Abstract
BackgroundLimb development and patterning originate from a complex interplay between the skeletal elements, tendons, and muscles of the limb. One of the genes involved in patterning of limb muscles is the homeobox transcription factor Pitx2 but its role in forelimb development is uncharacterized. Pitx2 is expressed in the majority of premature presumptive forelimb musculature at embryonic day 12.5 and then maintained throughout embryogenesis to adult skeletal muscle.ResultsTo further study the role of Pitx2 in forelimb development we have generated transgenic mice that exhibit a pulse of PITX2 over-expression at embryonic day 13.5 and 14.5 in the developing forelimb mesenchyme. These mice exhibit a distal misplacement of the biceps brachii insertion during embryogenesis, which twists the forelimb musculature resulting in severe skeletal malformations. The skeletal malformations have some similarities to the forearm deformities present in Leri-Weill dyschondrosteosis.ConclusionTaken together, the tendon, muscle, and bone anomalies further support a role of Pitx2 in forelimb development and may also shed light on the interaction between the skeletal elements and muscles of the limb during embryogenesis.
Highlights
Limb development and patterning originate from a complex interplay between the skeletal elements, tendons, and muscles of the limb
24 h later myogenic cells are identified at the base of the forelimb and at E11.5 the first hint of humerus is apparent [1]
Among the transcription factors involved in early myogenesis are Pax3 and Lbx1 whose expression precedes the expression of myogenic regulatory factors (MRFs)
Summary
Limb development and patterning originate from a complex interplay between the skeletal elements, tendons, and muscles of the limb. One of the genes involved in patterning of limb muscles is the homeobox transcription factor Pitx but its role in forelimb development is uncharacterized. Pitx is expressed in the majority of premature presumptive forelimb musculature at embryonic day 12.5 and maintained throughout embryogenesis to adult skeletal muscle. Limb development and patterning originate from close interactions between tendon, cartilage and muscle precursor cells. 24 h later myogenic cells are identified at the base of the forelimb and at E11.5 the first hint of humerus is apparent [1]. The homeobox transcription factor PITX2 was originally identified as one of the genes responsible for Axenfeld-Rieger syndrome, mainly (page number not for citation purposes)
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