Diffuse idiopathic skeletal hyperostosis as a cause for dysphagia in a patient with ankylosing spondylitis.

BACKGROUND: Diffuse idiopathic skeletal hyperostosis (DISH) is characterized by ankylosing spinal alterations which are often asymptomatic but may typically cause back pain and spinal stiffness. Presence of DISH may complicate spinal trauma and lead to unstable fractures requiring surgical intervention. Treatment options include physical activity, symptomatic treatment, local heat application, and optimization of metabolic comorbidities. CASE: A multimorbid older patient was admitted to the gastroenterological ward for the investigation of progressive dysphagia and weight loss. Gastroscopy revealed a dorsal impression of the esophagus at 25 cm from the incisor. Clinical work-up including computed tomography (CT) and magnetic resonance imaging (MRI) ruled out malignancy but showed ankylosing spondylophytes and non-recent fractures of vertebrae C5-C7, compatible with DISH of the cervicothoracic spine as a cause for the esophageal impression. Notably, imaging diagnostics showed ankylosing spine alterations extending to the lumbar spine and both sacroiliac joints, suggestive of ankylosing spondylitis (AS). Typical imaging characteristics, a history of psoriasis, and positive HLA*B27 status supported the diagnosis of underlying AS in this patient with dysphagia as an unusual primary symptom of DISH. Additionally, pulmonary alterations compatible with a usual interstitial pneumonia (UIP)-like pattern were seen on lung CT. CONCLUSION: Overlaps among AS, DISH and pulmonary abnormalities including UIP have been described previously; however, they represent unexpected findings in this older patient. This case underlines the importance of interdisciplinary collaboration and consideration of DISH as a differential diagnosis in patients with atypical symptoms.

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.

BACKGROUND: Cervical 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. METHODS: In 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. RESULT: Our 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. CONCLUSIONS: C-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.

Unusual cause of dysphagia and dysphonia.

Forestier's disease is an idiopathic noninflammatory condition associated with enthesopathy leading to hyperostosis of the vertebrae and peripheral skeletal system. The disease tends to affect elderly individuals and remains asymptomatic in most of the cases. Uncommonly, the patient may present with upper aerodigestive symptomatology, usually dysphagia. In elderly individuals, the disease may closely mimic upper aerodigestive tract malignancy, which should be actively excluded. In our patient, the hypopharyngeal soft tissue distortions created by the bony hypertrophy shifted the clinicoradiological suspicion towards malignant pathology. The current case presents the diagnostic dilemma associated with the disease and the need to keep the possibility of severe cervical bony hypertrophy as a cause of upper aerodigestive symptoms in mind.

Otolaryngologic manifestations of diffuse idiopathic skeletal hyperostosis (Forestier's disease): A systematic review of the literature.

OBJECTIVES: Forestier's disease or diffuse idiopathic skeletal hyperostosis (DISH) is a common, yet underreported, disease affecting the elderly population. From an otolaryngologic perspective, DISH may manifest with dysphagia, dysphonia, or even dyspnea. The purpose of this study was to identify all published cases of dysphagia and other associated upper airway symptoms resulting from DISH in the last decade and to establish subsequently a management algorithm. METHODS: A comprehensive review of the literature was conducted in May 2020 on Medline and Embase databases following the PRISMA statement for systematic reviews and meta-analysis. RESULTS: Sixty-three articles, consisting of 50 case-reports and 13 case-series, met the inclusion criteria. A total of 236 cases of DISH were reported from 2010 to date. Otolaryngology instrumental evaluation, by fiberoptic laryngoscopy and fiberoptic endoscopic evaluation of swallowing, was frequently reported. Surgery was the most common treatment strategy for the management of dysphagia in 58.9% of patients, while conservative treatment was used in 41.1%. Tracheotomy for acute airway obstruction relief was performed in 6% of patients. No correlation was found between the type of treatment and dysphagia improvement. CONCLUSIONS: Forestier's disease is currently a growing source of complications in elderly, mostly dysphagia and less commonly upper airway obstruction. The management of these complications requires a multidisciplinary team and a thorough approach, where the otolaryngologist plays a pivotal role.

Síndrome de forestier: caso clínico y revisión de la literatura / Forestier syndrome: clinical case and literature review

Antecedentes: El síndrome de Forestier también conocido como Hiperostosis Esquelética Idiopática Difusa (DISH, por sus siglas en inglés), es una enfermedad de etiología desco-nocida que se caracteriza por osificación del ligamento espinal anterior, siendo las porciones cervicales y torácicas las que se afectan más frecuentemente. Esta enfermedad es más frecuente en hombres y se asocia con diabetes, hipertensión arterial, disli-pidemia y trastornos endocrinos. Descripción del caso clínico:Paciente femenina de 63 años con antecedente de dolor cervical desde hace 32 años, que 6 años después del inicio del cuadro, presentó limitación en la movilidad del cuello; presentando va-rios episodios de disfonía desde hace 10 años; al momento de la consulta la paciente presentó limitación de la movilidad del cue-llo y dolor cervical. La imagen de resonancia magnética reportó: presencia de crecimiento óseo anterior de los cuerpos vertebra-les, este hallazgo está en relación con el síndrome de Forestier. Conclusiones: Por ser una enfermedad poco conocida es sub-diagnosticada y a menudo confundida con otras patologías. Los pacientes son diagnosticados muchos años después de que apa-recieron los primeros síntomas que incluyen dolor, limitación de la movilidad, disfagia y dificultad respiratoria. El tratamiento incluye manejo sintomático, terapia física y manejo quirúrgico...(AU)

[Dysphagia caused by osteophytes of the cervical spine]. / Dysfagie door osteofyten van de cervicale wervelkolom.

BACKGROUND: Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic condition characterised by ossification of the ligaments around the spinal column due to unknown aetiology. This is often asymptomatic, but it can cause dysphagia in rare cases. CASE DESCRIPTION: A 64-year-old man was referred to the ENT department with progressive dysphagia symptoms he had had for years. Extensive analysis by the ENT specialist did not reveal any abnormalities of the swallowing motor functions. However, osteophytes of the cervical spine of the patient were visible in the X-ray of the cervical spine. The patient was referred to an orthopaedic surgeon. The osteophytes were surgically removed from the cervical spine using a ventral approach. Immediately after the surgery, the patient no longer had problems swallowing. CONCLUSION: In rare cases, dysphagia can be caused by osteophytes of the cervical spine in patients with DISH. Surgical removal of these osteophytes usually has good results.

Atypical case of diffuse idiopathic skeletal hyperostosis mimicking ankylosing spondylitis in a young woman with polycystic ovary syndrome.

Both diffuse idiopathic skeletal hyperostosis and ankylosing spondylitis present with similar clinical manifestations of restricted spinal mobility and postural abnormalities, and radiographic resemblances including axial spine involvement and enthesopathy. Nonetheless, they are two entirely different diseases. We report an unusual case of DISH in a young woman whose diagnosis was established based on radiologic features. This case report aims to highlight the under-recognised radiologic aspects of the differential diagnosis between DISH and AS in order to avoid an inaccurate diagnosis.

Aortic pulsation prevents the development of ossification of anterior longitudinal ligament toward the aorta in patients with diffuse idiopathic skeletal hyperostosis (DISH) in Japanese: Results of chest CT-based cross-sectional study.

BACKGROUND: The development and etiology of diffuse idiopathic skeletal hyperostosis (DISH) were far from complete understanding. Even the precise mechanism of the development of its typical right-sided ossification of the anterior longitudinal ligament (OALL) frequently compared to 'flowing candle wax', a hallmark of DISH, remains unknown. METHODS: The participants of this study were 261 individuals (31 females and 230 males) diagnosed as DISH according to the criteria established by Resnick and Niwayama extracted from a consecutive 3013 patients who have undergone chest CT for the examination of pulmonary diseases in our institute. The patients with previous thoracic spine surgery and younger than 15 years old were excluded. Chest CT data were converted to the condition suitable for bone evaluation by the software application. The positional relationship between thoracic aorta and OALL, the morphology of the OALL adjacent to the aorta and the presence of calcification of the aortic wall adjacent to the OALL were studied. RESULTS: Of 261 individuals with DISH, we found that thoracic aorta was localized adjacent to the OALL (AD-group) in 123 cases (47%), whereas 138 cases (53%) were not (NAD-group). All OALL in AD-group was localized between T6 and T12. The shape of the OALL adjacent to the aorta was either flat or concave except for one. No case showed obvious calcification of the aortic wall adjacent to the OALL in AD-group. CONCLUSIONS: The aortic pulsation might play an important role in inhibit the development of the OALL toward the aorta in DISH. It is likely that establishment of the optimal condition of the pulsation stress simulating aortic pulsation and its delivery system can achieve arresting, slowing the progression and/or changing the morphology of the ossified lesions.

Management of Airway Obstruction due to Diffuse Idiopathic Skeletal Hyperostosis in the Cervical Spine: A Case Report and Literature Review.

Diffuse idiopathic skeletal hyperostosis (DISH) is a relatively common progressive noninflammatory entheses disease. Patients are often asymptomatic or are undiagnosed due to minor chronic symptoms. We herein report a rare case in which the primary symptom was sudden-onset upper airway obstruction due to exuberant osteophytosis in the cervical spine. Treatment was successful with careful airway management and surgical osteophyectomy. Most DISH cases in the literature with airway obstruction have been managed with tracheotomy. However, the safety and necessity of this approach remain questionable. We herein discuss the possibility of conservative management as a choice of airway control. Airway obstruction due to DISH may be underrecognized. This highlights the importance of including DISH in the differential diagnosis of airway obstruction. In addition, a detailed evaluation and personalized care for each individual case is essential.

Diffuse Idiopathic Skeletal Hyperostosis (DISH) and non small cell lung cancer: case presentation and review of the literature.

Diffuse idiopathic skeletal hyperostosis (DISH), also known as Forestier's disease, is a systemic non inflammatory disease of unknown cause. It is characterized by the presence of osteophytes due to calcification and ossification of spinal ligaments and entheses. Moreover, diffuse idiopathic skeletal hyperostosis has been associated with a variety of metabolic disorders. However, to the best of our knowledge no association with non small cell lung cancer (NSCLC) has been reported so far. In the present study we report a case of a patient with NSCLC and DISH.

Dysphagia Caused by Diffuse Idiopathic Skeletal Hyperostosis.

Diffuse idiopathic skeletal hyperostosis, or Forestier disease, is a relatively common disease in the elderly population. Although reported in the orthopedic and rheumatology literature, it has not, to date, been reported in the maxillofacial or dental literature. It is a disease entity that often presents with head and neck symptoms that might go unrecognized by the consultant oral and maxillofacial surgeon.

Proximal Junctional Failure After Long-Segment Instrumentation for Degenerative Lumbar Kyphosis With Ankylosing Spinal Disorder.

STUDY DESIGN: Case report. OBJECTIVE: We report a case of proximal junctional failure at the ankylosed, but not the mobile, junction after segmental instrumented fusion for degenerative lumbar kyphosis with ankylosing spinal disorder. SUMMARY OF BACKGROUND DATA: Proximal junctional failure (PJF) and proximal junctional kyphosis (PJK) are important complications that occur subsequent to long-segment instrumentation for correction of adult spinal deformity. Thus far, most studies have focused on the mobile junction as a site at which PJK/PJF can occur, and little is known about the relationship between PJK/PJF and ankylosing spinal disorders such as diffuse idiopathic skeletal hyperostosis. METHODS: The patient was an 82-year-old female with degenerative lumbar kyphosis. She had abnormal confluent hyperostosis in the anterior longitudinal ligaments from Th5 to Th10. The patient was treated operatively with spinal instrumented fusion from Th10 to the sacrum. RESULTS: Four weeks subsequent to initial surgery, the patient developed progressive lower extremity paresis caused by the uppermost instrumented vertebrae fracture (Th10) and adjacent subluxation (Th9). Extension of fusion to Th5 with decompression at Th9-Th10 was performed. However, the patient showed no improvement in neurological function. CONCLUSION: PJF can occur at the ankylosing site above the uppermost instrumented vertebrae after long-segment instrumentation for adult spinal deformity. PJF in the ankylosed spine may cause severe fracture instability and cord deficit. The ankylosed spine should be integrated into the objective determination of materials contributing to the appropriate selection of fusion levels. LEVEL OF EVIDENCE: 3.

Results after the surgical treatment of anterior cervical hyperostosis causing dysphagia.

PURPOSE: The objective of this study was to investigate the outcome of a case series of patients with dysphagia resulting from diffuse idiopathic skeletal hyperostosis (DISH) of the cervical spine who were treated surgically with resection and fusion. METHODS: A retrospective study was performed on all patients who presented (2005-2013) with complaints of dysphagia or respiratory compromise and who underwent anterior cervical osteophyte resection with fusion (polyether ether ketone cage and/or plate system) using an anterior approach. All patients were diagnosed with DISH and underwent preoperative esophageal and laryngoscopic examinations and a fluoroscopic swallowing study. Initial non-operative strategies were performed, including diet, change in head position during swallowing, non-steroidal anti-inflammatory drugs and pantoprazole. RESULTS: A total of six patients with DISH were included. The mean age was 67 ± 5 years. All patients were male and had symptoms of dysphagia and neck pain, one had simultaneous airway complaints, and another had regurgitation with a sleep disorder. All patients had significant improvements in dysphagia, respiratory complaints and regurgitation 6 weeks after surgery. The postoperative radiographs showed complete removal of the compressive structures. There were no postoperative complications. At the final follow-up (23 ± 8 months), the radiographic examinations showed no pathological regrowth, and the patients reported no recurrence of dysphagia. CONCLUSION: Diffuse idiopathic skeletal hyperostosis may lead to osteophyte-associated pathologies of the aerodigestive tract. Preoperative investigations with esophageal and laryngoscopic examinations combined with fluoroscopic swallowing tests are essential. Surgical decompression through osteophytectomy and fusion is an effective management strategy in selected patients and should be considered when non-operative strategies have failed.

Ultrasonography of peripheral entheses in the diagnosis and understanding of diffuse idiopathic skeletal hyperostosis (DISH).

The aim of this study was to investigate musculoskeletal ultrasound (MSUS) as a diagnostic modality in DISH and to explore whether it might help in elucidating its pathogenesis and events that precede the calcification/ossification process. Fifty patients with DISH and 34 patients with osteoarthritis of the lower limbs without DISH were investigated. Data regarding demographics and traditional cardiovascular risk factors were collected from all patients. An ultrasonography was performed according to the Glasgow Ultrasound Enthesitis Scoring System (GUESS) by observers who were blinded to the diagnosis or the clinical findings in the patients. The total mean GUESS score for patients with DISH was 14.12 ± 5.2 and for patients without DISH 5.32 ± 4.99 (P < 0.0001). Univariate logistic regression analysis found a strong association between the GUESS and the probability of having DISH (P < 0.0001). The area under the ROC curve (AUC) revealed that the GUESS accuracy in diagnosing DISH was 88.53% with sensitivity and specificity of 92 and 70.6%, respectively, at a cutoff value of 6.36. A stepwise logistic regression analysis of the statistically significant items in the GUESS isolated four items, and the presence of either all of them or the first three items yielded the likelihood of having DISH to be 98.8 and 90.6%, respectively. The GUESS and the stepwise logistic regression analysis of the GUESS items demonstrated a high likelihood of having DISH. MSUS might help to identify entheseal changes in DISH. Further studies are needed to confirm these results.

Doença de Forestier e suas implicações otorrinolaringológicas: revisão de literatura / Forestier's disease and its implications in otolaryngology: literature review

Introdução: A Doença de Forestier afeta a coluna vertebral, preferencialmente em homens Coluna vertebral; idosos. Não é rara, mas é frequentemente não reconhecida e pode levar a relevante morbimortalidade. Quando acomete a coluna cervical, pode gerar importantes manifestações otorrino-Transtornos de laringológicas. Objetivo: Destacar os sintomas faringolaríngeos da doença de Forestier. Método: Revisão da literatura nas bases de dados Web of Knowledge, PubMed e SciELO e entre os dez periódicos de maior número de citações na área de otorrinolaringologia e também busca manual por publicações nas listas de referências dos artigos selecionados, principalmente os de cunho histórico. Resultados: A doença não tem etiologia clara. Os sintomas das complicações são mais exuberantes que os da doença propriamente dita. A disfagia é o sintoma cervical mais conhecido. Outros sintomas discutidos são a apneia do sono, globus faríngeo, tosse, disfonia, dispneia, otalgia reflexa e sintomas medulares compressivos. O diagnóstico é efetuado com apropriado estudo radiológico. O tratamento é baseado em estratégia conservadora. Pacientes com disfagia refratária e comprometimento respiratório podem ser submetidos a tratamento cirúrgico. Conclusão: A doença de Forestier deve ser suspeitada em pacientes idosos com os principais sintomas das complicações, os quais são comuns na prática otorrinolaringológica, para o início precoce de acompanhamento multidisciplinar. .

Introduction: Forestier's disease affects the spinal column of primarily elderly men. It is not rare, but it is often undiagnosed and can lead to significant morbidity and mortality. When it affects the cervical spine, it can result in important otorhinolaryngological manifestations. Objective: To analyze the pharyngeal and laryngeal symptoms of the Forestier's disease. Methods: Literature review of the Web of Knowledge, PubMed, and SciELO databases and of the ten most frequently cited journals in the field of otorhinolaryngology. Additionally, a manual search was performed for publications in the reference lists of selected articles, mostly those of a historical nature. Results: The etiology of the disease is still unclear. Symptoms of complications are more significant than the disease itself. Dysphagia is the most common cervical symptom and has several involved mechanisms. Other symptoms are sleep apnea, pharyngeal globus, coughing, dysphonia, dyspnea, otalgia, and medullary compression. The diagnosis is verified by appropriate radiological study. Treatment is based on a conservative strategy. Patients with refractory dysphagia and respiratory impairment can be surgically treated. Conclusion: Forestier's disease should be suspected in elderly patients with the major symptoms of complications, which are common in otorhinolaryngology practice and when identified, a multidisciplinary approach should be instituted as soon as possible. .

Síndrome de Forestier: Reporte de un caso y revisión de la literatura / Forestier syndrome: Case report and literature review

Introducción: El síndrome de Forestier, también conocido como hiperostosis esquelética idiopática difusa, es una enfermedad de etiología desconocida, caracterizada por aumento en la masa ósea de diferentes partes del cuerpo, responsable de cervicalgia y dorsalgia crónicas. Métodos: Se presenta el caso de un paciente masculino de 40 años con diagnóstico mediante estudios de imagen de síndrome de Forestier desde el 2006. Inició su cuadro clínico con lumbalgia de intensidad progresiva irradiada a miembro pélvico derecho asociado a rigidez matutina de espalda. Se le realizó cirugía de columna lumbar en Junio de 2005 (laminotomía y disquectomía L4-L5, espaciadores intersomáticos rellenos de hueso y colocación de espaciador interespinoso dinámico L4-L5) con poca mejoría postquirúrgica, nosotros iniciamos tratamiento médico antiinflamatorio como se describe en la literatura presentando una evolución satisfactoria. Discusión: Este padecimiento se reporta como una entidad poco frecuente, por lo cual el médico no lo tiene presente dentro de sus diagnósticos diferenciales; se presenta con disfagia como síntoma principal además de condicionar cervicalgia o lumbalgia debido a hipertrofia y osificación ligamentaria, aumento de la masa ósea con la consiguiente formación de puentes óseos. El dolor lumbar o cervical es una causa frecuente de consulta médica neuroquirúrgica. Conclusiones: La finalidad de este trabajo es presentar un caso poco habitual de dolor lumbar aumentando la sensibilidad diagnóstica de este padecimiento y evitar así una posible cirugía innecesaria; se complementó además con una revisión de la literatura.

Introduction: Forestier syndrome, also known as diffuse idiopathic skeletal hyperostosis, is a disease of unknown etiology, characterized by an increase in the bone mass in different parts of the body, which results in chronic cervical and back pain. Methods: We report the case of a male 40 year-old patient with a diagnosis of Forestier syndrome in 2006 based on imaging studies. The clinical picture began with lumbar pain of progressive intensity irradiating to the right pelvic limb and associated with morning spine stiffness. Lumbar spine surgery was performed in June 2005 (laminotomy and L4-L5 diskectomy, interbody spaces filled with bone, and a dynamic interspinous spacer in L4-L5), but there was little postoperative improvement. We instituted antiinflammatory medical treatment as described in the literature and the patient did fine. Discussion: This condition is reported as an infrequent entity leading to physicians not considering it within their initial differential diagnoses. It presents with dysphagia as the main symptom, together with neck or lumbar pain due to ligament hypertrophy and ossification, and increased bone mass leading to the formation of bone bridges. Lumbar or cervical pain is a frequent cause for visiting the neurosurgeon. Conclusions: The purpose of this paper is to present an unusual case of lumbar pain, which increases the diagnostic sensitivity of this disease, and may contribute to avoid unnecessary surgery. This was complemented with literature review.