Document Detail


Situs inversus in a patient with nevoid basal cell carcinoma syndrome: a histogenetic relationship?
MedLine Citation:
PMID:  21712633     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Nevoid basal cell carcinoma syndrome (NBCCS) is an uncommon autosomal dominant inherited disorder with high penetrance and variable expressivity. It affects multiple organ systems, including the stomatological, skeletal, skin, eye, reproductive, and central nervous systems. It is caused by mutations in the patched tumor suppressor gene, PTCHI, located in the 9q22.3-q31 chromosome. To our knowledge, this is the first report of a patient with unusual radiological features, i. e. dextrocardia and situs inversus totalis, in conjunction with common features including multiple keratocystic odontogenic tumors, bifid ribs, palmar and plantar pits, bridging of the sella turcica and calcification of the falx cerebri. We examined whether these genetic conditions were associated, as both involve ciliary dysfunction.
Authors:
Monika Gupta; Debdutta Das; Amit Mittal; Harkamal Kaur; Aayush Malhotra
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of oral science     Volume:  53     ISSN:  1880-4926     ISO Abbreviation:  J Oral Sci     Publication Date:  2011 Jun 
Date Detail:
Created Date:  2011-06-29     Completed Date:  2011-11-03     Revised Date:  2012-01-24    
Medline Journal Info:
Nlm Unique ID:  9808942     Medline TA:  J Oral Sci     Country:  Japan    
Other Details:
Languages:  eng     Pagination:  253-6     Citation Subset:  D; IM    
Affiliation:
Department of Oral and Maxillofacial Surgery, MM College of Dental Sciences and Research, Haryana, India. monikabox@yahoo.com
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Basal Cell Nevus Syndrome / complications*,  genetics
Calcinosis / complications
Cranial Sinuses / pathology
Dextrocardia / complications,  genetics
Dura Mater / pathology
Female
Foot Deformities, Congenital / complications
Hand Deformities, Congenital / complications
Humans
Odontogenic Tumors / complications
Ribs / abnormalities
Sella Turcica / abnormalities
Situs Inversus / complications*,  genetics

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