TY - JOUR
T1 - Trismus-pseudocamptodactyly syndrome with bilateral hypoplastic mandibular condyles and shallow mandibular fossa
T2 - A case report
AU - Kusano, Chieko
AU - Hori, Naoaki
AU - Izawa, Kazumi
AU - Kosaki, Rika
AU - Nishimura, Gen
AU - Hasegawa, Tomonobu
N1 - Publisher Copyright:
© 2018 Japanese Stomatological Society
PY - 2018/7
Y1 - 2018/7
N2 - Trismus-pseudocamptodactyly syndrome (TPS) (OMIM#158300), also known as Hecht syndrome, is a rare autosomal dominant distal arthrogryposis. Bilateral hyperplasia of the coronoid processes has been reported to cause trismus. In this study, we report a case of TPS definitively diagnosed on the basis of clinical signs in a patient who had bilateral hypoplastic mandibular condyles and shallow mandibular fossa; TPS in such patients have never been reported in the literature. On the basis of the present case, we propose that a hypoplastic mandibular condyle and shallow mandibular fossa are also clinical features of TPS.
AB - Trismus-pseudocamptodactyly syndrome (TPS) (OMIM#158300), also known as Hecht syndrome, is a rare autosomal dominant distal arthrogryposis. Bilateral hyperplasia of the coronoid processes has been reported to cause trismus. In this study, we report a case of TPS definitively diagnosed on the basis of clinical signs in a patient who had bilateral hypoplastic mandibular condyles and shallow mandibular fossa; TPS in such patients have never been reported in the literature. On the basis of the present case, we propose that a hypoplastic mandibular condyle and shallow mandibular fossa are also clinical features of TPS.
KW - Hypoplastic mandibular condyle
KW - Shallow mandibular fossa
KW - Trismus
KW - Trismus-pseudocamptodactyly syndrome
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U2 - 10.1016/S1348-8643(18)30008-9
DO - 10.1016/S1348-8643(18)30008-9
M3 - Article
AN - SCOPUS:85046334690
SN - 1348-8643
VL - 15
SP - 90
EP - 92
JO - Oral Science International
JF - Oral Science International
IS - 2
ER -