Literature DB >> 22241869

Enlarged follicles and temporomandibular joint abnormalities in mucolipidosis Type III.

H Khalifa1, H S Grubisa, L Lee, E W N Lam.   

Abstract

Mucolipidosis Type III, or pseudo-Hurler polydystrophy, is a rare genetic abnormality, the result of a mutation to one of two genes that encode the hexameric protein N-acetylglucosaminyl-1-phosphotransferase (Glc-NAc-PT). The abnormality results in the accumulation of unprocessed macromolecules in cell and tissue compartments throughout the body. In this case report, we describe the clinical and radiographic findings of a 15-year-old male with this disorder. He presented with bilateral ectopically developing mandibular molar teeth with enlarged follicles and multiple joint involvement, including the temporomandibular joints. The patient underwent surgical removal of the molar teeth and curettage of the associated follicles. The subsequent histopathological examination of the tissues revealed hyperplastic follicles suggestive of dentigerous cysts. This report presents the plain film and cone beam CT examinations of the patient.

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Year:  2012        PMID: 22241869      PMCID: PMC3667515          DOI: 10.1259/dmfr/22822014

Source DB:  PubMed          Journal:  Dentomaxillofac Radiol        ISSN: 0250-832X            Impact factor:   2.419


  10 in total

1.  The natural history and osteodystrophy of mucolipidosis types II and III.

Authors:  Grace David-Vizcarra; Julie Briody; Jenny Ault; Michael Fietz; Janice Fletcher; Ravi Savarirayan; Meredith Wilson; Jim McGill; Matthew Edwards; Craig Munns; Melanie Alcausin; Sara Cathey; David Sillence
Journal:  J Paediatr Child Health       Date:  2010-03-29       Impact factor: 1.954

2.  Temporomandibular joint destruction in mucolipidosis type III necessitating gastrostomy insertion.

Authors:  Zarzuela Zolkipli; Lee Noimark; Maureen A Cleary; Cathy Owens; Ashok Vellodi
Journal:  Eur J Pediatr       Date:  2005-07-22       Impact factor: 3.183

3.  The osteodystrophy of mucolipidosis type III and the effects of intravenous pamidronate treatment.

Authors:  C Robinson; N Baker; J Noble; A King; G David; D Sillence; P Hofman; T Cundy
Journal:  J Inherit Metab Dis       Date:  2002-12       Impact factor: 4.982

4.  Orthopaedic management in four cases of mucolipidosis type III.

Authors:  C Hetherington; N J Harris; T W Smith
Journal:  J R Soc Med       Date:  1999-05       Impact factor: 5.344

5.  Clinical variability in mucolipidosis III (pseudo-Hurler polydystrophy).

Authors:  Anna Tylki-Szymańska; Barbara Czartoryska; Johanna E M Groener; Agnieszka Ługowska
Journal:  Am J Med Genet       Date:  2002-03-15

6.  Mucolipidosis III (pseudo-Hurler polydystrophy): Clinical and laboratory studies in a series of 12 patients.

Authors:  T E Kelly; G H Thomas; H A Taylor; V A McKusick; W S Sly; J H Glaser; M Robinow; L Luzzatti; C Espiritu; M Feingold; M J Bull; E M Ashenhurst; E J Ives
Journal:  Johns Hopkins Med J       Date:  1975-10

7.  Mucolipidosis III presenting as a rheumatological disorder.

Authors:  R Brik; H Mandel; A Aizin; D Goldscher; M Ziegler; V Bialik; M Berant
Journal:  J Rheumatol       Date:  1993-01       Impact factor: 4.666

8.  Phenotype and genotype in mucolipidoses II and III alpha/beta: a study of 61 probands.

Authors:  S S Cathey; J G Leroy; T Wood; K Eaves; R J Simensen; M Kudo; R E Stevenson; M J Friez
Journal:  J Med Genet       Date:  2009-07-16       Impact factor: 6.318

9.  Radiolucent lesions of the maxillofacial complex in a patient with mucolipidosis type II (MLSII): case report.

Authors:  Maico D Melo; George Obeid
Journal:  Oral Surg Oral Med Oral Pathol Oral Radiol Endod       Date:  2007-07-25

Review 10.  Combined tarsal and carpal tunnel syndrome in mucolipidosis type III. A case study and review.

Authors:  Izelle Smuts; Denise Potgieter; Francois Hendrikus van der Westhuizen
Journal:  Ann N Y Acad Sci       Date:  2009-01       Impact factor: 5.691

  10 in total
  1 in total

1.  Mucolipidosis Type III: A Rare Disease in Differential Diagnosis of Joint Stiffness in Pediatric Rheumatology.

Authors:  Çiğdem Seher Kasapkara; Meltem Akçaboy; Fehime Kara Eroğlu; Betül Emine Derinkuyu
Journal:  Arch Rheumatol       Date:  2017-05-25       Impact factor: 1.472

  1 in total

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