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PubMed2002

Cubonavicular coalition: a case report and literature review.

Tarsal coalitions are uncommon entities. Talocalcaneal and calcaneonavicular synostosis are present in most of the reported cases. Cubonavicular coalition represents only 1% of all the cases in the literature reviewed. The current authors report on a new case of bilateral cubonavicular coalition and present a literature review. The authors also report on the etiology, clinical and radiologic findings, and the treatment to be used. Tarsal coalitions are uncommon entities that must be suspected in an adolescent with painful feet who also has decreased mobility and deformity of the feet.

Adolescent↗
PubMed2003

Anthropometric study of synostotic frontal plagiocephaly: before and after fronto-orbital advancement with correction of nasal angulation.

Surgical correction of synostotic frontal plagiocephaly (unilateral coronal synostosis) focuses on the asymmetry of the forehead and orbits. However, there is controversy regarding whether nasal angulation should be addressed during primary fronto-orbital advancement in infancy. This prospective study was undertaken to answer that question. Preoperative and postoperative anthropometric measurements were obtained for 19 infants with nonsyndromic synostotic frontal plagiocephaly. The measurements included nasal angulation, nasion-to-endocanthion distance, nasion-to-exocanthion distance, and exocanthion-to-tragion distance. All patients underwent bilateral parallelogrammatic fronto-orbital correction. Closing wedge nasal ostectomy was performed for group I (n = 14) and was not performed for group II (n = 5). The average age at the time of follow-up assessments was 3 years 8 months (range, 1 to 14 years) in group I and 5 years 5 months (range, 2 to 15 years) in group II. A statistically significant change was observed for patients who underwent primary correction of nasal angulation; the change correlated with improved naso-orbital symmetry, as judged with nasion-to-endocanthion and nasion-to-exocanthion measurements (p < 0.01 and p < 0.05, respectively). Group I patients exhibited an average preoperative nasal angulation of 9.15 +/- 0.8 degrees that decreased to 3.1 +/- 0.6 degrees postoperatively (p < 0.01). Group II patients exhibited an average preoperative nasal angulation of 6.4 +/- 0.7 degrees that was unchanged postoperatively at 7.2 +/- 1 degrees. The improvement in nasal angulation in group I was particularly striking because the patients in group II exhibited, on average, a lesser degree of preoperative nasal deviation (p < 0.01). This prospective comparison of fronto-orbital correction of synostotic frontal plagiocephaly with and without nasal correction confirmed an earlier study and demonstrated that angulation of the nasal pyramid does not self-correct within 5 years after traditional bilateral fronto-orbital repair. Closing wedge nasal ostectomy results in improved nasal angulation and naso-orbital symmetry, without evidence of distortion or inhibition of nasal growth.

Anthropometry↗
PubMed1977

Congenital malformation of the feet with low body height. A new syndrome, caused by an autosomal dominant gene.

Among 75 members of a Danish family, 12 were found with a syndrome not previously described. Clinically, the syndrome consists of low body height and rigid flat feet, with weight-bearing pain in the feet. Radiologically, the deformation of the feet is a medial synostosis between the talus and the calcaneus combined with ankle joint dysplasia. The cause of the syndrome is most probably an autosomal dominant gene with complete penetrance. No linkage was found of the gene to 18 marker genes.

Adult↗
PubMed1982

Present nosology of the Cenani-Lenz type of syndactyly.

Synostoses of the carpals and metacarpals with oligodactyly were noted in a man whose brother was similarly affected. Since the proband's two children are normal, autosomal recessive transmission is probable. Although abnormalities of the feet, and particularly radioulnar synostosis, are lacking, this malformation is comparable to the Cenani-Lenz type of syndactyly. Interfamilial variations of published cases suggest subdivision of a new nosologic group.

Adult↗
PubMed2004

Abnormal steroidogenesis in three patients with Antley-Bixler syndrome: apparent decreased activity of 17alpha-hydroxylase, 17,20-lyase and 21-hydroxylase.

BACKGROUND: Antley-Bixler syndrome (ABS) is characterized mainly by abnormal skeletal morphogenesis such as craniosynostosis and radiohumeral synostosis, and by ambiguous genitalia in some cases. The mechanisms resulting in these deformities have not been determined. METHODS: The adrenal and gonadal function of three Japanese ABS patients were evaluated. Patient 1 (17-year-old-male) had bilateral cryptoorchidism, delayed puberty and symptoms of glucocorticoid deficiency. Patient 2 (14-year-old male) and patient 3 (4-year-old female) presented with emaciation. Additionally, patient 3 had partial labial fusion and common urogenital sinus. In each patient, blood sampling for steroid analysis before and after rapid adrenocorticotropic hormone (ACTH) stimulation was carried out. Additionally, urinary steroids were quantified. Molecular analysis of CYP17 and CYP21A2 were also performed. RESULTS: All patients showed elevated basal 17alpha-deoxysteroid levels. Although the 17alpha-deoxysteroid levels further increased after rapid ACTH stimulation, 17alpha-hydroxysteroids including cortisol did not respond, suggesting impaired 17alpha-hydroxylation. Patient 1 and patient 2 showed low adrenal androgen blood levels both before and after rapid ACTH stimulation. Patient 3 showed lower than normal excretions of urinary androgens. Additionally, a prolonged ACTH stimulation in patient 3 failed to elicit significant increase of adrenal androgens. These findings suggested impaired 17,20-lyase activity. In contrast to attenuated 17alpha-hydroxycorticosteroids, notably cortisol, elevated 17alpha-hydroxyprogesterone (17OHP) levels were observed, not only in pubertal patients (1 and 2) but also in prepubertal patient 3, indicating impaired 21-hydroxylation. This assumption was supported by increased urinary 21-deoxycortisol metabolite excretion in patients 2 and 3. With the exception of a heterozygous mutation of CYP17 in one of the patients, other mutations of this gene or CYP21A2 were identified in any of the patients. CONCLUSION: Combined decreased 17alpha-hydroxylation, 17,20-lyase activity and 21-hydroxylation was detected in three ABS patients. Considering that the enzymes responsible are all cytochrome P450 enzymes and that another cytochrome P450 enzyme, lanosterol 14alpha-demethylase, has recently been shown to be impaired in an ABS patient, we speculate that dysfunction of a system which commonly regulates cytochrome P 450 activity may be responsible for the ABS phenotype.

Abnormalities, Multiple↗
PubMed1987

Reactions of the condylar tissues to attempts to increase mandibular growth.

The relationship between the condyle and the glenoid fossa was changed in growing Long Evans/Turku rats by provoking an artificial cranial synostosis of the interparietal, temporoparietal and lambdoidal sutures. As a result the glenoid fossa was displaced posterosuperiorly, the length of the mandible was slightly increased and the condyle appeared flattened in shape by 50 days. The mesenchymal cell layer was thickened and chondrogenesis was disturbed. This disturbance was carried over to the cartilage in the course of growth, where it was seen in the presence of acellular and necrotic regions. The findings indicate that any attempt to increase the growth of the mandible by carrying it forwards should be executed gradually in order to allow the mesenchymal cells in the posterosuperior region of the condyle to alter their metabolism.

Animals↗
PubMed2003

Variations in calcaneonavicular morphology demonstrated with radiography.

PURPOSE: To determine and classify radiographically demonstrated variations in calcaneonavicular morphology and to estimate prevalence in a clinically relevant patient population. MATERIALS AND METHODS: Retrospective review was performed of foot radiographs obtained during diagnostic evaluation of 460 consecutive patients who presented to the emergency department with acute foot pain. Variations in calcaneonavicular morphology depicted on the medial oblique view (obtained at a 45 degrees angle) were classified into four groups according to morphologic type (types 1-4), and the prevalence of each type was calculated. Chi2 analysis was used to compare the prevalence of each type in male patients and in female patients. One-way analyses of variance were used to compare mean ages of patients for each type and mean calcaneonavicular gaps for each type. RESULTS: The prevalence of morphologic types 1, 2, and 3 was 94.3%, 2.8%, and 2.8%, respectively. The combined prevalence of types 2 and 3 (calcaneonavicular coalitions produced by synchondrosis and syndesmosis, respectively) was 5.6% (95% CI: 3.5%, 7.8%). There were no patients with type 4 morphology (synostosis). The numbers of male patients and female patients with morphologic types 1-3 were approximately equal (P =.9), and there was no statistically significant correlation between any of these three morphologic types and patient age (P =.2). The calcaneonavicular gap was significantly narrower in types 2 and 3 than in type 1 (P =.01), which was characterized as the normal morphology. CONCLUSION: The general prevalence of calcaneonavicular coalition (synchondrosis and syndesmosis) may be greater than previously reported, but further research is needed to prove the validity of this hypothesis.

Adolescent↗
PubMedDate not supplied

Total vertex craniectomy for the treatment of scaphocephaly.

Sutural stripping, morcellation, subtotal craniectomy, and even subtemporal decompression, have been employed in the treatment of sagittal synostosis. This report describes a further modification of past techniques, the total vertex craniectomy. This procedure, employed in 9 patients, has achieved uniformly excellent cosmetic results.

Craniotomy↗
PubMedDate not supplied

Total cranial vault reconstruction for the older child with scaphocephaly.

A technique is described for the surgical correction of primary of secondary sagittal synostosis in the older child. This involves placing the patient in the modified prone position, reshaping the supraorbital bar, and reconstructing virtually the entire cranial vault with microplates. This technique of total cranial vault reconstruction has been performed successfully on 5 patients without mortality or significant morbidity, although blood loss is significant, averaging 1.8 blood volumes. Further experience will be needed to establish the proper indications for the procedure, the best method of fixation, and the optimal timing.

Child, Preschool↗
PubMed1979

A study on the occurrence of alkaline phosphatase in the sutura interfrontalis of Wistar rats.

The aim of the present study was to determine the presence of alkaline phosphatase during various stages in development and closure of the sutura interfrontalis. The histological sections reveal that this enzyme could primarily be demonstrated in the dura mater of this suture. In further developmental stages, alkaline phosphatase could be observed within the intermediate zone as well as the pericranium. These findings are brought in relation with the occurrence of synostosis which can be induced under experimental conditions.

Age Factors↗
PubMedDate not supplied

Tarsal coalition in the young athlete.

Twenty young athletes with tarsal coalition were studied. There was a total of 26 feet including 15 cases of calcaneonavicular bars, 9 cases of talocalcaneal bars, 1 talonavicular bar, and 1 mixed. Twenty-five feet had bar resection. Calcaneonavicular bar resection is the recommended procedure for the adolescent who presents with a painful tarsal coalition. The talocalcaneal synostosis resection has been mentioned in the literature as yielding poor results. This was not true in my series. The eight cases of talocalcaneal coalition presented all had improvement in motion and decreased pain after surgical excision of the bar.

Adolescent↗
PubMed2003

Salvage technique for treatment of periplafond tibial fractures: the modified fibula-pro-tibia procedure.

We describe a technique of fixation for treatment of distal tibia periplafond fractures and nonunions that uses a modification of the principle of the fibula-pro-tibia procedure (fusing the tibia and fibula together to create a one-bone lower leg). The fibula is plated, and the screws are brought across to the medial tibial cortex. The procedure is accomplished with or without a tibial buttress plate and always includes iliac crest bone grafting of the nonunion site and synostosis. We have used this technique in five patients with satisfaction.

Adult↗
PubMed1996

Chronic Monteggia lesions in children. Complications and results of reconstruction.

We retrospectively reviewed the results of operative treatment of chronic Monteggia lesions (Bado type I or the equivalent) with anterior radiocapitellar dislocation in seven patients. The mean age at the time of the reconstruction was six years and nine months (range, eleven months to twelve years), and the mean time from the injury to the operation was twelve months (range, five weeks to thirty-nine months). The mean duration of follow-up was four years and six months (range, two years to eleven years and three months). There were fourteen complications, including malunion of the ulnar shaft in one patient; residual radiocapitellar subluxation in two patients (one anterior and one posterolateral); radiocapitellar dislocation (dynamic anterior subluxation of the radial head in supination) in one patient; transient ulnar-nerve palsy in three patients (with residual weakness in two); partial laceration of the radial nerve in one patient; loss of the fixation in two patients; and non-union of the ulnar osteotomy site, compartment syndrome, conversion reaction, and possible fibrous synostosis of the forearm in one patient each. The patients lost a mean of 36 degrees of pronation and a mean of 27 degrees of supination of the forearm compared with the contralateral, uninjured extremity. Two patients demonstrated a loss of flexion of the elbow of 8 and 13 degrees and three had a loss of extension (mean, 15 degrees) compared with the contralateral side. There were three good, two fair, and two poor results.

Age Factors↗
PubMed1979

Single layered periosteoplasty in experimental mid-palatal clefts. A histological study in the cat.

At the age of 2 months, the mid-palatal suture, with adjacent bone and nasal mucoperiosteum, was resected in 20 domestic cats leaving postoperatively only the oral side of the more than 5 mm wide osseous defect covered with mucoperiosteum. After cessation of general growth this single layered "palatal cleft periosteoplasty" had, an all animals, resulted in the formation of new bone extending from the resection edges, i.e. nearly butting in the midline in about 85% of the cases. Bone bridges interlocking the palatal halves were, as in normal untreated individuals, never observed. In 3 out of 20 other cats where merely an oral mucoperiosteal flap had been elevated and replaced, i.e. a shamoperation without resection of bone or mucoperiosteum, a beginning synostosis of the mid-palatal suture was found. In the remaining 17 cats of this last group and in further 20 unoperated controls, the suture was open even at 2 1/2 years of age. The results seem to indicate that soft tissue trauma inevitable in palatal surgery may interfere with periosteal and sutural integrity. In addition, the findings may render new informations in palatal/maxillary growth following cleft palate periosteoplasty.

Animals↗
PubMed1991

An unusual intermetatarsal coalition.

Synostoses commonly occur in the midfoot and rearfoot. However, metatarsal synostosis is a less common phenomenon. Several etiologies have been described. The authors present a case of a fourth-fifth intermetatarsal bar, which does not fit into one of the established categories, along with the surgical technique used in its correction.

Adult↗
PubMed1999

Musculoskeletal manifestations of the Antley-Bixler syndrome.

The Antley-Bixler syndrome is a rare disorder with many musculoskeletal anomalies that demand orthopedic assessment. The syndrome includes skeletal, craniofacial, and urogenital anomalies. The most common skeletal deformities are radiohumeral synostosis, craniosynostosis, multiple joint contractures, and arachnodactyly. Other orthopedic manifestations that may occur are femoral bowing, ulnar bowing, camptodactyly, synostoses of carpal and tarsal bones, clubfoot, vertebral body anomalies, perinatal fractures, and advanced skeletal age. The inheritance pattern is thought to be autosomal recessive. A patient with this syndrome is described, which is the 18th of 24 reports published in the world literature. This case is compared with the other reported cases.

Abnormalities, Multiple↗
PubMed1999

Juberg-Hayward syndrome: a new case report and clinical delineation of the syndrome.

We report a new case of Juberg-Hayward (orocraniodigital) syndrome (JHS). This 7-year-old Thai boy had characteristic features together with a number of newly recognized findings. Those findings are humeroradial synostosis (HRS), carpal anomalies, simian crease, brachydactyly A4, widely spaced nipples, seizures and myopia. Clinical delineation and correlation between the phenotypes of the syndrome are discussed.

Abnormalities, Multiple↗
PubMedDate not supplied

Remodeling of the sagittal suture in osteopetrotic (op/op) mice associated with cranial flat bone growth.

It is well known that cranial flat bone experiences growth and development at the sutural interface, which is regarded as a neutral zone to control mechanical stimuli. In osteopetrotic (op/op) mice, meanwhile, cranial deformation is produced by the deficiency of osteoclasts and the subsequent defect of bone resorption. It would be a reasonable assumption that such disturbance in bone remodeling affects sutural modification and the relevant cranial flat bone development. The present study was thus conducted to examine histological features of the sagittal sutures in op/op mice, with special reference to the relevant bone remodeling. The sagittal sutures in 10-, 15-, 30-, and 60-day-old normal and op/op mice were observed microscopically. Furthermore, osteoclastic activity was evaluated on the sections stained with tartrate-resistant acid phosphatase (TRAP). The sutures of 15-day-old op/op mice showed stenosis and synostosis, and less-developed collagen fibers associated with an irregular arrangement of fibroblasts, whereas these changes were rarely found in normal mice. Osteoclasts were hardly detected in the parietal bones around the sutures of op/op mice, although the number was numerous in normal mice. These results emphasize that congenital deficiency in osteoclast produces unbalanced bone remodeling at the sutural interface and on the surfaces of the cranial bones, which is assumed to be closely related to cranial bone deformity in op/op mice.

Animals↗