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Table of Contents
CLINICOPATHOLOGICAL PEARLS
Year : 2020  |  Volume : 3  |  Issue : 2  |  Page : 139-141

DISHphagia and DISHphonia DISHguised as a stroke


Department of Internal Medicine, King Abdulaziz Medical City, National Guard Health Affairs, Riyadh, Saudi Arabia

Date of Submission14-Oct-2019
Date of Decision02-Dec-2019
Date of Acceptance16-Dec-2019
Date of Web Publication02-Apr-2020

Correspondence Address:
Abdulrahman Khalid Alfadhel
PO Box 1291, Ar Rawdhah, Khurias Road, Riyadh
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JNSM.JNSM_47_19

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  Abstract 


Diffuse idiopathic skeletal hyperostosis (DISH) is a noninflammatory condition that usually affects the elderly patients with several metabolic derangements. It is characterized by symptoms that mimic more sinister conditions such as stroke or malignancy. Indeed, this diagnosis can easily be “DISHmissed” if not considered in the differential diagnosis. Dysphagia is the most common symptom reported by patients with DISH. Always due to cervical osteophytes, dysphagia due to DISH has been described as DISHphagia. Extrapolation of this terminology to DISHphonia, DISHpnea, DISHpepsia, and DISHesthesia may increase awareness of the plethora of symptoms that may be caused by DISHplacement of anatomy. This is clinically significant because the prevalence of DISH is likely to increase, and DISH can be managed with conservative, medical, and surgical therapies.

Keywords: Diffuse idiopathic skeletal hyperostosis, dysphagia, stroke, swallowing


How to cite this article:
Alfadhel AK, Almanea AK, Rajendram R, Alsaad AG. DISHphagia and DISHphonia DISHguised as a stroke. J Nat Sci Med 2020;3:139-41

How to cite this URL:
Alfadhel AK, Almanea AK, Rajendram R, Alsaad AG. DISHphagia and DISHphonia DISHguised as a stroke. J Nat Sci Med [serial online] 2020 [cited 2020 Jun 4];3:139-41. Available from: http://www.jnsmonline.org/text.asp?2020/3/2/139/275400




  Introduction Top


Diffuse idiopathic skeletal hyperostosis (DISH), first described as senile ankylosing hyperostosis by Forestier and Rotes-Querol, causes noninflammatory ossification and calcification of spinal ligaments.[1],[2] The abnormal ossification in Forestier disease disrupts the anatomy causing a plethora of symptoms including odynophagia, dysphagia, aspiration, dysphonia, and airway obstruction.[3],[4],[5]

Recent studies report more peculiar presentations such as sore throat, hoarseness, and total vocal cord paralysis.[6],[7] These presentations often divert clinicians to consider any of a vast array of alternative diagnoses. This exposes patients to a battery of unnecessary investigations, prolongs admission to hospital, and often results in diagnostic error. This is particularly concerning because DISH can be managed with conservative, medical, and surgical therapies. To highlight this, we, therefore, describe the presentation of a patient with cervical DISHphagia who was initially misdiagnosed with a stroke.


  Case Report Top


An 87-year-old obese Saudi man with hypertension, dyslipidemia, peripheral vascular disease, and hyperuricemia was admitted to hospital with “sudden onset” dysphagia to solids and liquids, which began 15 days before this presentation. This was associated with hoarseness of voice. His symptoms had been ascribed to a stroke at another hospital, although computed tomography (CT) of the brain was normal. Physical examination also revealed two smooth firm 1 cm × 1 cm mobile nodules on the right lower leg, an 8 cm × 6 cm ulcer on the medial aspect of the right thigh, packed with herbal medication and right inguinal lymphadenopathy. Full neurological examination, including assessment of the cranial nerves, was normal except for the patient's hoarse voice and inability to chew because of pain. However, there was oropharyngeal dysphagia with an obvious impediment to the elevation of the larynx on swallowing. On bedside swallowing assessment, the patient tolerated liquids well but had mild dysphagia with puree and so refused to try solids.

Imaging of the neck CT [Figure 1] revealed that the posterior pharynx was indented by exuberant anterior osteophytosis of C2, C3, C4, and C5 vertebrae. Anterior spinal surgery was not appropriate. The risk of perioperative mortality and morbidity was high, independent of the coincident diagnosis of metastatic melanoma. Hence, the DISHphagia was managed conservatively. He was advised on head maneuvers to facilitate swallowing. Liquid and pureed diet was recommended, and the formulations of his medications were changed from tablets to syrups.
Figure 1: (a and b) Imaging of the neck computed tomography [Figure 1] revealed that the posterior pharynx was indented by exuberant anterior osteophytosis of C2, C3, C4, and C5 vertebrae

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Investigations also revealed anemia and iron and folate deficiency. A chest X-ray revealed a 1 cm round pulmonary nodule. Chest CT revealed bilateral nodules of different sizes. The presence of right inguinal lymphadenopathy was confirmed with CT, and biopsy subsequently demonstrated metastatic melanoma. The patient was, therefore, also referred to oncology for the consideration for chemotherapy.


  What Is the Diagnosis? Top



  Discussion Top


DISH is characterized by superfluous hyperostosis of the anterolateral spinal column that expedites ankylosis and ossification of spinal ligaments. Epidemiological data on DISH are scarce, which may reflect the lack of a clear definition of DISH. However, some data on the prevalence of DISH are presented in [Table 1].
Table 1: The prevalence of diffuse idiopathic skeletal hyperostosis

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Dysphagia is the most common symptom and is reported by 15%–25% of patients with DISH. While always due to cervical osteophytes,[16],[17] there is no correlation between the size of the osteophytes and symptoms. However, age does correlate with symptom severity.[8],[18]

Patients with DISHphagia will initially cope subconsciously by adjusting the position of their neck while swallowing. Observation of this maneuver narrows the differential diagnoses substantially. This may also explain reports of “sudden onset” DISHphagia when the patient eventually consciously realizes that they must change the consistency of the food they eat.

These conscious and subconscious strategies often delay presentation until the patient can no longer cope with severe and often atypical symptoms. For example, one case report described vocal cord paralysis as a late presentation of DISH.[7] Such atypical presentations raise the suspicion of more sinister diagnosis such as stroke or malignancy. This often leads to a battery of unnecessary investigations and prolongs admissions. Misdiagnosis can be avoided by taking a careful history and performing a thorough physical examination to exclude more sinister diagnoses.

Risk factors for DISH include age, diabetes, cardiovascular diseases, hypertension, and obesity.[19],[20] As the prevalence of these risk factors is increasing, the prevalence of DISH is also likely to increase. Hence, clinicians should have a high index of suspicion for DISH in patients presenting with symptoms that may be related to the displacement of normal anatomy.

Anatomical imaging (e.g., X-ray or CT of the spine) is required to confirm the diagnosis. Plain X-ray has a high sensitivity and specificity for the diagnosis of DISH. However, the sensitivity of CT is higher than that of X-ray alone.[14] This anatomical assessment can be complemented such functional assessment as using barium swallow, videofluoroscopy, functional endoscopic evaluation of swallowing, nasopharyngoscopy, or laryngoscopy complements.[18],[21]

In the vast majority of cases, the initial management should be conservative. Specific advice from speech therapists, swallowing therapists, and dietary modifications may be guided by the functional diagnostic assessment. While nonsteroidal anti-inflammatory drugs and steroids may be beneficial, osteophytectomy is the definitive treatment. The surgical option is reserved for severe cases due the possibility of severe complication including esophageal perforation and injury of the superior and recurrent laryngeal nerve.[18],[21]

The term DISHphagia has been coined to describe dysphagia due to DISH.[22] However, almost any symptom can be caused by DISH. We, therefore, advocate the extrapolation of this terminology to include DISHphonia, DISHpnea, DISHpepsia, and DISHesthesia. This would highlight the plethora of other symptoms that may be caused by DISH. The description of the pathophysiology as DISHplacement of anatomical structures will help both physicians and patients understand the cause of the symptomatology and the approaches to treatment.

Final diagnosis

DISH causing dysphagia.


  Clinicopathological Pearls Top


  1. DISH is a common condition characterized by superfluous hyperostosis of the anterolateral spinal column
  2. Dysphagia is the most common symptom reported by patients with DISH
  3. Many symptoms can occur as a result of the displacement of normal anatomy caused by DISH
  4. The use of terminology such as DISHphonia, DISHpnea, DISHpepsia, and DISHesthesia would highlight the plethora of symptoms which may be caused by DISH
  5. The description of the pathophysiology as DISHplacement of anatomical structures may facilitate understanding of the etiology of the symptomatology and the approach to treatment.


Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Forestier J, Rotes-Querol J. Senile ankylosing hyperostosis of the spine. Ann Rheum Dis 1950;9:321-30.  Back to cited text no. 1
    
2.
Mader R, Verlaan JJ, Eshed I, Bruges-Armas J, Puttini PS, Atzeni F, et al. Diffuse idiopathic skeletal hyperostosis (DISH): Where we are now and where to go next. RMD Open 2017;3:e000472.  Back to cited text no. 2
    
3.
Mader R, Verlaan JJ, Buskila D. Diffuse idiopathic skeletal hyperostosis: Clinical features and pathogenic mechanisms. Nat Rev Rheumatol 2013;9:741-50.  Back to cited text no. 3
    
4.
Verlaan JJ, Boswijk PF, de Ru JA, Dhert WJ, Oner FC. Diffuse idiopathic skeletal hyperostosis of the cervical spine: An underestimated cause of dysphagia and airway obstruction. Spine J 2011;11:1058-67.  Back to cited text no. 4
    
5.
Thompson C, Moga R, Crosby ET. Failed videolaryngoscope intubation in a patient with diffuse idiopathic skeletal hyperostosis and spinal cord injury. Can J Anaesth 2010;57:679-82.  Back to cited text no. 5
    
6.
Pulcherio JO, Velasco CM, Machado RS, Souza WN, Menezes DR. Forestier's disease and its implications in otolaryngology: Literature review. Braz J Otorhinolaryngol 2014;80:161-6.  Back to cited text no. 6
    
7.
Goico-Alburquerque A, Zulfiqar B, Antoine R, Samee M. Diffuse idiopathic skeletal hyperostosis: Persistent sore throat and dysphagia in an elderly smoker male. Case Rep Med 2017;2017:2567672.  Back to cited text no. 7
    
8.
Cassim B, Mody GM, Rubin DL. The prevalence of diffuse idiopathic skeletal hyperostosis in African blacks. Br J Rheumatol 1990;29:131-2.  Back to cited text no. 8
    
9.
Weinfeld RM, Olson PN, Maki DD, Griffiths HJ. The prevalence of diffuse idiopathic skeletal hyperostosis (DISH) in two large American Midwest metropolitan hospital populations. Skeletal Radiol 1997;26:222-5.  Back to cited text no. 9
    
10.
Kim SK, Choi BR, Kim CG, Chung SH, Choe JY, Joo KB, et al. The prevalence of diffuse idiopathic skeletal hyperostosis in Korea. J Rheumatol 2004;31:2032-5.  Back to cited text no. 10
    
11.
Holton KF, Denard PJ, Yoo JU, Kado DM, Barrett-Connor E, Marshall LM, et al. Diffuse idiopathic skeletal hyperostosis and its relation to back pain among older men: The MrOS Study. Semin Arthritis Rheum 2011;41:131-8.  Back to cited text no. 11
    
12.
Zincarelli C, Iervolino S, Di Minno MN, Miniero E, Rengo C, Di Gioia L, et al. Diffuse idiopathic skeletal hyperostosis prevalence in subjects with severe atherosclerotic cardiovascular diseases. Arthritis Care Res (Hoboken) 2012;64:1765-9.  Back to cited text no. 12
    
13.
Kagotani R, Yoshida M, Muraki S, Oka H, Hashizume H, Yamada H, et al. Prevalence of diffuse idiopathic skeletal hyperostosis (DISH) of the whole spine and its association with lumbar spondylosis and knee osteoarthritis: The ROAD study. J Bone Miner Metab 2015;33:221-9.  Back to cited text no. 13
    
14.
Hirasawa A, Wakao N, Kamiya M, Takeuchi M, Kawanami K, Murotani K, et al. The prevalence of diffuse idiopathic skeletal hyperostosis in Japan – The first report of measurement by CT and review of the literature. J Orthop Sci 2016;21:287-90.  Back to cited text no. 14
    
15.
Mori K, Kasahara T, Mimura T, Nishizawa K, Nakamura A, Imai S. Prevalence of thoracic diffuse idiopathic skeletal hyperostosis (DISH) in Japanese: Results of chest CT-based cross-sectional study. J Orthop Sci 2017;22:38-42.  Back to cited text no. 15
    
16.
Bansilal S, Castellano JM, Fuster V. Global burden of CVD: Focus on secondary prevention of cardiovascular disease. Int J Cardiol 2015;201 Suppl 1:S1-7.  Back to cited text no. 16
    
17.
Mata S, Fortin PR, Fitzcharles MA, Starr MR, Joseph L, Watts CS, et al. A controlled study of diffuse idiopathic skeletal hyperostosis. Clinical features and functional status. Medicine (Baltimore) 1997;76:104-17.  Back to cited text no. 17
    
18.
Seidler TO, Pèrez Alvarez JC, Wonneberger K, Hacki T. Dysphagia caused by ventral osteophytes of the cervical spine: Clinical and radiographic findings. Eur Arch Otorhinolaryngol 2009;266:285-91.  Back to cited text no. 18
    
19.
Westerveld LA, van Ufford HM, Verlaan JJ, Oner FC. The prevalence of diffuse idiopathic skeletal hyperostosis in an outpatient population in the Netherlands. J Rheumatol 2008;35:1635-8.  Back to cited text no. 19
    
20.
Mader R, Lavi I. Diabetes mellitus and hypertension as risk factors for early diffuse idiopathic skeletal hyperostosis (DISH). Osteoarthritis Cartilage 2009;17:825-8.  Back to cited text no. 20
    
21.
Lecerf P, Malard O. How to diagnose and treat symptomatic anterior cervical osteophytes? Eur Ann Otorhinolaryngol Head Neck Dis 2010;127:111-6.  Back to cited text no. 21
    
22.
Curtis JR, Lander PH, Moreland LW. Swallowing difficulties from “DISH-phagia“. J Rheumatol 2004;31:2526-7.  Back to cited text no. 22
    


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