OBJECTIVES

To present a rare case of a 17-year-old girl with multiple odontogenic keratocysts, skeletal abnormalities, central nervous system and cutaneous anomalies.

METHODS

Design:Case Report

Setting:Tertiary Government Training Hospital

Patient: One

RESULTS

A 17-year-old Filipino girl presented with a one-year history of progressive left mandibular swelling. Orthopantomography revealed multiple cysts involving the mandible and maxillae. Histopathologic examination of incision biopsy specimens confirmed odontogenic keratocysts. Other physical examination findings included coarse face and multiple palmar and plantar pits. Radiologic investigations demonstrated calcification of the falx cerebri and tentorium cerebelli, bifid rib and cervicothoracic scoliosis. Based on clinical, radiological, and histopathological findings, a diagnosis of Gorlin-Goltz syndrome was established. The patient underwent enucleation and curettage of the cysts with peripheral ostectomy, and there was no recurrence on repeat orthopantomography at six months and two years post-operatively. However, on the fourth year, the patient claimed there was a mandibular cyst which was not verified as she was lost to follow-up.

CONCLUSION

This case highlights the importance of recognizing multiple odontogenic keratocysts as a potential manifestation of Gorlin-Goltz Syndrome. Early diagnosis enables appropriate management and long term surveillance to monitor for other manifestations of this syndrome that may occur later in life.

Human ; Female ; Adolescent: 13-18 Yrs Old ; Basal Cell Nevus Syndrome ; Mandible ; Radiography, Panoramic ; Focal Dermal Hypoplasia ; Ribs ; Scoliosis ; Spinal Cord ; Women ; History ; Lost To Follow-up ; Diagnosis

PURPOSE: To compare the diagnostic accuracy of cone-beam computed tomography (CBCT) with that of parallel (PPA) and oblique projected periapical (OPA) radiography for the detection of different types of peri-implant bone defects.MATERIALS AND METHODS: Forty implants inserted into bovine rib blocks were used. Thirty had standardized bone defects (10 each of angular, fenestration, and dehiscence defects), and 10 were defect-free controls. CBCT, PPA, and OPA images of the samples were acquired. The images were evaluated twice by each of 2 blinded observers regarding the presence or absence and the type of the defects. The area under the receiver operating characteristic curve (AUC), sensitivity, and specificity were determined for each radiographic technique. The 3 modalities were compared using the Fisher exact and chi-square tests, with P<0.05 considered as statistical significance.RESULTS: High inter-examiner reliability was observed for the 3 techniques. Angular defects were detected with high sensitivity and specificity by all 3 modalities. CBCT and OPA showed similar AUC and sensitivity in the detection of fenestration defects. In the identification of dehiscence defects, CBCT showed the highest sensitivity, followed by OPA and PPA, respectively. CBCT and OPA had a significantly greater ability than PPA to detect fenestration and dehiscence defects (P<0.05).CONCLUSION: The application of OPA radiography in addition to routine PPA imaging as a radiographic follow-up method for dental implantation greatly enhances the visualization of fenestration and dehiscence defects. CBCT properly depicted all defect types studied, but it involves a relatively high dose of radiation and cost.
Area Under Curve ; Cone-Beam Computed Tomography ; Dental Implantation ; Dental Implants ; Follow-Up Studies ; Methods ; Peri-Implantitis ; Radiography ; Radiography, Dental, Digital ; Ribs ; ROC Curve ; Sensitivity and Specificity

Delayed massive hemothorax requiring surgery is relatively uncommon and can potentially be life-threatening. Here, we aimed to describe the nature and cause of delayed massive hemothorax requiring immediate surgery. Over 5 years, 1,278 consecutive patients were admitted after blunt trauma. Delayed hemothorax is defined as presenting with a follow-up chest radiograph and computed tomography showing blunting or effusion. A massive hemothorax is defined as blood drainage >1,500 mL after closed thoracostomy and continuous bleeding at 200 mL/hr for at least four hours. Five patients were identified all requiring emergency surgery. Delayed massive hemothorax presented 63.6±21.3 hours after blunt chest trauma. All patients had superficial diaphragmatic lacerations caused by the sharp edge of a broken rib. The mean preoperative chest tube drainage was 3,126±463 mL. We emphasize the high-risk of massive hemothorax in patients who have a broken rib with sharp edges.
Chest Tubes ; Diaphragm* ; Drainage ; Emergencies ; Follow-Up Studies ; Hemorrhage ; Hemothorax* ; Humans ; Lacerations ; Radiography, Thoracic ; Rib Fractures* ; Ribs* ; Thoracic Injuries ; Thoracostomy ; Thorax

Supernumerary lumbar ribs are usually asymptomatic and discovered incidentally during routine diagnostic radiography. During a routine inventory of the research skeletal repository at the University of the Witwatersrand, a rare case of bilateral supernumerary ribs on the first lumbar vertebra was observed. The skeletal specimen belonged to a 70-year-old African male whose recorded cause of death was esophageal carcinoma. Plain radiography revealed bilateral joint cavities between the transverse processes of the first lumbar vertebra and the supernumerary ribs. Clinically, supernumerary lumbar ribs may present with pain of the renal angle, cause confusion during enumeration of the vertebral levels on radiographs, and may be misinterpreted as fractures, kissing osteophytes, and/or calcification of abdominal vasculature.
Adult* ; Aged ; Cause of Death ; Humans ; Joints ; Male* ; Osteophyte ; Radiography ; Ribs* ; Skeleton* ; Spine

A 2-year-old, female Pomeranian dog was referred for dyspnea. Thoracic radiographs revealed left-sided mediastinal shift, increased soft tissue opacity in the caudal aspect of left thorax with loss of the left diaphragmatic silhouette, and dorsal elevation of mediastinal structures and heart from the sternum by lung tissue. The left main bronchus was visualized as an air-bronchogram and observed to abruptly discontinue at the level of the 10th rib. Thoracic computed tomography (CT) revealed absence of the left lung parenchyma and left pulmonary vessels with a rudimentary left main bronchus. The case was congenital pulmonary aplasia diagnosed via radiography and CT.
Animals ; Bronchi ; Child, Preschool ; Diagnostic Imaging* ; Dogs* ; Dyspnea ; Female ; Heart ; Humans ; Lung ; Radiography ; Ribs ; Sternum ; Thorax

Fibrous dysplasia is a benign, bony abnormality that is usually asymptomatic. A 41-year-old male with minimal symptoms presented at this hospital with abnormal findings incidentally seen in his ribs on the chest radiograph. A skeletal survey showed numerous, osteolytic lesions throughout multiple bones. Diagnostic processes for malignancy of undefined primary origin (MUO) were performed in order to identify the underlying primary neoplasm, although abnormal findings were not seen except for multiple bone lesions. A computed tomography guided bone biopsy was performed on his left rib. The final diagnosis was fibrous dysplasia. This case demonstrates that fibrous dysplasia should be considered in the differential diagnosis in young patients with multiple, osteolytic lesions and without a prior history suggesting malignancy.
Adult ; Biopsy ; Bone Neoplasms ; Diagnosis ; Diagnosis, Differential ; Fibrous Dysplasia, Polyostotic* ; Humans ; Male ; Neoplasm Metastasis* ; Radiography, Thoracic ; Ribs

Periosteal osteosarcoma is an extremely rare chondroblastic osteosarcoma in the flat bone. There were authors reporting of two cases of periosteal osteosarcoma in the highly unusual sites. One of them arose from the rib, in a 17-year-old male, which appeared as a hypodense juxtacortical mass with periosteal reaction on CT. The other one arose from the scapula, in a 17-year-old female, which showed the intermediate signal intensity (SI) on T1-weighted image (WI), heterogeneous high SI on T2WI, and rim-enhancement on contrast-enhanced T1WI with cortical destruction on MRI.
Adolescent ; Bone Neoplasms/*radiography ; Contrast Media/diagnostic use ; Female ; Humans ; Male ; Osteosarcoma/*radiography ; Ribs/*radiography ; Scapula/*radiography

OBJECTIVESTo investigate the spatial structure of pedicle rib units in normal thoracic human spines and to compare the dimensions of the pedicle rib unit with corresponding dimensions.

METHODSThoracic spine specimens in four fresh adult cadaveric were used. Computerized tomographic (CT) images (including two-dimensional, three-dimensional reconstruction) of the thoracic spines were obtained. Measurement parameters include:the width, the height, the chord length and the sagittal angles of the pedicle rib unit compared with pedicle, especially for the pedicle-rib overlapping height.

RESULTSThe pedicle rib unit was not a simple two-dimensional structure but a three-dimensional structure. The shortest height of pedicle rib unit was (12.6 ± 0.8) mm (T(1)), while the longest was (16.9 ± 1.1) mm (T(11)). The shortest height of pedicle-rib overlap was (7.2 ± 0.3) mm (T(1)), while the longest was (11.8 ± 1.0) mm (T(10)). The height of pedicle rib unit and the height of pedicle were significantly larger than that of the pedicle-rib overlap (P < 0.05), while there was no significantly difference between the height of pedicle rib unit and the height of pedicle (P > 0.05).

CONCLUSIONSThe pedicle rib unit is a complicated spatial structure, and the longitudinal height of pedicle-rib overlap should be taken as the real height of the unit.

Adult ; Bone Screws ; Humans ; Male ; Radiography ; Ribs ; anatomy & histology ; diagnostic imaging ; Thoracic Vertebrae ; anatomy & histology ; diagnostic imaging ; surgery

Osteopetrosis is a rare bone disease characterized by systemic osteosclerosis due to an osteoclast dysfunction that decreases bone resorption. This report demonstrates two cases of adult osteopetrosis with secondary osteomyelitis of the maxilla, in siblings who are 43-year old female and 55-year old, male respectively. The common radiographic features of these cases were increased radiopacity in skull, rib and vertebra. The radiographic features that differed between these two cases were the osteosclerotic pattern of the jaw bones, that is, diffuse patterns in the female case, while the male case showed nodular patterns that were confined to the root apices. The diagnosis of osteopetrosis may be complicated due to the varying degree of osteosclerosis on panoramic radiograph. Additional radiographs such as the chest and skull radiograph may be helpful for the diagnosis of osteopetrosis.
Adult ; Bone Diseases ; Bone Resorption ; Female ; Humans ; Jaw ; Male ; Maxilla ; Osteoclasts ; Osteomyelitis ; Osteopetrosis ; Osteosclerosis ; Radiography, Panoramic ; Ribs ; Siblings ; Skull ; Spine ; Thorax

An epithelioid hemangioma involving three contiguous bones in continuity has, to the best of our knowledge, not been reported in the literature. A case of a 48-year-old man presented with radiating pain to the lower thoracic region for two years. A radiograph and CT scan revealed both permeative osteolytic and multiple trabeculated lesions involving the left posterior part of the 10th rib as well as the 9th and 10th vertebral bodies in continuity and was misled as a malignant or infectious lesion. The histopathology and immuno-histochemistry of the lesion confirmed the diagnosis of an epithelioid hemangioma. The lesion was still stable as of three years after surgery.
Bone Neoplasms/pathology/*radiography/surgery ; Diagnosis, Differential ; Hemangioendothelioma, Epithelioid/pathology/*radiography/surgery ; Humans ; Male ; Middle Aged ; Ribs/pathology/*radiography/surgery ; Spinal Neoplasms/pathology/*radiography/surgery ; Thoracic Vertebrae/pathology/*radiography/surgery ; *Tomography, X-Ray Computed