Coexisting
Internal and External Laryngoceles REPORT
OF A CASE
STANLEY L. LANE, M.D.,D.D.s., BERNARD COHEN, M.D.AND CARLO IPPOLITO, M.D., New York, New York From tbe Ear, Nose and Tbroat Department, Hospital, New York, New York.
Fordbam
gIands are tubular with mixed mucous and serous ceIIs. Incidence. LaryngoceIe is a reIativeIy rare condition, onIy approximately ninety-six cases being reported in the Iiterature to date. It is of interest to note that of these, forty-five were reported in the Iast ten years (I947 to 1957). Ten cases were associated with carcinoma of the Iarynx, eight were compIicated by infection and four were of the combined internaIexterna1 type. The remaining cases were pure air cysts of the external or interna type. Cases of biIatera1 externa1 IaryngoceIes were reported, but there were no cases of biIatera1 interna IaryngoceIes. Etiology. The saccuIus from which IaryngoceIe formation stems is considered by some authors to be an atavistic anaIogue of the air sacs normaIIy encountered in the orangutan and the chimpanzee, iIIustrating developmenta1 changes of evoIutionary degeneration [~,6,15]. Others beIieve that abnormai increase in the intraIaryngea1 air pressure, such as in coughing, singing, pIaying wind instruments and gIass bIowing distends the saccuIus so that it projects beyond the confines of the Iarynx [4,6,7]. Another view is that the vaIveIike cIosure occurs in the saccuIus so that air is aIIowed in but not out. Distention of the saccuIus is thus caused, which uItimateIy Ieads to the formation of a IaryngoceIe. It is probabIy a combination of a11 the aforementioned factors-a Iong saccuIus, increased intraIaryngea1 pressure, as we11 as a check vaIve mechanism-that produces a IaryngoceIe. A true IaryhgoceIe shouId not be confused with a simiIar mass containing air caused by trauma to the Iarynx. In this
ENTRICULAR IaryngoceIe,
named and deby Virchow [I] in 1863, is a cystic diIatation of the sac&us or appendix of the ventricIe of Morgagni. CIinicaIIy, this condition was recognized in 1829 by Larrey, a surgeon in NapoIeon’s army of occupation in Egypt. He observed that many of the men reciting prayers from the ternpIe minar’ets were afHicted with tumors of the neck. These tumors, he noted, were air cysts in the soft tissues that eventuaIIy Ied to the symptoms of aphonia and venous compression. AnatomicaIIy, it was GaIen (300 A.D.) who first noted a sIit between the true and the faIse voca1 cords of the larynx. Morgagni (1741) described it in more detaiI, and this ventricIe bears his name. In 1837 HiIton [2] drew attention to the anterior upward projection of Morgagni’s ventricIe, a vertica1 bIind pouch known as the IaryngeaI saccuIus or appendix. EmbryoIogicaIIy, the ventricIe is formed by the spIitting of the common thyroarytenoid sphincter into two parts, a Iower interna segment and an upper externa1 segment. The upper segment becomes the faIse cord and the Iower becomes the true vocaI cord. A recess is left between the two, representing the ventricIe. A saccule is present at the end of the second feta1 month, remaining reIativeIy Iarge in the newborn, with regression occurring at the age of six years [J]. HistoIogicaIIy, the IaryngeaI ventricIe and the saccuIus are covered with ciIiated coIumnar epitheIium. The stroma contains Iymphoid ceIIs, singIy or in groups. The submucosa1
V scribed
American Journal of iurgery,
Volume 96, December, 1958
810
InternaI
and
ExternaI
instance air may be caught between the tissues and heId by a baII-vaIve arrangement, and graduahy enIarge to appear as a Iaryngocele. An air sac is formed in this manner and is not lined by mucous membrane but rather by the tissue in which it forms [4]. Another condition which may be confused with a laryngocele is a true proIapse of the ventricle, a condition which results from inflammatory edema or hypertrophy with bulging of the ventricle [8,z8]. Classifxation. Various authors have presented cIass&cations of IaryngoceIes [6,7,1 I, 13,211. CIassifications are based on etiology, relation to the thyrohyoid membrane and relation to the larynx; the cyst contents have also been used as a basis. From the observations on the case reported herein, the following classification is suggested: (I j Internal Iaryngocele: The presence of a cystic dilatation within the lumen of the larynx with the base arising from the interna waII of the ventricle of Morgagni. (2) ExternaI Iaryngocele: The presence of a cystic diIatation outside the larynx with the base arising from the externa1 wall of the ventricIe of Morgagni. (3) Combined internal and external Iaryngocele: Th e presence of a cystic dilatation into the lumen of the Iarvnx as we11 as a cystic dilatation external to the wall of the ventricle of Morgagni. The content of a true IaryngoceIe is air. The histoIogica1 preparation shows ciIiated columnar epitheIium, and mixed mucous and serous tubular gIands. LaryngoceIes may be asymp$ymptoms. tomatic. The internal IaryngoceIe can produce voice changes, varying from hoarseness to complete aphonia. Partial respiratory obstruction with dyspnea to complete obstruction of the airway may occur, necessitating emergency tracheotomy. The externa1 IaryngoceIe produces a swelling in the neck that increases in size on coughing or straining. In the mixed type a combination of the previously mentioned symptoms are present. LocaI discomfort or venous compression with resuhant headaches may occur. Diagnosis. Palpation reveaIs a soft eIastic mass that may give a hissing sound of escaping air in the throat on pressure [6]. The presence of any of the aforementioned symptoms as we11 as the presence of a tumor or mass in the neck is an indication for roentgenoIogica1
Laryngoceles
studies. These wiII reveaI a typical, sharpIy defined, radiolucent area at the IeveI of the hyoid bone that is ovoid in shape. Mirror Iaryngoscopy reveaIs a swelling above the true vocal cords that may enlarge on straining. Direct laryngoscopic examination wiI1 present the same trndings in more detail. Aspiration of air vviI1 cause cohapse of the swelling and confirm the diagnosis. Treatment. Asymptomatic Iaryngocele requires no treatment. A IaryngoceIe, interna or externa1, producing symptoms shouId be removed surgicaIIy through an external incision. InternaI remova through the Iaryngoscope and the injection of trichIoroacetic acid into the c>;st has been attempted but this shouId be discouraged. CASE
REPORT
A twenty-two year oId white man was admitted to the hospital from the outpatient department with a swelling on the Ieft side of the neck of approximately five months’ duration. The swelling had appeared suddenly during the course of a normal conversation and disappeared three days later. A month later it reappeared and persisted to the time of admission to the hospital. A week after the swehing reappeared the patient’s voice became hoarse, hnaIIy resulting in aphonia. No medica treatment was sought during this time. Family and past histories were non-contributory. The patient liked to sing. Physical examination revealed a normal-appearing maIe of moderate build with a moderate swelIing of the left cervical region between the angIe of the mandibIe and the thyroid cartilage. (Fig. IA.) The mass was soft and eIastic in consistency and immediateIy resumed its origina shape after release of pressure. On coughing or straining the swelling increased in size. There were no other abnormal physica findings. Laboratory examinations were normal. Mirror Iaryngoscopic examination showed that just beIow a normal epigIottis a large, smooth swelling was present on the left Iateral hypopharyngea1 wal1 extending down to and obIiterating the left vocal cord. The right cord was norma in appearance and motility. On direct laryngoscopy, as the epiglottis was elevated anteriorIy, a sausage-shaped, avascuIar, bulging mass was visibIe on the left side extending into the Ieft aryepigIottic foId. Upon deep expiration another smaller-sized mass was seen beIow the Iarger mass; this smaher mass had a bIuish discoloration on its surface. No openings were noted in either mass. There was barely enough space between the Ieft side and the right vocal
Lane, Cohen and IppoIito hyoid bone and the thyroid cartilage. The incision was carried down through the platysma muscIe and the cystic mass was located. By bIunt and sharp dissection the cystic mass was freed intact. The superior laryngeal nerve was located and isolated. The cyst was then separated from the hyoid bone and was found to arise from the region of the thyrohyoid membrane to the left of the midline. The thyrohyoid membrane in this area was not present. The cyst was dissected free and found to bulge under the inner surface of the left side of the thyroid cartilage. The base of the cyst attached to the laryngeal wall was then partially opened and through this opening an internal cyst appeared, dilating on expiration and collapsing on inspiration. (Figs. zA and B.) The internal cyst was firmly attached to the external cyst. The internal cyst had a wide neck, about I cm. in diameter. With gentle retraction of the hyoid bone and the thyroid cartilage enough exposure was obtained to resect both cysts. The interna cyst was resected at its neck and removed attached to the larger external cyst. The opening into the Iarynx was then closed with inverting continuous mattress sutures. The defect in the thyrohyoid membrane was covered by suturing the constrictor muscle to the edge of the thyroid cartilage. The wound was cIosed in layers with a Penrose drain brought out through the lower angle of the wound. Grossly, the specimen consisted of a bulbousshaped cyst, measuring 7 cm. in length and 4 cm. in its greatest diameter. (Fig. 3A.) The external surface was reddish in color and the wall of the cyst was thin. The inner surface was smooth and contained a yellowish secretion. The cavity of the cyst contained air. There was another small cyst, about 2 cm. in diameter, located at one poIe of the larger cyst. This cyst wall was thin and the inner surface was smooth. Microscopically, the lining of the Iaryngoceles showed layered ciliated epithelium in most areas. (Fig. 3B.) The walls of both cysts were composed of loose connective tissue with areas of perivascular lymphocytic infiltration. A few glandular structures were found within the walls, consisting of narrow acini lined by high columnar epithelium. There were some wider ducts lined with cuboidal epithelium and a Iayer of myoepithelial cells was present. The patient’s postoperative course was uneventful. The tracheotomy tube was removed on the fourth postoperative day. On the Fifth day laryngoscopic examination revealed a slightly edematous and reddened epiglottis. The vocal cords were normaI in color and motility. There was slight hypertrophy of the right ventricular band with marked visualization of the left vocal cord, indicating obliteration of the left ventricular
IA
FIG. I. A, preoperative view of patient showing location of external swelling. B, x-ray of neck showing cystic cavity.
cord to allow for respiratory exchange. The skin on the left side of the neck over the area of the bulging mass was pierced with a No. 18 needle and air was obtained upon aspiration. The larger mass was collapsed when the laryngoscope was reintroduced. The Iower bluish colored mass was still present. Roentgenological examination showed a large ovoid shadow of increased illumination on the left side of the neck at the level of the hyoid bone. (Fig. IB.) In view of these hndings a diagnosis of coexisting internal and external IaryngoceIes was made. On September I I, 1937, with the patient under IocaI anesthesia, tracheotomy was performed through a vertical midline incision. Anesthesia was started with intravenous Pentothalm and an endotracheal tube was placed through the tracheal opening. Anesthesia was maintained with nitrous oxide, oxygen and ether. A left curved oblique incision was made z cm. below the angle of the mandibIe from the anterior border of the sternocleidomastoid muscle to the midline between the
812
InternaI
and ExternaI
LaryngoceIes
2B
ZA Flc.. 2. A, operative appearance B, operative appearance during
during expiration showing both internal and cxtcrnal Iaryngocelcs. inspiration showing bulge of internal largngocclc into larynx.
3A
3B
FIG. 3. A, resected specimen showing intact external Iaryngocele goceIe. B, microscopic section through the common walI between showing epithelial linings of both sacs.
band. The patient was discharged postoperative day with a normal SURGICAL
on the fourteenth voice.
TECHNIC
AI1 authors agree that IaryngoceIes which produce symptoms shouId be excised. The exte.rnaI approach, paraIIe1 to the ramus of the mandibIe at a IeveI between the hyoid bone and the thyroid cartiIage, has been found to be adequate for external as we11 as interna IaryngoceIes. Various methods of exposing the Iarynx have been described in the Iiterature. Some spIit the hyoid bone [ro], others enlarged the opening in the thyrohyoid membrane by cutting the thyroid cartiIage paralIe1 to the midIine [r6,17], and some recommended laryngofissure [21]. It was found in the case
and cut surface of internal laryninternal and external Iaryngocele
reported herein that adequate exposure could be obtained by gentIe retraction of the hyoid bone and the thyroid cartiIage. The base of the transected interna IaryngoceIe staIk shouId be inverted. The defect in the thyrohyoid membrane should be cIosed whenever possible and covered by muscIe in an attempt to strengthen this weakness [20]. Tracheostomy is advisabIe for interna or combined laryngoceIes. In the externa1 type tracheostomy is usuahy unnecessary [23J. SUMMARY
A review of the history and Literature of IaryngoceIes is presented. Ninety-six cases have been reported to date, approximateIy 50 per cent in the Iast ten years. Four cases
Lane,
Cohen
of combined interna and externa1 IaryngoceIes are reported in the Iiterature. The fifth case is presented herein. A simpIe cIassification of IaryngoceIe is suggested. The surgica1 technic for excision of IaryngoceIe is discussed, and an approach for combined IaryngoceIes without sectioning either the hyoid bone or the thyroid cartiIage is presented. We wish to thank Dr. Acknowledgment: Louis J. MiIIman, Director of the PathoIogy Department, Fordham HospitaI, and Dr. Henry Poster, Director of the Department of RoentgenoIogy, Fordham HospitaI, for their assistance. REFERENCES I.
2.
3.
4. 5. 6.
VIRCHOW, R. Die Krankhaften Geschwiilste, vol. 3, p. 35. BerIin, 1863. A. HirschwaId. HILTON, J. Guy’s Hosp. Rep., 2: 519, 1837. NEGUS, V. E. The Comparative Anatomy Bnd Physiology of the Larynx, pp. 165-187. New York, 1949. Grune & Stratton. KERMAN. J. D. Surgerv of Nose and Throat. London, 1942. Thomas NeIson & Sons. MCLAURIN, J. W. LaryngoceIe. Laryngoscope, 57: 616, 1947. JACKSON, C. L. LaryngoceIe. Laryngoscope, 57: u
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R. Laryngocele ventricuIaris. Ann. Otol., Rbin. ti Laryng., 49: 661, 1940. 8. MOORE, L. So-caIIed prolapse of the IaryngeaI
and
IppoIito
ventricIe and eversion of the sacculus. J. Laryng. & Otol., 37: 265, 333, 381, 1922. 9. SHAMBAUGH. G. VentricIe of the Iargnx. Ann. Otol., Rbin. EdLaryng., 24: 129-138, rb15. IO. KEIM, W. F. and LIVINGSTON, R. G. Internal IaryngoceIe. Ann. Otol., Rbin. @ Laryng., 60: 39-50, ‘951. II. LAWSON, H. P. SurgicaI treatment of Iaryngocele. J. Laryng. ti Otol, 64: 779-781. 1950. 12. NEUMAN. H. BiIateraI combined externa1 and Wien med. Wcbnscbr., internal Iaryngocele. 81: 1548, 1931. 13. STRANGE, T. LaryngoceIe uniIatera1 mixed. Nord. med., 53: 160-161, 1955. -. R. Mixed Iarvnsocele. , J. and DUCROCO. ” 14. PIOUET. Lille cbir., 7: I 18-123, 1952. J. Laryng. @ Otol., 15. HOROWITZ, S. Laryngoceles. 65: 724-734,
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acid in therapy of 17. BOURDON, E. Trichloroacetic IaryngoceIe. Ann. oh-hry,?tg., 71: 588-593, 1954. 18. GARLAND. W. E. LarvnnoceIe: reoort of a bilateral case. A&b. 0tolaryng.r 65 : 1’48,’ I 957. ‘9. NEW, G. B. Treatment of cysts of the Iarynx. Arch. Otolaryng., 36: 687, 1942. 20. HILL, M. R., JR. and KIPEN, C. S. LaryngoceIe-an unusuaI tumor of the neck. Surgery, 35: 273, ‘954. 21. HOOVER, W. B. Laryngocele; report of two cases and discussion of surzzica1 treatment. S. Clin. Nortb America, 32: 954. 1952. 22. MEDA, P. Symptomatic IargngoceIe in cancer of _ ” the Iarynx. Arch. Otolaryng., 56: 5 12-520, ig52. 23. JOHNSTON,J. H., JR. ExternaI Iaryngocele. Surgery, 34: 307-3’2,
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