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Case report
Forestier syndrome and obstructive sleep apnea: Surgical treatment D.K. Ribeiro ∗ , J.A. Pinto , G.S. Freitas Department of otolaryngology, núcleo de otorrinolaringologia e cirurgia de Cabec¸a e Pescoc¸o de São Paulo, São Paulo, Brazil
a r t i c l e
i n f o
a b s t r a c t
Keywords: Forestier Sleep apnea Skeletal hyperostosis Surgery
Introduction: We report a case of obstructive sleep apnea that occurred as a result of Forestier disease and describe the surgical treatment that was performed. Summary: The patient is a 56-year-old man who presented dysphagia for solids and liquids, snoring (score 10) and excessive daytime sleepiness for 5 years. On fiber optic laryngoscopy examination, there was interarytenoid edema and protrusion of the posterior wall of the larynx. The cervical X-Ray showed protrusion of intervertebral disc between C3–C5 (skeletal hyperostosis) and the polysomnography revealed apnea-hypopnea index (AHI) of 56 events/h. Surgery was performed by the otorhinolaryngology and orthopedic teams. The patient evolved with complete symptom resolution and an AHI of 3,9 events/h on the control polysomnography. Discussions: This is the first reported case of Forestier Syndrome (FS) associated with Obstructive Sleep Apnea (OSA) that was proposed surgical treatment and the patient evolved with complete symptom improvement. © 2018 Elsevier Masson SAS. All rights reserved.
1. Introduction
during examination with fiber optic laryngoscopy, he presented a supraglottic posterior wall protrusion of the larynx. Muller’s Maneuver in the hypopharynx (anteroposterior closing 4 + /4 sitting and lying) and he was classified as type III Fujita and Friedman II. Prompted X-ray of cervical-evidenced protrusion of intervertebral disc between C3–C5 (Fig. 1). On polysomnography, the apnea-hypopnea index (AHI) was of 56 events/h; O2 average saturation: 93%; minimum O2 saturation: 82%, during 12% of total sleep time the O2 saturation remained below 90% and rapid eye movement (REM) in 9% of the exam; evidencing severe OSA. The diagnosis was DISH associated with OSA and the proposed treatment was a surgical approach with removal of the osteophytes by the orthopedic and otorhinolaryngological (ENT) associated teams. A cervical approach was performed with resection of the osteophytes. Cervical X-Ray taken immediately after surgery-evidenced no injury (Fig. 2) three months after surgery, the patient showed full recovery and evolved with complete remission of dysphagia, neck pain and excessive daytime sleepiness. However, still remaining with mild snoring (score 4), Epworth 5, FOSQ-10: 18 and BMI: 28.5. Laryngoscopy showed an absence of supraglottic protrusion of the posterior wall of the larynx. Two years follow-up after surgery, the polysomnography showed IAH: 3,9 events/h; O2 average saturation: 93%, minimum O2 saturation: 89% and in 2,8% of total sleep time the O2 saturation remained below 90% with REM in 22% of the exam. The patient recovered fully and remained asymptomatic.
Diffuse Idiopathic Skeletal Hyperostosis (DISH), also called Forestier’s syndrome (FS) is a common skeletal disease of unknown etiology and pathophysiology [1]. This syndrome was first described by Forestier and Rotes-Querol in 1950 [2]. Later, in 1975, the acronym DISH was introduced by Resnick et al. [3]. This osteophytes growth in rare cases may facilitate airway collapse leading to OSA [4]. In non-obese individuals, airway and craniofacial abnormalities that narrow the upper airway may predispose respiratory disturbances during sleep [5]. The current study reports the first case of FS associated with OSA that after surgical treatment evolved with complete symptom improvement. 2. Case report A male patient, 56 years-old, complaining of progressive dysphagia to solids evolving to liquids, associated with cervical pain, loud snoring (score 10) and excessive daytime sleepiness for 5 years. Presented Epworth: 18 and Functional Outcomes of Sleep Questionnaire (FOSQ-10): 11. Physical examination showed no craniofacial alterations (Angle – I), palatine tonsils grade I, Mallampati grade II and Body Mass Index (BMI): 28. Moreover,
∗ Corresponding author. Department of otolaryngology, Alameda dos Nhambiquaras, 159, Moema São Paulo, São Paulo 04090-010, Brazil. E-mail address:
[email protected] (D.K. Ribeiro). https://doi.org/10.1016/j.anorl.2017.05.004 1879-7296/© 2018 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Ribeiro DK, et al. Forestier syndrome and obstructive sleep apnea: Surgical treatment. European Annals of Otorhinolaryngology, Head and Neck diseases (2017), https://doi.org/10.1016/j.anorl.2017.05.004
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Fig. 1. Lateral neck radiograph showing ossification anterior to C3, C4 and C5 vertebral bodies. Note the edema of the posterior hypopharyngeal wall in this phase.
3. Discussion There are three radiological criteria for the diagnosis of Forestier’s disease: bone bridge of at least four contiguous vertebral bodies, preservation of intervertebral disc height and absence of sacroiliac ankyloses. Clinically, the disease can be completely asymptomatic or manifest with pain, neck stiffness, myelopathy, dysphagia, globus, dysphonia, dyspnea and cough [6]. ENT symptoms are diverse and the most frequent complaints are discomfort when swallowing that can progress to dysphagia. Studies indicate that 2.4–5.4% of individuals over 40 years of age have Forestier’s disease. This condition is associated with Type 2 diabetes, obesity and it is also known to be related to gallstones, systemic hypertension, atherosclerotic vascular disease and other metabolic disorders [3]. OSA is known to be associated with changes in the anatomy of the upper airways. In patients with OSA, fat deposits in the lateral pharyngeal walls, is the most important sites of collapse during sleep [3]. In this case, it is highly likely that the obstruction of the upper airway is caused by bone mass that contributes to the occlusion of the hypopharynx during sleep. Analyses of the main publications show that a variety of therapies are available: in most cases, medical treatment is initiated with hygiene and diet. Intake of soft foods, semi-liquids or the use of food supplements are highly helpful solutions for the management of elderly patients with significant comorbidities [6]. Four case reports documented in the literature associate FS and OSA, all treated without a surgical procedure. First to report was Hughes et al. [7] in 1994, where a patient had a history of dysphagia and hoarseness for 12 months that after continuous positive airway pressure (CPAP) adaptation evolved with improvement of symptoms. Machado et al. [8] in 2003 described a case with limited cervical movement and severe OSA being proposed CPAP reporting significant clinical improvement, the same occurred with Kawauchi
Fig. 2. Postoperative radiologic result. Note that all bone excess has been removed at the level of C3, C4, and C5 vertebrae.
et al. [3] in 2012. In 2009 Ando et al. [9] indicated oral appliance for a mild OSA without cervical complaints with partial improvement of the symptoms. Regardless of the reports, all were male patients over 50 years old and only Kawauchi et al. [3] mentioned the possibility of a surgical procedure if there were other symptoms associated. Table 1 describes the case reports and their outcomes. Transcervical treatment includes anterolateral (AL) and posterolateral (PL) approaches. The AL way provides better exposure of the carotid sheath, but recurrent laryngeal nerve injury is expected to be more frequent. PL approach requires more retraction and damage to the sympathetic plexus is more common. The transoral approach has the advantages of not having skin incision and less risk of neurovascular injury [6]. The transcervical AL approach used in our patient was the procedure of choice with no complications.
4. Conclusion This was the first case of FS found in a patient with typical symptoms of OSA who after surgery evolved with complete remission of symptoms. The case shows how an X-ray of the lateral cervical spine may be useful in the diagnosis of patients with sleep apnea and dysphagia. In this case, Forestier’s disease appears to have been the cause of upper airway obstruction. Thus, sleep apnea due to DISH, although rare, should be kept in mind in the differential diagnosis of patients with OSA. Future studies need to be done in order to correlate the symptoms of DISH with OSA and surgical approach.
Please cite this article in press as: Ribeiro DK, et al. Forestier syndrome and obstructive sleep apnea: Surgical treatment. European Annals of Otorhinolaryngology, Head and Neck diseases (2017), https://doi.org/10.1016/j.anorl.2017.05.004
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Table 1 Description of case reports on Forestier syndrome associated with OSA. Source Hughes et al., 1994 [7]
Age/Gender 72 years/Male
Machado et al., 2003 [8]
59 years/Male
Ando et al., 2009 [9]
75 years/Male
Kawauchi et al., 2012 [3]
75 years/Male
Symptoms Painless dysphagia, and apnea Limitation of cervical mobility, hoarseness and excessive daytime sleepiness Snoring and cough during conversation Snoring and apnea
AIH Pre 40 events/h
Treatment CPAP
Outcome Improvement of OSA symptoms however remained with dysphagia Remained with cervical limitation but with improvement of OSA symptoms
AIH Post Not reported
38 events/h
CPAP
9.4 events/h
Oral appliance
Partial improvement of symptoms
6.1 events/h
35.5 events/h
CPAP
Sleep quality improved with CPAP, no other complaints
0.7 events/h
28 events/h
CPAP: continuous positive airway pressure; OSA: obstructive sleep apnea; AIH: apnea-hypopnea index.
Disclosure of interest The authors declare that they have no competing interest. References [1] Castellano DM, Sinacori JT, Karakla DW. Stridor and dysphagia in diffuse idiopathic skeletal hyperostosis (DISH). Laryngoscope 2006;116:341–4. [2] Ozgursoy OB, Salassa JR, Reimer R, Wharen RE, Deen HG. Anterior cervical osteophyte dysphagia: manofluorographic and functional outcomes after surgery. Head Neck 2010;32(5):588–93. [3] Kawauchi E, Yamagata T, Tohda Y. A case of Forestier disease with obstructive sleep apnea syndrome. Sleep Breath 2012;16:603–5.
[4] Ferguson KA, Ono T, Lowe A. The relationship between obesity and craniofacial structure in obstructive sleep apnea. Chest 1995;108:375–81. [5] Morrison DL, Launois SH, Isono S. Pharyngeal narrowing and closing pressure in patients with obstructive sleep apnea. Am Rev Respi Dis 1993;148:606–11. [6] Lacerf P, Malard O. How to diagnose and treat symptomatic anterior cervical osteophytes? Eur Ann Otorhinolaryngol Head Neck Dis 2010;127(3):111–6. [7] Hughes TAT, Wiles CM, Lawrie BW, Smith AP. Dysphagia and sleep apnoea associated with cervical osteophytes due to diffuse idiopathic skeletal hyperostosis (DISH). J Neurol Neurosurg Psychiatr 1994;57:384. [8] Machado A, Winck JC, Almeida J. Obstructive sleep apnea syndrome associated with cervical osteophyte due to diffuse idiopathic skeletal hyperostosis – clinical case. Rev Port Pneumol 2003;9(5):427–33. [9] Ando E, Ogawa T, Shigeta Y, Hirai S, Ikawa T, Ishikawa C, et al. A case of obstructive sleep apnoea with anterior cervical osteophytes. J Oral Rehab 2009;36:776–80.
Please cite this article in press as: Ribeiro DK, et al. Forestier syndrome and obstructive sleep apnea: Surgical treatment. European Annals of Otorhinolaryngology, Head and Neck diseases (2017), https://doi.org/10.1016/j.anorl.2017.05.004