RESUMO
Diffuse idiopathic skeletal hyperostosis (DISH) is a noninflammatory progressive disease resulting in ossification of the anterior longitudinal ligament of the spine and tendons. Herein, we describe a case of DISH in a patient on long-term hemodialysis. The patient was a 79-year-old man undergoing hemodialysis for chronic kidney disease due to diabetic nephropathy. He presented to the emergency department complaining of back pain after a slip and fall. Radiographs revealed bamboo spine-like findings, extending from the cervical to the lumbar spine. Computed tomography and magnetic resonance imaging revealed a compression fracture of thoracic vertebra 12, with abnormal ossification of the anterior longitudinal ligament. Inter-vertebral vertical osseous bridges were also observed at the cervical 7 and lumbar 2 vertebrae. There was no obvious spinal cord compression. Leukocytosis and C-reactive protein levels were not elevated and the human leukocyte type antigen HLA-B27 test was negative. Based on this finding, a diagnosis of DISH was made. In the absence of neurological findings, the patient was treated conservatively. Our findings show an overlap between the clinical features of DISH and those of hemodialysis patients, including older age, male sex, and diabetes.
Assuntos
Hiperostose Esquelética Difusa Idiopática , Compressão da Medula Espinal , Humanos , Masculino , Idoso , Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Hiperostose Esquelética Difusa Idiopática/patologia , Tomografia Computadorizada por Raios X , Radiografia , Osso e Ossos/patologiaRESUMO
Diffuse idiopathic skeletal hyperostosis (DISH) is a condition characterised by calcification and ossification of ligaments and entheses. The condition usually affects the axial skeleton, in particular, at the thoracic segment, though also other portions of the spine are often involved. DISH often involves also peripheral tendinous and/or entheseal sites either alone, or in association with the involvement of peripheral joints. At times, new bone formation involves the bone itself, but sometimes it involves joints not usually affected by osteoarthritis (OA) which result in bony enlargement of the epiphysis, joints space narrowing and a reduced range of motion. Because of the entheseal involvement, DISH can be mistaken for seronegative spondyloarthropathies or for a "simple" OA. Furthermore, other implications for the recognition of DISH include spinal fractures, difficult intubation and upper endoscopies, decreased response rates in DISH with concomitant spondyloarthritides, and increased likelihood to be affected by metabolic syndrome and cardiovascular diseases. This Atlas is intended to show the imaging finding in DISH in patients diagnosed with the condition by the Resnick classification criteria.
Assuntos
Diagnóstico por Imagem/métodos , Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Osteoartrite/complicações , Coluna Vertebral/diagnóstico por imagem , Calcinose/complicações , Doenças Cardiovasculares/complicações , Estudos de Casos e Controles , Entesopatia/patologia , Humanos , Hiperostose Esquelética Difusa Idiopática/patologia , Ligamentos/patologia , Síndrome Metabólica/complicações , Osteogênese/fisiologia , Amplitude de Movimento Articular/fisiologia , Coluna Vertebral/patologia , Espondiloartropatias/imunologiaRESUMO
BACKGROUND: Diffuse idiopathic skeletal hyperostosis (DISH) is characterized by the ossification of vertebral bodies and peripheral entheses. However, variations in sacroiliac (SI) joint change in patients with DISH have not been fully clarified. The purpose of this study was to evaluate SI joint variation in patients with DISH in comparison with a non-DISH population. METHODS: A total of 342 SI joints in 171 patients (DISH+, n = 86; DISH-, n = 85) who had undergone lumbar spine surgery were analyzed by computed tomography examination. SI joint variations were classified into four types: Type 1, normal or tiny peripheral bone irregularity; Type 2, subchondral bone sclerosis and osteophytes formation; Type 3, vacuum phenomenon; and Type 4, bridging osteophyte and bony fusion. The type of bridging osteophyte in SI joints and the prevalence of ossification in each spinal segment from C1 to SI joint were also examined. RESULTS: The most common SI joint variation in the DISH+ group was bony fusion (Type 4), with 71.6% exhibiting anterior paraarticular bridging. On the other hand, SI joint vacuum phenomenon (Type 3) was the most frequent change (57.1%) in the DISH- group. The middle to lower thoracic spine and SI joints were highly affected in DISH and caused bony ankylosis. CONCLUSIONS: Anterior paraarticular bridging was the most common type of SI joint change in patients with DISH who underwent lumbar spine surgery. The present results regarding variations of SI joint changes in DISH should help understand the etiology of DISH.
Assuntos
Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Articulação Sacroilíaca/diagnóstico por imagem , Idoso , Idoso de 80 Anos ou mais , Variação Anatômica , Feminino , Humanos , Hiperostose Esquelética Difusa Idiopática/etiologia , Hiperostose Esquelética Difusa Idiopática/patologia , Masculino , Estudos Retrospectivos , Articulação Sacroilíaca/patologia , Tomografia Computadorizada por Raios XRESUMO
BACKGROUND: The epidemiology and cause of ossification of the spinal ligaments (OSL) remains obscure. To date, there is no study that comprehensively evaluates the prevalence, distribution, and concomitance of each type of OSL by CT among general Chinese population. We therefore aimed to comprehensively investigate epidemiological characteristics of OSL using whole spine CT in the Chinese population and examine the factors that correlate with the presence of OSL. METHODS: Ossification of the posterior longitudinal ligament (OPLL), ligamentum flavum (OLF), anterior longitudinal ligament (OALL), nuchal ligament (ONL), and diffuse idiopathic skeletal hyperostosis (DISH) were evaluated from the subjects who underwent PET/CT for the purpose of cancer screening in our hospital. Prevalence, distribution, and concomitance of OSL were reviewed. Logistic regression analysis was performed to identify the risk factors of OSL. RESULTS: A total of 2000 subjects (1335 men and 665 women) were included. The prevalence rate of cervical OPLL (C-OPLL) was 4.1%, thoracic OPLL (T-OPLL) 2.25%, lumbar OPLL (L-OPLL) 0.8%, thoracic OLF (T-OLF) 37.65%, lumbar OLF (L-OLF) 1.45%, ONL 31.5%, DISH 3.85%. The most commonly involved level was C5 for C-OPLL, T1 for T-OPLL, T10 for T-OLF, and T8/9 for OALL. 21% of subjects with C-OPLL had T-OPLL, 44% of C-OPLL had T-OLF, 38% of T-OPLL had C-OPLL, 53% of T-OPLL had T-OLF, 44% of L-OPLL had T-OPLL, and 56% of L-OPLL had T-OLF. The average age of OSL-positive subjects was significantly higher than that of OSL-negative subjects. The results of the multiple regression analysis revealed that males had a strong association with DISH (odds ratio, 3.15; 95% confidence interval, 1.27-7.78; P = 0.013). CONCLUSION: The prevalence of OSL in the Chinese was revealed. Tandem ossification is not uncommon in people with OSL. There is a high incidence of multiple-regional OPLL in the whole spine. Approximately half of the subjects with OPLL coexist with T-OLF. For patients with clinical symptoms induced by OPLL, thorough evaluation of whole spine using CT is recommended.
Assuntos
Hiperostose Esquelética Difusa Idiopática/epidemiologia , Ligamentos Articulares/patologia , Ossificação do Ligamento Longitudinal Posterior/epidemiologia , Coluna Vertebral/patologia , Adulto , China , Estudos Transversais , Feminino , Humanos , Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Hiperostose Esquelética Difusa Idiopática/patologia , Incidência , Ligamentos Articulares/diagnóstico por imagem , Masculino , Pessoa de Meia-Idade , Ossificação do Ligamento Longitudinal Posterior/diagnóstico , Ossificação do Ligamento Longitudinal Posterior/patologia , Tomografia por Emissão de Pósitrons combinada à Tomografia Computadorizada , Prevalência , Estudos Retrospectivos , Fatores de Risco , Coluna Vertebral/diagnóstico por imagemRESUMO
Background Degenerative intervertebral disease (DID) is an exclusion criterion in the Resnick and Niwayama radiographic classification for diffuse idiopathic skeletal hyperostosis (DISH). However, although DID was previously described in DISH, no systematic computed tomography (CT) analysis has been reported so far. Purpose To assess for the presence and prevalence of such changes on CT examinations of the thoracic spine of individuals with DISH. Material and Methods Intervertebral space (D1-L1) on chest CT examinations of DISH patients was retrospectively evaluated for the presence of DID. Parameters evaluated were disc space height, disc protrusion, subchondral cysts/sclerosis, Schmorl nodes, vacuum phenomenon, and posterior elements including costovertebral and facet joints. Parameters were compared with two age- and gender-matched control groups of individuals whose entire spine CT lacked evidence of DISH (Control 1 individuals < 2 flowing osteophytes, Control 2 individuals < 4 and ≥ 2 flowing osteophytes). Results A total of 158 participants (DISH/Control 1/Control 2 = 54/54/50; 106 men, 52 women; average age = 70.6 years) were evaluated. Average intervertebral disc height was significantly lower in the DISH group compared with both control groups (DISH/Control 1/Control 2 = 4.55/5.13/5.01 mm, P < 0.001). Costovertebral degenerative changes were more prevalent in DISH patients ( P < 0.05) and, except for vacuum phenomenon (more prevalent in controls), other DID changes were as prevalent in DISH as in controls. Conclusion The presence of degenerative intervertebral changes on thoracic CT should not deter from diagnosing DISH. Thus, the radiographic Resnick and Niwayama DISH criteria cannot be directly adapted to CT.
Assuntos
Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Hiperostose Esquelética Difusa Idiopática/patologia , Vértebras Torácicas/diagnóstico por imagem , Vértebras Torácicas/patologia , Tomografia Computadorizada por Raios X/métodos , Idoso , Idoso de 80 Anos ou mais , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estudos RetrospectivosRESUMO
Equilibrative nucleoside transporter 1 (ENT1) mediates passage of adenosine across the plasma membrane. We reported previously that mice lacking ENT1 (ENT1(-/-)) exhibit progressive ectopic mineralization of spinal tissues resembling diffuse idiopathic skeletal hyperostosis (DISH) in humans. Here, we investigated mechanisms underlying aberrant mineralization in ENT1(-/-) mice. Micro-CT revealed ectopic mineralization of spinal tissues in both male and female ENT1(-/-) mice, involving the annulus fibrosus of the intervertebral discs (IVDs) of older mice. IVDs were isolated from wild-type and ENT1(-/-) mice at 2months of age (prior to disc mineralization), 4, and 6months of age (disc mineralization present) and processed for real-time PCR, cell isolation, or histology. Relative to the expression of ENTs in other tissues, ENT1 was the primary nucleoside transporter expressed in wild-type IVDs and mediated the functional uptake of [(3)H]2-chloroadenosine by annulus fibrosus cells. No differences in candidate gene expression were detected in IVDs from ENT1(-/-) and wild-type mice at 2 or 4months of age. However, at 6months of age, expression of genes that inhibit biomineralization Mgp, Enpp1, Ank, and Spp1 were reduced in IVDs from ENT1(-/-) mice. To assess whether changes detected in ENT1(-/-) mice were cell autonomous, annulus fibrosus cell cultures were established. Compared to wild-type cells, cells isolated from ENT1(-/-) IVDs at 2 or 6months of age demonstrated greater activity of alkaline phosphatase, a promoter of biomineralization. Cells from 2-month-old ENT1(-/-) mice also showed greater mineralization than wild-type. Interestingly, altered localization of alkaline phosphatase activity was detected in the inner annulus fibrosus of ENT1(-/-) mice in vivo. Alkaline phosphatase activity, together with the marked reduction in mineralization inhibitors, is consistent with the mineralization of IVDs seen in ENT1(-/-) mice at older ages. These findings establish that both cell-autonomous and systemic mechanisms contribute to ectopic mineralization in ENT1(-/-) mice.
Assuntos
Calcificação Fisiológica , Hiperostose Esquelética Difusa Idiopática/patologia , Hiperostose Esquelética Difusa Idiopática/fisiopatologia , Coluna Vertebral/patologia , Coluna Vertebral/fisiopatologia , 2-Cloroadenosina/metabolismo , Fosfatase Alcalina/metabolismo , Animais , Anel Fibroso , Calcificação Fisiológica/genética , Calcinose/genética , Calcinose/patologia , Células Cultivadas , Modelos Animais de Doenças , Transportador Equilibrativo 1 de Nucleosídeo/metabolismo , Feminino , Regulação da Expressão Gênica , Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Disco Intervertebral/diagnóstico por imagem , Disco Intervertebral/patologia , Disco Intervertebral/fisiopatologia , Masculino , Camundongos Endogâmicos C57BL , Caracteres Sexuais , Coluna Vertebral/diagnóstico por imagem , Microtomografia por Raio-XRESUMO
OBJECTIVE: To report femoral neuropathy caused by nerve entrapment associated with diffuse idiopathic skeletal hyperostosis (DISH). STUDY DESIGN: Case report. ANIMAL: Seven-year-old female spayed Boxer dog. RESULTS: Entrapment of the right femoral nerve due to DISH caused a femoral nerve deficit and atrophy of muscle groups associated with the affected nerve. A combination of computed tomography and magnetic resonance imaging was performed to provide a diagnosis. Amputation of the right transverse process of the sixth lumbar vertebra at the level of nerve entrapment relieved the neurological abnormality. CONCLUSIONS: Nerve entrapment leading to neurapraxia may occur concurrently with DISH and surgery in this case was successful in restoring function. CLINICAL RELEVANCE: Peripheral neuropathy from nerve entrapment should be considered in patients with DISH. Surgical amputation of impinging osseous structures may be indicated for relief of femoral neuropathy.
Assuntos
Doenças do Cão/patologia , Neuropatia Femoral/veterinária , Hiperostose Esquelética Difusa Idiopática/veterinária , Síndromes de Compressão Nervosa/veterinária , Animais , Cães , Feminino , Neuropatia Femoral/cirurgia , Hiperostose Esquelética Difusa Idiopática/complicações , Hiperostose Esquelética Difusa Idiopática/patologia , Hiperostose Esquelética Difusa Idiopática/cirurgia , Síndromes de Compressão Nervosa/patologia , Síndromes de Compressão Nervosa/cirurgiaRESUMO
We aimed to assess the prevalence of diffuse idiopathic skeletal hyperostosis (DISH) and its association with lumbar spondylosis (LS) and knee osteoarthritis (KOA) using a population-based cohort study entitled Research on Osteoarthritis/osteoporosis Against Disability (ROAD). In the baseline ROAD study, which was performed between 2005 and 2007, 1,690 participants in mountainous and coastal areas underwent anthropometric measurements and radiographic examinations of the whole spine (cervical, thoracic, and lumbar) and both knees. They also completed an interviewer-administered questionnaire. Presence of DISH was diagnosed according to Resnick criteria, and LS and KOA were defined as Kellgren-Lawrence (KL) grade ≥3. Among the 1,690 participants, whole-spine radiographs of 1,647 individuals (97.5%; 573 men, 1,074 women; mean age, 65.3 years) were evaluated. Prevalence of DISH was 10.8% (men 22.0%, women 4.8%), and was significantly higher in older participants (presence of DISH 72.3 years, absence of DISH 64.4 years) and mainly distributed at the thoracic spine (88.7%). Logistic regression analysis revealed that presence of DISH was significantly associated with older age [+1 year, odds ratio (OR): 1.06, 95% confidence interval (CI): 1.03-1.14], male sex (OR: 5.55, 95% CI: 3.57-8.63), higher body mass index (+1 kg/m(2), OR: 1.08, 95% CI: 1.02-1.14), presence of LS (KL2 vs KL0: 1, OR: 5.50, 95% CI: 2.81-10.8) (KL ≥3 vs KL0: 1, OR: 4.09, 95% CI: 2.08-8.03), and presence of KOA (KL ≥3 vs KL0: 1, OR: 1.89, 95% CI: 1.14-3.10) after adjusting for smoking, alcohol consumption, and residential area (mountainous vs coastal). This cross-sectional population-based study clarified the prevalence of DISH in general inhabitants and its significant association with LS and severe KOA.
Assuntos
Hiperostose Esquelética Difusa Idiopática/epidemiologia , Hiperostose Esquelética Difusa Idiopática/patologia , Vértebras Lombares/patologia , Osteoartrite do Joelho/patologia , Coluna Vertebral/fisiopatologia , Espondilose/patologia , Idoso , Índice de Massa Corporal , Estudos Transversais , Feminino , Humanos , Masculino , Osteoporose/patologia , Prevalência , Estudos ProspectivosRESUMO
The principal objective of this investigation was to analyze the association between diffuse idiopathic skeletal hyperostosis (DISH) and the presence of aortic valve sclerosis (AVS). For this study we used results from 1000 consecutive outpatients (473 males), older than 50 years of age (average 67.6 years), that had been examined with Doppler echocardiogram and anterior and lateral chest radiographs. Overall, 195 patients (19.5%) were diagnosed with DISH and 283 (28.3%) with AVS. DISH was more prevalent than AVS in males (66.7% vs. 42.6%, p< 0.0001) and in older patients (73.6 ± 9 years vs. 66.1 ± 9 years, p < 0.0001). Furthermore, 55.4% of patients with dorsal DISH presented aortic sclerosis calcification vs. 21.7% of patients free of DISH (OR = 4.47; 95% CI = 3.22-6.21). The adjusted odds ratio (OR) was calculated by sex and age resulting in 3.04 (95% CI = 2.12-4.36; p < .0001). A statistically significant association was found between DISH and AVS in accordance to age and sex. The biological plausibility of this association is based on similar risk factors, pathogenic mechanisms and vascular complications.
El objetivo principal fue analizar la asociación entre la hiperostosis esquelética idiopática difusa (DISH) y la presencia de esclerosis valvular aórtica (AVS). Se evaluaron los resultados de 1000 pacientes ambulatorios consecutivos (473 varones), mayores de 50 años (promedio, 67.6 años), que habían sido examinados con un ecocardiograma Doppler y radiología torácica anterior y lateral. Globalmente, 195 pacientes (19.5%) tuvieron diagnóstico de DISH y 283 (28.3%) de AVS. DISH fue más prevalente que AVS en varones (66.7% vs. 42.6%, p < 0.0001) y en pacientes de mayor edad (73.6 ± 9 años vs. 66.1 ± 9 años, p < 0.0001). Además, 55.4% de los pacientes con DISH dorsal presentaron AVS vs. 21.7% de los pacientes sin DISH (OR = 4.47; 95% CI = 3.22-6.21). El odds ratio (OR) ajustado por sexo y edad fue 3.04 (95% CI = 2.12-4.36; p < 0.0001). Se encontró una asociación estadísticamente significativa entre DISH y AVS, que se mantuvo después de ajustar por sexo y edad. La plausibilidad biológica de esta asociación se basa en los factores de riesgo, mecanismos patogénicos y complicaciones vasculares compartidos.
Assuntos
Idoso , Idoso de 80 Anos ou mais , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estenose da Valva Aórtica/etiologia , Valva Aórtica/patologia , Hiperostose Esquelética Difusa Idiopática/complicações , Hiperostose Esquelética Difusa Idiopática/patologia , Calcificação Vascular/complicações , Fatores Etários , Ecocardiografia Doppler , Hiperostose Esquelética Difusa Idiopática/epidemiologia , Modelos Logísticos , Valva Mitral/patologia , Razão de Chances , Prevalência , Radiografia Torácica , Fatores de Risco , Esclerose , Fatores SexuaisRESUMO
INTRODUCTION: Osteochondromas are common benign bone tumors, rarely involving the spine. BACKGROUND: Osteochondroma account for about 35% of bone benign tumors and 9% of all bone tumors. Spinal involvement is described in 1%-4% of cases and their origin from the anterior surface of cervical vertebral bodies is exceedingly rare. AIM: We describe the rare case of an osteochondroma arising from the anterior surface of the C4 and C5 vertebral bodies, and not involving the spinal canal, in a 68-year-old male patient suffering from Diffuse Idiopathic Skeletal Hyperostosis (DISH). MATERIALS AND METHODS: The patient presented with acute onset of respiratory distress due to laryngeal compression exerted by the lesion, dysphagia and paralysis of left vocal cord. Imaging revealed the unusual lesion compressing and dislocating the air ways. An anterior approach to cervical spine was performed to remove the tumor. RESULTS: Postoperatively, the patient sustained a progressive improvement of respiratory function and recovery of the vocal cord paralysis. DISCUSSION: We believe that this case holds some interesting peculiarities: firstly, the anterior location of a cervical osteochondroma could be considered exceptional; yet, the resulting clinical picture should be remembered for differential diagnosis. Secondly, we hypothesized a possible correlation between the tumorigenesis of osteochondroma and the co-existence of DISH. Indeed, the abnormal bone turnover in cervical segments due to DISH could explain the unusual occurrence of osteochondromas in adult age. CONCLUSIONS: The occurrence of an osteochondroma should be considered in patients suffering from DISH and harbouring large osteophytes.
Assuntos
Vértebras Cervicais , Transtornos de Deglutição/etiologia , Hiperostose Esquelética Difusa Idiopática/complicações , Osteocondroma/complicações , Neoplasias da Coluna Vertebral/complicações , Paralisia das Pregas Vocais/etiologia , Idoso , Vértebras Cervicais/patologia , Humanos , Hiperostose Esquelética Difusa Idiopática/patologia , Doenças da Laringe/etiologia , Laringoscopia , Imageamento por Ressonância Magnética , Masculino , Osteocondroma/patologia , Doenças Raras/complicações , Doenças Raras/patologia , Neoplasias da Coluna Vertebral/patologiaRESUMO
Diffuse idiopathic skeletal hyperostosis (DISH) is considered an underdiagnosed and mostly asymptomatic nonprimary osteoarthritis. The etiology of DISH remains unknown and the validated diagnostic criteria are absent. This condition is still recognized radiologically only. Rarely, large projecting anterior osteophytes result in esophageal impingement and distortion leading to dysphagia. We report the case of progressive dysphagia and neck pain due to DISH of the cervical spine in a 70-year-old man, which was surgically removed with excellent postoperative results and complete resolution of symptoms. Imaging studies, surgical findings, and histopathological examinations were used to support the diagnosis. The patient was successfully treated with total excision of the anterior osteophytes with no evidence of recurrence 12 months after surgery. In this report, we also discuss the clinical features and perioperative considerations in combination with a literature review. Our patient illustrates that clinicians should be aware of this rare clinical manifestation as the presenting feature of DISH in cervical spine. Surgical decompression through osteophytectomy is effective for patients who fail conservative treatment.
Assuntos
Vértebras Cervicais , Transtornos de Deglutição/etiologia , Hiperostose Esquelética Difusa Idiopática/complicações , Cervicalgia/etiologia , Idoso , Vértebras Cervicais/diagnóstico por imagem , Humanos , Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Hiperostose Esquelética Difusa Idiopática/patologia , Masculino , Tomografia Computadorizada por Raios XRESUMO
Diffuse idiopathic skeletal hyperostosis (DISH) is a noninflammatory spondyloarthropathy, characterized by ectopic calcification of spinal tissues. Symptoms include spine pain and stiffness, and in severe cases dysphagia and spinal cord compression. The etiology of DISH is unknown and there are no specific treatments. Recent studies have suggested a role for purine metabolism in the regulation of biomineralization. Equilibrative nucleoside transporter 1 (ENT1) transfers hydrophilic nucleosides, such as adenosine, across the plasma membrane. In mice lacking ENT1, we observed the development of calcified lesions resembling DISH. By 12 months of age, ENT1(-/-) mice exhibited signs of spine stiffness, hind limb dysfunction, and paralysis. Micro-computed tomography (µCT) revealed ectopic mineralization of paraspinal tissues in the cervical-thoracic region at 2 months of age, which extended to the lumbar and caudal regions with advancing age. Energy-dispersive X-ray microanalysis of lesions revealed a high content of calcium and phosphorus with a ratio similar to that of cortical bone. At 12 months of age, histological examination of ENT1(-/-) mice revealed large, irregular accumulations of eosinophilic material in paraspinal ligaments and entheses, intervertebral discs, and sternocostal articulations. There was no evidence of mineralization in appendicular joints or blood vessels, indicating specificity for the axial skeleton. Plasma adenosine levels were significantly greater in ENT1(-/-) mice than in wild-type, consistent with loss of ENT1--a primary adenosine uptake pathway. There was a significant reduction in the expression of Enpp1, Ank, and Alpl in intervertebral discs from ENT1(-/-) mice compared to wild-type mice. Elevated plasma levels of inorganic pyrophosphate in ENT1(-/-) mice indicated generalized disruption of pyrophosphate homeostasis. This is the first report of a role for ENT1 in regulating the calcification of soft tissues. Moreover, ENT1(-/-) mice may be a useful model for investigating pathogenesis and evaluating therapeutics for the prevention of mineralization in DISH and related disorders.
Assuntos
Calcificação Fisiológica , Transportador Equilibrativo 1 de Nucleosídeo/fisiologia , Hiperostose Esquelética Difusa Idiopática/patologia , Coluna Vertebral/patologia , Animais , Transportador Equilibrativo 1 de Nucleosídeo/genética , Expressão Gênica , Camundongos , Camundongos Endogâmicos C57BL , Espectrometria por Raios X , Tomografia Computadorizada por Raios XRESUMO
A six-year-old, spayed female Weimaraner dog was first presented with the complaint of hindlimb paresis and then hindlimb paralysis two years later after colliding with a tree. Radiographs and computed tomography revealed spinal fractures at lumbar vertebrae (L)2-3 and at L4-5. In addition, the spinal column was affected by new bone formation along the vertebral bodies, bridging the disc spaces, as seen in diffuse idiopathic skeletal hyperostosis (DISH). Open reduction and internal fixation was achieved with standard vertebral body plating. This is the first report of DISH-associated spinal fractures after minor trauma in a dog. Surgery resulted in return of the full function after the first, and in improvement of neurologic function after the second incident.
Assuntos
Doenças do Cão/patologia , Hiperostose Esquelética Difusa Idiopática/veterinária , Fraturas da Coluna Vertebral/veterinária , Animais , Cães , Feminino , Hiperostose Esquelética Difusa Idiopática/patologia , Fraturas da Coluna Vertebral/patologia , Fraturas da Coluna Vertebral/cirurgiaRESUMO
Diffuse idiopathic skeletal hyperostosis is a condition of unknown cause, characterized by calcification and ossification of entheseal sites, which generally causes ankylosis of the spinal column and ossification of the extra-spinal ligaments or tendons. The case reported is from the cervical region of the spinal column of skeletal remains. Knowledge of the multiple clinical and functional implications analyzed is of great importance when approaching patients with said manifestations.
La hiperostosis esquelética difusa idiopática es una afección de causa desconocida, caracterizada por calcificación y osificación de sitios de entesis, que generalmente ocasiona anquilosis de la columna vertebral y osificación de ligamentos o tendones. El caso reportado es a partir de la región cervical de la columna vertebral de un resto esquelético. El conocimiento de las múltiples implicaciones clínicas y funcionales analizadas es de gran importancia para el momento del abordaje de pacientes con dichas manifestaciones.
Assuntos
Humanos , Adulto , Hiperostose Esquelética Difusa Idiopática/patologia , Osteófito , Vértebras Cervicais/patologia , AnquiloseRESUMO
Diffuse idiopathic skeletal hyperostosis (DISH) is difficult to distinguish from various forms of inflammatory arthritis, including psoriatic arthritis (PsA), rheumatoid arthritis, and ankylosing spondylitis. A 67-year-old Japanese male had been treated for psoriasis vulgaris for 13 years. Numbness of his right arm and lower limbs and spinal stiffening had developed 7 years prior to his initial evaluation at our facility. He noticed pain mainly while exercising. There were symmetrical marginal syndesmophytes in the spine, from the thoracic vertebrae to the upper lumbar vertebrae, on radiological examinations. We therefore suspected DISH. Furthermore, ossifications of the posterior and anterior longitudinal ligaments were noted in the cervical spine. Laboratory examinations revealed a normal peripheral white blood cell count, serum C-reactive protein, and erythrocyte sedimentation rate, and he was negative for rheumatoid factor. We detected human leukocyte antigen B39 but not B27. All distal interphalangeal joints were swollen but without pain. X-ray imaging showed narrowing of the joint space, and the consolidation of the joint was recognized, but there was no new juxta-articular bone formation. Based on clinical and radiological findings, we concluded that he had DISH and not PsA. DISH was indicated by marked radiological features of the axial skeleton, particularly the thoracic spine, but may also have involved the peripheral joints. DISH is one of the entheseal disorders, and 10% of Japanese middle-aged and elderly men have DISH. Therefore, the differentiation of DISH from PsA is necessary in psoriasis patients with spinal involvement.
Assuntos
Hiperostose Esquelética Difusa Idiopática/complicações , Ligamentos Longitudinais/patologia , Ossificação do Ligamento Longitudinal Posterior/etiologia , Ossificação Heterotópica/etiologia , Psoríase/complicações , Idoso , Artrite Psoriásica/diagnóstico , Artrite Psoriásica/etiologia , Biomarcadores/sangue , Diagnóstico Diferencial , Humanos , Hiperostose Esquelética Difusa Idiopática/sangue , Hiperostose Esquelética Difusa Idiopática/diagnóstico , Hiperostose Esquelética Difusa Idiopática/diagnóstico por imagem , Hiperostose Esquelética Difusa Idiopática/tratamento farmacológico , Hiperostose Esquelética Difusa Idiopática/patologia , Imunossupressores/uso terapêutico , Ligamentos Longitudinais/diagnóstico por imagem , Masculino , Ossificação do Ligamento Longitudinal Posterior/sangue , Ossificação do Ligamento Longitudinal Posterior/diagnóstico , Ossificação do Ligamento Longitudinal Posterior/diagnóstico por imagem , Ossificação do Ligamento Longitudinal Posterior/tratamento farmacológico , Ossificação Heterotópica/sangue , Ossificação Heterotópica/diagnóstico , Ossificação Heterotópica/diagnóstico por imagem , Ossificação Heterotópica/tratamento farmacológico , Ossificação Heterotópica/patologia , Valor Preditivo dos Testes , Psoríase/diagnóstico , Psoríase/tratamento farmacológico , Psoríase/patologia , RadiografiaRESUMO
OBJECTIVE: To investigate the degree of nonarticular tenderness and functional status in patients with diffuse idiopathic skeletal hyperostosis (DISH). We assessed these variables' correlation with their clinical, radiographic, and constitutional measurements and with metabolic syndrome (MS). METHODS: Eighty-seven patients with DISH were compared with 65 controls without DISH. Examination of nonarticular tenderness was performed by thumb palpation. Tenderness was scored for the 18 fibromyalgia tender points (TP), and 4 control points. Nonarticular tenderness was expressed by the number of TP and by the total tenderness score (TTS). The Short Health Assessment Questionnaire (HAQ II) was administered to all participants. Clinical and laboratory data were collected from all patients. Patients were classified as having MS by both the National Cholesterol Education Program and World Health Organization definitions. RESULTS: There was a statistically significant difference in TTS between controls and patients with DISH. The mean tenderness of many individual TP was significantly higher in the DISH group compared with the control group. TP counts, TTS, and body mass index (BMI) positively correlated with the HAQ II. There was a linear trend in intensity of T-spine bony bridges (BB) and the total number of TP as well as many individual TP. Patients with DISH were more likely to be affected by MS. No correlation was found between TP count, TTS, and MS. CONCLUSION: Patients with DISH have a lower pain threshold than patients who do not have DISH. TP count and TTS correlate with the functional status, BMI, waist circumference, and high-grade BB. No correlation was observed between pain threshold and MS.
Assuntos
Atividades Cotidianas , Hiperostose Esquelética Difusa Idiopática , Dor , Fibromialgia/fisiopatologia , Humanos , Hiperostose Esquelética Difusa Idiopática/complicações , Hiperostose Esquelética Difusa Idiopática/patologia , Hiperostose Esquelética Difusa Idiopática/fisiopatologia , Dor/etiologia , Dor/fisiopatologia , Medição da Dor , Limiar da Dor , Palpação , Inquéritos e QuestionáriosRESUMO
Hyperostosis of anterior cervical vertebral osteophytes can produce otolaryngological symptoms ranging from mild dysphagia, dysphonia, and foreign body sensation to severe food impaction and stridulous dyspnea. Airway compromise necessitating a tracheostomy is very rare. We discuss the case of an elderly man who presented with progressive dysphagia and a large hypopharyngeal mass as his initial manifestations of hypertrophic anterior cervical osteophytes. After a biopsy of the mass, the patient went into airway distress due to bilateral vocal fold fixation by the enlarging mass and consequently required a surgical airway. A combined team approach to the removal of the osteophytes successfully resolved his symptoms. The clinical, diagnostic, radiologic, and therapeutic principles involved in this case are presented and discussed. The recognition of hypertrophic osteophytes as a potential cause of common otolaryngological symptoms in the elderly population is paramount, as these symptoms can rapidly progress and lead to life-threatening airway obstruction. Medical and surgical interventions can be employed for the treatment of hypertrophic anterior cervical osteophytes, and they often result in favorable outcomes.
Assuntos
Obstrução das Vias Respiratórias/etiologia , Vértebras Cervicais , Transtornos de Deglutição/etiologia , Hiperostose Esquelética Difusa Idiopática/complicações , Idoso de 80 Anos ou mais , Vértebras Cervicais/patologia , Humanos , Hiperostose Esquelética Difusa Idiopática/diagnóstico , Hiperostose Esquelética Difusa Idiopática/patologia , Hiperostose Esquelética Difusa Idiopática/cirurgia , Hipertrofia , Laringoscopia , Masculino , TraqueostomiaRESUMO
STUDY DESIGN: Case report and clinical discussion. OBJECTIVE: To describe technical pitfall to treat 2 cervical cord injuries, including dislocations in patients with ankylosed spine due to diffuse idiopathic skeletal hyperostosis (DISH) or ossification of the posterior longitudinal ligament (OPLL). SUMMARY OF BACKGROUND DATA: DISH and OPLL are disease processes similar in pathology, which can lead to unexpected fractures due to low-energy trauma. In reported cases of fracture of the ankylosed spine in patients with DISH or OPLL, increasing lever arm and a grossly unstable fracture occurred. However, the actual surgical intervention for these fractures and spinal cord injuries was not discussed. METHODS: We report 2 cervical cord injuries, including dislocations in patients with ankylosed spine due to DISH or OPLL. RESULTS: Two patients underwent posterior fusion without decompression; however, postoperative progressive paraplegia still occurred. There were 3 points in common: these patients had ankylosed spines due to DISH or OPLL; they were elderly and had spinal canal stenosis; and after undergoing posterior fusion without decompression, their bilateral, lower extremity palsies worsened after surgery. Cervical alignment was slightly different after posterior fusion, and this change concentrated in one segment because adjacent vertebral bodies were ankylosed, and thus, immoveable. Additionally, this stress caused infolding of the ligamentum flavum with resultant spinal cord compression. CONCLUSION: In these cases, we recommend posterior fusion and decompression such as laminoplasty to avoid worsening palsy.
Assuntos
Anquilose/cirurgia , Hiperostose Esquelética Difusa Idiopática/cirurgia , Ossificação do Ligamento Longitudinal Posterior/cirurgia , Paraplegia/etiologia , Traumatismos da Medula Espinal/cirurgia , Fusão Vertebral/efeitos adversos , Coluna Vertebral/cirurgia , Acidentes por Quedas , Idoso , Anquilose/complicações , Anquilose/patologia , Vértebras Cervicais/patologia , Vértebras Cervicais/cirurgia , Progressão da Doença , Evolução Fatal , Humanos , Hiperostose Esquelética Difusa Idiopática/complicações , Hiperostose Esquelética Difusa Idiopática/patologia , Masculino , Ossificação do Ligamento Longitudinal Posterior/complicações , Ossificação do Ligamento Longitudinal Posterior/patologia , Traumatismos da Medula Espinal/complicações , Traumatismos da Medula Espinal/patologia , Coluna Vertebral/patologia , Resultado do TratamentoRESUMO
The authors describe the main clinical and radiological findings of common enthesopathic disorders-spondylarthritis (SpA), chondrocalcinosis/calcium pyrophosphate dehydrate crystal deposition disease (CPPD CDD) and diffuse idiopathic skeletal hyperostosis (DISH), stressing similarities and differences which may help in the differential diagnosis. They emphasize the clinical presentation of the "pseudoankylosing spondylitis" forms of CPPD CDD. They also review the most relevant genes and molecular mechanisms associated with these conditions and with another enthesopathic disorder with high prevalence in the Japanese population-ossification of the posterior longitudinal ligament (OPLL).