Joint shape morphogenesis precedes cavitation of the developing hip joint.

The biology and mechanobiology of joint cavitation have undergone extensive investigation, but we have almost no understanding of the development of joint shape. Joint morphogenesis, the development of shape, has been identified as the 'least understood aspect of joint formation' (2005, Birth Defects Res C Embryo Today 75, 237), despite the clinical relevance of shape morphogenesis to postnatal skeletal malformations such as developmental dysplasia of the hip. In this study, we characterise development of early hip joint shape in the embryonic chick using direct capture 3D imaging. Contrary to formerly held assumptions that cavitation precedes morphogenesis in joint development, we have found that the major anatomical features of the adult hip are present at Hamburger Hamilton (HH)32, a full day prior to cavitation of the joint at HH34. We also reveal that the pelvis undergoes significant changes in orientation with respect to the femur, despite the lack of a joint cavity between the rudiments. Furthermore, we have identified the appearance of the ischium and pubis several developmental stages earlier than was previously reported, illustrating the value and importance of direct capture 3D imaging.

Avian pelvis originates from lateral plate mesoderm and its development requires signals from both ectoderm and paraxial mesoderm.

The pelvic girdle is composed of three skeletal elements: ilium, pubis, and ischium. In comparison with other parts of the postcranial skeleton, its development is not well known to date. To elucidate the embryonic origin of the avian pelvic girdle and the signaling centers that control its development, we have performed extirpation and quail-to-chick grafting experiments. The results reveal that the entire pelvic girdle originates from the somatopleure at somite levels 26 to 35. No somitic cell contribution to skeletal elements of the pelvis has been detected. Removal of the surface ectoderm covering the lateral plate mesoderm has revealed that ectodermal signals control the development of the pelvic girdle, especially the formation of the pubis and ischium. The impaired development of the ischium and pubis correlates with the downregulation of Pax1 and Alx4, two transcription factors that control the normal development of the ischium and pubis. Although of somatopleural origin, the development of the ilium depends on somitic signals. Insertion of a barrier between somites and somatopleure disrupts the expression of Emx2 and prevents normal development of the ilium but does not affect the expression of Pax1 or Alx4 and the development of the pubis and ischium. Thus, the development of the ilium, but not of the pubis and ischium, depends on somitic and ectodermal signals.

Fetal bony pelvis in the bladder exstrophy complex: normal potential for growth?

OBJECTIVES: To provide the first look at the bony histologic features of fetuses with the exstrophy complex, specifically evaluating the endochondral ossification, stage of development, and microscopic potential for normal growth. METHODS: Three fetuses between 28 and 30 weeks of gestation, one with classic bladder exstrophy, one with cloacal exstrophy, and one control, were obtained from France. The bony pelves were dissected and preserved in formalin, and multiple representative sections were sliced from all pelvic areas: pubis, ischium, ilium, and sacrum. These slices were sequentially processed as slides, stained with hematoxylin-eosin, and evaluated microscopically for histologic changes, developmental stage, and degree of endochondral ossification. RESULTS: All slides from the three specimens showed cartilage analogue with endochondral ossification. Histologically the exstrophy specimens were identical to the control and appeared completely normal; bone development was occurring at an expected rate with the potential for continued normal growth. CONCLUSIONS: These new findings illustrate that fetal bone in the exstrophy complex displays normal microscopic growth patterns and unhindered endochondral ossification at 28 weeks of gestation, well beyond the embryologic period. With no evident microscopic bony defect, the gross bony anomalies in exstrophy should be surgically correctable, leading us to conclude that early reapproximation of the physiologic shape of the pelvis could lead to more normal gross bone growth, decreased shortage of bone, and a more appropriate distribution of the mechanical and developmental forces on a closed, normally functioning pelvic ring.

Connections of the bladder plate and bladder neck with the bony pelvis in a fetus with classic bladder exstrophy.

OBJECTIVES: To demonstrate that the bladder plate and bladder neck in classic bladder exstrophy are laying on smooth muscle fibers that extend laterally to the pubic bones. METHODS: We compared a male fetus of 28 weeks' gestational age with classic bladder exstrophy with a normal fetus of the same age. The specimens were divided into two parts by a midsagittal section, from the bladder neck to the membranous urethra. Thin transverse slices were also obtained on one part, and longitudinal slices on the other part. RESULTS: The smooth musculature of the bladder is normally differentiated cytologically and extends laterally to the bony structures of the pelvis. The musculature of the bladder neck and urethra are normally present compared with the control. CONCLUSIONS: The results of this study demonstrate the musculoskeletal organization of the urogenital system in classic bladder exstrophy. They also indicate that these structures should be clearly individualized and repaired in the reconstruction of classic bladder exstrophy.

MR imaging of the ischiopubic synchondrosis.

The ischiopubic synchondrosis (IPS) is a temporary joint, occurring in childhood prior to fusion of the ischial and pubic bones. On conventional radiographs this tumor-like appearance is a well known normal anatomic variant, however, there are no reports in the literature of the appearance of IPS on MRI. Therefore the purpose of this study was to evaluate typical magnetic resonance features of the IPS. All pelvic MRIs from 1/1992 to 4/1998 of children ranging in age from 4 to 16 years, who were scanned for reasons other than bone disorders, were retrospectively investigated. Twenty-eight children were included and the morphologic appearance of IPS on MRI was evaluated. Seventeen (61%) of these 28 children had the following findings of IPS on MRI. Listed in order of frequency we found signal alteration of the ischiopubic fusion zone [hyperintense on T(2) with fat-saturation (89%) or STIR (74%), hypointense on T(1) (71%)], fibrous "bridging" (68%) [hypointense band on all sequences in perpendicular orientation to the axis of the inferior pubic ramus], fusiform swelling (68%), signal alteration of the adjacent soft tissue (57%) and irregular margins (56%). In 10 children contrast enhanced scans were available for evaluation, showing contrast enhancement of the bone marrow in 83.3% and of the adjacent soft tissue in 66.7%. Fusiform swelling of the ischiopubic fusion zone, signal alteration and contrast enhancement of both the bone marrow and the adjacent soft-tissue are characteristic features of the IPS on MRI, which may be due to mechanical stress at this temporary joint. These features are nonspecific and may resemble tumor, infection or trauma. Fibrous "bridging" was the only finding on MRI, which has not been described for any other entity and thus, it seems to be a characteristic MRI-feature.