Pseudocyst of the Auricle Case Report and World Literature Review Philip

R.

Cohen, MD, Marc E. Grossman, MD

\s=b\ We treated a patient with pseudocyst of the auricle and reviewed the 113 cases previously published in the world literature. Pseudocyst of the auricle is an

asymptomatic, noninflammatory cystic swelling that involves the anthelix of the ear, results from an accumulation of fluid within an unlined intracartilaginous cavity, and occurs predominantly in men (93% of

patients). Characteristically, only

one ear

is involved (87% of patients), and the lesion is usually located within the scaphoid or triangular fossa of the anthelix. Previous trauma to the involved ear is uncommon. The diagnosis may be suggested by the clinical features, and analysis of the aspirated cystic fluid and/or histologic examination of a lesional biopsy specimen will confirm the diagnosis. Therapeutic intervention that maintains the architecture of the patient's external ear should be used in the treatment of this benign condition. (Arch Otolaryngol Head Neck Surg.

1990;116:1202-1204)

Pseudocyst commonly reported

of the auricle is an un¬ condition that

typically presents as an asymptomat¬ ic, unilateral cystic swelling of the up¬ per anthelix of the ear in middle-aged men.117 Although only 113 cases of pseudocyst of the auricle have been Accepted for publication January 29, 1990. From the Department of Dermatology, College of Physicians and Surgeons of Columbia University, New York, NY. Dr Cohen is now with the Department of Dermatology, The University of

Texas Health Science Center at Houston. Reprint requests to 7900 Cambridge, #252C, Houston, TX 77054 (Dr Cohen).

published, this disorder likely is more prevalent than the number of reported cases implies.3 The purpose of this re¬ port of pseudocyst of the auricle is to review the epidemiologie features, clinicopathologic characteristics, pos¬ tulated pathogenesis, differential di¬ agnosis, and appropriate treatment of this benign condition. REPORT OF A CASE A 33-year-old white man presented with a 1-month history of an asymptomatic swelling within the auricle of his left ear. Straw-colored fluid had been aspirated 2 weeks earlier by an otolaryngologist, but the lesion had promptly recurred. Although no recent antecedent trauma was reported, the patient had had a chronic neurologic movement disorder (dystonia musculorum deformane) since age 8 years, manifested by unusual body positioning and posturing. On examination, an erythematous, pain¬ less 4.0 X 2.0-cm cystic swelling that in¬ volved the entire anthelix of the left ear was observed (Figure) that was neither warm nor tender to palpation. The right ear was normal. A clinical diagnosis of pseudocyst of the auricle was made, and the possible additional diagnostic evaluations and/or

therapeutic approaches were discussed. The patient decided against any further in¬ tervention. COMMENT

Pseudocyst of the auricle has also been referred to as an intracartilagi¬ nous cyst,4 an endochondral pseudo¬ cyst,3·17 and cystic chondromalacia617 of

the auricle. It has been described most often in Chinese (48 patients)2·3·8 and whites (31 patients)1·4·6·11·17 and less fre¬ quently in Japanese,101116 French,9 Indian,515 Malaysian,8 black,6·7 and Puerto Rican7 patients. Pseudocyst of the auricle has been observed to occur predominantly in men (93% [106/114] of pa¬ tients). «*>« " Only 8 women with the disorder have been described in the literature.3·5·7·817 The age at onset in pa¬ tients with pseudocyst of the auricle ranges from 16 to 73 years,617 with most patients between the ages of 30 and 39 years when the condition ap¬ 17 Pseudocyst of the pears (Table l).1 auricle seldom occurs before the age of 20 years4·617 and is uncommon after age 60 years.13·6·17 Clinically, pseudocyst of the auricle presents as an enlarging lesion on the upper portion of the anterior aspect of the external ear. The lesion is cystic, ranges from 1 to 4 cm, and usually is

asymptomatic (Figure). Occasionally,

minor discomfort from pressure on the surrounding tissues is noted. The sca¬ phoid and triangular fossae of the an¬ thelix are the most common sites of this lesion.15·813 A solitary, unilateral pseudocyst is the typical presentation, reported in 87% (99/114) of these patients.1"5·9·1116 Lesions are located on the right ear (59% [36/61])36·8'9'"-13·17 1.5 times more frequently than on the left ear (41%

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Table 1.—Distribution of Age at Onset in Patients With Pseudocyst of the Auricle* No.

Age Range,

(%) of

y 20 20-29 30-39

Patients

40-49

10(16) 9(14) 5 (8)

2 12

25

(3) (19) (40)

References 4, 6, 17 2-4, 7, 9, 11, 14 2-5, 8, 9, 11, 13,

present report 50-59 >60

*The tients. '

Direct

(left) and posterior (right) views of a pseudocyst of the auricle on the anthelix of the left ear.

[25/61]).2"6'8·9·11'15·17 Only 13% (15/114) of the patients with pseudocyst of the

auricle have bilateral lesions.13·7·10·11·17 The simultaneous appearance of bilat¬ eral pseudocysts of the auricle is rare (2% [2/114] of patients).710 Between 0.54 and 10.03 mL of fluid has been observed to fill the pseudocyst. The liquid is water soluble yet insolu¬ ble in ether and petroleum benzin.2 The viscous fluid varies from clear to serosanguineous; usually it is straw colored2'81011·1315 and similar to olive 0jj 2,6.11,17 rpjjg 0smolarity and glucose, protein, and cholesterol concentra¬ tions of the fluid are similar to those of the patient's serum2·3·8·11·16; albumin2·5·10 is also present in the fluid. Bac¬ terial3·8·12·1315 and fungal12 cultures of the fluid collected from patients' pseudocysts are sterile. The distinctive histologie feature of pseudocyst of the auricle is the absence of an epithelial layer of cells lining the inner surface of the intracartilaginous cavity; hence, the lesion is described as a pseudocyst. In early lesions, a cystic space with surrounding fibrosis is noted to replace the central portion of the involved cartilage. Irregular thin¬ ning and hyalinization of the cartilage peripheral to the cavity also may be present. In some areas, necrosis and total dissolution of the cartilage can be observed. Intracavitary foci of granu¬ lation tissue and more extensive in¬ tracartilaginous fibrosis may be found in these lesions in later stages.2'2-12,14,15,17 ,

Minimal changes occur in the epi¬ dermis and dermis overlying the pseudocyst. Mild capillary ectasia and a sparse lymphocytic perivascular in¬ filtrate are noted in the dermis of early lesions. Changes seen later include (1) fibrosis and edema, which result in thickening of the dermis, and (2) a mild dermal infiltrate consisting of lympho¬ cytes and plasma cells.3·1 u7 The pathogenesis of pseudocyst of the auricle remains undefined. Two

possible, compatible, not mutually ex¬ clusive, mechanisms for this disorder involve (1) embryologie malformation

of the auricle and lesions that occur at the locus minoris resistentiae2·4·6·7·16 and (2) cartilaginous degeneration second¬ ary to the release of chondrocyte lysosomal enzymes.2·4·6·15 During approx¬ imately the sixth week of embryonic life, six knoblike outgrowths from the first and second branchial arches ap¬ pear. By the third month, these out¬ growths have gradually fused and folded around the first branchial groove to form the auricle. If residual planes are created during the folding process of the auricle's embryologie development, then, theoretically, these potential spaces of lowered structural resistance might subsequently serve as the sites of an intracartilaginous cavity.2·4·6·7 The observed pseudocystic lesion of the auricle may result from an additional nonspecific, ischemie,2·11 and/or traumatic3·11·1315 stimulus to the involved cartilage.

2,3,8,11,12,14,15 3,9 1, 3, 6, 17

age at onset was not

reported

in 51 pa¬

"*'· "

Other investigators have postulated that an abnormal release of lysosomal enzymes from local chondrocytes with subsequent degeneration of cartilage may produce the intracartilaginous cavity seen in pseudocyst of the auricle.2'4'6-15 Although this is an inter¬ esting possibility, elevated lysosomal enzyme levels have not yet been dem¬ onstrated in the fluid aspirated from the pseudocysts. Also, the electron mi¬ croscopic evaluation of a series of le¬ sions from patients with pseudocysts of the auricle failed to show an in¬ crease in the number of lysosomes within the involved chondrocytes.3 Pseudocyst of the auricle is not com¬ monly preceded by trauma to the in¬ volved ear. More than two thirds of patients with pseudocyst of the auricle (80 of 114 persons) had not sustained prior trauma to the lesionai area.11317 In contrast, several authors have hy¬ pothesized that a localized, traumatic stimulus may have been an important causative factor in the initiation of

pseudocyst development.3·11·13·1516 Histologically, pseudocyst of the au¬ ricle was most frequently misdiag¬ nosed as inflammatory disorders of cartilage,17 including relapsing poly-

chondritis, chondrodermatitis nodularis chronica helices, and cauliflower ear. Chondroma, invasive hemangio¬ ma, angiosarcoma, and chondrosar-

other disorders included in histologie differential diagnosis of pseudocyst of the auricle.6 " The clini¬ cal differential diagnosis of pseudocyst of the auricle includes benign and ma¬ lignant tumors, cystic lesions, inflam¬ matory and vascular disorders, and metabolic and systemic diseases (Ta¬ ble 2).2·3·6·11·13"15'1718 Associated clinical characteristics and histologie features coma are

the

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Table 2.—Clinical Differential Diagnosis of Pseudocyst of the Auricle Malignant tumors Angiosarcoma

Benign tumors Chondroma Fibroma Cystic lesions Epidermal inclusion cyst Dermoid cyst Inflammatory disorders Cauliflower ear Cellulitis Chondrodermatitis nodularis chronica helices Perichondritis (traumatic)

Chondrosarcoma

Metabolic/systemic diseases Rheumatoid nodule

Tophus (gout) Xanthoma

Vascular disorders

Hemangioma Othematoma Other disorder Seroma

Relapsing polychondritis

enable these conditions to be distin¬

guished from pseudocyst of the auricle. The treatment of pseudocyst of the auricle should accomplish two goals: (1) successful resolution of the lesion without subsequent recurrence and (2) preservation of the normal architec¬

tural structure and of the aesthetic appearance of the external ear. Needle aspiration3'5·7·12·13·15 and/or incision and drainage4·7·11 of the pseudocyst without additional intervention almost always results in the prompt reaccumulation of fluid within the lesion. The addi¬ tional application of a pressure dress¬ ing does not eliminate the possibility of recurrence of the pseudocyst,3·4·8 yet this simple approach has been therapeutically successful in a few patients.8·11·13 A modification of this technique produced a favorable func¬ tional and cosmetic result in one pa¬ tient: immediately after aspiration of the pseudocyst, cotton bolsters were firmly sutured to the anterior and pos¬ terior aspects of the auricle for 7

days.12

Several successful therapeutic alter¬ natives involve the induction of fibro¬ sis and scarring of the intracartilaginous cavity of the pseudocyst. Cu¬ rettage of the pseudocyst walls fol¬ lowing incision and drainage, and subsequent contour pressure dress¬ ings, may be sufficient to prevent re¬ currence of the lesion.4·5 Several pa¬ tients with pseudocyst of the auricle have had their lesions successfully treated without subsequent external ear deformities when 1% iodine tinc¬ ture or iodoform gauze was introduced into the cavity of the pseudocyst fol¬ lowing aspiration and/or incision and

drainage.2·4·7

A series of 31 patients with pseudocyst of the auricle were surgi-

cally treated by (1) elevating the skin

of the anterior anthelix of the ear off the pseudocyst, (2) excising only the anterior wall of the pseudocyst, (3) su¬ turing the skin flap back into place, and (4) applying a contour pressure dress¬ ing to the anterior and posterior as¬ pects of the auricle for 7 days.3 There were no recurrences, and this treat¬ ment

approach gave cosmetically sat¬

isfactory results in 90% of these patients.3 The posterior wall of the pseudocyst was inadvertently excised in one patient, with subsequent devel¬ opment of a floppy ear.3 Corticosteroids do not play a role in the management of pseudocyst of the auricle. Systemic corticosteroids failed to alter the course of one pa¬ tient's pseudocyst,11 and another pa¬ tient developed a permanent defor¬ mity of the ear after being treated with intralesional corticosteroids (0.4 mL of triamcinolone [10 mg/mL])." We recommend a relatively nonin¬ vasive initial approach to the treat¬ ment of pseudocyst of the auricle. Nee¬ dle aspiration of the lesion should be followed by the firm application (with tape and/or sutures) of a contour pres¬ sure bandage. The introduction of a few drops of 1% iodine tincture into the intracartilaginous cavity after the lesion has been aspirated is optional but will probably ensure the resolution of the lesion without subsequent re¬ currence. If the pseudocyst recurs, ex¬ cision of the anterior wall may be nec¬ essary.

In conclusion, pseudocyst of the au¬ ricle is a benign, asymptomatic cystic swelling of the anthelix of the ear. It most often appears as a solitary, uni¬ lateral lesion on the upper half or third of the anterior part of the ear of mid¬ dle-aged men. The intracartilaginous

cavity is without

an

epithelial lining,

and the fluid it contains is sterile, wa¬ ter soluble, rich in albumin, and usu¬ ally straw colored. Abnormal embryologic development of the auricle and/ or the abnormal release of chondrocyte lysosomal enzymes with subsequent cartilage degeneration may be in¬ volved in the pathogenesis of pseudocyst of the auricle. Benign and malignant tumors, cystic lesions, in¬ flammatory and vascular disorders, and metabolic and systemic diseases are included in the differential diag¬ nosis of pseudocyst of the auricle. Suc¬ cessful treatment of this condition re¬ quires (1) resolution of the lesion with¬ out recurrence and (2) structural and cosmetic preservation of the architec¬ ture of the external ear. References 1. Harmann A. \l=U"\berCystenbildung in der Ohrenmuschel. Arch Otorhinolaryngol. 1885; 15:156-166. 2. Engel D. Pseudocyst of the auricle in Chinese. Arch Otolaryngol. 1966;83:197-202. 3. Choi S, Lam K-h, Chan K-w, Ghadially FN, Ng ASM. Endochondral pseudocyst of the auricle in Chinese. Arch Otolaryngol. 1984;110:792-796. 4. Hansen JE. Pseudocyst of the auricle in Caucasians. Arch Otolaryngol. 1967;85:13-14. 5. Job A, Bhanu TS, Mathai R, Raghaven R. Pseudocyst of the auricle. J Laryngol Otol. 1988; 102:344-345. 6. Lazar RH, Heffner DK, Hughes GB, Hyams VK. Pseudocyst of the auricle: a review of 21 cases. Otolaryngol Head Neck Surg. 1986;94:360-361. 7. Santos VB, Polisar IA, Ruffy ML. Bilateral pseudocysts of the auricle in a female. Ann Otol Rhinol Laryngol. 1974;83:9-11. 8. Shanmugham MS. Pseudocyst of the auricle. J Laryngol Otol. 1985;99:701-703. 9. Botella-Anton R, Matos-Mula M, Jimenez\x=req-\ Martinez A, Pinazo-Canales MI, Castells-Rodellas A, Moragon-Gordon M. Pseudo- kystes du pavillon auriculaire: etude clinique et histologique de trois cas. Ann Dermatol Venereol. 1984;111:919\x=req-\ 923. 10. Fukamizu H, Imaizumi S. Bilateral pseudocysts of the auricles. Arch Dermatol.

1984;120:1238-1239. 11. Glamb R, Kim R. Pseudocyst of the auricle. J Am Acad Dermatol. 1984;11:58-63. 12. Karakshian GV, Lutz-Nagey LL, Anderson R. Pseudocyst of the auricle: compression suture therapy. J Dermatol Surg Oncol. 1987;13:74-75. 13. Lapins NA, Odom RB. Seroma of the auricle. Arch Dermatol. 1982;118:503-505. 14. Mendelson DS, Lund HZ. The histology of

pseudocyst of the auricle: resemblance and possible relation to cauliflower ear. Arch Dermatol. 1978;114:1831. Abstract. 15. Ramesh V. Pseudocyst of the auricle. Der-

matologica. 1986;172:125-126. 16. Saito M, Yagi S, Morishima T. Four cases of pseudocyst of the auricle. Jpn J Clin Dermatol. 1983;37:81-85. 17. Heffner DK, Hyams VJ. Cystic chondromalacia (endochondral pseudocyst) of the auricle. Arch Pathol Lab Med. 1986;110:740-743. 18. Cohen PR, Rapini RP. Relapsing polychondritis. Int J Dermatol. 1986;25:280-285.

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Pseudocyst of the auricle. Case report and world literature review.

We treated a patient with pseudocyst of the auricle and reviewed the 113 cases previously published in the world literature. Pseudocyst of the auricle...
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