|Year : 2013 | Volume
| Issue : 4 | Page : 208-210
Bilateral recurrent auricular pseudocyst: Importance of fine-needle aspiration cytology and lactate dehydrogenase estimation
Kalyan Khan, Krishnendu Mondal, Mamata Guha Mallick Sinha, Palash Kumar Mandal, Rupali Mandal, Piyali Ghosh
Department of Pathology, North Bengal Medical College, Sushrutanagar, Darjeeling, West Bengal, India
|Date of Web Publication||7-Jan-2014|
Flat No. 11, 'Bela' Apartment, Netaji Subhas Road, Subhaspally, Siliguri, Darjeeling - 734 001, West Bengal
Source of Support: None, Conflict of Interest: None
Auricular pseudocyst or Idiopathic cystic chondromalacia is a rare, benign condition characterized by a focal noninflammatory cystic swelling on the pinna, occurring usually in young male patients. Bilaterality and recurrence have been reported rarely. We report a case of bilateral, recurrent auricular pseudocyst in a young male patient, where fine needle aspiration cytology coupled with fluid lactate dehydrogenase level estimation was diagnostic. Repeated surgery was avoided and conservative treatment was proved to be effective.
Keywords: Auricular pseudocyst, fine needle aspiration cytology, Idiopathic cystic chondromalacia, lactate dehydrogenase
|How to cite this article:|
Khan K, Mondal K, Sinha MG, Mandal PK, Mandal R, Ghosh P. Bilateral recurrent auricular pseudocyst: Importance of fine-needle aspiration cytology and lactate dehydrogenase estimation. Indian J Otol 2013;19:208-10
|How to cite this URL:|
Khan K, Mondal K, Sinha MG, Mandal PK, Mandal R, Ghosh P. Bilateral recurrent auricular pseudocyst: Importance of fine-needle aspiration cytology and lactate dehydrogenase estimation. Indian J Otol [serial online] 2013 [cited 2020 Apr 8];19:208-10. Available from: http://www.indianjotol.org/text.asp?2013/19/4/208/124527
| Introduction|| |
Pseudocyst of the auricle or Idiopathic cystic chondromalacia is a relatively uncommon condition with distinct clinical, pathological, and biochemical features. It was originally described back in 1846, when Hartmann reported auricular cystic swellings in 12 patients.  Later in 1866, after performing detailed histopathological examinations, Meyer interpreted similar lesions as ''chondromalacia. '' Further observations revealed, it is often induced by minimal recurrent trauma,  although contradictory evidence also remains there at large. Patients commonly present with long-standing unilateral painless, fluctuant swelling over the auricle, secondary to intracartilaginous accumulation of serous fluid within the external ear. Right ear is affected more frequently and bilaterality is observed in upto 13% of cases only.  Its rarity, insidious clinical presentation, and uncertainty in the etiopathogenesis makes ''pseudocysts of auricle'' an enigmatic disease for clinicians and pathologists.
We report a rare case of recurrent but synchronous, bilateral pseudocysts of auricles in a 42-year-old man.
| Case Report|| |
A 42-year-old military personnel, presented to the outpatient department of ENT with bilateral painless slowly growing firm nodular swellings involving the scaphoid fossa of auricles for last 1 year. The lesions measured 2 × 1.5 cm on left side and 0.5 × 0.5 cm on the right [Figure 1]. The lesions were not associated with fever, ulceration, or induration of the overlying skin or any other associated symptoms. Identical lesions could not be found on any other parts of the body. Further inspection revealed an old surgical scar on the under surface of his right helix, which on subsequent enquiry was proved to be a postoperative mark of similar lesion, treated 8 years back and the details of that lesion could not be retrieved. Traumatic exposure of the affected parts from his army-uniform cap was evitable at the time of presentation as well as pointed out by the patient himself.
|Figure 1: Bilateral auricular pseudocyst: Left-sided lesion is larger than the right one|
Click here to view
Clinically, the lesions mimicked various auricular lesions, including relapsing polychondritis, chondrodermatitis nodularis helicis, traumatic perichondritis, hemangioma, chondroma, and angiosarcoma. The attending otorhinolaryngologist could not provide a definitive diagnosis clinically and advised a fine-needle aspiration cytology (FNAC) examination from the nodules.
On needle aspiration, a thick and viscous straw-colored fluid was obtained. Microscopically, the smears were devoid of any cellular elements with abundant myxoid material in the background, which often featured characteristic drying artefacts [Figure 2]. The aspirated fluid was sterile on culture too. Provisionally, a diagnosis of ''pseudocysts of bilateral auricles''was rendered.
|Figure 2: Auricular pseudocyst: Abundant acellular proteinaceous myxoid matrix material (H and E, ×40) with characteristic drying artifacts (inset) (H and E, ×40)|
Click here to view
Biochemical profile of the aspirate was comparable to that of the serum, especially with regard to total protein, albumin, and glucose concentration; except the LDH level, which measured 6130 U/L, several folds higher than the serum, where the level was within the normal range.
The typical history, location, and clinicopathological findings, corroborated with biochemical profile of the aspirated fluid actually confirmed the diagnosis of auricular pseudocyst. Residual part of the fluid was then aspirated from both lesions, followed by administration of intralesional triamcinolone injection and the lesions completely resolved by 4 weeks without any residual deformity. Thereby, repeat surgery was avoided in this case.
| Discussion|| |
''Pseudocyst'' or ''Idiopathic cystic chondromalacia'' of auricle is a rare benign condition, typically presents as a painless, fluctuant cystic swelling, located commonly in the triangular and scaphoid fossa of the right pinna.  It occurs predominantly in young adults and 93% of them are male. Asynchronous involvement of the contralateral pinna is seen in upto10%-20% of successfully treated patients. But, as in our patient, bilaterality is encountered in 13% of cases only. , Chronic minor trauma inflicted by hard pillows, stereo headphones, and caps or helmets has been commonly implicated as an inducing factor of this condition, but this etiology is not undoubtedly proven. , In this present case, the patient was serving Indian military and his uniform-cap was a continuous source of irritation over the ears.
Clinical differential diagnoses of auricular pseudocyst commonly include, relapsing polychondritis, chondrodermatitis nodularis helicis, or traumatic perichondritis; and rarely hemangioma, chondroma, or angiosarcoma also poses potential differentials. But, its long-standing asymptomatic presentation, sparing of the overlying skin, frequent association with chronic trauma alongside characteristic cytological and biochemical properties differentiates it from its mimickers.
Karabulut et al., described the cytological features as aggregates of foamy macrophages in a proteineous background.  But as in the reported case, Salib et al.,  also experienced that aspirates from the pseudocyst yielded only a sterile, pale straw-colored viscid fluid, which on microscopic examination appeared to be a myxoid proteinaceous substance with no cellular component in it.
Auricular pseudocyst is a degenerative disease of the cartilage. Possible pathogenetic mechanism that has been proposed is, repeated trauma causing perichondral ischemia and cartilaginous degeneration, leading to the massive release of hemosiderin and serum lactate dehydrogenase (LDH) into the cartilaginous fluid. For this reason, similar to the reported case, many other studies have also acknowledged the usefulness of the cyst fluid LDH level in diagnosing it. , Although among all LDH isoenzymes-LDH 4 and LDH 5 , specifically predominate in the cystic fluid, even when serum LDH levels have been normal, but still the importance of total LDH level should not be underestimated. 
Various therapeutic modalities exist for auricular pseudocyst. However, irrespective of the method, the principle aim should always have to be the provision of best cosmetic outcome for the patient while using minimal invasive technique that provides no or minimal chance of recurrence. Surgical deroofing followed by compression dressing under local anesthesia is said to be the most effective treatment option with almost zero recurrence rate. But, this invasive method is associated with complications like, postoperative pain, pressure necrosis of the skin, and has poor patient compliance.  Incisional drainage or needle aspiration followed by compressive dressing is also one of the most commonly practised methods, but this is associated with a recurrence rate of about 38%.  Job and Raman  and Miyamoto et al.,  successfully applied aspiration followed by intracystic injection of steroids to treat their patients. Recurrent auricular pseudocysts have also been effectively treated by several authors with aspiration and oral/intralesional steroid administration. , The present case had one denovo lesion in his left ear and the other was a recurrent one. Both of these lesions were treated effectively by the above-mentioned method.
But, intralesional steroid administration is associated with disadvantages like, skin pigmentation; skin, soft tissue, and cartilage atrophy and systemic side effects.  But, these were not observed in the reported patient within 4 months following therapy.
| Conclusion|| |
Idiopathic cystic chondromalacia or pseudocyst of auricle is a rare and benign disease. Recurrent trauma is a controversial but yet noteworthy etiological factor. It can be diagnosed easily by experienced pathologists with the help of proper history, clinical presentation, FNAC features of the lesion as well as the raised LDH level in the aspirate. Needle aspiration and intralesional steroid administration is a simple and minimally invasive therapeutic modality, which can be a useful substitute for surgical procedures and in the reported case it gave an optimal cosmetic result. Moreover, this technique can also be applied confidently and successfully in bilateral and recurrent cases. Above all, the present case underlines the fact that FNAC with adjunctive fluid LDH estimation can lead to an early and definitive diagnosis of auricular pseudocyst which in turn can save the patient form unnecessary and in certain cases repeated surgery. Both patient satisfaction and quality of care can, thus, be improved substantially.
| References|| |
|1.||Kopera D, Soyer HP, Smolle J, Kerl H. "Pseudocyst of the auricle", othematoma and otoseroma: three faces of the same coin? Euro J Dermatol 2000;10:451-4. |
|2.||Engel D. Pseudocyst of the auricle in Chinese. Arch Otolaryngol 1966;83:197-202. |
|3.||Cohen RR, Grossman ME. Pseudocyst of the auricle. Case report and world literature review. Arch Otolaryngol Head Neck Surg 1990;116:1202-4. |
|4.||Wenig BM. The ear and temporal bone. In: Mills SE, editor. Sternberg′s Diagnostic Surgical Pathology. 5 th ed. Baltimore: Lipincott Williams and Wilkins, a Wolters Kluwer business; 2010. p. 935-6. |
|5.||Lee JA, Panarese A. Endochondral pseudocyst of the auricle. J Clin Pathol 1994;47:961-3. |
|6.||Glamb R, Kim R. Pseudocyst of the auricle. J Am Acad Dermatol 1984;11:58-63. |
|7.||Karabulut H, Acar B, Tuncay KS, Tanyildizli T, Karadag AS, Güresçi S, et al. Treatment of the non-traumatic auricular pseudocyst with aspiration and intralesional steroid injection. New J Med 2009;26:117-9. |
|8.||Salib RJ, Kaul GH, Oates J. Benign idiopathic cystic chondromalacia. CME Bull Otorhinilaryngol Head Neck Surg 2003;7:94-5. |
|9.||Miyamoto H, Okajima M, Takahashi I. Lactate dehydrogenase isozymes in and intralesional steroid injection therapy for pseudocyst of the auricle. Int J Dermatol 2001;40:380-4. |
|10.||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;59:1450-2. |
|11.||Kanotra SP, Lateef M. Pseudocyst of pinna: A recurrence-free approach. Am J Otolaryngol 2009;30:73-9. |
|12.||Job A, Raman R. Medical management of pseudocyst of the auricle. J Laryngol Otol 1992;106:159-61. |
|13.||Miyamoto H, Oida M, Onuma S, Uchiyama M. Steroid injection therapy for pseudocyst of the auricle. Acta Derm Venereol 1994;74:140-2. |
[Figure 1], [Figure 2]