Journal of Cutaneous and Aesthetic Surgery
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   Table of Contents     
LETTER  
Year : 2011  |  Volume : 4  |  Issue : 2  |  Page : 158-159
Cutaneous ciliated cyst over knee


Department of Pathology, Shrimati Kashibai Navale Medical College and General Hospital, Narhe, Pune, India

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Date of Web Publication17-Sep-2011
 

How to cite this article:
Ashturkar AV, Pathak GS, Joshi AR. Cutaneous ciliated cyst over knee. J Cutan Aesthet Surg 2011;4:158-9

How to cite this URL:
Ashturkar AV, Pathak GS, Joshi AR. Cutaneous ciliated cyst over knee. J Cutan Aesthet Surg [serial online] 2011 [cited 2019 Sep 15];4:158-9. Available from: http://www.jcasonline.com/text.asp?2011/4/2/158/85051


Sir,

Cutaneous ciliated cysts are very unusual benign lesions exclusively occurring on the lower extremity of young females shortly after puberty. [1] They have been widely regarded as Müllerian heterotopias because of the morphological similarity of the cyst lining cells to the epithelium of  Fallopian tube More Detailss. [2] We report a case of an 18-year-old female presented in orthopaedic outpatient department with gradually increasing, painless swelling, over right knee joint since 4 years. On examination, a soft to cystic, solitary, movable, nontender, fluctuant swelling, measuring 4 × 4 cm in size was noted. There was no abnormality of the overlying skin. Ultrasonography confirmed the cystic nature of the lesion. There was no continuation between the cyst and knee joint. Surgical removal of the cyst was carried out under local anaesthesia. Grossly, we received a skin covered, cystic specimen measuring 3.9 × 3.5 × 3.0 cm in size. On cutting open, an uniloculated cyst, measuring 3 cm in diameter was identified, which contained serous fluid. The cyst wall was thin, smooth, and greyish white in colour. Light microscopy revealed a cyst in the deep dermis, which was predominantly lined by single layer of ciliated cuboidal to columnar cells [Figure 1]. At places the lining epithelium showed stratification and squamous metaplasia [Figure 2]. These lining epithelial cells did not contain mucin. The cyst wall was thin, fibrocollagenous without any inflammatory infiltrate. On immunohistochemical staining, the lining epithelial cells showed strong membrane positivity for epithelial membrane antigen and cytoplasmic positivity for cytokeratin (PanCK AE1/AE3) [Figure 3]. Strong nuclear positivity for oestrogen receptor (ER) and progesterone receptor (PR) was noted within the epithelial cells [Figure 4]. Immunohistochemical staining for carcinoembryonic antigen (CEA) and S-100 was negative.
Figure 1: Photomicrograph showing a thin-walled cyst in the deep dermis lined by ciliated cuboidal to columnar cells (H and E, ×40). Inset: closer view of the ciliated epithelium (H and E, ×400)

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Figure 2: Focal squamous metaplasia of the lining epithelium of cyst (H and E, ×100)

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Figure 3: Lining epithelial cells showing (a) cytoplasmic positivity for cytokeratin (×100) and (b) strong membrane positivity for epithelial membrane antigen

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Figure 4: Lining epithelial cells showing nuclear positivity for (a) progesterone receptor and (b) oestrogen receptor (×100)

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Cutaneous cysts are rare benign lesions, lined by a simple cuboidal to columnar ciliated epithelium, seen typically on lower extremity in females in the second or third decade of life. [3] One case has been reported in a 51-year-old postmenopausal female patient. [4] Ciliated cutaneous cysts have also been reported at unusual sites, such as abdominal wall and posterior mediastinum. [1],[5] Cases of cutaneous ciliated cysts in males in perianal and inguinal area have also been documented in the literature. [6] In our case, the patient was an 18-year-old female with a cyst over right knee joint. [Table 1] summarises the previous reports of cutaneous ciliated cysts highlighting the age and sex of the patient with their sites.
Table 1: Summary of the previous reports on cutaneous ciliated cysts

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On light microscopy, the cysts are uniloculated and are lined by ciliated cuboidal to columnar epithelium without mucous cells, morphologically similar to the epithelium of fallopian tubes. [2] Immunohistochemical staining for PR, ER, cytokeratin, and epithelial membrane antigen were positive, whereas it was negative for CEA, which supports the theory of heterotopia of the ciliated epithelium from the Müllerian epithelium in its histopathogenesis. [12] The positivity of lining epithelium for ER and PR and occurrence of this lesion in second decade after puberty also suggests Müllerian origin. This supports the hypothesis, which suggests that the cells from the fimbrial ends of the fallopian tubes developing from the Müllerian ducts possibly detach and become incorporated into the lateral mesoderm where the lower limb buds arise. These arrested cells then remain dormant until puberty after which, under the influence of ovarian hormone stimulation, the heterotopic Müllerian epithelium produces serous fluid, resulting in cystic formation. [2] Ciliated metaplasia of eccrine or apocrine duct epithelial is another hypothesis documented for histogenesis of ciliated cyst, which explains rare occurrence of cutaneous ciliated cysts in male. [1],[4],[11] There is marked similarity between the cutaneous ciliated cyst lining and normal salpingeal epithelium in the mode of staining for dynein. [13]

This lesion shares its cutaneous origin with other cutaneous cysts, such as bronchogenic cyst and thyroglossal cyst. [14] However, the location of lower extremity, absence of mucous glands, cartilage and inflammation, positivity for ER and negativity for CEA establishes the diagnosis of cutaneous ciliated cyst.

Surgical removal under local anaesthesia is the recommended treatment for cutaneous ciliated cyst. [4] The recurrence has not been reported in the literature. In the present case also we followed-up the patient for 6 months without any signs of recurrence.

In conclusion, although rare, the possibility of ciliated cutaneous cyst should be considered in a young female presenting with cystic lesion on lower extremity because of its distinct Müllerian histogenesis.

 
   References Top

1.Fontaine DG, Lau H, Murray SK, Fraser RB, Wright JR. Cutaneous ciliated cyst of the abdominal wall: A case report with a review of the literature and discussion of pathogenesis. Am J Dermatopathol 2002;24:63-6.  Back to cited text no. 1
    
2.Al-Nafussi AI, Carder P. Cutaneous ciliated cyst: A case report and immunohistochemical comparison with fallopian tube. Histopathology 1990;16:595-8.  Back to cited text no. 2
    
3.Farmer ER, Helwig EB. Cutaneous ciliated cysts. Arch Dermatol 1978;114:70-3.  Back to cited text no. 3
    
4.Torisu-Itakura H, Itakura E, Horiuchi R, Matsumura M, Kiryu H, Takeshita T, et al. Cutaneous ciliated cyst on the leg in a woman of menopausal age. Acta Derm Venereol 2009;89:323-4.  Back to cited text no. 4
    
5.Businger AP, Frick H, Sailer M, Furrer M . A ciliated cyst in the posterior mediastinum compatible with a paravertebral Mullerian cyst. Eur J Cardiothorac Surg 2008;33:133-6.  Back to cited text no. 5
    
6.Ashton MA. Cutaneous ciliated cyst of the lower limb in a male. Histopathology 1994;26:467-9.  Back to cited text no. 6
    
7.Yokozaki H, Yanagawa E, Harada M, Tahara E. Cutaneous ciliated cyst of the right lower leg. Pathol Intl 1999;49:354-7.  Back to cited text no. 7
    
8.Tachibana T, Sakamoto F, Ito M, Ito K, Kaneko Y, Takenuchi T. Cutaneous ciliated cyst: A case report and histochemical, immunohistochemical and ultrastructural study. J Cutan Pathol 1995;22:33-7.  Back to cited text no. 8
    
9.Ohba N, Tsuruta D, Muraoaka M, Haba T, Ishii M. Cutaneous ciliated cyst on the cheek in a male. Intl J Dermatol 2002;41:48-9.  Back to cited text no. 9
    
10.Santos LD, Mendelson G. Perineal cutaneous cyst in a male. Pathology 2004;36:369-70.  Back to cited text no. 10
    
11.Valcárcel PJ, Currás PG, Arca SM, Gómez RI, Escandón A. Cutaneous ciliated cyst of the scrotal skin. A case report with discussion of pathogenesis. Actas Urol Esp 2008;32:843-6.  Back to cited text no. 11
    
12.Chong SJ, Kim SY, Kim HS, Kim GM, Kim SY, Jung JH. Cutaneous ciliated cyst in a 16-year-old girl. J Am Acad Dermatol 2007;56:159-60.  Back to cited text no. 12
    
13.Dini M, Russo GL, Baroni G, Colafranceschi M. Cutaneous ciliated cyst: a case report with immunohistochemical evidence for dynein in ciliated cells. Am J Dermatopathol 2000;22:519-23.  Back to cited text no. 13
    
14.Scott GA. Cutaneous cysts and related lesions. In: Barnhill RL, editor. Textbook of dermatopathology. 2 nd ed. New York, NY: McGraw-Hill; 2004. p. 561-73.  Back to cited text no. 14
    

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Correspondence Address:
Amrut V Ashturkar
Department of Pathology, Shrimati Kashibai Navale Medical College and General Hospital, Narhe, Pune
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0974-2077.85051

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]

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