orthop Surg. 2005 Jan-Feb;13(1):77-86
pubmed: 15712985
Clin Oral Implants Res. 2006 Aug;17(4):359-66
pubmed: 16907765
Int J Periodontics Restorative Dent. 2006 Dec;26(6):543-51
pubmed: 17243327
Rofo. 2007 Jun;179(6):618-26
pubmed: 17492539
Otolaryngol Head Neck Surg. 2007 Jul;137(1):88-92
pubmed: 17599572
Anticancer Res. 2007 Jul-Aug;27(4A):1783-7
pubmed: 17649773
Am J Orthod. 1976 Jun;69(6):611-9
pubmed: 179326
Beijing Da Xue Xue Bao Yi Xue Ban. 2008 Feb 18;40(1):15-8
pubmed: 18278130
J Oral Maxillofac Surg. 2008 May;66(5):1025-36
pubmed: 18423297
J Oral Sci. 2008 Jun;50(2):205-12
pubmed: 18587212
Postgrad Med J. 2008 Jun;84(992):330-2
pubmed: 18644927
Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008 Nov;106(5):e1-7
pubmed: 18805714
Orphanet J Rare Dis. 2008 Nov 25;3:32
pubmed: 19032739
Eur J Paediatr Dent. 2008 Dec;9(4):195-8
pubmed: 19072008
Arch Oral Biol. 2009 Oct;54(10):893-7
pubmed: 19679296
J Craniomaxillofac Surg. 2010 Jul;38(5):358-64
pubmed: 19897381
J Craniofac Surg. 2010 May;21(3):833-6
pubmed: 20485063
Clin Implant Dent Relat Res. 2012 Mar;14(1):41-50
pubmed: 20491816
Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2010 Aug;110(2):e41-6
pubmed: 20659694
J Oral Implantol. 2012 Sep;38 Spec No:485-8
pubmed: 21073299
Int J Surg Pathol. 2012 Apr;20(2):154-60
pubmed: 22108498
Int J Periodontics Restorative Dent. 2012 Jun;32(3):e82-6
pubmed: 22408782
J Oral Pathol Med. 2012 Oct;41(9):697-701
pubmed: 22582837
J Appl Oral Sci. 2012 Mar-Apr;20(2):282-5
pubmed: 22666848
J Oral Pathol Med. 2013 Feb;42(2):162-5
pubmed: 22742575
Oral Surg Oral Med Oral Pathol Oral Radiol. 2012 Dec;114(6):725-32
pubmed: 22858021
J Oral Pathol Med. 2013 Mar;42(3):275-80
pubmed: 22882291
Int J Dent. 2012;2012:737262
pubmed: 22899930
PLoS One. 2012;7(8):e43827
pubmed: 22952776
Br J Oral Maxillofac Surg. 2013 Jun;51(4):358-62
pubmed: 22981336
J Oral Implantol. 2012 Sep;38 Spec No:519-26
pubmed: 23072285
J Oral Maxillofac Surg. 2013 Feb;71(2):e137-42
pubmed: 23351767
J Burn Care Res. 2013 Sep-Oct;34(5):576-84
pubmed: 23966114
J Oral Maxillofac Surg. 2013 Nov;71(11):1886-92
pubmed: 23992784
PLoS One. 2013 Oct 21;8(10):e77305
pubmed: 24204797
Clin Oral Implants Res. 2015 Apr;26(4):e22-e27
pubmed: 24383943
Contemp Clin Dent. 2013 Oct;4(4):547-50
pubmed: 24403808
Aust Dent J. 2014 Mar;59(1):48-56
pubmed: 24588310
J Craniomaxillofac Surg. 2014 Sep;42(6):966-75
pubmed: 24629798
J Prosthet Dent. 2014 Sep;112(3):439-43
pubmed: 24831743
Head Face Med. 2014 May 28;10:21
pubmed: 24885136
J Maxillofac Oral Surg. 2014 Sep;13(3):341-5
pubmed: 25018611
Med Oral Patol Oral Cir Bucal. 2014 Nov 01;19(6):e605-11
pubmed: 25129241
Indian J Endocrinol Metab. 2014 Nov;18(Suppl 1):S63-71
pubmed: 25538880
Contemp Clin Dent. 2015 Mar;6(Suppl 1):S102-5
pubmed: 25821359
J Oral Maxillofac Surg. 2015 Dec;73(12 Suppl):S77-86
pubmed: 26608157
J Pediatr Neurosci. 2015 Oct-Dec;10(4):359-61
pubmed: 26962343
Oral Surg Oral Med Oral Pathol Oral Radiol. 2016 Jun;121(6):629-35
pubmed: 27050804
J Oral Biol Craniofac Res. 2016 May-Aug;6(2):93-100
pubmed: 27195205
J Maxillofac Oral Surg. 2016 Dec;15(4):521-527
pubmed: 27833347
J Craniomaxillofac Surg. 2017 Jan;45(1):131-144
pubmed: 27955959
Angle Orthod. 1967 Apr;37(2):134-43
pubmed: 4290545
N Z Dent J. 1994 Jun;90(400):56-9
pubmed: 8058219
J Med Genet. 1993 Jun;30(6):460-4
pubmed: 8326488

Auteurs

Manfred Nilius (M)

Niliusklinik Dortmund, Londoner Bogen 6, 44269, Dortmund, Germany. manfrednilius@niliusklinik.de.

Jürgen Kohlhase (J)

Humangenetik Freiburg GmbH, Heinrich-von-Stephan-Str. 5, D-79100, Freiburg, Germany.

Johann Lorenzen (J)

Department of Pathology, Klinikum Dortmund gGmbH, Beurhausstraße 40, D-44123, Dortmund, Germany.

Günter Lauer (G)

Department of Oral and Maxillofacial Surgery, University Hospital "Carl Gustav Carus", Technische Universität Dresden, Fetscherstr. 74, D-01307, Dresden, Germany.

Matthias C Schulz (MC)

Department of Oral and Maxillofacial Surgery, University Hospital "Carl Gustav Carus", Technische Universität Dresden, Fetscherstr. 74, D-01307, Dresden, Germany. Matthias.Schulz@uniklinikum-dresden.de.

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Classifications MeSH