Cysticercosis Jan

in

Otolaryngology

Kinnman, MD, PhD; Chung Hee Chi, MD, PhD; Jae Hoon Park, MD

\s=b\ Cysticercosis is not likely to be the first diagnosis the otologist has in mind when regarding tumors in the head and neck area. The fact that this disease may present a diagnostic and therapeutic problem is illustrated by three cases, appearing in the tongue, buccal mucosa, and midline of the neck, respectively. The local finding of a soft nontender mass, the typical calcifications in the soft tissues visible on roentgenograms, and the histopathological findings are described and illustrated. Attention is drawn to the fact that cerebral cysticercosis, which is a very grave disease, may cause tinnitus, nystagmus, and hearing disturbances.

(Arch Otolaryngol 102:144-147, 1976)

few exist manifestations of

reports concerning Veryotolaryngological cysticercosis field, in

complications such as arteritis.n Cysticercosis is an infestation with

the cyst stage of Taenia solium. The adult Taenia solium is strictly a para¬ site of man, but, unlike Taenia saginata, the larval cysticercoid stage, although it normally occurs in the pig, can also develop in man.'1 Infestation with the adult worm follows the consumption of viable cysticerci (Cysticercus cellulosae) in uncooked pork. The infestation of the tissues may also be caused by autoinfestation, in which an individual harboring the adult parasite transfers egg-laden feces from the anus to fingers to the mouth or in which

cases, to

our

trate the wall of the stomach and

disseminate systemically through the and arteriovenous chan¬ nels.4-5 The heads of the cysticerci attach themselves to the mucosa of the upper part of the small intestine, and gravid segments grow from the neck. The segments are passed through the anus; it is these eggs that are consumed by pigs that ensure the maintenance of the parasite. A human develops cysticercosis if he swallows eggs liberated from disin¬ tegrating gravid segments of his own

lymphatic

Fig 1.—Larvae (arrow) from Taenia solium become enclosed within fibrous capsule, forming cyst that varies in diameter from 0.5 to 3 cm (hematoxylin-eosin, 12.5).

the and this disease is not mentioned in standard textbooks of otolaryngology. The only

reported

peristalsis carries mature proglottides from the small intestine to the stomach. The oncospheres pene¬ reversed

knowledge,

include one occurring in the tongue1 and one in the nose.2 Vestibular and hearing disorders have also been reported.3 The central nervous system is one of the chief sites of localization of the cysticerci,4 and focal symptoms are due to either intracerebral localization of the parasite or to vascular

Accepted

for publication Nov 17, 1975. From the departments of otolaryngology (Drs Kinnman and Park) and pathology (Dr Chi), National Medical Center, Seoul, Korea. Reprint requests to Department of Otolaryngology, Karolinska Institutet, Huddinge sjukhus, S 141 86, Huddinge, Sweden (Dr Kinnman).

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another's intestinal worm. The larvae liberated from the eggs pene¬ trate the mucosa of the small intestine and are carried by the blood stream to various tissues, where they encyst and form cysticerci. The cysticerci in man lodge in the connective tissue, the voluntary muscles, the eye, and the central nervous system. The larvae at first cause a local, surrounding, inflammatory reaction with infiltration by neutrophils, eosi¬ nophils, and lymphocytes and a stimu¬ lation to fibroblast production. Subse¬ quently, the larvae become enclosed within a fibrous capsule, or necrosis may occur followed by caseation or calcification. Giant cells may be found around the lesion.7 The resulting cysts may vary in diameter from 0.5 to 2 or 3 cm. The membrane that forms the wall of the cyst consists of an outer row of cilia set on a basal amorphous layer, a dense fibrocanalicular layer, a layer of cells with scant cytoplasm and eccentric nuclei, and an inner fibroreticular layer with canalicular spaces and scattered cells.4 Sections may show the hooks and suckers in the scolex or the internal structures of the body of the parasite (Fig 1 through or

Fig 2.—Capsule that forms cyst consists of row of cilia set on basal amorphous layer (1 ), dense fibrocanalicular layer (2), layer of cells with scant cytoplasm and eccentric nuclei (3), and fibroreticular layer with canalicular spaces and scattered cells (4). Fibrocollagenous membrane infiltrated with lymphocytes and eosinophils (5) separates parasitic cyst from surrounding muscle (6) (hematoxylin-eosin, original magnification x200).

4).

Fig

3.—Section from larva

lin-eosin, X800).

showing part

of intestines

Fig 4.—Section from larva showing one (2) (hematoxylin-eosin, 800).

sucker

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hooklet

(hematoxy¬

(1) and

one

Fig 5.—Cystic

tumor measuring 0.5 1 upper lateral surface of tongue. Histopathological examination gave evidence of cysticercosis (case 2). cm

observed

on

at surgery were almost identical in all three cases, regardless of where in body cysticerci were lodged.

Fig 6.—Findings

Thick-walled, mass

(arrow)

inal wall in

REPORT OF CASES

for five months. At admission in November 1965, the local finding was a rather firm, freely movable tumor, 0.5 1 cm, in the midline of the neck at the level of the cricoid, free from the skin and apparently also from the thyroid gland. The erythrocyte sedimentation rate as determined by the Westergren method was 4 mm/hr and the eosinophil count was 2%. Routine stool tests for parasites showed Trichuris trichiura and hookworm but no Taenia. The tentative diagnosis was a nonspecific swelling of the lymph node or a possible thyroid cyst, and the small node situated in the sternohyoid muscle was removed. The histopathological examination showed Cys¬ ticercosis cellulosae (the findings were identical with those in case 2, Fig 1-4). Roentgenograms of the chest did not demonstrate calcified cysts, and repeated stool tests gave negative findings for Taenia. Medication was not given, but the patient was informed about prophylactic measures.

Case 2.-A 55-year-old, previously healthy woman had a slightly tender, cystic the tongue for three years. She admitted that she had eaten uncooked pork on several occasions 15 years previously. On admission in March 1966, the local mass on

case

cystic

removed from abdom¬

2.

findings

included a cyst measuring far back on the dorsum linguae (Fig 5) and a somewhat larger, welllimited, movable tumor deep in the tongue. The erythrocyte sedimentation rate was 30 mm/hr with 5% eosinophils. A routine stool test gave positive findings for Trichiuris trichiura. Our preliminary diagnosis was that of a myoblastoma of the tongue asso¬ ciated with a superficial retention cyst. On surgical removal, the superficial cyst was found to be firmer than expected. After incision of the frenulum linguae, the deep tumor could also be removed, and this revealed the same consistency as the superficial one. The histopathological find¬ ing was identical in the two tumors. There was a large cyst with small aggregates of lymphocytes and some eosinophilic polymorphonuclear leukocytes in the wall. The lumen contained parts of a parasite identified as Cysticercus cellulosae (Fig 1-4). After this histopathological report, roentgenograms of the skull, chest, and soft tissues were taken, but no calcified cysts were demonstrable. Stool examinations were performed several times, and on one occasion Taenia solium was also found. Accordingly, quinacrine

0.5 Case I-A 30-year-old, previously healthy man suffered from a slowly growing nodule in the midline of the neck

half-transparent,

was

x

1

cm

hydrochloride therapy was given success¬ fully. By careful palpation of the soft tissues, one additional cyst was found rather deep in the abdominal wall and was also removed. The finding was exactly the same as

Fig 7—Cysticerci dying in muscles often calcify and may be demonstrated roentgenographically. Cysticerci are seen here in medial aspects of right and left thighs.

that in the

tongue and in the

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two

other

thick-walled, half(Fig 6). Case 3.-A 32-year-old man suffered from a nontender mass measuring 0.5 1 cm in the right buccal mucosa for four years. The past history disclosed that cases—a

rather

transparent, cystic

mass

two years earlier he had been examined at

Department of Neurology and had complained of dull headache on the left side for lVè months. The neurological examina¬ tion, including spinal tapping, was normal the

except for

inconstant nystagmus on The patient, however, was not referred to the Department of Otolaryngology. The headache lessened with meprobamate therapy. Ascaris lumbricoides was found in the stool, and piperazine tartrate was given. On admission in May 1966, the local finding was a rather firm, movable tumor measuring 0.5 1 cm situated deep in the right buccal mucosa. The erythrocyte sedi¬ mentation rate was 2 mm/hr and eosinophil count was 6%. The tumor was removed and showed Cysticercosis cellulosae, with essentially the same findings as in Fig 1 through 4. Several stool examinations were made and showed Ascaris lumbricoides, Trichuris trichuria, hookworm, and Trichostrongylus orientalis in some of the specimens, but no Taenia eggs were found. Specific treatment against Taenia was, therefore, not given. Roentgenograms of the chest, skull, and soft tissues, and the otoneurological examination were normal. an

looking to the right.

COMMENT The clinical features of cysticercosis are extremely variable and depend on the location and the number of cysti¬ cerci in the host. Light human infesta¬ tions, the result of swallowing a few eggs, may escape notice unless a cyst is so located as to cause physical signs. Heavy infestations with hundreds of cysticerci cause very grave manifesta¬ tions, chiefly due to the presence of cysticerci in the central nervous system. While alive, these parasites rarely cause trouble, but they begin to die and degenerate within three to five years. The cysticerci that die in the muscle often calcify and can then be seen roentgenographically (Fig 7). Those dying in the central nervous system very rarely calcify and cannot be seen on roentgenograms, but as they degenerate, they swell and cause disintegrating cellular changes in the surrounding nerve tissue. This results in a wide range of clinical manifesta¬ tions such as Jacksonian attacks, physical disturbances, and mental deterioration.6 The onset of symp¬ toms, associated with the death of the parasite, occurs after an incubation period of a few months to as long as 30

years.8

Otoneurological

examinations may

reveal a clinical picture characteristic of brain stem lesions of neoplastic or other origin but of changeable course. Tinnitus, vertigo, and spontaneous

nystagmus

may

develop during

exa¬

cerbations. Another characteristic feature of cysticercosis of the poste¬ rior cranial fossa is the relative mild¬ ness of the auditory and vestibular symptoms, even when the generalized cerebral features are very prominent; this is especially true in cysticercosis of the cerebellopontine angle.3 A diagnosis of cysticercosis is made by palpation of cysticerci, by biopsy of the cyst, and by identification of the hooked head of the future worm in each of these cysts. Roentgenograms of soft tissues may be helpful in demonstrating the typical calcified cysts in the muscles (Fig 7), while roentgenograms of the skull are usual¬ ly normal. Alterations of the sella turcica indicative of intracranial hy¬ pertension may occur.4 The comple¬ ment fixation test in the cerebrospinal fluid is of great importance in the differential diagnosis of the disease.5 Eradication of the adult worms from the intestine should not be delayed when infestation of Taenia solium is detected. Early precautions

must be taken to

ensure that dissemi¬ nation of the eggs cannot occur. It is important that the head of the worm is released from its hold on the intes¬ tine. If it remains, even though all the existing segments of the worm are removed, formation of new segments will restore the worm to its full size within a few months. The drug tradi¬ tionally used is extract of aspidium, but more consistently effective is

quinacrine hydrochloride."

There is however no specific treat¬ ment for cysticercosis. Surgical re¬ moval may be indicated in some cases

when

a

localized lesion

symptoms. The

causes

obvious

treatment of cerebral

cysticercosis is palliative,

but indi¬ vidual cysts may, in selected cases, be removed from the brain. Preventive measures are important and include the thorough cooking of pork and the early detection and complete removal of the worm, including the head, as outlined previously. Mild infestations cause little trou¬ ble, but in cases of cerebral cysticerco¬ sis, the prognosis is very poor.

Nonproprietary Name and Trademark of Drug Piperazine tartrate-Piperai

Tartrate.

References 1. Celis San Felix A, San Miguel Bertrand L: Cisticerco de la lengua (observaci\l=o'\ncl\l=i'\nica).An Esp Odontoestomat 24:35-40, 1965. 2. Sheksuvarli MA, Alieva SI, Ba\l=i%u\ramalibekova RT: Redkaia lokalizatsiia bych'ego tsepnia s otkhozhdeniem proglottid cherez nos. Med Parazit 33:354-355, 1964. 3. Antonov IP, Sklyut IA: O vestibularynkh i slukhovykh narusheniiakh pri tsistitserkose zadnei cherepnoi iamki. Vestn Otorinolaringol 27:33,

1965. 4. Garbutt GD, Courville CB: Cysticercosis cerebri: A retrospective study including 14 new cases personally investigated. Bull Los Angeles Neurol Soc 32:6-16, 1967. 5. Pilz H, M\l=u"\llerD: Cerebrale Cysticercose des Menschen. Klinische Sympomatik, Differential\x=req-\ diagnose und Therapie an Hand von 14 eigenen Beobachtungen. Z Neurol 201:241-260, 1972. 6. Adams ARD, Maegraith BG: Clinical Tropi-

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cal Diseases, ed 3. Philadelphia, WB Saunders Co, 1960, p 551. 7. Hunter GW, Frye WW, Swartzwelder JC: A Manual of Tropical Medicine, ed 3. Philadelphia, WB Saunders Co, 1960, p 551. 8. Dixon JBF, Lipscomb FM: Cysticercosis: An analysis and follow-up of 450 cases. Med Res Counc Spec Rep Ser 299:1, 1961.

Cysticercosis in otolaryngology.

Cysticercosis is not likely to be the first diagnosis the otologist has in mind when regarding tumors in the head and neck area. The fact that this di...
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