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Kelemen 1953
Kelemen 1953
CONTENTS
General considerations Autopsy
Materials and methods Microscopic anatomy
Case histories Birth trauma
Character of the vocal performance
Comment
Antomy Comparative phonetics
Prenatal and postnatal development Therapy
Comparative anatomy Conclusions
GENERAL CONSIDERATIONS
The present study covers the histological data offered by examination of laryngeal specimens
provided by patients with congenital stridor.5 Eight specimens, four from infants with and four
from infants without stridor, were studied. After being embedded in celloidin, three specimens
from each group were sectioned in the horizontal and one in the frontal plane. Sections of 18
Case 3 (Massachusetts General Hospital Path. 12,269) (Figs. 25 to 30).—A baby boy was
admitted three and one-half hours after a breech delivery. Although his cry at birth was good,
difficult breathing with stridor was noted shortly afterwards. There was severe sternal, subcostal,
and supraclavicular retraction, with a fairly good pulse but with severe cyanosis. An x-ray
revealed an unexpanded right lung. There was no anatomical abnormality and no evidence of
subglottic obstruction on direct laryngoscopy. With the glottis thus exposed, the infant's color
became pink, retraction ceased, and air could be heard coming in and out of the lungs. With a
straight Mosher lifesaver, breathing was maintained for two to three hours. Tracheotomy was
6. The histological work was done by Miss Dorothy Linden and the microphotography by
Mr. Donald C. Whitee.
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simply changing the position of the patient point to a loose structure of the tissues,
with gravitation rather than elasticity playing the main role. In 7 of his 12 cases,
Lorin was able, like many others, to stop the stridor by lowering the infant's head
and turning the body to the right or left. The situation was similar in our Cases
1 and 3.
The complicated conditions in the upper part of the quadrangular membrane,
which ends in the aryepiglottic folds in man and animals, have been discussed in
detail by Némai.12
The constant striking of a long epiglottis against the posterior pharyngeal wall
results, finally, according to Mosher, in a permanent forward bend of the tip. One
of his cases illustrates the occlusion of the laryngeal entrance by a reclining epiglottis
with an asymmetric position of this organ. By x-ray he studied the movements of
the epiglottis in swallowing and found that it actually turns downward to cover the
larynx ; it rises with the base of the tongue, becomes applied to it, and finally is
forced back against the posterior pharyngeal wall.
With stridor present before and after lifting of the epiglottis by the tube of the
laryngoscope, the pathological structures are found located deeper than the vestibule.
Prenatal and Postnatal Develo piment.—The inconstancy of the relation between
the epiglottis and the musculature of the vestibule is well documented by the fact
that Frazer,13 in the 6 mm. embryo, found no trace of these muscles, though the
epiglottic cartilage was fairly well developed.
8. Braus, H.: Anatomie des Menschen, Berlin, Springer-Verlag, 1924, Vol. 2.
9. Jackson, C., and Jackson, C. L.: Diseases of the Nose, Throat and the Ear, Including
Bronchoscopy and Esophagoscopy, Philadelphia, W. B. Saunders Company, 1945.
10. Mosher, H. P.: X-Ray Study of the Tongue, Epiglottis and Hyoid Bone in Swallowing,
Followed by a Discussion of Difficulty in Swallowing Caused by Retropharyngeal Diverticulum,
Postcricoid Webs and Exostoses of Cervical Vertebrae, Laryngoscope 37:235-262 (April) 1927.
Wien. med. Ztg. 35:579-580 (Dec. 9) 1890.
12. N\l=e'\mai,J.: Vergleichend-anatomische Studien am Kehlkopfe der Saeugetiere, Arch.
Laryng. u. Rhinol. 26:451-508, 1912.
13. Frazer, J. E.: The Development of the Larynx, J. Anat. & Physiol. 44:155-191 (Jan.)
1910.
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16. Hasslinger, F.: Zur Pathogenese, Diagnostik und Therapie des Stridor congenitus,
Ztschr. Hals- Nasen- u. Ohrenh. 21:223-235 (May 10) 1928.
17. Finkelstein, H.: Lehrbuch der S\l=a"\uglingskrankheiten,Berlin, H. Kornfeld, 1912, Vol. 3,
pp. 45-47.
18. Angyal, F.: Zur Pathogenese des Stridor inspiratorius congenitus, Pract. oto-rhino\x=req-\
laryng. 3:129-144, 1940.
19. Bagg, C.: Beitr\l=a"\gezur \l=A"\tiologiedes Stridor congenitus, Monatsschr. Kinderh. 45:102\x=req-\
115 (Oct.) 1929.
20. Eley, R. C., and Farber, S.: Hypoplasia of the Mandible (Micrognathy) as a Cause of
Cyanotic Attacks in the Newly Born Infant: Report of 4 Cases, Am. J. Dis. Child. 39:1167\x=req-\
1175 (June) 1930.
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our four cases a normal picture twice presented itself, and in the remaining two
a larger than normal epiglottis was noted (Table 3). Hasslinger16 pointed to the
pitfalls in evaluating the autopsy records : The curving and declination of the epi¬
glottis, generally present in the living child, are most accentuated in the cadaver;
approximation of the aryepiglottic folds may result from rigor mortis, while, on
the other hand, edema disappears.
As the suspension apparatus from the two sides (pharynx and trachéal pull) is
absent in isolated specimens of the larynx, the position in which the elements of the
vestibule are found will not yield reliable information. The same must be said
regarding the angle of inclination between the pharynx and the laryngeal vestibule.
Microscopic Anatomy.—In cases of congenital laryngeal stridor even autopsy
records are rare, histological observations are conspicuous by their absence, and the
number of biopsy reports is minimal. Iglauer 27 found the amputated part of the epi¬
glottis to be normal. Hasslinger on excising of flabby ridges from the aryepiglottic
16
folds saw beneath the squamous epithelium edematous tissue with few cells. Beneke
(quoted by Bagg19) found structural displacement of the cartilaginous cells through
25. Elza, K. : Anatomie des Kehlkopfes, in Denker, A., and Kahler, O., Editors : Handbuch
der Hals- Nasen- Ohren-heilkunde, Berlin, Springer-Verlag, 1925, Vol. 1, pp. 225-260.
26. Heiss, R.: Der Atmungsapparat, in von M\l=o"\llendorf,W., Editor : Handbuch der mikro-
skopischen Anatomie des Menschen, Berlin, Springer-Verlag, 1936, Vol. 5, pp. 728-742.
27. Iglauer, S.: Epiglottidectomy for the Relief of Congenital Laryngeal Stridor, with
Report of a Case, Laryngoscope 32:56-59 (Jan.) 1922.
entrance of the larynx almost completely, one originating from the region of the right ventricular
band and the other, on the left, from the summit of the arytenoids ; the flaps were sucked inward
with each respiration. There were no inflammatory signs, and respiration was completely free
when a tube was inserted. Autopsy showed a small, tubular epiglottis, with conspicuously loose
aryepiglottic folds covered by a hyperemic mucous membrane, overhanging the laryngeal
entrance in such a way that no free airway was left except immediately behind the epiglottis.
A horizontal series of sections was prepared for microscopy. The tunica propria was thick, as
was the submucosa, especially in the aryepiglottic folds. These folds showed lymph cell infiltra¬
tion in some areas, but this nowhere penetrated the muscles or the corpus glandulare. Lymph
and blood vessels were enlarged. Besides an increase in collagenous fibers, a similar condition
was found regarding the elastic fibers. Fibers of the thyromembranosus (ventricularis) origin¬
ating from the upper edge of the thyroarytenoideus were practically absent, while they were
found regularly in the controls. In the free edge of the aryepiglottic folds the aryepiglotticus and
the pars aryepiglottica of the stylopharyngeus were completely absent. Above the corpus
glandulare the aryepiglottic folds were formed by a mere duplication of the thickened mucous
membrane.
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Aryepiglottic folds void of musculature down to level of roof of ventricle_ 20, 28, 29 8, 9,14,18
Bulky ventricular bands. 27 .
higher in the quadrangular membrane (Fig. 24), and a few fibers connected with
the hyothyroid ligament in the thyroid notch, ending in a loose, edematous area
(Fig. 23). In Case 3 the most delicate fibers were observed in cross sections, none
above the level of the upper rim of the thyroid (Fig. 29). In Case 4 there was no
muscle in bulk above a level where the arytenoids are seen in horizontal sections as
frontal bars.
One of the controls presented the following picture : Muscle bundles entered
the aryepiglottic fold at the level where the edges of the flattening-out epiglottis
consisted merely of soft tissues (Fig. 15) ; a few bundles approached the epiglottis
without reaching it (Fig. 16). Fibers of the aryepiglotticus and arymembranosus
almost reached the edge of the epiglottis (Fig. 17), a few bundles being seen on both
sides of the ventricle in the quadrangular membrane, immediately under the ary¬
epiglottic fold.
Thus, while in the cases of stridor the aryepiglottic folds were always utterly
void of muscles, this situation was also found, though not regularly, in the controls.
Luchsinger,32 discussing congenital hoarseness and asymmetry of the larynx,
found in three cases unilateral hyperplasia of the musculature of the vocal cord.
31. Jackson, C., and Jackson, C. L.: Dysphonia Plicae Ventricularis : Phonation with
Ventricular Bands, Arch. Otolaryng. 21:157-167 (Feb.) 1935.
32. Luchsinger, R.: Angeborene Heiserkeit und die Asymmetrie des Kehlkopfes, Ztschr.
Hals- Nasen- u. Ohrenh. 50:107-112, 1944.
Fig. 31.—Tubular epiglottis in an adult, "a not infrequent variation" (from von Luschka 7).
Regarding the pseudosyrinx, experiments on the avian syrinx, though it is
located at the trachéal bifurcation, have shown that sound can be produced there
in the same way by an inspiratory and by an expiratory air current.
Therapeutical Considerations.—In this short series of cases in which autopsy
was performed, death occurred from conditions not directly connected with the
stridor. Operative measures could be only palliative. With temporary relief given by
laryngoscopy and tracheotomy, direct attack by excision of obstructive parts was
forbidden by the general condition of the patient. On the basis of histological findings,
44. Kelemen, G., and Hassk\l=o'\,A.: Das Stimmorgan des Seeloewen (Otaria jubata), Ztschr.
Anat. 95:497-511, 1931.
The musculature of the laryngeal vestibule, having lost its role around the air
sac, ison the way to a new orientation around the epiglottis, the latter structure
being thus freed from its retrovelar rigid position for a new kind of motility.
Variability with frequent insufficiency points to this new role, not yet fully achieved
in the course of evolution.
Thus, no effective dilator is formed for the laryngeal vestibule, which opens
only by movements of the epiglottis executed to a very minor degree by a muscula¬
ture of its own. No muscular action is responsible for the inward curling of the
epiglottic margins.
The suspension of the epiglottis is extremely loose. This fact is demonstrated
(1) histologically by the areolar-adipose preepiglottic cushion, which continues
downward intralaryngeally between the inner aspect of the thyroid lamina and the
upper part of the quadrangular membrane, until it reaches the top of the ventricular
appendix, and (2) by clinical experience, according to w'hich embarrassing positions
of the epiglottis can be overcome by changing the position of the body. Here gravi¬
tation surmounts elasticity, with the help of looseness in the periepiglottic tissues.
Besides swinging forward and backward en masse, the epiglottis can bend along
a midhorizontal axis at the boundary of the suprathyroid and the infrathyroid
parts.
The tubai form of the upper part has little pathological significance, even in pulling
up the anterior end of the aryepiglottic folds. The lower half offers little resistance,
its substance being honeycombed by glandular masses. This shovel-shaped lower
part can protrude into the lumen, wrinkling the aryepiglottic folds and exerting
pressure over the ventricular bands. In contrast to conditions in anthropoids, it
is withdrawn from the body of the ventricular band. This means a gain in the
phonetical sense, but a loss in securing patency, by spreading the ventricular bands
rigidly apart.
The anterior, horizontal leg of the T-shaped laryngeal lumen of the infant is
easily obstructed by even a slight declination of the epiglottis, but it is easily liberated
by the laryngoscope. The purpose of the laryngoscope at this point is both to push
back the tube-shaped upper half, and, in a more important role, to correct the angle
within the epiglottis itself between the suprathyroid and the infrathyroid parts.
Correlation of the histological findings with the clinical symptoms shows that
all characteristic changes in cases of stridor—protruding, sometimes edematous, or
wrinkled folds, occurring at the epiglottic base, along the aryepiglottic plicae, at
the anterior attachment of the quadrangular membrane and above the arytenoids—
can be duplicated in controls but, seemingly, with less regularity, the difference
being merely one of frequency. The plica aryepiglottica can be as completely void of
musculature in a normal control
in a case with stridor.
as