Diffuse idiopathic skeletal hyperostosis of the cervical spine: an underestimated cause of dysphagia and airway obstruction

Background context

Diffuse idiopathic skeletal hyperostosis (DISH) is a common but underdiagnosed condition relating to ossification of spinal ligaments that can cause compression of the esophagus and trachea. According to case reports, dysphagia or airway obstruction resulting from DISH is a rare occurrence.

Purpose

This study was intended to identify all published cases of dysphagia and/or airway obstruction resulting from DISH to increase the epidemiologic/clinical knowledge of these related conditions.

Study design

A systematic review of the literature was performed.

Methods

The articles resulting from the systematic PubMed/EMBASE search of the literature were closely read, and predefined parameters were scored.

Results

The search yielded a total of 118 articles (95 case reports and 23 case series) describing 204 patients with dysphagia and/or airway obstruction resulting from DISH. The number of cases demonstrated a steady increase from 1980 to 2009. This might be a real effect not ascribable to publication bias or expansion of the medical literature alone.

Conclusions

Diffuse idiopathic skeletal hyperostosis as a cause of dysphagia and/or airway obstruction may be an increasing and underappreciated phenomenon. Diffuse idiopathic skeletal hyperostosis should be included in the differential diagnosis of dysphagia and airway obstruction.     

Postsurgical recurrence of osteophytes causing dysphagia in patients with diffuse idiopathic skeletal hyperostosis

Although cervical anterior osteophytes accompanying diffuse idiopathic skeletal hyperostosis (DISH) are generally asymptomatic, large osteophytes sometimes cause swallowing disorders. Surgical resection of the osteophyte has been reported to be an effective treatment; however, little study has been given to the recurrences of osteophytes. A prospective study was performed for seven patients who underwent surgical resection of cervical anterior osteophytes for the treatment of recalcitrant dysphagia caused by osteophytes that accompanied DISH. The seven patients were six men and one woman ranging in age from 55 to 78 years (mean age = 65 years). After a mean postoperative follow-up period of 9 years (range: 6–13 years), surgical outcomes were evaluated by symptom severity and plain radiographs of the cervical spine. On all operated intervertebral segments, the effect of postoperative intervertebral mobility (range of movement >1 degree) on the incidence of recurrent osteophytic formation (width >2 mm) was analyzed by Fisher’s exact test. Complete relief of the dysphagia was obtained within one month postoperatively in five patients, while it was delayed for 3 months in two patients. All of the patients developed recurrent cervical osteophytic formation, with an average increase rate of approximately 1 mm/year following surgical resection. Of the 20 operated intervertebral segments, the incidence of recurrent osteophytes was significantly higher (P = 0.0013) in the 16 segments with mobility than in the four segments without mobility. Five of the seven patients remained asymptomatic, although radiological recurrence of osteophytes was seen at the final follow-up. The two remaining patients complained of moderate dysphagia 10 and 11 years after surgery, respectively; one of these two required re-operation due to progressive dysphagia 11 years postoperatively. In patients with cervical DISH and dysphagia, surgical resection of osteophytes resulted in a high likelihood of the recurrence of osteophytes. Therefore, attending surgeons should continue to follow these patients postoperatively for more than 10 years in order to assess the regrowth of osteophytes that may contribute to recurrent symptoms.     

弥漫性特发性骨质增生症伴腰椎管狭窄症的影像学特征

目的：探讨弥漫性特发性骨质增生症（diffuse idiopathic skeletal hyperostosis,DISH）伴腰椎管狭窄症的影像学特征。方法：回顾性分析2017年6月～2021年10月在我院手术治疗的96例DISH合并L4/5椎管狭窄症患者的术前影像学资料,男58例,女38例;年龄51～77岁（65.9±6.1岁）;将骨化仅限于胸椎节段的患者纳入T-DISH组（n=49）,骨化累及胸椎和腰椎节段的患者纳入L-DISH组（n=47）;选取同期在我院手术治疗的性别、年龄及狭窄节段相匹配的无DISH的腰椎管狭窄症患者作为对照组（N-DISH组,n=125）。测量各组术前胸椎后凸角（thoracic kyphosis,TK）、腰椎前凸角（lumbar lordosis,LL）、骶骨倾斜角（sacral slope,SS）、骨盆入射角（pelvic incidence,PI）、骨盆倾斜角（pelvic tilt,PT）和矢状位平衡距离（sagittal vertical axis,SVA）,计数骨化韧带节段数目,按照Pfirrmann分级和Weishaupt分级分别评估椎间盘与...     

Cervical hyperostosis: A rare cause of dysphagia

Summary Dysphagia can be caused by disorders of the cervical spine. Very seldomly, prominent osteophytes of the ventral spine are responsible. The case of a 63-year-old patient with large anterior osteophytes from C3 to C7 is presented. The successful ablation of these spondylophytes relieved the patient of his swallowing difficulties. Up to now there have been many different opinions about the etiology of this disease. In this special case, a diffuse idiopathic skeletal hyperostosis, also known as Forestier's disease or diffuse idiopathic skeletal hyperostosis, seems to be the most likely cause.     

Presentation and treatment of anterior cervical hyperostosis

We report a case of severe anterior cervical hyperostosis presenting with dysphagia.     

伴与不伴弥漫性特发性骨质增生症的腰椎管狭窄症患者脊柱骨盆参数的比较

目的 :探讨弥漫性特发性骨质增生症(diffuse idiopathic skeletal hyperostosis,DISH)对腰椎管狭窄症(lumbar spinal stenosis,LSS)患者脊柱骨盆矢状面参数的影响。方法 :回顾性分析2014年1月~2017年6月于南京鼓楼医院行腰椎后路全椎板切除减压椎间融合手术的伴DISH的LSS患者40例,其中男23例,女17例,年龄51~75岁(65.1±7.3岁)。同时选取年龄及性别匹配且接受相同术式的不伴DISH的LSS患者40例作为对照组,其中男23例,女17例,年龄51~75岁(64.7±7.1岁)。两组患者性别、年龄、责任节段分布均无统计学差异(P0.05)。分别测量两组患者术前胸椎后凸角(thoracic kyphosis,TK)、腰椎前凸角(lumbar lordosis,LL)、矢状面平衡(sagittal vertical axis,SVA)、骨盆入射角(pelvic incidence,PI)、骨盆倾斜角(pelvic tilt,PT)、骶骨倾斜角(sacral slope,SS)、腰骶角(lumbar-sacral angle,LSA)、腰5入射角(L5 incidence,L5I)等脊柱骨盆参数,并用术前VAS评分和ODI评分评估入选对象的生活质量。采用独立样本t检验比较两组患者脊柱骨盆参数的差异及生活质量的差异。结果:伴DISH的LSS患者的术前SVA显著低于不伴DISH的LSS患者(20.0±38.7mm vs.40.0±46.3mm,P=0.039),而TK(27.2°±10.7°vs 25.5°±16.0°)、LL(48.1°±13.7°vs 47.1°±13.5°)、PI(51.4°±14.1°vs 52.5°±13.9°)、PT(18.0°±8.0°vs 19.0°±7.6°)、SS(33.6°±9.5°vs 34.4°±7.8°)、LSA(17.0°±12.0°vs 18.4°±6.7°)及L5I(22.4°±9.8°vs 24.7°±11.9°)两组均无统计学差异(P0.05)。伴DISH患者术前腰痛和下肢痛VAS评分及ODI评分(分别为5.6±1.0,6.5±1.3,36.9±4.9)均高于不伴DISH组患者(分别为5.4±1.2,6.2±1.7,36.8±5.0),但差异均无统计学意义(P0.05)。结论:与不伴DISH的LSS患者相比,伴DISH的LSS患者SVA显著降低,TK、LL、PI、PT、SS、LSA及L5I等其他脊柱骨盆矢状面参数无显著差异。     

弥漫性特发性骨肥厚症（DISH）的手术治疗

报告２８例弥漫性特发性骨肥厚症（ＤＩＳＨ），其中２７例伴有脊髓病或神经根病，１例伴有食道压迫症。全部病例均进行了手术治疗。ＤＩＳＨ伴有颈脊髓压迫的手术优良率为８３．３％；伴有胸脊髓压迫的手术优良率为６６％。对手术治疗有关的问题进行了讨论。     

胸腰椎过伸骨折合并弥漫性特发性骨肥厚症的临床研究

目的分析合并弥漫性特发性骨肥厚症(DISH)胸腰椎过伸骨折的损伤机制、损伤特点,以指导治疗方案的选择。方法回顾性分析自2007-01—2015-12诊治的95例合并DISH胸腰椎过伸骨折,根据X线片、CT及MRI评估脊柱损伤类型:前柱经椎间盘损伤44例(46.3%),经骨损伤41例(43.2%),混合型损伤10例(10.5%)。所有患者均行脊柱后路切开复位椎弓根钉内固定手术治疗。结果术后3例出现切口愈合不良,给予换药、抗生素治疗后痊愈。术后X线片显示8枚椎弓根钉位置不佳。在治疗期间,4例出现贫血,5例出现全身炎症性反应综合征(SIRS),4例抗甲氧西林金黄色葡萄球菌感染(MRSA),5例肺或肾脏衰竭,2例肺部感染,1例泌尿系感染,1例出现褥疮。4例在创伤后3个月内死亡,死亡患者年龄75~88(81.7±4.1)岁。结论随着高龄人口及脊柱代谢疾病的增多,DISH患者中出现的胸腰椎过伸损伤也会随之增加。临床骨科医师要充分认识合并DISH胸腰椎过伸骨折的特点,以便制定较为合理的治疗方案。     

Quantitative analysis of the anterolateral ossification mass in diffuse idiopathic skeletal hyperostosis of the thoracic spine

Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic condition leading to ossification of spinal ligaments and has been shown to behave similarly to ankylosing spondylitis (AS) often leading to unstable hyperextension fractures. Currently, no quantitative data are available on the spatial relationship between the bridging anterolateral ossification mass (ALOM) and the vertebral body/intervertebral disc to explain the propensity in DISH to fracture through the vertebral body instead of through the intervertebral disc as more often seen in AS. Furthermore, no reasonable explanation is available for the typical flowing wax morphology observed in DISH. In the current study, a quantitative analysis of computed tomography (CT) data from human cadaveric specimens with DISH was performed to better understand the newly formed osseous structures and fracture biomechanics. Additionally, the results were verified using computed tomography angiography data from ten patients with DISH and ten controls. Transverse CT images were analyzed to obtain ALOM area and centroid angle relative to the anteroposterior axis; intervertebral disc and adjacent cranial and caudal levels. The ALOM area at the mid-vertebral body level averaged 57.9 ± 50.0 mm²; at the mid-intervertebral disc space level it averaged 246.4 ± 95.9 mm². The mean ALOM area at the adjacent level caudal to the mid-vertebral body level was 169.6 ± 81.3 mm²; at the adjacent cranial level, it was 161.7 ± 78.2 mm². The main finding was the significant difference between mean ALOM area at the mid-vertebral body level and other three levels (p < 0.0001). The subsequent verification study showed the presence of vertebral segmental arteries at the mid-vertebral body level in nearly all images irrespective of the presence of DISH. A larger area of ALOM seemed associated with increased counter-clockwise rotation (away from the aorta) of the centroid relative to the anteroposterior axis. The results from the present study suggest a predisposition for fractures through the vertebral body and a role for the arterial system in the inhibition of soft tissue ossification.     

Diffuse idiopathic skeletal hyperostosis as a cause of acute respiratory distress in early postoperative period of total knee arthroplasty

Diffuse idiopathic skeletal hyperostosis (DISH) is a common disorder among the elderly. The diagnosis is made on radiological findings and most of the cases are asymptomatic. In the cases with spinal involvement, bridging ossification of the anterior longitudinal ligament is a common finding. We report a case of DISH, who developed respiratory distress after bilateral total knee arthroplasty operation. The acute occurrence of symptoms after general anesthesia is a rare manifestation of the disease. Radiological findings and the result of surgical resection of anterior cervical osteophytes are discussed.     

Clinical outcome after traumatic spinal fractures in patients with ankylosing spinal disorders compared with control patients

Background contextThe clinical outcome of patients with ankylosing spinal disorders (ASDs) sustaining a spinal fracture has been described to be worse compared with the general trauma population.PurposeTo investigate clinical outcome (neurologic deficits, complications, and mortality) after spinal injury in patients with ankylosing spondylitis (AS) and diffuse idiopathic skeletal hyperostosis (DISH) compared with control patients.Study designRetrospective cohort study.Patient sampleAll patients older than 50 years and admitted with a traumatic spinal fracture to the Emergency Department of the University Medical Center Utrecht, the Netherlands, a regional level-1 trauma center and tertiary referral spine center.Outcome measuresData on comorbidity (Charlson comorbidity score), mechanism of trauma, fracture characteristics, neurologic deficit, complications, and in-hospital mortality were collected from medical records.MethodsWith logistic regression analysis, the association between the presence of an ASD and mortality was investigated in relation to other known risk factors for mortality.ResultsA total of 165 patients met the inclusion criteria; 14 patients were diagnosed with AS (8.5%), 40 patients had DISH (24.2%), and 111 patients were control patients (67.3%). Ankylosing spinal disorder patients were approximately five years older than control patients and predominantly of male gender. The Charlson comorbidity score did not significantly differ among the groups, but Type 2 diabetes mellitus and obesity were more prevalent among DISH patients. In many AS and DISH cases, fractures resulted from low-energy trauma and showed a hyperextension configuration. Patients with AS and DISH were frequently admitted with a neurologic deficit (57.1% and 30.0%, respectively) compared with controls (12.6%; p=.002), which did not improve in the majority of cases. In AS and DISH patients, complication and mortality rates were significantly higher than in controls. Logistic regression analysis showed the parameters age and presence of DISH to be independently, statistically significantly related to mortality.ConclusionsMany patients with AS and DISH showed unstable (hyperextension) fracture configurations and neurologic deficits. Complication and mortality rates were higher in patients with ASD compared with control patients. Increasing age and presence of DISH are predictors of mortality after a spinal fracture.     

Coexistence of flavum ligament ossification with diffuse idiopathic skeletal hyperostosis in the cervical spine: Review of literature and technical note starting from a rare case

BackgroundCervical flavum ligament ossification (C-OLF) is very rare source of myeloradiculopathy. Less than 100 cases have been reported in modern English literature up to 2020. Association between C-OLF and Diffuse Idiopathic Skeletal Hyperostosis (DISH) at cervical level has never been described.MethodsIn this article we performed a systematic review about epidemiology, physiopathology, clinical and surgical management of C-OLF. Moreover, we research its possible association with other cervical spine ligament ossification and in particular with anterior longitudinal ligament ossification. We report a case of 73 years-old woman experiencing mild cervical myeloradiculopathy caused by C6-C7 C-OLF compression and coexistence of DISH at cervico-thoracic level. A brief technical note about intraoperative management of C-OLF has also been described.ResultOur research found 81 previous reported case of C-OLF. The coexistence of Posterior longitudinal ligament ossification has been reported in 21.3% of C-OLF case. Conversely, we reported the first case describing the association between DISH and C-OLF. Posterior surgical decompression is the only useful treatment providing good long-term functional outcome. Instrumentation should be tailored according to pre-operative findings.ConclusionsC-OLF is a rare source of myeloradiculopathy and it may coexists with DISH probably due to alteration in the cervical mechanical stress and tendency of bone formation in patients harboring coexistent ligament ossifications. According to our result, skip en-bloc microsurgical laminectomy is safe and less invasive method to avoid complication and to provide optimal cervical spinal cord and nerve decompression avoiding CSF-leak.