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CASE REPORT  
Year : 2011  |  Volume : 54  |  Issue : 1  |  Page : 150-151
Cutaneous ciliated cyst in the subcutaneous area


Department of Pathology, Region Education and Research Hospital, Erzurum, Turkey

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Date of Web Publication7-Mar-2011
 

   Abstract 

A 25-year-old woman was seen for a painless subcutaneous mass of 2 years duration. On excisional biopsy, a collapsed cystic structure lined by stratified, ciliated, columnar epithelium was noted. These linning cells did not produce mucin. Immunohistochemical staining for progesterone receptor, estrogen receptor and epithelial membrane antigen was positive, whereas it was negative for carcinoembryonic antigen. Findings were consistent with cutaneous ciliated cyst (CCC). CCCs are rare, predominantly occurring on the lower extremities of young women. Most of them have been regarded as Mullerian remnants. A case of a CCC in the subcutaneous area is reported.

Keywords: Cutaneous ciliated cyst, immunohistochemistry, subcutaneous area

How to cite this article:
Gelincik I. Cutaneous ciliated cyst in the subcutaneous area. Indian J Pathol Microbiol 2011;54:150-1

How to cite this URL:
Gelincik I. Cutaneous ciliated cyst in the subcutaneous area. Indian J Pathol Microbiol [serial online] 2011 [cited 2019 Nov 14];54:150-1. Available from: http://www.ijpmonline.org/text.asp?2011/54/1/150/77380



   Introduction Top


Cutaneous ciliated cyst (CCC) is an unusual cystic lesion of the skin, which preferentially affects the lower limbs of young females. [1] Cutaneous cysts lined by ciliated epithelium are very rare. [2] Only 40 well-documented cases of cutaneous ciliated cysts have been reported in the published work. CCC typically presents as a single unilocular or multilocular subcutaneous cyst lined by epithelium, histologically reminiscent of  Fallopian tube More Details mucosa, on the lower extremity or buttock of young girls, shortly after puberty. [3]


   Case Report Top


A 25-year-old woman presented with a 2-year history of a mass in the subcutaneous area. According to the patient, the mass was painless, but had gradually increased in size. Laboratory investigation results including complete blood cell count and blood chemistry were normal. After surgical removal, examination of the cyst wall resulted in the escape of clear serous fluid from the cystic cavity. Histopathologic examination revealed a uni-loculated cyst located in the deep dermis. Scanning magnification revealed a collapsed cystic structure lined by stratified, ciliated, columnar epithelium, resembling the fimbriated portion of the fallopian tube [Figure 1]. Immunohistochemical analysis revealed positive staining for epithelial membrane antigen and cytoplasmic staining for keratin, which proved the epithelial origin of the tumor. The cyst did not stain for carcinoembryonic antigen and S-100. No evidence of muscular differentiation was observed, as evidenced by the negative staining of desmin and actin. There was a strong nuclear staining for progesterone receptor and estrogen receptor [Figure 2] in many of the epithelial cells. Inflammatory infiltrate was absent in and around the cyst.
Figure 1: Histopathologic features of a cystic structure lined by stratified, ciliated, columnar epithelium in the dermis [H and E, (a) ×100; (b) ×100]

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Figure 2: Cutaneous ciliated cyst. (a) Epithelial cells are positive for progesterone receptors (IHC, ×400); (b) epithelial cells are positive for estrogen receptors (IHC, ×400)

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   Discussion Top


Since the original report of CCC by Hess [4] in 1890, there have been only 40 reported cases of CCCs. [5],[6],[7] On electron microscopy, the ciliary shaft contains an array of nine peripheral microtubule pairs arranged in a circle around two single microtubules. [8],[9] This finding is identical to that of normal human ciliated cells found in the epithelium of the airway, fallopian tube, middle ear and in the ependymal lining of the brain and spinal cord. Also, similar immunohistochemical staining patterns for dynein, which functions as microtubule-based molecular motors, are seen in both cutaneous ciliated cysts and fallopian tube epithelium. [10] Negative carcinoembryonic antigen and S-100 with positive staining for estrogen receptor and/or progesterone receptor verified the CCCs' nature to be of fallopian tube epithelium. [11] In the present case, immunohistochemically, there was a strong nuclear staining for progesterone receptor and estrogen receptor in many of the epithelial cells. The cyst did not stain for carcinoembryonic antigen and S-100.

A CCC is a benign cystic lesion that is often a palpable subcutaneous mass without any symptoms. In this case, immunohistochemical analysis for the presence of progesterone receptors and estrogen receptor was positive, which supports the theory of heteropia of the ciliated epithelium from the Mullerian epithelium because the Mullerian duct is the precursor of the fallopian tubes and the body and cervix of the uterus, which are known to stain for progesterone receptors and estrogen receptors. [12]

It is believed that cutaneous ciliated cysts are a distinct entity occurring in females, probably arising from hormonal activation of a Mullerian rest.

 
   References Top

1.Farmer ER, Helwing EB. Cutaneous ciliated cysts. Arch Dermatol 1978;114:70-3.  Back to cited text no. 1
    
2.Sidoni A, Bucciarelli E. Ciliated cyst of the perineal skin. Am J Dermatopathol 1997;19:93-6.  Back to cited text no. 2
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3.Fontaine DG, Lau H, Murray SK, Fraser RB, Wright Jr 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. 3
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4.Hess K. Ueber eine subcutane flimmerzyste. Beitr Pathol 1890;8:98-109.  Back to cited text no. 4
    
5.Fontaine DG, Lau H, Murray SK, Fraser RB, Wright JR 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. 5
[PUBMED]  [FULLTEXT]  
6.Vadmal MS, Makarewicz K, Fontaine DG, Wright JR Jr. Cutaneous ciliated cyst of the abdominal wall. Am J Dermatopathol 2002;24:452-3.  Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.Santos LD, Mendelsohn G. Perineal cutaneous ciliated cyst in a male. Pathology 2004;36:369-70.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.Tachibana T, Sakamoto F, Ito M, Ito K, Kaneko Y, Takenouchi 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
[PUBMED]    
9.Fontaine DG, Lau H, Murray SK, Fraser RB, Wright Jr 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. 9
[PUBMED]  [FULLTEXT]  
10.Dini M, Lo Russo G, 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. 10
[PUBMED]  [FULLTEXT]  
11.Lee JS, Kim YC, Lee ES. Cutaneous ciliated cyst of the inguinal area in a man. J Dermatol 2006;2:146-9.  Back to cited text no. 11
    
12.Coppens MT, Boever JG, Dhont MA, Serreyn RF, Vandekerckhove DA, Roels HJ. Topographic distribution of estrogen and progesterone receptors in the human endometrium and fallopian tube. Histochemistry 1993;99:127-31.  Back to cited text no. 12
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Correspondence Address:
Ibrahim Gelincik
Bolge Egitim ve Arastirma Hastanesi, Patoloji Klinigi Cat Yolu cad, Yildizkent-Erzurum
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.77380

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    Figures

  [Figure 1], [Figure 2]

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