II

Obstructive

sleep apnoea syndrome

A. G. Smyth, R. P. Ward-Booth,

_-

I

presenting

as lingual ulceration

A. F. Henderson

Department of Oral and Maxillofacial Surgery, Sunderland District General Hospital, Sunderland Department of Chest Medicine, The Royal Infirmury, Sunderland

and

SUMMARY. Two patients with obstructive sleep apnoea syndrome are reported where the initial presenting complaint was of lingual ulceration. This unusual presentation has not been reported previously. Both patients experienced frequent apnoeic episodes during sleep with a profound fall in the arterial oxygen saturation. It is postulated that the lingual ulceration resulted from repeated trauma to the tongue by the teeth as the patient made violent inspiratory efforts at the termination of an apnoeic episode. The diagnosis of sleep apnoea syndrome was based upon suggestive symptoms of snoring, morning fatigue and day-time somnolence plusa minimum of 15 apnoeic episodes per hour of sleep. The first-line investigations of this condition are available in all district general hospitals and a diagnosis of sleep apnoea syndrome obtained. Referral to a regional sleep study centre may be appropriate prior to the commencement of therapy. Management is predominantly medical, consisting of weight loss and the administration of nocturnal nasal continuous positive airways pressure.

INTRODUCTION

Britain and certainly it was a very rare diagnosis more than 10 years ago (Shapiro et ul., 1981). The prevalence of the condition is estimated to be about 2% of the adult population (Gislason et al., 198X). Sleep apnoea is defined as a cessation of breathing during sleep which lasts for 10 s or more (Stradling & Phillipson, 1986) while sleep hypopnoea is defined as a 50% or more reduction in tidal volume for at least 10 s (Gould et ul., 1988). The episodes of apnoca/hypopnoea must be accompanied by a reduction in the arterial oxygen saturation of at least 4% below the baseline value. The apnoea/hypopnoea index (AHI) is the number of apnocic or hypopnoeic episodes that occur per hour of sleep. ‘Obstructive’ sleep apnoea is caused by upper airway obstruction and is accompanied by continued respiratory effort. ‘Central’ sleep apnoea is not accompanied by continued respiratory effort. Many patients with this condition feature both types. The characteristic symptoms of this condition are snoring, excessive daytime sleepiness with occasional episodes of falling asleep during the day, morning tiredness, fatigue and headaches. Other less common symptoms are enurcsis and decreased libido. Whytc et al. (1989) also alluded to the frequent symptom of awakening from sleep with attacks of choking which occurred in 21 of their SO patients with sleep apnoea syndrome. The diagnosis of sleep apnoea syndrome is established by suggestive symptoms plus confirmed evidencc of at lcast 15 episodes of apnoea/hypopnoca occurring per hour of sleep (Gould et al., 1988)) that is AH1 >1.5. The sleep apnoea syndrome predominantly affects males and there is also a strong relationship with obesity defined as a body mass index (BMI) greater

The sleep apnoea/hypopnoea syndrome is a condition in which recurrent episodes of apnoea occur during sleep, resulting in frequent periods of hypoxacmia and restless sleep. Although the medical profession have recognised this condition .for only the last 50 years, descriptions of sleep apnoea appear in antiquity. Dionysius, born in 360 BC was a tyrant of Heracleia in Pontus in the era of Alexander the Great. A description of him appears in Smith’s book of 1880. ‘He was an unusually fat man, which increased at length to such a degree that he could take no food which was not introduced into his stomach by artificial means. At last, however, he was choked by his own fat.’ Dionysius had difficulty breathing during sleep and the treatment ordered by the physicians was the insertion of long, fine needles which were thrust through his sides into his belly whenever he fell into a sound sleep, resulting in a state of thorough arousal! Later Charles Dickens described a character with classical features; we would now recognisc him as suffering from the sleep apnoea syndrome. ‘Damn that boy,’ said the old gentleman, ‘he’s gone to sleep again.’ ‘Very extraordinary boy, that,’ said Mr. Pickwick, ‘does he always sleep in this way?’ ‘Sleep!’ said the old gentleman, ‘he’s always asleep. Goes on errands fast asleep, and snores as he waits at table.’ ‘How very odd!' said Mr. Pickwick. (Referring to ‘the fat boy’ Joe in The Pickwick Papers by Charles Dickens.) Only recently has the condition

been diagnosed in 263

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than 27 Kg/m*. Occasionally an underlying systemic disorder occurs in association with sleep apnoea such as hypothyroidism, acromegaly, uracmia or sickle cell anacmia. Treatment of these disorders usually results in abolition of the sleep apnoea syndrome, or at least a significant improvement. As a direct consequence of the profound nocturnal dips in arterial oxygenation, these patients may develop polycythaemia, pulmonary hypertension and heart failure. Ultimately features of hypertension and ischaemic heart disease may supervene (Hing et al., 1990). Untreated sleep apnoea syndrome is associated with an increased mortality, especially in older patients with an apnoea index greater than 20 per h (He et al., 1988; Partinen et al., 1988). The mortality figures in these retrospective studies were 11% and 13% respectively during a 5-year period.

Fig. 2-Ulcerated

right lateral margin of tongue (patient

Fig. 3 -Ulcerated

left lateral margin of tongue (patient

1)

Case reports Cuse 1

A 4%year-old male butcher weighing 135 kg (Fig. 1) was referred by his medical practitioner to the Oral and Maxillofacial Surgery Unit of Sundcrland District General Hospital with a 3-month history of ulceration of his tongue. The practitioner was concerned regarding the possible malignant nature of the lesion. The painful ulcers affected both sides of the tongue and were present continuously. The patient reported that the ulcers were often larger and more painful after awakening from sleep and that on occasions he had noticed the presence of blood on his pillow. Prior to this current episode there was no past history of oral ulceration. The ulcerated lateral margins of the tvngue are shown in Figures 2 and 3. I).

Traumatic ulceration was suspected and indeed biopsy of the ulcers revealed only chronic inflammatory changes. with no evidence of malignancy. Closer questioning revealed a history of poor, restless sleep with snoring, morning fatigue, daytime somnolence, headaches and episodes of apnoea during sleep as described by his wife. A diagnosis of sleep apnoea syndrome was considered and initial investigations included pulmonary function tests (Table 1) which did not reveal an obstructive pattern and early morning blood gases (Table 2) which revealed a significant degree of hypoxaemia. The diagnosis of sleep apnoea was confirmed following an overnight pulse oximetry study which produced a severely abnormal Table I Patient I

Results .~.

of pulmonary -_

butcher weighing 135 kg (patient

1).

tests (spiromctry).

.__.

.~

Fig. I-4%year-old

function

_..-

Forced expiratory volume in one second (FEV,) Forced vital capacity (FVC) Fl3,/FV(’ Funcrional residual capacity (FRC) ‘l‘o(al lung capacity (‘II.(‘) Residual volume (RV) RVil-1-C: -_. _

_.

.--_

Measured (Predicted mean in brackets)

2.751. 3.MlL 76.0% 2.h4L 6. 131. 2.471. 40.22% --.._

~

(3.47) (4.25) (r(S)

(6.30) (I .9,x) (.3..3) .__

Ohstruclivc Table 2-

Arterial

POZ “CO? 1‘COz ABE

Table 3-

-

265

ulceration

DISCUSSION

Results of lung function laboratory

-

svndrome

at present to suffer from intermittent lingual despite the provision of a soft mouthguard.

blood gas analysis (on air). Patient 1 7.39 6.56 KPa 7.73 KPa 29.9 MM/L 31.4 MM/I, 4.1 MM/L

P” PC02

sleep apnoea

-.

Duration of study (hours) Number of apnoeic episodes Apnoea index Mean SAOz (%) Maximum &saturation (%) Minimum SAO? (%) ‘%I time below 90”/0 saturation

_-

sleep study

Patient 6 459 76.5 90 30 60 50

1 -

Patient 2 3x: 77.4 xs 44 51 69

Results in both patients demonstrate severe obstructive sleep

apnoea.

trace with numerous episodes of apnoea associated with profound arterial oxygen desaturation. As a prelude to treatment, a full sleep study was subsequently performed when a total of 459 apnoeic episodes were recorded during a 6-hour sleeping period (Table 3). Thus an apnoeic index of 76.5 was obtained. The mean oxygen saturation (SaOz) was 90% with a minimum reading of 60% occurring during one apnoeic episode. These results were consistent with severe obstructive sleep apnoea. The patient was encouraged to lose weight and a trial course of protriptylinc was prescribed? but no improvement was seen. A trial of nasal continuous positive airways pressure (CPAP) was commenced which dramatically improved his clinical condition with fewer apnoeic episodes and resolution of the lingual ulceration. The patient was commenced on regular domiciliary CPAP.

Case 2 A 46year-old policeman, weighing 127 kg was referred with a similar history of chronic ulceration of his tongue suspected of malignancy. The painful ulceration of his tongue had been present intermittently, occurring every few weeks for the previous year, with no previous history of oral ulceration. The patient confirmed that the ulceration was much worse on awakening from sleep. Examination revealed the presence of deep chronic ulceration of the lateral borders of the tongue. A biopsy supported the clinical diagnosis of traumatic ulceration of the tongue. Classical symptoms of sleep apnoea were present with loud snoring. poor quality sleep, headaches, fatigue and daytime somnolence. Indeed this latter symptom was so severe that on one occasion he fell asleep at the wheel of his police car! A sleep study was performed which confirmed severe obstructive sleep apnoea. The patient was observed to have a restless night’s sleep with a total of 388 apnocic episodes during a S-hour sleep period, giving a apnoeic index of 77.4 associated with severe oxygen desaturation (Table 3). Again weight loss was encouraged and a trial of nasal CPAP commenced. but this was unsuccessful as the patient could not tolerate the treatment. He is due to undergo surgery for a deviated nasal septum and hypertrophic turbinatcs to provide a better nasal airway. He continues

Contrary to earlier reports (Shapiro et al., 1981), the sleep apnoea syndrome is not a rare disorder in Britain. There is now no doubt that obstructive sleep apnoca results from abnormalities in the structure and function of the upper airway. However, structural abnormalities which may be correctable with surgery arc only present in 15% of patients with sleep apnoea (Hoffstein & Zamel, 1990). Methods of assessing the patency of the upper airway include the flow-volume curve, which is a simple inexpensive test but suffers from a low sensitivity, requiring at least a 50% reduction in upper airway diameter before abnormal loop patterns are seen (Miller & Hyatt, 1973). The standard lateral radiograph of the head can be assessed ccphalometrically (Partinen et al., 1988; Bacon et al., 1990) to localisc anatomical abnormalities which may be amenable to surgical correction. Conventional CT scans have not proved to be useful in assessing sleep apnoea patients, principally because they do not provide dynamic study of the airway; however, ultra-fast tine-CT may be useful in this respect (Calvin et al., 1989). The cross-sectional area of the upper airway can be measured using an acoustic reflection technique. An airway echogram can be produced giving area measurements from the mouth to the carina. This technique has been applied to sleep apnoea patients (Bradley et al., 1986; Rubenstein et al., 1988), but it is confined to only a few specialist laboratories and the technique also by-passes the nasopharyngeal airway which may be important in the aetiology of sleep apnoea. Many treatment modalities exist for sleep apnoea, but the efficacy of the treatment options vary widely. Patients with sleep apnoea are a danger when driving due to the daytime somnolence (George et al., 1987). Therefore they should be advised to avoid driving until satisfactory relief from symptoms is achieved. All patients who are overweight must be strongly advised to lose weight because this invariably produces symtomatic improvement and a dramatic reduction in the number of nocturnal apnoeic episodes (Harman et ul., 1982). Patients with obstructive sleep apnoca must also avoid alcohol, scdativcs, hypnotics and tranquillizers, including hospital prc-medication. About 15 years ago, the only effective treatment for obstructive sleep apnoea was a tracheostomy which reduces the morbidity and mortality from obstructive sleep apnoea but is inconvenient for the patient and has an associated morbidity with the procedure. However other medical and surgical treatment options arc now available. Protriptyline, a tricyclic antidepressant, is occasionally of value in reducing symptoms and improving

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arterial oxygen saturation. Protriptylinc suppresses the phase of rapid eye movement (REM) sleep, which is the stage of sleep during which the majority of apnoeic/hypopnoeic episodes occur and thereby reduces the apnoea/hypopnoca index. Acetazolamide, a respiratory stimulant, has also been studied, but the effects of both protriptyline and acctazolamide have been generally disappointing (Whyte et al. , 1988). The most impressive advance in the treatment of obstructive sleep apnoea was discovered by Sullivan et uf. (1981) who demonstrated complete abolition of symptoms in most patients with low continuous positive airway pressure (CPAP). A low pressure of between 2.5-20 cm H20 is applied by nasal mask. CPAP is thought to work by a ‘pneumatic splint effect’ which maintains the airway open by virture of the positive intra-luminal pressure. The compliance rate with CPAP is of the order of 80%. Nasal CPAP has also been shown to reduce the mortality from vascular complications (He et al., 1988). Surgical intervention for sleep apnoea is indicated in about 15% of cases who have a demonstrable abnormality of the upper airway. The most frequent surgical procedure performed is a uvulopalatopharyngoplasty, as described by Fujita et al. (1981). This involves excision of the uvula and tonsils with partial resection of the soft palate. The effectiveness of this procedure varies widely due to the different criteria applied by authors to assess outcome. For instance Conway et al. (1985) reported a successful outcome in 50% of their patients, but a postoperative reduction in the apnoea/hypopnoea index of SO% was considered a successful. reponse to treatment. It is difficult to recommend this form of surgical management when one takes into account the limited success of the procedure, the adverse effects upon speech and swallowing and that no reduction in mortality has been demonstrated following this operation. Orthognathic surgery for maxillary or mandibular hypoplasia has been successful in alleviating symptoms in some sleep apnoca patients. Indeed Riley et al. (1986) described a himaxillary advancement procedure for a patient with severe retrognathia which dramatically relieved the sleep apnoea syndrome. Later, Riley et al. (1989) presented 55 patients who were treated by mandibular osteotomy and hyoid myotomy suspension, with a good response in 67% of patients. The two patients presented here were both subsequently diagnosed as severe obstructive sleep apnoea syndrome, but the clinical presentation was unusual in that the main complaint was lingual ulceration. As both patients were unaware of any trauma to the tongue, it is reasonable to assume that the traumatic ulceration occurred during sleep, especially as the ulceration was noted to be worse after sleep. Obstructive sleep apnoea, like many chronic illnesses, often pursues a progressively deteriorating course and trauma within the mouth may only be associated with the more severe forms of the condition. This would explain why in our two patients the lingual ulceration had been present for a

finite period prior to presentation. Indeed. Whytc et al., 1989 stressed the importance of cpisodcs of nocturnal choking. Twenty one of 80 patients in their study complained of awakening from sleep with attacks of choking. We presume that trauma to the tongue from the teeth is prone to occur during these choking attacks as the patient makes vigorous efforts to breathe before adequate tone has returned to the upper airway which establishes patency and allows inspiration. Acknowledgements We would like to thank the Sleep Study Laboratory of Freeman ffospital, Newcastle Upon Tync. for the data shown m Table 3.

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60,115. Bradley,D.T.,Brown, I. G..Grossman.R. F.,%amel,N.. Martinez, D., Phillipson. E. A. & I loffstcin, V. (lY86). Pharyngealsizcinsnorers, non-snorersandpatientswith obstructivesleepapnea. NewE~~~lundJournulofMedicine.

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sleeo annoea svndromc

The Authors A.G.SmythMBBS,FDSRCS,FRCS Registrar DepartmentofOralandMaxillofacialSurgery Sunderland District General Hospital Kayll Road Sunderland SR4 7TP R. P. Ward BoothMBChB, FDSRCS, FRCS ConsultantOraland MaxillofacialSurgeon Depatmentof Oral and Maxillofacial Surgery Sunderland District General Hospital Kayll Road SunderlandSR4Tf’P A. F. HendersonMD, MA, MRCP

Consultant Physician Department of Chest Medicine The Royal Infirmary New Durham Road SunderlandSR27JE

CorrespondenceandrequestsforoffprintstoMrA.G.Smyth, Senior Registrar in Oral and Maxillofacial Surgery. Queen ElizabethHospital,Edgbaston,BirminghamB152TH

Paper received 16 July 19yl Accepted 29 November 191

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