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PubMed1998

Pediatric peripheral neuropathy in proteus syndrome.

Proteus syndrome is a rare congenital disorder comprised of subcutaneous and internal hamartomas, pigmented skin nevi, skull exostoses, hemihypertrophy, and macrodactyly of the hands and feet. A 5-year-old girl diagnosed with Proteus syndrome presented with distal median compression neuropathy with the primary complaint of severe pain involving the left hand. Surgical exploration of the hand revealed a lipofibromatous hamartoma of the median nerve. The transverse carpal ligament was released and epineurectomy of the median nerve was performed. The patient remains symptom free at the 9-month follow-up. This report is the first description of a hamartoma directly involving a peripheral nerve in Proteus syndrome. Decompression of the nerve with the removal of the fibrofatty neural sheath resulted in the resolution of the symptoms in this patient. The surgeon should consider this approach as a potential first line of treatment before a more radical resection of the nerve is contemplated.

Child, Preschool↗

PubMed1998

Osteosarcoma and other bone cancers.

In this review, recent advances in the clinical therapy of osteosarcoma, including results from the European Osteosarcoma Intergroup trial demonstrating the efficacy of a short intensive two drug protocol are discussed as well the evolving role of ifosfamide. Biologically, the area of interest on chromosome 3q, which may contain an osteosarcoma tumor suppressor gene, is being narrowed, and several promising new therapeutic approaches including tumor vaccine have been explored. In chondrosarcoma research, abnormalities in hereditary multiple exostoses genes, which encode protein products essential for normal cartilage development, and a potential mechanism for the characteristic chemotherapy resistance of cartilaginous tumors (overexpression of P-glycoprotein) have been described. Surgical advances include testing of total en bloc spondylectomy for vertebral tumors as well as a noninvasively extendable long bone endoprosthesis. Finally, new insights in diagnostic imaging, including the evolving role of 201Tl, 99mTc-MIBI (methoxyisobutylisonitrile), and newer variations on magnetic resonance imaging are reviewed.

Animals↗

PubMed2002

Utility of planar bone scintigraphy to distinguish benign osteochondromas from malignant chondrosarcomas.

PURPOSE: The current study was designed to evaluate the role of planar bone scintigraphy in the diagnosis of sarcomatous change in osteochondromas (cartilaginous exostoses). MATERIALS AND METHODS: Histologically verified cases of chondrosarcoma and osteochondroma, in which Tc-99m bone scintigraphy was performed, were reviewed in a retrospective study. RESULTS: Twenty-two cases were evaluated. Of 11 cases with chondrosarcomas, 8 (73%) had clearly increased tracer uptake, whereas the remaining cases had normal or decreased tracer uptake. Of the 11 cases with osteochondromas, 8 (73%) had clearly increased tracer uptake, whereas the remaining cases had normal or decreased tracer uptake. No significant difference in the distribution of scintigraphic results was noted between the two pathoanatomic groups (p = 1). CONCLUSION: In conclusion, single standing planar bone scintigraphy has no value in distinguishing benign osteochondromas from malignant chondrosarcomas.

Adult↗

PubMed1999

Fibro-osseous lesions of the external auditory canal.

OBJECTIVES: To differentiate a novel type of benign circumscribed bone lesion of the external auditory canal from those described previously, such as exostoses and osteomas. STUDY DESIGN: Information was obtained from computed tomography (CT) images, surgical findings, and pathologic study. METHODS: Five patients (26 to 82 years old) who presented a hard, round, unilateral, skin-covered mass occluding the external auditory canal to varying degrees were studied. A CT study carried out before resection of the lesions by curettage disclosed the absence of a bony connection to the underlying structures. All the tissue specimens underwent pathologic study. RESULTS: CT and surgical findings demonstrated the absence of a connective pedicle. The pathologic findings showed lesions consisting of an osteoma-like bone formation with sparse osteoblastic areas; mature lamellar bone was observed in three cases, bone marrow containing adipose tissue and hematopoietic remnants in two, and a dense, collagenous stroma in another. They all showed irregular trabeculae, bordered by osteoid osteoblasts. In no case was there evidence of a relationship to the cartilaginous tissue or to the bony structures of the external auditory canal. CONCLUSIONS: The data obtained from the clinical, CT, surgical, and pathologic findings suggest the existence of a lesion unlike those previously known, possibly related to ossifying reactions in other parts of the organism.

Adult↗

PubMed1999

Relationship of the facial nerve to the tympanic annulus: a direct anatomic examination.

OBJECTIVE: To examine the relation of the facial nerve to the only identifiable surgical landmark in the external auditory canal. INSTITUTION: Community-based teaching hospital. STUDY DESIGN: Examination of formalin-fixed human temporal bones. BACKGROUND: The transcanal approach is often used in tympanoplasty, canaloplasty, hypotympanotomy, and removal of tumors of the external auditory canal (EAC), such as exostoses and osteomas. Surgery of the EAC places the facial nerve at risk for injury as the nerve courses vertically in the posterior canal wall. Few articles have described the relation of the facial nerve's course to the tympanic annulus, the only identifiable landmark in the EAC. This study is the first to document the relationship of the course of the facial nerve with respect to the tympanic annulus by direct anatomic measurement of the temporal bone. METHODS: Thirty-seven formalin-fixed cadaver temporal bones were studied after skeletonization of the facial nerve and tympanic annulus. RESULTS: The facial nerve coursed lateral to the plane of the annulus in 70% of specimens, always in the posteroinferior quadrant. The nerve also coursed anterior to a plane through the most posterior point of the annulus in 73.1% of specimens, also exclusively in the posteroinferior quadrant. The course of the nerve was quite variable with respect to the annulus. CONCLUSIONS: The facial nerve is most vulnerable to injury in the posteroinferior quadrant in transcanal surgery. The annulus is not a reliable landmark for the facial nerve. Anecdotal evidence is cited and recommendations are offered.

Culture Techniques↗

PubMed1998

Costal osteochondroma. A rare cause of spinal cord compression.

STUDY DESIGN: Report of a rare cause of spinal cord compression: costal osteochondroma. OBJECTIVE: To describe a very rare cause of spinal cord compression, costal osteochondroma, which was present in a 16-year-old girl with a history of hereditary multiple exostoses. SUMMARY OF BACKGROUND DATA: Only four cases of expansion of costal osteochondroma into the spinal canal through an intervertebral foramen have been reported previously. METHODS AND RESULTS: The origin of the osteochondroma at the head of the right 12th rib, the invasion of the spinal canal through the right T12-L1 intervertebral foramen, and the compression of the spinal cord were shown on computed tomography and magnetic resonance imaging. The exact extent of the osteochondroma, particularly the cartilage cap, was delineated accurately by magnetic resonance imaging. Complete excision followed by full recovery occurred 19 months after surgery. CONCLUSION: Magnetic resonance imaging is the preferred method of investigation in cases of osteochondroma related to spine, because it allows for better pre-operative planning and helps to prevent incomplete excision of the tumor.

Adolescent↗

PubMed2000

Osteochondroma of the C5 lamina with cord compression: case report and review of the literature.

STUDY DESIGN: Case report of a solitary osteochondroma of the cervical spine causing myelopathy in a 66-year-old woman. OBJECTIVES: To review the relevant literature and describe a highly unusual clinical manifestation of solitary osteochondroma. SUMMARY OF BACKGROUND DATA: Osteochondromas are common benign bony lesions that seldom occur in the axial skeleton. These lesions are more commonly reported with neural compression in cases of hereditary multiple exostoses (Bessel-Hagel syndrome, diaphyseal aclasis). METHODS: Chart review, review of relevant radiographic examinations and histopathologic specimens, clinical follow-up with examination, and literature review. RESULTS: Manifestation with new neurologic deficit in a 66-year-old patient was singular. CONCLUSIONS: Osteochondromas are unusual in the axial skeleton, and are rarely signaled by neural compression. Occurrence is generally in young adults in the second and third decades. Initial manifestation with a new neurologic deficit in a 66-year-old patient was highly unusual.

Aged↗

PubMed2003

Secondary chondrosarcoma in osteochondroma: report of 107 patients.

Secondary chondrosarcomas are rare; recognition and diagnosis are difficult. Slow growth and late recurrence require long-term followup to understand the clinical course. In the current study, 107 patients had secondary chondrosarcoma arising in a solitary osteochondroma (61 patients) or multiple exostoses (46 patients). All histologic slides were reviewed without knowledge of the outcome, and radiologic studies were available for review in 71 cases. Patients with secondary chondrosarcoma were one to two decades younger than those with primary chondrosarcoma. Male preponderance and a predilection for flat bones were observed. The radiologic signs of sarcomatous degeneration included irregularity of the margin, inhomogeneous mineralization, and an associated soft tissue mass. The tumors generally were well-differentiated. Only 10 tumors were classified as Grade 2. Five-year and 10-year local recurrence rates were 15.9% and 17.5%, respectively, and 5- and 10-year mortality rates were 1.6% and 4.8% for patients having initial treatment at the authors' institution. Metastasis developed in five patients: in the lung in four patients and in the groin region in one patient. Most patients who died of tumor died of local recurrence. Wide excision had the lowest local recurrence rate. With successful surgical treatment, patients may have long-term disease-free survival.

Adolescent↗

PubMed2006

Medical illnesses and injuries encountered during surfing.

Surfing is an exciting sport enjoyed in many coastal communities around the globe. Participants are prone to various conditions ranging from acute injuries to conditions borne from chronic environmental exposure. Lacerations, contusions, sprains, and fractures are the common types of acute traumatic injury. Injury from the rider's own surfboard is the prevailing mechanism of injury. Interaction with marine animals may lead to injury through envenomation. Although jellyfish stings are common, no definitive treatment strategy has been proven most effective in dealing with such stings. Exposure to jellyfish and other nematocyst-containing larvae can cause a reaction known as seabather's eruption. Stingrays and coral reefs present further hazards to the surfboard rider. Infection of wounds is often seen and should be treated with fluoroquinolones or third-generation cephalosporins to cover Vibrio species, along with Staphylococcus and Streptococcus species. Otologic sequelae of surfing include auditory exostoses, ruptured tympanic membrane, and otitis externa.

Athletic Injuries↗

PubMed2004

A rare brachial artery pseudoaneurysm 13 years after excision of a humeral osteochondroma.

Pseudoaneurysms resulting from vascular impingement by osteochondromas are extremely rare. The authors detail the case of a 19-year-old man who represents the third known report in the English literature of a brachial artery pseudoaneurysm associated with a humeral osteochondroma. In patients presenting with a painful upper arm mass and a history of multiple hereditary exostoses, one must have a high index of suspicion for pseudoaneurysm.

Adult↗

PubMed2004

Vascular anomalies in Proteus syndrome.

Proteus syndrome (PS) is a complex hamartomatous disorder defined by local overgrowth (macrodactyly or hemihypertrophy), subcutaneous tumours and various bone, cutaneous and/or vascular anomalies (VA). VA are manifold in PS, but their prevalence is unknown so far. In order to further characterize PS, we studied the prevalence of VA in 22 PS patients presenting to our outpatient clinic and reviewed 100 PS patients previously reported between 1983 and 2001. The diagnosis of vascular abnormalities was made on clinical grounds and supported with imaging studies and/or histology in 12 and seven patients out of 22, respectively. Thirty-five VA were identified in 22/22 (100%) of our patients, and more than one type of VA were present in 10 of them. Vascular tumours, portwine stains (PWS), and venous anomalies (varicosities, prominent veins) were equally common. A total of 118 VA were previously reported in 70/100 (70%) PS patients; vascular hamartomas were more prevalent (56/118 = 47.5%), whilst PWS (21.2%) and venous anomalies (22.9%) were slightly less common than in our series, but there is the possibility of under-reporting. Unlike Klippel-Trenaunay syndrome, where VA are mostly confined to the hypertrophic limb, major arteriovenous anomalies are rare, and - similar to the other hamartomas and naevi observed in PS (pigmentary naevi, epidermal naevi, subcutaneous tumours, exostoses) - VA appear to be distributed at random sites on the body. We conclude that VA are among the most common findings in PS. Their varying type and distribution lend further support to the concept of somatic mosaicism.

Abnormalities, Multiple↗

PubMed1995

Unusual distribution of enamel hypoplasia in an 11-year-old child with Proteus syndrome.

An 11-year-old girl with Proteus syndrome attended our dental department because her teeth were sensitive to cold stimuli and because of the poor appearance of her anterior teeth. Extraoral examination revealed several of the previously reported craniofacial features of Proteus syndrome: facial and skull asymmetry, exostoses of the nasal bridge, and mandibular prognathism. Intraoral examination revealed enamel hypoplasia of primary and permanent teeth. The distribution of hypoplasia was unusual in that only teeth on the right side of the mouth were affected, and also in that only the distal half of the crown of the maxillary right permanent central incisor was affected. Behavioural problems necessitated treatment under general anaesthesia; the affected primary teeth were extracted and the hypoplastic permanent teeth were restored, relieving the patient's symptoms and greatly improving aesthetics.

Child↗

PubMedDate not supplied

Palliative irradiation of Scottish Fold osteochondrodysplasia.

This report describes palliative irradiation as treatment for Scottish Fold osteochondrodysplasia. A 3-year-old female spayed Scottish Fold cat suffering from osteochondrodysplasia was referred to the Veterinary Teaching Hospital, University of Zurich. Based on the breed, history, clinical signs, radiographic findings, and the histologic diagnosis of a biopsy specimen, Scottish Fold osteochondrodysplasia was confirmed. To control the exostoses leading to inflammatory processes and pain, radiotherapy was initiated as palliative treatment. This was successful in relieving the clinical signs within a few weeks. The short- and long-term results after radiotherapy are presented and discussed with a comparable human disease, the plantar heel spur, which also responds well to radiotherapy.

Animals↗

PubMed1978

Changes in the sustentaculum tali associated with distension of the tarsal sheath (thoroughpin).

Results of the clinical and radiographic examination of 8 lame horses with tarsal sheath distension are described. In chronic cases pathological exostoses were identified radiographically in the sustentaculum tali and were demonstrated at post mortem in 4 of the horses which were destroyed. The prognosis and the feasibility of treatment are discussed in the light of these changes and the associated damage found at post mortem in the deep flexor tendon and its sheath. Trauma to the hock was known to have occurred in half the cases and was suspected in the others.

Animals↗

PubMed1976

The effect of aminoacetonitrile on calcium metabolism and bone in the rat.

1. The effect of the osteolathyrogen aminoacetonitrile (AAN) on plasma calcium, phosphate and alkaline phosphatase, on bone, on growth rates, on absorption of calcium and phosphate in the gut and on their urinary excretion, has been examined in rats. 2. AAN caused a general deterioration in the health of the rat and reduced its rate of growth. 3. AAN reduced plasma calcium and increased plasma alkaline phosphatase but did not affect plasma phosphate. 4. AAN caused obvious deformity of the long bones with large exostoses. The femurs of the lathyritic rats had an increased fat-free weight and increased fat-free weight to ash weight ratio. 5. AAN increased true and apparent absorption of calcium but did not consistently affect urinary excretion of phosphate or apparent absorption of phosphate. 6. AAN did not produce a significant reduction in the plasma calcium of parathyroidectomized or thyroparathyroidectomized rats. 7. Absorption of calcium from ligated jejunal loops was increased in AAN-treated parathyroidectomized rats. 8. In some experiments parathyroidectomy or thyroparathyroidectomy protected the bones from the effect of AAN.

Acetonitriles↗

PubMed1987

Chondrosarcoma of the calcaneum and massive soft tissue calcification in a patient with hereditary and acquired connective tissue diseases.

We describe the first case of the coexistence of the hereditary connective tissue disorder multiple exostoses (HME) and an acquired connective tissue disorder manifest by the overlap of dermatomyositis (DM), scleroderma (PSS), high titre speckled pattern antinuclear antibodies, and increased antibodies to double stranded deoxyribonucleic acid (DNA). Furthermore this patient developed chondrosarcoma of the calcaneum (an unusual site for this malignancy) and massive soft tissue calcification (an unusual feature of PSS, adult DM, and systemic lupus erythematosus (SLE)).

Bone Neoplasms↗

PubMed1986

Surgical treatment of patellar tendon pain in athletes.

A series of surgically treated patellar tendon lesions among athletes is presented. The material was collected during 5 years from three sports injury clinics and from two hospitals. During this period the authors treated about 150 cases of jumper's knee, of which 34 cases were treated by operation. The athletes were mostly volley ball players, jumpers or runners. The operation revealed a necrotic focus of the patellar tendon in 21 cases, the retinaculum was thick and adherent in 16 patients and an exostosis of the patellar insertion was seen in two cases. The necrotic areas were excised, the thick and adherent retinaculum was divided and the exostoses were excised and drilled. Surgical treatment of chronic patellar tendon pains may give good results in selected cases.

Adolescent↗

PubMed1978

Cervical cord compression by solitary osteochondroma of the atlas.

A 28 year old woman presented a syndrome of slow spinal compression evolving over four and a half years at the level C1--C2 vertebrae due to a solitary osteochondroma of the posterior arch of the atlas which was excised by laminectomy. Radiographys of the skeleton did not show the presence of other osteogenic tumours. This is the fifth case of solitary osteochondroma to be published. Elsewhere, in six cases out of 14, the vertebral lesion formed part of generalised exostoses.

Adult↗