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Lingual Tonsil Hypertrophy Producing

Obstructive Sleep Apnea


Ahmet Dundar, MD; Adnan Ozunlu, MD; Murat Sahan, MD; Fuat Ozgen, MD

The hypertrophy of the lingual tonsil is a rare oc- identification of the nature of apnea and the determi-
currence in adults. This disorder may cause obstruc- nation of physiopathologic changes due to apnea.
tive sleep apnea (OSA) and also may lead to a life-
threatening obstruction of the upper airway. Surgical removal of the lingual tonsils is a thera-
Diagnosis of lingual tonsil hypertrophy in patients peutic option in patients with recurrent infection.
with OSA requires a careful examination by ad- However, when chronic adenotonsillitis, adenotonsil-
vanced technologic methods. The sleep apnea is suc- lar hypertrophy, and especially lingual tonsil hyper-
cessfully treated by lingual tonsillectomy, with or trophy are associated with OSA, surgery is absolutely
without tracheotomy. indicated. Lingual tonsillectomy is performed with the
In this study the case of an adult with OSA patient under general anesthesia, using various tech-
caused by lingual tonsil hypertrophy which was niques including sharp dissection, snare, cryosurgery,
treated with lingual tonsillectomy is presented, and or the CO, laser in children or adults. The tracheoto-
the diagnosis and the treatment procedure of such my is carried out if necessary.
cases are discussed.
In this study a report concerning an adult patient
LARYNGOSCOPE,
1061167-1169,1996 with OSA caused by lingual tonsil hypertrophy is pre-
sented, and the diagnosis and the treatment of such
INTRODUCTION cases are discussed by reviewing the literature.
Obstructive sleep apnea (OSA)is a clinical entity
occurring as a consequence of many anatomic and
physiologic diseases of the upper airway. One of the CASE REPORT
causes is lingual tonsil abnormality such as hyper- A 58-year-old man presented with a 3-year history of
tropy, hyperplasia, or lingual tonsillitis. Hypertrophy sleep disturbance and a 2-month history of pain in the
of the lingual tonsil was first described by Vesalius in suprahyoid region. At night, he awoke because of apneic
1543.The true incidence of lingual tonsillitis has been episode three or four times, and he snored intermittently
underestimated for the last hundred years. This fact and loudly. Symptoms were worse when he was supine. He
may be explained by the position of the lingual tonsils, had dysphagia that was eased by protruding his chin and ex-
since they are difficult to visualize on routine physical tending his neck. Swallowing provoked pain in the suprahy-
oid region. He had been treated with palatine tonsillectomy
examination.1 Improved visualization and physiologic in another hospital in 1961. He had not smoked in 10 years
evaluation of the upper respiratory tract made possi- and denied drinking alcoholic beverages.
ble by developing technology permitted detection of
disease in this region.2.3 On physical examination, about 5 x 4 cm of pale, soft,
extremely hypertrophic lingual tonsil tissue was noted on
The upper airway of patients with OSA has been the base of the tongue (Fig. 1).This was more evident when
evaluated anatomically and physiologically for the traction was applied to the tongue. Scar tissue from the for-
last two decades, using advanced techniques such as mer palatine tonsillectomy was observed between front and
direct and fiberoptic visualization, cephalometric back plicas. Indirect laryngoscopy revealed the hypertrophic
roentgenograms, fluoroscopy, computerized tomogra- lingual tonsil tissue extending into the aryepiglottic plicas
phy, magnetic resonance imaging, and polysomnogra- and vallecula, displacing the epiglottis posteriorly and pro-
ducing a nearly total obstruction of the glottis. Because of
phy.2 Polysomnography is especially important in the lingual tonsil hypertrophy and overhanging epiglottis, the
larynx could not be evaluated. After topical anesthesia
From the Departments of Otorhinolaryngology-Head and Neck Sur- with 2% pantocaine, fiberoptic laryngoscopic examination
gery (A.D., A.o., M.s.) and Psychiatry (F.o.), Gulhane Military Medical Acade- showed that the larynx was normal. During Muller’s ma-
my, Etlik-Ankara, Turkey.
neuver the hypopharynx was almost completely obstructed
Editor’s Note: This Manuscript was accepted for publication April 9,
1996.
because of hypertrophy of the lingual tonsil. Allergy was not
found. Cephalometry was normal. The patient underwent
Send Reprint Requests to Adnan Oziinlii, MD, Giilhane Military Med-
ical Academy, Department of Otolaryngology & Head and Neck Surgery, Et- polysomnographic studies in the preoperative and postoper-
lik-Ankara, 06018, Turkey. ative periods.

Laryngoscope 106:September 1996 Dundar et al.:Obstructive Sleep Apnea


1167
Fig. 2. On macroscopic study the right lingual tonsil is brown and 6.5
x 4.5x 1.5cm, and the left lingual tonsil is gray-brownand 6 x 4.5 x
1.2 cm after the surgery.

Fig. 1. Lingual tonsil tissue on the base of the tongue: pale, soft, and
about 5 x 4 cm. Extremely hypertrophic. (T = tongue; It = lingual the surgery but decreased to 12 within an 8-hour sleep after
tonsil.) the surgery. Preoperative and postoperative apnea condi-
tions are shown in Figure 3.
Surgical treatment was elected. The severity of the air- At 2 months after surgery the patient was completely
way compromise and the potential for postoperative airway relieved of his symptoms of sleep disturbance, snoring, and
distress led us to tracheotomy with local anesthesia. The pa- dysphagia. Scar tissue was noted on the tongue base.
tient subsequently underwent a lingual tonsillectomy with
general anesthesia, using sharp dissection and a Boyle
Davis mouth gag for exposure. Hemostasis was achieved
DISCUSSION
with bipolar cautery, and the surgery was concluded by plac- The lingual tonsil is lymphoid tissue located on
ing a nasogastric tube. There were no bleeding or airway the base of the tongue. It has no capsule. Recurrent in-
complications in the immediate postoperative period. On fection and hyperplasia of this tissue may be the caus-
macroscopic examination the right lingual tonsil was brown es of respiratory problems and, rarely, OSA. The
and its dimensions were 6.5 x 4.5 x 1.5cm, whereas the left
lingual tonsil was gray-brown and its dimensions were 6 X pathologic condition of lingual tonsil hypertrophy is
4.5 X 1.2 cm (Fig. 2). Lymphoid hyperplasia was evident on usually hyperplasia, but papillary hyperplasia is
microscopic study. Postoperatively the patient’s alimenta- rarely ~een.4~5 Although the lymphoid tissue in
tion was managed by the nasogastric tube for 5 days. The pa- Waldeyer’sring tends to decrease with advancing age,
tient was decannulated on the sixth postoperative day, and the lingual tonsil may increase in size. Research has
the tracheotomy was surgically closed. Clinically, his com- shown that the most important cause of lingual tonsil
plaint was dysphagia that persisted for a few days after the hypertrophy is the occurrence of compensatoryhyper-
surgery. His sleep returned to normal. plasia following adenotonsillectomy. It was first de-
The polysomnographic studies were conducted in the scribed by Brown (in 1899),175Elia6 reported in 1959
preoperative period and at the end of the first postoperative that 70% of 43 patients had lingual tonsil hypertrophy
month for 2 consecutive nights. The second nights of these after adenotonsillectomy. On the other hand, allergy,
studies were evaluated. The preoperative study showed a alcohol, smoke, and chronic infections are cited as oth-
91% sleep efficiency index, 20-minute initial sleep latency, er possible etiologic factors.5 The most common symp-
87-minute rapid eye movement (REM)latency, and 0%third toms of lingual tonsil enlargement are pain, dyspha-
and fourth sleep stage rate. Postoperative values were 94% gia, otalgia, and sensation of a lump in the throat.7
sleep efficiency index, absence of initial sleep latency, 37-
minute REM latency, and 12%third and fourth sleep stage Five cases of OSA due to lingual hyperplasia have
rate. Increasing third and fourth sleep stage rate and de- been recorded in the prior literature. These include a
creasing initial sleep latency after the surgery were consid- case of lingual tonsil hypertrophy and a case of large
ered as signs of improving sleep structure of the patient. Be- lingual cyst reported by Olsen et a1.8 A 12-year-oldgirl
fore the surgery the patient could not sleep deeply by night with Down’s syndrome was reported by Phillips and
because of apnea and felt tired in the day. After the surgery
these symptoms resolved. All the determined apneas were of
Rogers.1A 5-year-oldboy with lingual tonsil hypertro-
obstructive type. There were 22 apneic episodes of 40 sec- phy and a 9-year-old boy with Down’s syndrome were
onds and longer, all during REM sleep. Preoperatively the reported by Guarisco et al.7 Elia6 in a study of 43 pa-
apnea index was 45.5, and the longest apnea was 92 seconds. tients with lingual tonsil hypertrophy and Joseph et
Postoperativelythe apnea index was 2.5, and the longest ap- al.5 in a study of 11patients with lingual tonsillitis did
nea was 33 seconds. The incidence of apnea was 212 before not report any patients with OSA due to lingual tonsil-

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1168
100 accomplished by adopting various techniques includ-
90 ing sharp dissection, snare, cryosurgery, or the CO,
80
laser. Olsen et a1.8 reported the use of tracheotomy be-
IC 70
fore tonsillectomy in their cases. Joseph et al.5 noted
that they preferred nasotracheal intubation but per-
60
c
0 formed tracheotomy in one case because of respiratory
50
8 distress. Other authors preferred nasotracheal intu-
5 40 bation during the surgery because OSA was not pres-
.-
‘0
ent in their cases.1,7*8In other words, most of their
-e
0 30
20 parents were not faced with serious respiratory dis-
10 tress. We preferred to establish tracheotomy before
0 the surgery in our patient because of partial obstruc-
0-20 21-30 31-40 41-50 51+ tion. It was obvious that the tissue edema occurring in
the postoperative period would cause respiratory dis-
Apnea duration (sec) tress in the patient.
Fig. 3. Preoperativeand postoperative apnea conditions. In all reported cases treated with surgery, namely,
in patients with OSA, surgery resulted in a consider-
litis or lingual tonsil hypertrophy. Among these cases able improvement in regard to respiratory distress
and recurrent infections. Examination and laboratory
only one patient is a 13-year-oldyouths; the others are
findings confirmed an adequate upper airway interval
children. We could not find any case of lingual tonsil
in the postoperative period.1,5,7,8Jo
disease causing OSA in adults in the review of litera-
ture except the above-mentionedpatient described by
Olsen et a1.8 Consequently, one can state that the oc- CONCLUSION
currence of OSA due to lingual hyperplasia in adults is Lingual tonsil hypertrophy is a rare cause of OSA.
extremely rare. Nonetheless, lingual tonsil abnormalities may pro-
duce severe airway compromise or OSA in adults. The
Preoperative and postoperative evaluation of pa-
tients with OSA is very important. Polysomnography patient with OSA should be examined carefully to rule
out anatomic abnormalities such as lingual tonsil hy-
is important to document and characterize the ap-
pertrophy. A tracheotomy may be required before sur-
neas.2.9.10 In OSA apneic episodes of 40 seconds and
longer and apneic episodes in REM sleep are frequent. gery when a serious respiratory problem is present.
We found that apneic episodes of 40 seconds and
longer appeared 22 times in our patient, all recorded BIBLIOGRAPHY
during REM sleep. Besides apnea, the most important 1. PhilliDs DE. Rogers JH. Down’s svndrome with linrmal tonsil
problems in the patients with OSA are sleep distur- hypertrophy iroducing sleep ainea. J Laryngol6tol. 1988;
bance, tiredness, and snoring. After surgery, these pa- 102:1054-1055.
tients feel better because of tranquil sleep. We ob- 2. Shepard JW, Gefter WB, Guilleminault C, et al. Evaluation of
the upper airway in patients with obstructive sleep apnea.
served that our patient’s apnea index decreased, the Sleep. 1991;14:361-371.
longest apnea shortened, and the apnea number di- 3. Forrest Jv,Lester PD. Roentgenographic evaluation in lingual
minished after the surgery. The patient was comfort- tonsillitis. Arch Otolaryngol. 1973;97:482-483.
able and active, and his sleep was regular. 4. Schantz A, Goodman M, Miller D. Papillary hyperplasia of the
lingual tonsil. Arch Otolaryngol. 1972;95:272-273.
The differential diagnosis of lingual tonsil hyper- 5. Joseph M, Reardon E, Goodman M. Lingual tonsillectomy: a
trophy includes lingual thyroid tissue, thyroglossal treatment for inflammatory lesions of the lingual tonsil.
duct cysts, dermoid cysts, lymphagiomas, angiomas, LARYNGOSCOPE. 1984;94:179-184.
6. Elia JC. Lingual tonsillitis. A n n N YAcad Sci. 1959;82:52-56.
adenomas, fibromas, papillomas, lymphomas, squa- 7. Guarisco JL, Littlewood SC, Butcher I11 RB. Severe upper air-
mous cell carcinomas, and minor salivary gland tu- way obstruction in children secondary to lingual tonsil hy-
mors on the base of the tongue.5 pertrophy. Ann Otol Rhinol Laryngol. 1990;99:621-624.
8. Olsen KD, Suh KW, Staats BA. Surgically correctable causes of
The treatment of OSA due to lingual tonsil hyper- sleep apnea syndrome. Otolaryngol Head Neck Surg. 1981;
trophy consists of lingual tonsillectomy performed 89:726-731.
with the patient under general anesthesia. The surgi- 9. Rundell OH, Jones RK. Polysomnography methods and inter-
pretations. Otolaryngol Clin North Am. 1990;23:583-592.
cal technique should be chosen according to equip- 10. Rosenfeld RM, Green RP. Tonsillectomy and adenoidectomy:
ment, the patient’s clinical features, and the prefer- changing trends. Ann Otol Rhinol Laryngol. 1990;99:
ence of the surgeon. Lingual tonsillectomy can be 187-191.

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