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1.
Br J Plast Surg ; 57(4): 295-302, 2004 Jun.
Article in English | MEDLINE | ID: mdl-15145731

ABSTRACT

Chest wall reconstruction following extensive resection is greatly facilitated by the use of vascularised flaps and prosthetic material. Plastic surgeons are often asked to assist with coverage of large chest wall defects. However, in addition to soft tissue coverage, we need to address other important issues such as the status of the pleural cavity, and the requirement for skeletal support. The purpose of this report is to analyse our experience, provide a reconstructive algorithm following the ablative procedure and review the literature. Two hundred chest wall resections were performed from 1975 to 2000. Defect location was divided into anterior (n = 73) lateral (n = 36) anterior-lateral (n = 36) posterior-lateral (n = 19) posterior (n = 22) and forequarter (n = 14) Average number of ribs resected was four. One hundred and fifty-eight patients (79%) required chest wall reconstruction with either prosthetic material and/or flap closure. Mesh closure was required in 85 cases (43%), being highest for lateral defects (61%), and lowest for anterior defects (31%). Vascularised flaps were needed in 112 patients (56%), more common in anterior defects (79%), and less common for the posterior-lateral defects (26%). Inpatient complication rate was 27% (43/158) following reconstruction, with a mortality of 6% (10/158). Chest wall reconstruction is common following extensive resection. This includes management of the pleural cavity, skeletal support and soft tissue coverage. A better understanding of the respiratory mechanics and local thoracoabdominal anatomy is crucial for managing these complex defects. The need for skeletal support was more prevalent in lateral and posterior-lateral defects. Flap reconstruction was required more often to cover large anterior defects, with regional flaps predominating.


Subject(s)
Plastic Surgery Procedures/methods , Thoracic Wall/surgery , Adolescent , Adult , Aged , Aged, 80 and over , Algorithms , Clinical Protocols , Female , Humans , Length of Stay , Male , Middle Aged , Postoperative Complications/etiology , Surgical Flaps , Surgical Mesh , Treatment Outcome
2.
J Craniofac Surg ; 12(5): 451-5; discussion 456-7, 2001 Sep.
Article in English | MEDLINE | ID: mdl-11572250

ABSTRACT

A retrospective review was performed on 63 patients at Childrens Healthcare of Atlanta at Scottish Rite who underwent correction of single-suture craniosynostosis using a resorbable fixation system. Included in the series were 24 patients with metopic synostosis, 15 with sagittal synostosis, and 24 with unicoronal synostosis. The average age at operation was 22.7 months (range: 2.8 months-18 years), and mean follow-up time was 30.7 months (range: 7.1-10 years). Reoperation equal to or exceeding the magnitude of the original procedure occurred in 4.76% of the patients. This was comparable to the reoperation rate observed at our institution using traditional fixation systems. Minor complications related to the use of resorbable plates were also identified, and the final outcome for single-suture synostosis was favorable. Results suggest that resorbable plates and screws are as effective as titanium-based systems in the treatment of single-suture synostosis.


Subject(s)
Absorbable Implants , Craniosynostoses/surgery , Craniotomy/instrumentation , Adolescent , Biocompatible Materials , Bone Plates , Bone Screws , Child , Child, Preschool , Craniotomy/adverse effects , Craniotomy/economics , Female , Health Care Costs , Humans , Infant , Lactic Acid , Male , Polyglycolic Acid , Polylactic Acid-Polyglycolic Acid Copolymer , Polymers , Reoperation , Retrospective Studies , Titanium , Treatment Outcome
3.
Plast Reconstr Surg ; 108(2): 361-9, 2001 Aug.
Article in English | MEDLINE | ID: mdl-11496176

ABSTRACT

Numerous procedures are available for nipple reconstruction with no true universal favorite. This study presents long-term follow-up data for nipple reconstruction using the C-V flap technique. Patients were identified by searching the Breast Reconstruction Database, and they were asked to return for a follow-up visit. All those who underwent nipple reconstruction using the C-V flap technique between January of 1992 and December of 1996 were reviewed in an attempt to conduct a long-term follow-up evaluation. The response was poor, and 11 patients participated in the study and returned for follow-up. They all completed a questionnaire, which focused on patient satisfaction using a visual analogue scale. Nipple measurements were taken with a caliper and compared with the opposite breast for symmetry. Fourteen nipple reconstructions were evaluated in 11 patients with an average follow-up of 5.3 years. All patients had undergone transverse rectus abdominis musculocutaneous (TRAM) flap reconstructions. Patient satisfaction was 42 percent with nipple projection, 62 percent with pigmentation, and 26 percent with sensation. Overall patient satisfaction with the procedure was 81 percent. Average nipple projection of the reconstructed nipple was 3.77 mm and was not statistically different when compared with the opposite nipple. Long-term subjective evaluation of the C-V flap technique does report a loss in nipple projection; however, overall patient satisfaction at 5.3 years is good, as is the ability to restore symmetry with the opposite breast.


Subject(s)
Mammaplasty , Nipples/surgery , Surgical Flaps , Adult , Breast Neoplasms/surgery , Female , Follow-Up Studies , Humans , Mammaplasty/methods , Middle Aged , Patient Satisfaction , Reoperation , Tattooing
6.
Ann Plast Surg ; 46(3): 222-8, 2001 Mar.
Article in English | MEDLINE | ID: mdl-11293510

ABSTRACT

Skin sparing mastectomy (SSM) removes the breast, nipple-areolar complex, previous biopsy incisions, and skin overlying superficial tumors. Preservation of the native skin envelope facilitates immediate breast reconstruction. The procedure has been adopted for the treatment of breast cancer. All cases of SSM and immediate breast reconstruction performed by the senior author (G.W.C.) from January 1, 1993, through December 12, 1997, were reviewed. Patient demographics, cancer staging, treatment, types of surgery performed, and postoperative outcomes were examined. Aesthetic outcomes were measured using four 3-point subscales. A total of 100 patients underwent 118 SSMs during the study period. The American Joint Committee on Cancer staging was as follows: stage 0, 27 patients; stage I, 25 patients; stage II, 39 patients; stage III, 7 patients; stage IV, 3 patients; recurrent, 2 patients; and cystosarcoma phylloides, 1 patient. The mean follow-up was 42.7 months. Local recurrence occurred in 2 patients (2.7%). Reconstructive methods included the transverse rectus abdominis musculocutaneous flap (N = 82; pedicled, 73; free, 9), the latissimus flap (N = 18), and tissue expansion (N = 20). Two patients underwent contralateral delayed reconstruction. The aesthetic results achievable with the three methods were similar. The failure rate was higher for expander reconstruction (10%) than those observed for transverse rectus abdominis musculocutaneous (4.9%) and latissimus (5.6%) flaps. SSM can be used in the treatment of invasive breast cancer without compromising local control. The aesthetic results of the three methods were similar, but tissue expander reconstruction had a higher failure rate.


Subject(s)
Breast Neoplasms/surgery , Mammaplasty/methods , Mastectomy/methods , Adult , Aged , Esthetics , Female , Humans , Middle Aged , Postoperative Complications , Statistics, Nonparametric , Surgical Flaps , Tissue Expansion Devices , Treatment Outcome
7.
Ann Plast Surg ; 46(3): 242-9, 2001 Mar.
Article in English | MEDLINE | ID: mdl-11293514

ABSTRACT

This study provides a retrospective analysis of 60 patients who underwent thoracic reconstruction with the omentum. Patients were identified by searching several databases to determine demographics, indications for surgery, operative technique, and postoperative course, including donor and recipient site morbidity. From January 1975 to May 2000, the authors harvested and transferred the omentum successfully (57 pedicled, 3 free) in 60 patients (mean age, 60 years; age range, 21-86 years) for sternal wound infections (N = 34), chest wall resections (N = 17), pectus deformities (N = 2), intrathoracic defects (N = 4), and breast reconstruction (N = 3). The omentum was used as a primary flap in 39 patients and as a salvage flap in 21 patients. Average operative time was 3.9 hours and average hospital stay was 34.3 days. Partial flap loss occurred in 7 patients, with no total flap failures. Morbidity included six abdominal wound infections and seven epigastric hernias. Mortality was 11.7%. The omentum can be harvested safely and used reliably to reconstruct varying thoracic wounds and defects. Specific indications from this series include osteoradionecrosis, chest wall tumors, massive sternal wounds, and refractory mediastinitis. Hultman CS, Culbertson JH, Jones GE, et al. Thoracic reconstruction with the omentum: indications, complications, and results.


Subject(s)
Omentum/transplantation , Plastic Surgery Procedures/methods , Surgical Flaps , Thoracic Diseases/surgery , Adult , Aged , Female , Humans , Male , Middle Aged , Postoperative Complications , Retrospective Studies , Treatment Outcome
9.
Ann Plast Surg ; 44(2): 147-53, 2000 Feb.
Article in English | MEDLINE | ID: mdl-10696040

ABSTRACT

Abdominal wall reconstruction of ventral hernia defects with loss of visceral domain and inadequate soft-tissue coverage presents a surgical challenge. Four patients with large, skin grafted ventral hernia defects were treated by staged abdominal wall reconstruction. During the initial stage, tissue expanders were placed under the skin and subcutaneous tissue lateral to the defects. After adequate interval expansion, the second stage was performed. The expanders were removed, the visceral contents reduced easily, and the fascia reapproximated with polypropylene mesh. The expanded skin was closed easily over the fascial repair. All four patients were reconstructed successfully without complications. Tissue expansion can restore abdominal domain and allow soft-tissue closure in complicated ventral hernia defects.


Subject(s)
Abdominal Muscles/surgery , Hernia, Ventral/surgery , Tissue Expansion/methods , Humans , Male , Middle Aged , Plastic Surgery Procedures/methods , Reoperation
10.
Ann Thorac Surg ; 67(3): 845-7, 1999 Mar.
Article in English | MEDLINE | ID: mdl-10215247

ABSTRACT

Thoracic vertebral body hemicorpectomy and chest wall resection was performed in a 17-year-old male patient with a posterior mediastinal tumor thought to be neurogenic in origin. No preoperative tissue diagnostic endeavor was made. Final pathologic diagnosis showed this tumor to be Ewing's sarcoma. This communication alerts the thoracic surgeon to the need for definitive diagnosis of posterior mediastinal masses with vertebral body involvement, particularly in children. Induction chemotherapy is the accepted standard of management of these sarcomas.


Subject(s)
Bone Neoplasms/diagnosis , Mediastinal Neoplasms/diagnosis , Sarcoma, Ewing/diagnosis , Adolescent , Diagnosis, Differential , Humans , Male , Mediastinal Neoplasms/therapy , Ribs , Sarcoma, Ewing/therapy , Soft Tissue Neoplasms/diagnosis , Spinal Neoplasms/diagnosis , Spinal Neoplasms/therapy , Thoracic Vertebrae
11.
Ann Surg ; 229(3): 421-7, 1999 Mar.
Article in English | MEDLINE | ID: mdl-10077056

ABSTRACT

OBJECTIVE: The goals of this work were to develop animal models of lymphedema and tissue flap transfer, and to observe physiologic changes in lymphatic function that occur in these models over time, both systemically with lymphoscintigraphy (LS) and locally using fluorescence microlymphangiography (FM). SUMMARY BACKGROUND DATA: Although lymphedema has been managed by a combination of medical and surgical approaches, no effective long-term cure exists. Surgical attempts aimed at reconnecting impaired lymphatic channels or bypassing obstructed areas have failed. METHODS: The tails of rats (A groups) and mice (B groups) were used because of their different features. Lymphedema was created by ligation of the lymphatics at the tail base and quantified by diameter measurements there. In the experimental group, rectus abdominis myocutaneous flap was transferred across the ligation. In addition to the ligation (A1 and B1) and ligation + flap (A2 and B2) groups, three control groups were included: sham flap with ligation (B4), sham flap alone (B5), and normal (A3 and B3) animals. Observations were made at weekly time points for lymphatic function and continuity. RESULTS: Lymphedema was successfully created in the mouse ligation groups (B1 and B4) and sustained for the entire length of observation (up to 14 weeks). Lymphatic continuity was restored in those animals with transferred flaps across the ligation site (A2 and B2), as seen both by LS and FM. Sham flaps did not visibly affect lymphatic function nor did they cause any visible swelling in the tail. CONCLUSIONS: Acute lymphedema developing after ligation of tail lymphatics in mice can be prevented by myocutaneous flap transfer. Restored lymphatic continuity and function were demonstrable using lymphoscintigraphy and fluorescence microlymphangiography.


Subject(s)
Disease Models, Animal , Lymphedema/surgery , Surgical Flaps , Acute Disease , Animals , Female , Lymph Nodes/diagnostic imaging , Lymph Nodes/physiology , Lymphography , Mice , Mice, Nude , Radionuclide Imaging , Rats , Rats, Inbred F344
12.
Ann Plast Surg ; 41(2): 171-9, 1998 Aug.
Article in English | MEDLINE | ID: mdl-9718151

ABSTRACT

Midfacial hypoplasia following primary cleft lip repair is a common clinical correlate. Recent experimental work has suggested that increased lip pressure following an undermined lip repair may contribute to midfacial growth inhibition. Soft-tissue expansion has been used to generate additional soft tissue for reconstruction in the cranial region. The use of this technique in the labial region may allow lip repair to be performed with less tension (pressure) and thus facilitate midfacial growth. To test this hypothesis, 40 4-week-old rabbits were randomly divided into four groups. Two groups had surgically created lip and alveolar defects. Ipsilateral labial tissue expanders were placed in all four groups. One cleft and one normal group underwent expansion. The other two groups served as a control. The expanders were removed at 4 weeks, and an undermined lip repair was performed in both cleft groups. Findings revealed that soft-tissue expansion increased labial surface area significantly by approximately 96% (p < 0.001). Labial soft-tissue expansion alone had no effect on midfacial growth during the observation period. In contrast, cleft animals undergoing tissue expansion exhibited significantly reduced (p < 0.05) postoperative lip pressure and increased midfacial growth compared with cleft animals without expansion through 36 weeks of age. Results suggest that preoperative tissue expansion reduced postoperative lip pressure and improved midfacial growth in a rabbit cleft lip model.


Subject(s)
Cleft Lip/surgery , Lip/surgery , Maxillofacial Development , Tissue Expansion , Animals , Disease Models, Animal , Evaluation Studies as Topic , Female , Humans , Male , Rabbits , Random Allocation
13.
Am Surg ; 63(11): 975-8, 1997 Nov.
Article in English | MEDLINE | ID: mdl-9358785

ABSTRACT

Acute torsion of the gallbladder is a rare and poorly understood entity. Since it was first described by A.V. Wendel in 1898, approximately 300 cases have been reported in the literature. The treatment of choice remains immediate cholecystectomy, and most cases are diagnosed intraoperatively. There are typical clinical and radiological findings consistent with torsion of the gallbladder that should raise the index of suspicion for this condition preoperatively. We present a case of gallbladder torsion and discuss the pertinent literature. A re-emphasis is placed on the salient clinical features, and the availability of diagnostic tests is stressed. Given the possibility of laparoscopic cholecystectomy and the increasing incidence with which gallbladder torsion is being witnessed today, the importance of a preoperative suspicion is discussed.


Subject(s)
Gallbladder Diseases/diagnosis , Aged , Aged, 80 and over , Cholecystitis/diagnosis , Female , Gallbladder Diseases/diagnostic imaging , Gallbladder Diseases/surgery , Humans , Torsion Abnormality , Ultrasonography
14.
Plast Reconstr Surg ; 96(3): 689-98, 1995 Sep.
Article in English | MEDLINE | ID: mdl-7638294

ABSTRACT

This study was designed to assess the effects of overdistraction of an experimentally immobilized coronal suture using an internal appliance on craniofacial growth in rabbits. Fifty-three, 1.5-week-old rabbits were used. Markers were placed on either side of the calvarial sutures. Thirty-nine rabbits had bilateral coronal suture immobilization using methyl methacrylate; 14 rabbits served as normal controls. At 6 weeks of age, the 39 immobilized rabbits were randomly assigned to four groups: (1) immobilized controls (n = 14); (2) suturectomy (n = 6); (3) suturectomy with distraction (n = 9); and (4) suturectomy with overdistraction (n = 10). Lateral head radiographs were taken at 1.5, 6, 12, and 18 weeks of age. Results revealed that, by 18 weeks of age, rabbits with overdistraction exhibited significant compensatory growth abnormalities in the cranial vault, midface, and anterior cranial base compared with the other groups. Results indicate that overdistraction may contribute to craniofacial anomalies through altered growth vectors and compressive tension-stress forces at adjacent sutures and suggest that it may be important to keep "pace" with the growing coronal suture and neurocapsular matrix during distraction to reestablish normal craniofacial morphology.


Subject(s)
Cranial Sutures/surgery , Craniosynostoses/surgery , Internal Fixators , Animals , Postoperative Complications , Rabbits , Skull/growth & development
15.
J Craniofac Surg ; 6(1): 40-8, 1995 Jan.
Article in English | MEDLINE | ID: mdl-8601006

ABSTRACT

The effects of long-term afferent denervation on midfacial and nasal capsule growth after craniofacial or skull base surgery are unknown. Experimental studies suggest that afferent nerves may play a role in regulating craniofacial growth through neurotrophic influences. The present study was designed to assess the effects of nasal capsule afferent (V1 and V2) denervation on nasal capsule growth in the rabbit. Eighteen 1 1/2-week-old rabbits were evenly divided into three groups: unoperated controls, sham controls, and animals with unilateral anterior ethmoid and sphenopalatine nerve lesions. The nerves were lesioned at the skull base with electrocautery through an intraorbital approach at 1 1/2 weeks of age. Lateral head radiographs and dental study models were taken at 1 1/2, 3 1/2, 6, and 12 weeks of age. By 3 1/2 weeks of age, sham controls and animals with nerve lesions exhibited significant (p < 0.05) reductions in nasal capsule area and height compared with unoperated control animals; the lesioned animals were more severely affected. However, by 6 weeks no differences were noted between the sham and lesioned groups, which were both smaller than unoperated controls through 12 weeks of age. Results suggest that acute nasal capsule growth deficits are a consequence of surgery as well as afferent denervation, with a greater deficit in the latter group, possibly through neurotrophic interruption. Such deficits may not be a permanent effect in the rabbit model.


Subject(s)
Maxillofacial Development/physiology , Nose/growth & development , Nose/innervation , Skull/surgery , Trigeminal Nerve/physiology , Afferent Pathways/physiology , Afferent Pathways/surgery , Age Factors , Analysis of Variance , Animals , Cephalometry , Craniotomy/adverse effects , Craniotomy/methods , Denervation , Electrocoagulation , Female , Male , Rabbits , Trigeminal Nerve/surgery
16.
Int J Oral Maxillofac Surg ; 23(6 Pt 2): 443-5, 1994 Dec.
Article in English | MEDLINE | ID: mdl-7890996

ABSTRACT

A recently developed biodegradable system made of DL-polylactic acid (DL-PLA) for internal fixation of non-weight-bearing bones of the craniofacial skeleton was investigated. The plates were used for rigid fixation of experimental nasal bone fractures in 20 New Zealand white rabbits. In addition, prebent plates were placed in subcutaneous pockets in the backs of the animals. The material was removed after 7, 14, 28, and 42 days, and bending angles, plate stability, molecular weights (MW), and histologic analyses were studied. A significant decrease of MW over time and a difference in MW loss, showing a faster degradation subcutaneously, were observed. Plate stability did not decrease during the interval of 6 weeks, but a loss of bending angle was found in all prebent implants. This effect was caused by memory of DL-PLA. The results suggest that memory of biodegradable materials should be investigated before clinical application and that degradation rates differ according to the site of implantation.


Subject(s)
Facial Bones/surgery , Fracture Fixation, Internal/instrumentation , Lactates/chemistry , Lactic Acid , Polymers/chemistry , Prostheses and Implants , Skull Fractures/surgery , Animals , Biodegradation, Environmental , Bone Plates , Bone Screws , Disease Models, Animal , Elasticity , Hydrolysis , Kinetics , Molecular Weight , Nasal Bone/injuries , Nasal Bone/surgery , Polyesters , Rabbits
17.
J Craniofac Surg ; 5(5): 318-26, 1994 Nov.
Article in English | MEDLINE | ID: mdl-7833414

ABSTRACT

Correction of craniosynostosis involves removal of the coronal suture to allow for expansion of the developing brain and normal craniofacial growth. Frequently, however, the site reossifies, restricts growth, and requires additional surgery. The present study was designed to assess the effects of an internal, subperiosteal calvarial distractor on suturectomy site patency and compensatory craniofacial growth changes in an experimental rabbit model of coronal suture synostosis. In the present study, 43 1 1/2-week-old rabbits were used. Amalgam markers were placed across the frontonasal, coronal, and anterior lambdoidal sutures in all animals. Twenty-nine rabbits underwent bilateral coronal suture immobilization using methyl methacrylate. Fourteen rabbits were left untreated and served as sham controls. At 6 weeks of age, the immobilized rabbits were divided into three groups: (1) immobilized (controls), (2) suturectomy, and (3) suturectomy and distraction. The distractors were activated percutaneously at an average of 0.6 mm/week for 5 weeks (3 mm total). Lateral head radiographs were taken at 1 1/2, 6, 12, and 18 weeks of age. Results revealed that, by 6 weeks of age, animals with coronal suture immobilization exhibited significantly (p < 0.01) reduced growth across the coronal suture, resulting in shorter and inferiorly rotated cranial vaults compared with sham controls. By 12 weeks of age, rabbits with distraction returned to normal (sham control) coronal suture marker separation, whereas rabbits with immobilized sutures and suturectomy only showed significantly reduced marker distances. Rabbits with distraction also exhibited greater anterior cranial base lengths compared with the other experimental groups.(ABSTRACT TRUNCATED AT 250 WORDS)


Subject(s)
Bone Lengthening/instrumentation , Craniosynostoses/surgery , Animals , Bone Lengthening/methods , Craniosynostoses/physiopathology , Female , Internal Fixators , Male , Maxillofacial Development , Prostheses and Implants , Rabbits , Skull/growth & development , Skull/surgery
18.
J Craniofac Surg ; 5(4): 247-52; discussion 253, 1994 Sep.
Article in English | MEDLINE | ID: mdl-7833399

ABSTRACT

Little is known about the minimum bacterial dose required to induce infections by superficial contamination alone (i.e., not direct inoculation). This study quantified superficial rabbit calvarial bone graft contamination by exposing parietal bone grafts to Staphylococcus aureus. Two 5 x 5-mm full-thickness calvarial bone grafts were harvested from 40 rabbits and contaminated with different concentrations (range = 10(7)-10(11) colony-forming units [CFUs]/mL) of S. aureus by immersion. One graft from each rabbit was cultured immediately, and the other was inset into a calvarial donor site, fixed in place with wire, and observed for evidence of infection. At harvest (28 days postoperatively), a bacterial exposure of greater than 10(8) CFUs/mL of S. aureus was required to induce infections in the rabbit calvarial grafts (p < 0.001, Student's t-test), transmitting an infectious dose of approximately more than 10(4) CFUs per graft. These results validate the previously known dogma regarding the number of organisms needed to manifest infection [1,2] and highlight a multifold increment in bacterial count needed to transmit this infectious dose by superficial contamination alone. Such a model may be useful in investigating the efficacy of various treatment modalities of contaminated bone.


Subject(s)
Bone Transplantation/adverse effects , Osteomyelitis/microbiology , Staphylococcal Infections/microbiology , Surgical Wound Infection/microbiology , Air Microbiology , Analysis of Variance , Animals , Chi-Square Distribution , Colony Count, Microbial , Disease Models, Animal , Male , Osteomyelitis/etiology , Parietal Bone/transplantation , Rabbits , Staphylococcus aureus , Surgical Wound Infection/etiology
19.
Ann Plast Surg ; 32(6): 606-11, 1994 Jun.
Article in English | MEDLINE | ID: mdl-8074368

ABSTRACT

Biodegradable plates and screws have many advantages over metal plates and screws. Plates were injection-molded and made of DL-polylactic acid. Four-hole plates were bent with the use of a heated tip to angles of 30, 45, and 60 degrees. They were inserted in the subcutaneous space in the back of rabbits. After 2 weeks, the plates straightened, suggesting that the plates had a memory. The results were confirmed by similar in vitro investigations. The in vitro experiment was repeated with tempering of the plates. The plates were heated to a temperature of 200 degrees C for 45 seconds, cooled, and then again heated to 200 degrees C for 40 seconds. This process resulted in loss of memory of the plates. After 4 weeks, there was a change of the bending angle of less than 2.5 degrees.


Subject(s)
Bone Plates , Lactates , Lactic Acid , Polymers , Animals , Biodegradation, Environmental , Chemical Phenomena , Chemistry, Physical , Molecular Weight , Polyesters , Rabbits
20.
Cleft Palate Craniofac J ; 31(1): 1-7, 1994 Jan.
Article in English | MEDLINE | ID: mdl-8130237

ABSTRACT

The lack of an animal model of congenital coronal suture (CS) synostosis has prompted the widespread use of an experimental rabbit model using adhesive immobilization of the CS. Such postnatal models have helped make significant scientific contributions but may still not fully represent all aspects of the human congenital condition. In the March 1993 issue of The Cleft Palate-Craniofacial Journal we reported a female rabbit born in our laboratory with complete bilateral CS synostosis. This follow-up study presents our attempts to breed this animal and establish a strain of craniosynostotic rabbits. To date, we have accomplished 10 back- and intercrosses with these animals and have produced a total of 71 live offspring; 10 animals exhibited complete nonsyndromic unilateral (plagiocephalic) or bilateral (brachycephalic) CS synostotic deformities at birth, and 19 animals exhibited partial CS synostosis that showed more than 75% growth retardation across the CS (well below the 95% confidence interval for normals). Results revealed that gestational time and litter size averages were consistent with those reported for the strain, although the average litter size decreased with increased inbreeding. By 1.5 weeks of age the completely synostosed animals already exhibited brachycephalic cranial vaults and midfacial hypoplasia compared to unaffected siblings. Initial pedigree analysis suggested an autosomal dominant inheritance pattern with incomplete penetrance and variable expressivity. The development of such a congenital rabbit model may prove useful in helping to understand the etiopathogenesis of this condition in human populations.


Subject(s)
Craniosynostoses/genetics , Disease Models, Animal , Facial Bones/abnormalities , Frontal Bone/abnormalities , Inbreeding , Parietal Bone/abnormalities , Rabbits/abnormalities , Skull/abnormalities , Animals , Craniosynostoses/pathology , Facial Asymmetry/genetics , Facial Asymmetry/pathology , Facial Bones/pathology , Female , Frontal Bone/pathology , Genes, Dominant/genetics , Male , Malocclusion/genetics , Malocclusion/pathology , Mutation/genetics , Occipital Bone/abnormalities , Occipital Bone/pathology , Parietal Bone/pathology , Pedigree , Prostheses and Implants , Rabbits/genetics , Skull/pathology
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