Case Report

Auricular Pseudocyst in an Older Man

Alexandra Miniati, MD; Christine O. Urman, MD; Michael S. Krathen, MD; and Alice B. Gottlieb, MD, PhD

An accumulation of fluid within a small chamber but with no epithelial or other membrane lining is called a pseudocyst. When this cyst-like structure develops within the cartilage of the auricle of the ear, also called the pinna, it is commonly referred to as an auricular pseudocyst, although it has also been referred to as cystic chondromalacia, intracartilaginous cyst, or endochondral pseudocyst in the medical literature.1 Because auricular pseudocysts are benign and rare, they are easily overlooked or misdiagnosed by clinicians. When the condition is diagnosed correctly, medical or surgical interventions do not guarantee a successful outcome. We report a case of an auricular pseudocyst in an older man who we did not treat with medical or surgical intervention, but who was successfully monitored clinically. A literature review of the topic as well as typical therapeutic options and their usual outcomes are provided.

Case Report

lesion in the earDuring physical examination of a 74-year-old white man who was seen in our dermatology clinic for follow-up of bullous pemphigoid, we noted a noninflammatory fluctuant mass at the left superior antihelix that extended to the concha (Figure 1). The mass was not tender, had no drainage, and was not associated with tinnitus. The patient stated he had no history of trauma, including chronic rubbing or scratching of the ear or sleeping on the left side. We determined that the mass was an auricular pseudocyst and elected not to treat, as the condition is not associated with disability, the treatments may cause discomfort, and there is a high incidence of recurrence following treatment. In addition, our patient was immunosuppressed secondary to his methotrexate and steroid use for bullous pemphigoid, so any interventional treatment at that time would have had an uncertain outcome.

The patient had two follow-up visits in our clinic within a 6-month time period for bullous pemphigoid. We recorded that the auricular pseudocyst remained stable; it did not change in shape, there were no signs of inflammation, and no other symptoms had developed (Figure 2). Based on these findings, we decided the best option would be continued clinical monitoring.

To our knowledge, this is the first reported case of a patient with an auricular pseudocyst being followed clinically with great success, without any medical or surgical intervention. Because of the immunosuppressed state of our patient, any interventional procedure could result in significant complications. Physicians should be aware of the additional comorbidities of their patients, such as diabetes mellitus or immunosuppression from medications, when deciding whether to proceed with interventional therapy or to monitor the patient clinically.

Discussion

Pseudocyst of the auricle is an uncommon benign condition that typically presents as a painless, fluctuant cystic mass commonly located at the triangular and scaphoid fossa of the ear.2 The right ear is affected more frequently than the left, and only 13% of cases are bilateral.2 Since auricular pseudocysts were first reported in the English literature by Engel3 in 1966 in a group of Chinese men,cases in white men,4 women, and other races have been reported, demonstrating no racial or gender predisposition to this clinical entity. The most common age of onset is the third decade of life, and it is uncommonly encountered in those younger than 20 years and in those older than 60 years.

Histologically, a pseudocyst of the auricle is characterized by an intracartilaginous cavity unlined by epithelium, hence the name pseudocyst. The most prominent histologic findings are intracartilaginous fibrosis and excessive granulation tissue along with a perivascular mononuclear infiltrate, which is lymphocyte predominant.5,6 The lymphocytic infiltrate suggests an underlying inflammatory process in the pathogenesis of this condition. Aspiration of the lesion produces a viscous, sterile, straw-colored, albumin-rich fluid with an osmolarity and a glucose and protein concentration similar to that of plasma.7

Pathogenesis
The most prominent theory regarding the pathogenesis of an auricular pseudocyst is that of chronic minor trauma, which was first suggested by Engel,3 who thought the Chinese habit of sleeping on the right side against a hard pillow may have contributed to the occurrences he observed. Later evidence has shown that individuals who wear motorcycle helmets or frequently use stereo headphones are more prone to developing an auricular pseudocyst.8 Children with atopic dermatitis who constantly rub their ears have also been described as having this condition.9 It is possible that chronic trauma to the tissues from those activities can result in perichondral ischemia and cartilaginous degeneration, leading to the release of high amounts of hemosiderin and serum lactate dehydrogenase (LDH) into the cartilaginous fluid.8,10 Two of the LDH isoenzymes—LDH-4 and LDH-5, in particular—have been reported to predominate in the cystic fluid, even when serum LDH levels have been normal.10

Congenital embryonic dysplasia of the auricular cartilage has been suggested to be the main predisposing factor in the formation of a pseudocyst. During the complex auricular developmental process, some residual tissue planes may be formed within the mesenchyme of the immature auricle, which may lead to the formation of an intracartilaginous pseudocyst upon reopening.7

High levels of interleukin-1 (IL-1) in the fluid have also been shown to play a significant role in the formation of an auricular pseudocyst in susceptible individuals.11 IL-1 is well known for its central role in inflammation; it induces chemokine production to recruit leukocytes, activates endothelium to express adhesion molecules that are involved in cartilaginous inflammation, and triggers the synthesis of matrix metalloproteinases, leading to cartilage destruction.12

Diagnosis and Treatment Options
The diagnosis of an auricular pseudocyst is usually clinical. The differential diagnosis includes relapsing polychondritis, chondrodermatitis nodularis helicis, subperichondral hematoma, and cellulitis. Except for cellulitis, all of these conditions are characterized by subperichondral lesions, whereas the lesion in an auricular pseudocyst is intracartilagenous.7

Auricular pseudocyst is a benign disorder that does not cause clinical symptoms. As such, treatment is usually unnecessary; however, various treatment modalities have been proposed for this clinical entity for cosmesis and to address physician and patient fear that the condition will progress to unusual deformity of the auricle if left untreated. Auricular pseudocyst treatments have shown variable success rates. After aspiration alone, there is a high rate of recurrence and the likelihood of permanent ear deformities.13 Aspiration followed by intralesional injection or oral administration of steroids has been described by Job and Raman14 and Miyamoto and colleagues,15 but atrophy of both skin and cartilage and the potential systemic side effects of steroid use limit its effectiveness. Intralesional injection of minocycline hydrochloride (1 mg/mL 2-3 times at 2-week intervals) or curettage and use of fibrin glue, both used as sclerosing agents, have been shown to be effective, but the number of patients involved in these treatments was too small to produce significant results.16,17

Incision and drainage followed by a compression dressing using bolstered pressure sutures, button sutures, clips, or casts showed better results compared with aspiration followed by intralesional steroid injection.18-22 The aim of the compression dressing is to keep the two leaves of the cartilage attached during the healing process and prevent recurrence of the intracartilaginous cavity. Even in the case of incision and drainage followed by buttoning, the recurrence rate is about 38%.23 Surgical deroofing under local anesthesia followed by buttoning seems to be the most effective treatment option, as it has been used in 29 patients and has been associated with no recurrence.23 Buttoning seems to be the best form of compression because it is easy to apply, provides constant pressure, has good cosmetic results, and is well tolerated by patients.23

Nevertheless, surgical excision followed by compression techniques has been associated with some complications; there is a potential risk for pressure necrosis of the skin if the compression prosthetic device is too tight.13 Proper application and patient adherence to removing and checking the compression prosthetic device are two important measures to avoid that unfavorable outcome. Another complication is postsurgical perichondritis. Lim and colleagues20 reported perichondritis in a diabetic woman who was treated with antibiotics. The perichondritis resolved, but she developed a cosmetic failure called cauliflower ear
a few months after surgery.

Conclusion

Our case report shows that watchful waiting may result in a successful outcome for patients with an auricular pseudocyst. Therapy can be fraught with complications that may be worse than the disorder itself. Patients should be educated about the risks and benefits of follow-up only versus medical or surgical intervention so they can make a fully informed decision regarding treatment options.

The authors report no relevant financial relationships.

Dr. Miniati is Visiting Physician, Department of Dermatology, Tufts Medical Center; Dr. Urman is Chief Resident, Department of Dermatology, Tufts Medical Center; Dr. Krathen is Resident, Department of Dermatology, Boston Medical Center–Tufts Medical Center Combined Program; and Dr. Gottlieb is the Dermatologist-in-Chief and Chair, Department of Dermatology, Tufts Medical Center, Boston, MA.

References

1. Heffner DK, Hyams VJ. Cystic chondromalacia (endochondral pseudocyst) of the auricle. Arch Pathol Lab Med. 1986;110(8):740-743.

2. Cohen RR, Grossman ME. Pseudocyst of the auricle. Case report and world literature review. Arch Otolaryngol Head Neck Surg. 1990;116(10):1202-1204.

3. Engel D. Pseudocysts of the auricle in Chinese. Arch Otolaryngol. 1966;83(3):197-202.

4. Hansen JE. Pseudocysts of the auricle in Caucasians. Arch Otolaryngol. 1967;85(1):13-14.

5. Zhu L, Wang X. Histological examination of the auricular cartilage and pseudocyst of the auricle. J Laryngol Otol. 1992;106(2):103-104.

6. Lim CM, Goh YH, Chao S, Lim LH, Lim L. Pseudocyst of the auricle: a histological perspective [published correction appears in Laryngoscope. 2005;115(4):759]. Laryngoscope. 2004;114(7):1281-1284.

7. Lee JA, Panarese A. Endochondral pseudocyst of the auricle. J Clin Pathol. 1994;47(10):961-963.

8. Glamb R, Kim R. Pseudocyst of the auricle. J Am Acad Dermatol. 1984;11(1):58-63.

9. Devlin J, Harrison CJ, Whitby DJ, David TJ. Cartilaginous pseudocyst of the external auricle in children with atopic eczema. Br J Dermatol. 1990;122(5):699-704.

10. Miyamoto H, Okajima M, Takahashi I. Lactate dehydrogenase isozymes in and intralesional steroid injection therapy for pseudocyst of the auricle. Int J Dermatol. 2001;40(6):380-384.

11. Yamamoto T, Yokohama A, Umeda T. Cytokine profile of bilateral pseudocyst of the auricle. Acta Derm Venereol. 1996;76(1):92-93.

12. Jacques C, Gosset M, Berenbaum F, Gabay C. The role of IL-1 and IL-1Ra in joint inflammation and cartilage degradation. Vitam Horm. 2006;74:371-403.

13. Salgado CJ, Hardy JE, Mardini S, Dockery JM, Matthews MS. Treatment of auricular pseudocyst with aspiration and local pressure. J Plast Reconstr Aesthet Surg. 2006;59(12):1450-1452.

14. Job A, Raman R. Medical management of pseudocyst of the auricle. J Laryngol Otol. 1992;106(2):159-161.

15. Miyamoto H, Oida M, Onuma S, Uchiyama M. Steroid injection therapy for pseudocyst of the auricle. Acta Derm Venereol. 1994;74(2):140-142.

16. Oyama N, Satoh M, Iwatsuki K, Kanek F. Treatment of recurrent auricle pseudocyst with intralesional injection of minocycline: a report of two cases. J Am Acad Dermatol. 2001;45(4):554-556.

17. Tuncer S, Basterzi Y, Yavuzer R. Recurrent auricular pseudocyst: a new treatment recommendation with curettage and fibrin glue. Dermatol Surg. 2003;29(10):1080-1083.

18. Ophir D, Marshak G. Needle aspiration and pressure sutures for auricular pseudocyst. Plast Reconstr Surg. 1991;87(4):783-784.

19. Schulte KW, Neumann NJ, Ruzicka T. Surgical pearl: the close-fitting ear cover cast—a noninvasive treatment for pseudocyst of the ear. J Am Acad Dermatol. 2001;44(2):285-286.

20. Lim CM, Goh YH, Chao SS, Lynne L. Pseudocyst of the auricle. Laryngoscope. 2002;112(11):2033-2036.

21. Kim TY, Kim DH, Yoon MS. Treatment of a recurrent auricular pseudocyst with intralesional steroid injection and clip compression dressing. Dermatol Surg. 2009;35(2):245-247.

22. Göktay F, Aslan C. Successful treatment of auricular pseudocyst with clothing button bolsters alone [published online ahead of print September 2, 2010]. J Dermatol Treat. 2011;22(5):285-287.

23. Kanotra SP, Lateef M. Pseudocyst of pinna: a recurrence-free approach. Am J Otolaryngol. 2009;30(2):73-79.