Diffuse Idiopathic Skeletal Hyperostosis: A Rare Cause of Dysphagia, Airway Obstruction, and Dysphonia

doi:10.1016/j.jamcollsurg.2006.02.030

Journal of the American College of Surgeons

Volume 202, Issue 6, June 2006, Pages 938-942

https://doi.org/10.1016/j.jamcollsurg.2006.02.030 Get rights and content

Background

We retrospectively query the clinical records of patients with cervical osteophytes to distinguish the clinical features of those presenting with symptomatic dysphagia and airway obstruction.

Study design

Retrospective review of all patients presenting over a 20-year period (1985 to 2005) with the diagnosis of cervical osteophytes and dysphagia with or without airway compromise. Two hundred thirty-four patients were identified at Marshfield Clinic between 1985 and 2005; 9 (3.8%) met criteria for inclusion.

Results

Eight of nine patients presented with dysphagia. Three of nine patients presented with acute airway obstruction requiring intubation and tracheotomy. Osteophytes occurred at multiple levels, with C4, C5, and C6 being most commonly involved. Surgical decompression resulted in complete resolution of symptoms in four of five patients.

Conclusions

Although commonly found and usually asymptomatic in the older population, anterior cervical osteophytes can be a source of considerable morbidity and potential life-threatening airway obstruction. Recognizing this clinical entity is imperative in establishing a diagnosis and initiating appropriate treatment. Surgical decompression appears to be beneficial in relieving symptoms.

Section snippets

Methods

Institutional review board approval was obtained and a retrospective chart review was done using our electronic medical record. The records of patients seen at Marshfield Clinic over a 20-year period (1985 to 2005) were reviewed for a diagnosis of cervical osteophytes. Those with associated dysphagia with or without airway obstruction were included. All patients had undergone evaluation to rule out other causes of their symptomatology. Data were collected with respect to age, duration of

Results

Two hundred thirty-four patients were identified with a diagnosis of cervical osteophytes. Nine (3.8%) met criteria for inclusion (Table 1). Mean age of the 9 patients was 77 years; there were 7 men and 2 women.

Eight patients presented with dysphagia (eight of nine), with solid food being the most problematic. Three patients developed acute airway obstruction and required tracheotomy, with subsequent percutaneously placed endoscopic gastrostomy tube for nutrition. Two patients developed marked

Discussion

Our patient population reveals that this is a disease of the elderly. The most common complaint was solid-food dysphagia (n = 8), followed by stridor (n = 3), regurgitation (n = 2), globus (n = 1), and dysphonia (n = 1). Patients typically had a long history of dysphagia or other complaints before diagnosis was confirmed, in some cases it was years. Barium esophagram and computed tomography scans were helpful in confirming the diagnosis. C4 to C5 was the cervical level most commonly involved,

Acknowledgment

We thank Marshfield Clinic Research Foundation for its support through the assistance of Graig Eldred and Alice Stargardt in the preparation of this article.

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Acute airway obstruction in a patient with forestier disease. Case report
2019, Revista Espanola de Anestesiologia y Reanimacion
La osificación del ligamento anterior longitudinal vertebral, conocida como enfermedad de Forestier o hiperostosis esquelética idiopática difusa, es una enfermedad que suele cursar de forma asintomática. Se produce sobre todo en la región torácica, seguida por la lumbar y cervical. Cuando existe afectación cervical, en caso de producir clínica, los síntomas más frecuentes son disfagia, disnea o disfonía y, de forma excepcional, obstrucción aguda de la vía aérea. Este proceso patológico supone un reto anestésico en el manejo de la vía aérea de estos pacientes. Por ello presentamos el caso de un paciente de 85 años con obstrucción aguda de vía aérea asociada a enfermedad de Forestier, al que se le realizó intubación mediante fibroscopio bajo anestesia inhalada con sevoflurano manteniendo ventilación espontánea para la posterior realización de traqueotomía por los otorrinolaringólogos.
Ossification of the anterior longitudinal ligament of the spine, known as Forestier disease or diffuse idiopathic skeletal hyperostosis, is usually an asymptomatic disorder. The area most frequently affected is the thoracic spine, followed by lumbar and cervical regions. In the case of cervical involvement with clinical manifestations, the most common symptoms include dysphagia, dyspnoea, dysphonia, and can exceptionally cause an acute airway obstruction. The airway management of these patients represents a great anaesthetic challenge. The case is reported of an eighty-five-year-old patient who had an acute airway obstruction associated with Forestier disease. A fibre-optic-assisted intubation was accomplished under sevoflurane inhaled anaesthesia, maintaining spontaneous ventilation, with subsequent tracheostomy performed by ENT surgeons.
Diffuse idiopathic skeletal hyperostosis (DISH) – A common but less known cause of back pain
2017, Journal of Clinical Orthopaedics and Trauma
Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic non-inflammatory disease of an unknown aetiology. It is characterized by ossification of the enthesis and may significantly affect the quality of life of the affected individuals. Despite the fact that this disease was first described more than 60 years ago and is a relatively common condition in the society, its awareness among clinician is still limited. An awareness of this entity and its mimicking conditions is essential so as to reach to an early diagnosis and avoid unnecessary investigations and treatment.
Dysphagia with diffuse idiopathic skeletal hyperostosis/forestier's disease
2014, Journal for Nurse Practitioners
Citation Excerpt :
Swallowing may be accompanied by muscle spasms and pain as a bolus of food passes through the esophagus and lower esophageal sphincter.11 Other consequences of DISH in the cervical spine include stridor, globus pharyngeus, respiratory compromise, sleep apnea, and difficulty with intubation.19,20,21 Unfortunately, nonoperative measures are rarely effective in addressing dysphagia symptoms caused by DISH, as exemplified in the case report presented here.
Diffuse idiopathic skeletal hyperostosis (DISH), also known as Forestier’s disease, is a systemic bone-forming condition prevalent in older adults. Abnormal radiographic findings of the spine are essential to a DISH diagnosis. The disease is often not included in the differential diagnosis of dysphagia in primary-care practices. Disorders of the cervical spine rarely cause dysphagia but are in fact common manifestations of DISH. The purpose of this brief report is to: (1) provide an overview of DISH; (2) examine dysphagia in the context of DISH through presentation of a case report; and (3) broaden the recognition of DISH for diagnostic purposes.
Diffuse idiopathic skeletal hyperostosis of the cervical spine: An underestimated cause of dysphagia and airway obstruction
2011, Spine Journal
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.
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.
A systematic review of the literature was performed.
The articles resulting from the systematic PubMed/EMBASE search of the literature were closely read, and predefined parameters were scored.
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.
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.
Dysphagia due to anterior cervical hyperosteophytosis
2009, Surgical Neurology
Citation Excerpt :
Dysphagia due to ACH is considered an uncommon entity and has been described mainly in case reports. The osteophytic overgrowth causing dysphagia has been attributed to multiple etiologies including DISH, trauma, postlaminectomy syndromes, and cervical spondylosis [6,23,31,37-39,48]. Although various mechanisms have been suggested, the predominant cause of dysphagia is thought to be mechanical compression of the esophagus by overgrown osteophytes.
Anterior cervical hyperosteophytosis describes the excessive formation of osteophytes along the ventral spine. Dysphagia due to ACH is considered an uncommon entity described mainly in case reports. Symptomatic ACH has been attributed to multiple etiologies including DISH, trauma, postlaminectomy syndromes, and cervical spondylosis. We report one of the largest series of patients with ACH-induced dysphagia requiring surgery.
After IRB approval, a retrospective chart review was completed. From 2001 to 2006, 9 patients presented with dysphagia due to ACH requiring surgical treatment.
Eight patients were male, and the mean age was 65.1 years. Cervical spine x-rays and CT clearly demonstrated ACH in each case. Esophagram or a video fluoroscopic swallowing study was used to verify that dysphagia was caused by osteophytic overgrowth in all instances but one. In 2 patients, a focal osteophyte had formed adjacent to a previously fused segment. Of the remaining 7 patients, osteophytic formation was attributed to cervical spondylosis in 2 patients and DISH in 5 patients. All patients underwent osteophytectomy without spinal fusion. Average follow-up was 9.8 months. Although all 9 patients experienced resolution of dysphagia, improvement was delayed in 2 patients.
Diffuse idiopathic skeletal hyperostosis and spondylosis are the most common etiologies accounting for ACH-induced dysphagia. Adjacent segment disease may also be a potential cause of symptomatic ACH and has not been previously reported. Regardless of etiology, surgical resection is highly successful if conservative measures fail.
Stiffness and axial pain are associated with the progression of calcification in a mouse model of diffuse idiopathic skeletal hyperostosis
2023, Arthritis Research and Therapy

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Original scientific articleDiffuse Idiopathic Skeletal Hyperostosis: A Rare Cause of Dysphagia, Airway Obstruction, and Dysphonia

Background

Study design

Results

Conclusions

Section snippets

Methods

Results

Discussion

Acknowledgment

Am J Otolaryngol

Oral Surg Oral Med Oral Pathol

Spine J

Evaluation and correction of dysphagia-producing cervical osteophytosis

Laryngoscope

Radiographic and pathologic features of spinal involvement in diffuse idiopathic skeletal hyperostosis (DISH)

Radiology

Diffuse skeletal abnormalities in Forestier’s disease

Arch Intern Med

Physiologic vertebral ligamentous calcificationan aging process

Am J Roentgenol Radium Ther Nucl Med

A case of dysphagia due to an osteochondroma of the cervical spine-osteotomy-recovery

Ann Otol Rhinol Laryngol

Dysphagia and hypertrophic spurring of the cervical spine

N Engl J Med

Original scientific article
Diffuse Idiopathic Skeletal Hyperostosis: A Rare Cause of Dysphagia, Airway Obstruction, and Dysphonia