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Ossification of the posterior longitudinal ligament of the spine. A case-control risk factor study.

STUDY DESIGN: A case-control study. OBJECTIVES: To determine the risk factors for ossification of the posterior longitudinal ligament (OPLL) of the spine. SUMMARY OF BACKGROUND DATA: Previous epidemiologic studies conducted in Japan showed consuming vegetable protein and salt was a risk factor for OPLL. Dietary habits of the Taiwanese and Japanese people are similar. Whether the similar dietary habits play an important role in the high prevalence of OPLL in Taiwan was of interest. METHODS: A case-control study was conducted in a tertiary teaching hospital. The study included 98 consecutive cervical spine patients with OPLL, with 98 age-matched patients with cervical spondylosis serving as control subjects. Radiologic examinations, clinical interviews, physical examinations, and risk factor questionnaires were administered to all the participants. RESULTS: Compared with incidence in the control patients, the frequency of the ossification of the anterior longitudinal ligaments was significantly higher in OPLL patients with OPLL (31% vs. 19%; P = 0.049), but there was no difference in incidence of ossification of the ligamentum flavum (13.3% vs. 16.3%; P = 0.546). More OPLL patients preferred a high-salt diet (OPLL/CS, no:somewhat:yes = 23/38:18/25:57/35;, X2 for trend = 6.3; P = 0.001) and pickled foods (OPLL/CS, no:somewhat:yes = 39/56:11/11:48/31; X2 for trend = 6.7; P = 0.0099). Fewer patients with OPLL consumed meat daily (63% vs. 79%; P = 0.018). High-salt diet (odds ratio = 2.62) and daily meat intake (odds ratio = 0.39) showed persistent association with OPLL in a multivariate logistic regression. CONCLUSIONS: The similar dietary habits, particularly a high-salt and low meat intakes, may partially explain the high prevalence of OPLL in Taiwan and Japan. Modifying dietary habits may help prevent this disease, especially in those high-risk populations.

Aged↗

Heterotopic mesenteric ossification: a distinctive pseudosarcoma commonly associated with intestinal obstruction.

Heterotopic mesenteric ossification (HMO) is a rare intraabdominal bone-producing pseudosarcoma with fewer than 14 reported cases in the literature. We report our experience with 6 additional cases, all of which were referred to us with a diagnostic consideration of extraskeletal osteosarcoma (EO) or "sarcoma" and emphasize features which distinguish HMO from EO. Six intraabdominal lesions coded as "heterotopic mesenteric ossification," "ossifying pseudotumor," or "reactive myofibroblastic proliferation with ossification" were retrieved from our consultation files. Clinical follow-up information was obtained. Lesions occurred exclusively in males, with a mean patient age of 49 years (range, 22-72 years). The tumors occurred in the mesentery (N = 4), omentum (N = 1), or both (N = 1) and were preceded by significant abdominal surgery (4 cases) or trauma (1 case) in all but 1 case. Five patients presented with bowel obstruction and 1 with abdominal sepsis. Tumors were difficult to precisely measure; the mean size of the resection specimens was 11.8 cm (range, 3.5-20 cm). Grossly, the tumors resembled fat necrosis and often cut with a gritty sensation. Microscopically, all lesions demonstrated an exuberant, reactive (myo)fibroblastic proliferation resembling nodular fasciitis, with extensive hemorrhage and fat necrosis. All tumors produced abundant bone and osteoid, often "lace-like," and 2 contained cartilage. The proliferating (myo)fibroblasts, osteoblasts, and chondroblasts were mitotically active but cytologically bland. Follow-up (4 cases; mean, 47.3 months; range, 5-120 months) showed 3 patients alive without disease and 1 dead of unrelated causes. One case was recent. HMO is a distinct intraabdominal ossifying pseudotumor that typically occurs in males, almost always after surgery or abdominal trauma, and frequently presents with symptoms of intestinal obstruction. This clinical history, presence of clearly reactive zones resembling nodular fasciitis, thick osteoid, and absence of nuclear atypia, necrosis, and atypical mitotic figures allow the distinction of HMO from its most important morphologic mimic, EO.

Adult↗

Nonepiphyseal ossification and pseudoepiphysis formation.

Direct ossification extending from the metaphysis into the epiphysis preceded and continued to be more mature than formation and expansion of the typical epiphyseal ossification center at the opposite end of each longitudinal bone of the hand and foot. Direct metaphyseal to epiphyseal ossification usually started centrally and expanded hemispherically, replacing both physeal and epiphyseal cartilage simultaneously. When remnants of the "physis" were retained, however, while juxtaposed epiphyseal cartilage was replaced, a pseudoepiphysis formed. Three basic patterns of pseudoepiphysis formation were evident: (a) a central osseous bridge, (b) a peripheral osseous bridge, and (c) multiple bridging. In each condition, the remnant of the "physis" lacked typical cell columns capable of contributing to the postnatal longitudinal growth of the involved bone. Pseudoepiphyses were well formed by 4-5 years and coalesced with the rest of the bone months to years before skeletal maturation was attained at the opposite epiphyseal end.

Adolescent↗

Cutaneous mixed tumor containing ossification, hair matrix, and sebaceous ductal differentiation.

A 58-year-old Japanese male presented with a cutaneous mixed tumor containing ossification and hair matrix differentiation on the left side of the chin. Histologically, the tumor consisted almost exclusively of apocrine-type epithelial ductal structures and chondroid stroma. Strands and aggregation of basaloid cells which contained keratinous cystic structures with a column of shadow cells arising from basophilic basaloid cells, sebaceous duct-like structures, and ossification in the stroma were also evident. These findings suggest that cutaneous mixed tumors with ossification and hair matrix differentiation are related to both the whole hair follicle and the sweat apparatus.

Adenoma, Pleomorphic↗

Cortical dysplasia with ossification.

We present the first case of cortical dysplasia with extensive intracerebral ossification. An eight-year-old epilepsy patient with a calcified lesion was successfully treated by surgical intervention. Pathological examination revealed a number of bizarre dysplastic cells in the whole lesion, which consisted of an epileptogenic cerebral cortex and a nonepileptogenic hamartomatous lesion with adipose tissue, vascular tissue, calcification, and ossification. The patient was diagnosed as having cortical dysplasia with ossification. Our findings support the notion that cortical dysplasia has a multipotentiality of cellular differentiation, including various hamartomatous tissues. We suggest that cortical dysplasia should be considered as a potential cause for epileptogenicity of a hamartomatous lesion even when magnetic resonance imaging (MRI) fails to disclose cortical dysplasia.

Brain Diseases↗

Cataracta ossea and other intraocular ossifications. A case report and a thirty-year Danish material.

Cataracta ossea is described in a 5-year-old Danish boy, representing a sequal to a unilateral measles-endophthalmitis 3 years earlier. Ossification was not demonstrated elsewhere in the eye. Further, cases of intraocular ossification over a 30-year period were reviewed, based on the files of the Eye Pathology Institute. The total of 155 made up about 3% of enucleated eyes from Danish material received for examination. Regarding age at the initial eye lesion, more than half appeared within the first decade of life. The most frequent underlying lesions were trauma (32%) and uveitis (25%). Among the less frequent causes, emphasis is given to three cases of malignant uveal melanoma. Ossification of the lens was not encountered in the series - except for the case which motivated the review.

Cataract↗

Benign mixed tumour of the skin with extensive ossification and marrow formation: a case report.

Benign mixed tumour of the skin (chondroid syringoma) is an uncommon skin adnexal tumour, usually presenting as a slow growing solitary painless nodule. The morphological appearances are similar to those of a pleomorphic adenoma of the salivary gland. Hair matrix and sebaceous differentiation can be seen in some lesions. Focal ossification is a rare finding. This report presents a case of a similar tumour arising in the cheek of a 43 year old white man, showing extensive ossification. Clinical, radiological, and pathological correlation and diagnosis proved to be difficult preoperatively. Only two cases of a benign mixed tumour with pronounced ossification have been reported so far, both in Japanese patients. This is the first reported case seen in a white man. Awareness of these lesions will avoid potential diagnostic pitfalls.

Adenoma, Pleomorphic↗

Endometrial ossification in a postmenopausal woman.

A case of endometrial ossification in a 62 year old woman is reported. The patient presented with increased vaginal discharge. On transvaginal ultrasonography, a hyperechoic area within the uterine cavity, suggestive of an intrauterine foreign body, was noted. Histological examination of the endometrial curettage showed mature bone with neutrophilic infiltration. There was no evidence of malignancy. Endometrial ossification in postmenopausal women is very rare; most women presenting with this condition are between 20 and 40 years of age. Therefore, clinicians should consider the possibility of endometrial ossification as a differential diagnosis of intrauterine foreign body on ultrasound, even in older patients. In addition, pathologists should be aware of this rare entity to avoid a misdiagnosis of malignant mixed müllerian tumor in the endometrial curettage specimen, which may result in unnecessary hysterectomy.

Endometrium↗

Familial ectopic ossification.

We report a family with dominantly inherited ectopic ossification. It is characterised by childhood onset of multifocal subcutaneous ossifications (primary osteoma cutis), which are of trivial clinical significance. One family member had extensive ectopic ossification involving one limb. We speculate that this may reflect somatic mutation having caused conversion to homozygosity.

Bone and Bones↗

Idiopathic heterotopic ossification in the intensive care setting.

Heterotopic ossification is characterised by the periarticular deposition of ectopic bone. It typically occurs after trauma, neurogenic injury, or congenital causes. Idiopathic heterotopic ossification has been rarely reported. A patient who developed idiopathic heterotopic ossification in the intensive care unit without any known predisposing conditions is presented.

Adult↗

Pelvic heterotopic ossification: MR imaging characteristics.

PURPOSE: To evaluate the magnetic resonance (MR) signal intensity characteristics of pelvic heterotopic ossification (HO) in various stages of maturation. MATERIALS AND METHODS: Thirty-six patients with HO proved at computed tomography (CT) (n = 17) or radiography (n = 19) who underwent 1.5-T pelvic MR imaging within 3 months were included. HO was defined at CT or radiography as grade 1, fluid attenuation without calcification at CT; grade 2, calcification; grade 3, immature ossification; or grade 4, mature ossification. The location and MR signal intensity of all HO sites were noted. RESULTS: HO was determined to be grade 1 at 20 of 141 sites, grade 2 at 39, grade 3 at 30, and grade 4 at 52. With increasing HO grade, the following findings were observed: (a) decreasing T2 signal intensity (grade 1, 70%; grade 2, 58%; grade 3, 44%; grade 4, 4%), (b) increasing fat and cortical bone signal intensity at T1-weighted imaging (grade 1, 0%; grade 2, 3%; grade 3, 13%; grade 4, 86%), and (c) decreasing contrast enhancement (from 100% for grade 1 to 20% for grade 4). Fifteen (88%) patients with CT correlation had HO in the anatomic area of the trochanteric or iliopsoas bursa (55 [60%] of 91 sites). CONCLUSION: With progressive maturity of HO, T2 signal intensity and contrast enhancement decrease, but fat and cortical bone-equivalent signal intensity increases.

Adult↗

Ossification in the cephalic attachment of the ligamentum flavum. An anatomical and CT study.

A review of 50 anatomical specimens and 100 routine CT scans of the chest and abdomen demonstrated that ossification in the cephalic attachment of the ligamentum flavum is a common anatomical finding, but not as common on CT. Examination of hospital charts and plain radiographs of 26 patients with such ossification suggests that it is probably a normal variant; there was no evidence that it is related to diffuse idiopathic skeletal hyperostosis (DISH) or inflammatory arthropathy. Marked ossification may cause or contribute to neurological symptoms.

Adolescent↗

Congenital ossification of the stapedius tendon: diagnosis with CT.

PURPOSE: To describe the features of congenital ossification of the stapedius tendon at thin-section computed tomography (CT). MATERIALS AND METHODS: Thin-section CT scans, clinical records, and surgical findings were retrospectively evaluated in nine ears of five patients with surgically proved congenital ossification of the stapedius tendon. Thin-section CT scans of 50 control ears were also reviewed. RESULTS: On thin-section CT scans, a linear area of soft-tissue attenuation was demonstrated between the monopod stapes and the pyramidal eminence (nine ears), as well as thickening of the stapes footplate (two ears). In the 50 control ears, the normal stapes tendon was not clearly seen on thin-section CT scans. CONCLUSION: At CT, a linear area of soft-tissue attenuation that extends from the pyramidal eminence to the monopod stapes is suggestive of congenital ossification of the stapedius tendon or a bony bar adjacent to it.

Adolescent↗

Cutaneous ossification in Albright's hereditary osteodystrophy.

A 23-year-old woman presented with subcutaneous ossification, which together with short stature, stocky physique, round face and brachydactyly suggested Albright's hereditary osteodystrophy (AHO). Serum calcium and phosphorus levels were normal. AHO refers to the phenotype of the syndromes of pseudo-hypoparathyroidism (PHP) type Ia and pseudopseudohypoparathyroidism (PPHP), both considered genetically related variants with a defect of the alpha subunit of the stimulatory G protein of adenylate cyclase, necessary for the action of parathyroid and other hormones using cyclic AMP as an intracellular second messenger. PPHP differs from PHP in that it lacks parathyroid hormone resistance manifesting itself as hypocalcemia. Other endocrine end organ unresponsiveness, e.g. hypothyroidism and hypogonadism, may also be found with PHP. Both PHP and PPHP usually exhibit characteristic phenotypic abnormalities, of which subcutaneous ossification may be a presenting feature. The differential diagnosis of cutaneous calcification and ossification is outlined.

Adult↗

Penile ossification and acquired penile deviation.

We report on 3 patients with penile deviation during erection caused by ossification in the corpora cavernosa. In each case hard plaques could be palpated. These indurations were removed through a dorsal longitudinal incision. Histologically, solid bone was demonstrable. Two patients were able to resume normal sexual intercourse, but one became impotent following postoperative cavernitis. Penile ossification is rare in man, and its etiology is unknown. It bears no relationship to the os penis normally present in many other mammals. Diagnosis is best made by palpation and X-ray examination. The treatment of choice for symptomatic ossification is surgical excision.

Adult↗

Periarticular heterotopic ossification after multiple knee ligament reconstructions. A report of three cases.

Heterotopic ossification is a frequently encountered clinical and radiographic entity. There are no previous reports in the English literature of heterotopic ossification after arthroscopically assisted ligament reconstructions for knee dislocations. Further, a link between the PCL reconstruction and posterior capsular ossification has not been heretofore recognized. Our three cases should raise the clinical awareness of such an entity.

Accidents, Traffic↗

Ossification of the Achilles tendon: a case report.

Ossification of the Achilles tendon is a rare condition. We recently treated a patient with ossification of bilateral Achilles tendons. The patient was a 50 year old male whose chief complaint was discomfort around the Achilles tendon. He had a previous history of treatment of bilateral club feet. On the roentgenograms, the length of the bony mass in the Achilles tendon was 5.5 cm on the right side and 1.5 cm on the left side. The left side was treated by surgical removal of a bony mass and suture of the tendon. Microscopic examination of the extirpated specimen revealed bone formation through enchondral and intramembranous ossification in the Achilles tendon.

Achilles Tendon↗

Ossification of the posterior longitudinal ligament: an update on its biology, epidemiology, and natural history.

Significant progress has been achieved in basic research during the past decade on the pathogenesis of ossification of the posterior longitudinal ligament (OPLL), a multifactorial disease in which complex genetic and environmental factors interact. A review of the literature was conducted to update recent findings on the biology, epidemiology, natural history, and related diseases of OPLL. Gene analysis studies found specific polymorphisms that may be associated with OPLL in several collagen genes, which encode for extracellular matrix proteins. Polymorphisms in the nucleotide pyrophosphate gene, which is involved in regulation of calcification in chondrocytes, may also be associated with OPLL. However, the results of the gene analysis studies have not always been consistent. Involvement of many growth factors and cytokines, including bone morphogenic proteins and transforming growth factor-beta, has been demonstrated in various histochemical and cytochemical analyses. Several transcription factors involved in cellular differentiation may also have a role. Recent epidemiological studies reaffirmed an earlier finding that diabetes mellitus is a distinct risk factor for OPLL. The long-term follow-up studies of OPLL patients are disclosing the natural history, as well as the frequency and rate of progression, of OPLL after surgical intervention. Further knowledge on the factors responsible for progression of OPLL may predict its behavior in each patient, and treatment may be tailored accordingly. The coexistence of OPLL with other diseases of ectopic ossification of the spine, such as ossification of the ligamentum flavum and diffuse idiopathic skeletal hyperostosis, is not uncommon. Scientific breakthrough in those diseases may, in turn, give insights into the pathogenesis of OPLL.

Animals↗