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Yalçın AC, Yalçın N, Çakmak Hİ, Bedir R. Cutaneous Ciliated Cysts: 2 Case Reports and Review of Literature. Am J Dermatopathol 2024; 46:774-780. [PMID: 38941541 DOI: 10.1097/dad.0000000000002777] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/30/2024]
Abstract
ABSTRACT Cutaneous ciliated cysts are rare benign lesions most commonly seen in the lower extremities of young female patients. There is still ongoing debate about the origin of these cysts, with the most common theories being Mullerian heterotopia and eccrine gland metaplasia. To further increase the data pool, we present 2 new cutaneous ciliated cyst cases and gathered data about all 67 cases of literature. We want to highlight that immunohistochemistry and demographic information may play a key role in distinguishing between the 2 possible origins of this cyst, namely estrogen receptor, progesterone receptor, and carcinoembryonic antigen immunohistochemistry. Estrogen receptor and progesterone receptor nuclear positivity is proof of Mullerian origin, but when they are negative with a positive carcinoembryonic antigen, the most likely origin is eccrine gland metaplasia. We advocate using the terms "Cutaneous Mullerian Cyst" and "Ciliated Cutaneous Eccrine Cyst" instead of "Cutaneous Ciliated Cyst" to accurately indicate their distinct origins.
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Affiliation(s)
- Anıl Can Yalçın
- Department of Pathology, Medical Faculty, Recep Tayyip Erdogan University, Rize, Turkey; and
| | - Nazlıcan Yalçın
- Department of Pathology, Medical Faculty, Recep Tayyip Erdogan University, Rize, Turkey; and
| | - Halil İbrahim Çakmak
- Department of Pediatric Surgery, Medical Faculty, Recep Tayyip Erdogan University, Rize, Turkey
| | - Recep Bedir
- Department of Pathology, Medical Faculty, Recep Tayyip Erdogan University, Rize, Turkey; and
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Kim YH, Lee J. Cutaneous ciliated cyst on the anterior neck in young women: A case report. World J Clin Cases 2020; 8:4481-4487. [PMID: 33083407 PMCID: PMC7559661 DOI: 10.12998/wjcc.v8.i19.4481] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/01/2020] [Revised: 08/07/2020] [Accepted: 08/31/2020] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND A cutaneous ciliated cyst (CCC) is a rare, benign tumor in young female adults, which is usually found on the lower extremities.
CASE SUMMARY We found an uncommon location of CCC in the anterolateral cervical area and reviewed the literature. A 20-year-old female complained of a well-defined, painless, palpable mass that started several years ago. The mass was tense and movable and located at the anterolateral aspect of the neck. Imaging showed a non-enhancing round mass. Surgical excision biopsy was performed, and the cystic mass was revealed to be a CCC.
CONCLUSION The rare location of CCC can be found in anterior neck area, which should be another diagnostic option for mass on anterior neck.
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Affiliation(s)
- Yon Hee Kim
- Department of Pathology, Soonchunhyang University Seoul Hospital, Seoul 04401, South Korea
| | - Jihyoun Lee
- Department of Surgery, Soonchunhyang University Seoul Hospital, Seoul 04401, South Korea
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The Cutaneous Ciliated Cyst in Young Male: The Possibility of Ciliated Cutaneous Eccrine Cyst. Case Rep Med 2015; 2015:589831. [PMID: 26491452 PMCID: PMC4605210 DOI: 10.1155/2015/589831] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/06/2015] [Accepted: 09/15/2015] [Indexed: 12/19/2022] Open
Abstract
Cutaneous ciliated cyst was described as a painless cyst occurring on the lower limbs of women between the ages of 15 and 30 years. The cysts are typically lined by ciliated cuboidal to columnar epithelium with pseudostratified areas and focal squamous metaplasia is occasionally present. Immunohistochemical studies have demonstrated that the cysts are PR and ER positive, similar to the epithelia of the fallopian tubes. However, outliers of cutaneous ciliated cysts, including those in male patients and in unexpected locations such as the scalp, finger, and scapular area, have been reported. Thus, some hypotheses have been proposed including the Mullerian heterotopias, ciliated metaplasia of eccrine sweat glands, and embryonic remnants of the cloacal membrane. We report a rare case of cutaneous ciliated cyst on the left shoulder of a 7-year-old boy and this is the eighth case of cutaneous ciliated cyst in male patients. Moreover, through reviewing the articles, we try to propose the classification of the cutaneous ciliated cysts into the cutaneous Mullerian cysts and the ciliated cutaneous eccrine cysts.
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Lezcano C, Chaux A, Velazquez EF, Cubilla AL. Clinicopathological features and histogenesis of penile cysts. Semin Diagn Pathol 2015; 32:245-8. [DOI: 10.1053/j.semdp.2014.12.014] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/28/2022]
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Cutaneous ciliated cyst of the scalp: a case report of a cutaneous ciliated eccrine cyst and a brief review of the literature. Am J Dermatopathol 2015; 36:679-82. [PMID: 25051045 DOI: 10.1097/dad.0b013e3182a00c09] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
Cutaneous ciliated cysts (CCC) are rare benign cysts known to occur in the lower extremities of females of reproductive age. Currently, there are 2 theories that attempt to explain the histogenesis of this rare entity. The theory of Mullerian heterotopia provides a plausible histogenetic explanation for the vast majority of CCC. A proposed alternative theory is the ciliated metaplasia of eccrine glands. We believe that previously reported cases of CCC include 2 distinct entities. We report, herein, the first case reported in the literature of a cutaneous ciliated eccrine cyst occurring on the scalp.
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Abstract
Cutaneous ciliated cysts (CCC) are exquisitely rare, benign cystic lesions demonstrating simple, ciliated epithelial linings reminiscent of fallopian tube epithelium. Most commonly, CCC show a predilection for the lower extremities of young reproductive age women and demonstrate immunohistochemical positivity for estrogen and progesterone receptors, supporting the theory that they are derived from ectopic Müllerian rests. PAX-8 is a paired box gene, important in the development of Müllerian and thyroid organs and has utility in the identification of tumors of Müllerian, renal, and thyroid origin. Prompted by the precedent studies on PAX-8 immunohistochemical expression in tumors of Müllerian origin, this article aimed to explore the utility of this antibody in defining the histogenesis of 2 bona fide cases of CCC, both occurring in young reproductive age women. Herein, 2 prototypic index cases of CCC with strong nuclear positivity for estrogen and progesterone receptors are shown to also have positive nuclear staining for PAX-8, further supporting their likely Müllerian origin. These data support the designation of these lesions as cutaneous Müllerian cysts, distinct from potential ciliated cysts of eccrine origin.
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Abstract
As previously recognized by various authors, "cutaneous ciliated cyst" is a confusing term. Typically, the term refers to rare cystic lesions, commonly found on the lower limbs of women in their reproductive years. To date, 40 cases diagnosed as "cutaneous ciliated cyst" have been reported in the literature. Histologically, the cysts are composed of a simple layer of ciliated columnar cells along with nonciliated columnar cells, cuboidal cells, and round "peg-like" cells, resembling fallopian tube epithelium. This histology has been described in cysts found in males and females and in locations other than the lower limbs. Controversy has thus arisen over the etiology of these lesions, with some believing that the cysts arise from heterotopic Mullerian rests and others advocating for ciliated metaplasia of eccrine glands. We herein describe the first case of cutaneous ciliated cyst of Mullerian origin occurring on the dorsal thumb of a 16-year-old female. A review of literature shows that 2 groups of cysts are covered under the umbrella term "cutaneous ciliated cysts." We thus propose the abandonment of the confusing term "cutaneous ciliated cyst" and the adoption of "cutaneous Mullerian cysts" for estrogen receptor/progesterone receptor-positive lesions resembling simple fallopian tube epithelium and "Cutaneous ciliated eccrine cyst" for estrogen receptor/progesterone receptor-negative lesions usually occurring in males, which are immunohistochemically compatible with an eccrine origin.
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Affiliation(s)
- Amrut V Ashturkar
- Department of Pathology, Shrimati Kashibai Navale Medical College and General Hospital, Narhe, Pune, India. E-mail:
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Cutaneous ciliated cyst: a case report with focus on mullerian heterotopia and comparison with eccrine sweat glands. Am J Dermatopathol 2011; 32:731-4. [PMID: 20644463 DOI: 10.1097/dad.0b013e3181d43f01] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/14/2023]
Abstract
Cutaneous ciliated cyst is an exceedingly rare, benign lesion most commonly found in the dermis or subcutis of the lower extremities of young female patients in their second and third decades. The pathogenesis of the cyst is unknown. We report a cutaneous ciliated cyst in the lower extremity of a 13-year-old female patient. On histologic examination, clusters of eccrine sweat glands were observed adjacent to the cyst. Upon comparison of the immunohistochemical profile of the cutaneous ciliated cyst and the eccrine sweat glands, they appeared almost completely unrelated. The histologic, immunohistochemical, and ultrastructural findings of this case and the literature provide evidence in favor of the Mullerian heterotopia theory.
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Pérez Valcárcel J, Peón Currás G, Sánchez Arca ME, Rodríguez Gómez I, Sousa Escandón A. [Cutaneous ciliated cyst of the scrotal skin. A case report with discussion of pathogenesis]. Actas Urol Esp 2009; 32:843-6. [PMID: 19013984 DOI: 10.1016/s0210-4806(08)73944-5] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/15/2023]
Abstract
Cutaneous ciliated cyst (CCC) is a rare benign lesion predominantly occurring in the lower limbs of young women and exceedingly rare in males. Here, we report a case involving a previously unreported site (i.e., scrotal skin) in a 15-year-old male. We also describe pathologic and immunonohistochemical findings, review the pertinent literature and discuss their pathogenetic mechanisms. We propose that CCC could represent a morphologic pattern encompassing several pathogenetically different entities. Data we provide support the hypothesis that at least a part of CCC, specially those occurring in males, could have their origin in ciliated metaplasia of apocrine sweat glands.
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Abstract
Cutaneous ciliated cysts are rare, predominantly occurring on the lower extremities of young women. Most of them have been regarded as müllerian remnants. Cases in men are rarely reported. We report a case of a cutaneous ciliated cyst involving the inguinal area of a 56-year-old man and briefly review the published work.
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Affiliation(s)
- Joong Sun Lee
- Department of Dermatology, Ajou University School of Medicine, Suwon, Republic of Korea
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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. [PMID: 11803285 DOI: 10.1097/00000372-200202000-00013] [Citation(s) in RCA: 25] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
A cutaneous ciliated cyst is a rare lesion typically found on the lower extremity of young girls shortly after puberty. Here, we report a case involving a previously unreported site (i.e., abdominal wall) in a 14-year-old girl. We also describe immunohistochemical and ultrastructural findings, review and analyze the world literature, and offer insights as to the pathogenesis of this lesion.
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Affiliation(s)
- Daniel G Fontaine
- Department of Pathology, IWK Health Center and Queen Elizabeth II Health Center, Dalhousie University Faculty of Medicine, Halifax, Nova Scotia, Canada
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Affiliation(s)
- Norihiro Ohba
- Department of Plastic and Reconstructive Surgery, Osaka City University Medical School, Osaka, Japan.
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Abstract
We report a case of the unusual location of a cutaneous bronchogenic cyst on the abdominal wall. The patient was a 9-month-old boy who had presented with a 1.5 cm-sized polypoid mass, present since birth. Pathological examination of the excised mass revealed multiple small cystic structures surrounded by the fibroadipose tissue. The lining epithelium consisted of either pseudostratified ciliated columnar epithelium with goblet cells or a single layer of ciliated or non-ciliated cuboidal to columnar cells. The cystic walls contained a well-developed smooth muscle bundle, mucous glands and hyaline cartilage plate. This lesion was adherent to the peritoneum, but there was no direct communication with the abdominal cavity. Cutaneous bronchogenic cyst located in the abdominal wall has not been described in the English literature. The present case suggests a possible origin from a downward migration, from the sequestered bud of a tracheobronchial tree primordium along the midline of the body surface, during embryonic development.
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Affiliation(s)
- N R Kim
- Department of Pathology, Kangnam General Hospital Public Corporation, Korea
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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. [PMID: 11190444 DOI: 10.1097/00000372-200012000-00007] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
Cutaneous ciliated cysts (CCCs) are rare benign lesions predominantly occurring in the lower limbs of young women. We observed such a lesion in the perineal region of a 12-year-old girl. The histogenetic interpretation of CCCs is controversial. The similarity of the epithelial lining of the cyst to the salpingeal epithelium supports the hypothesis of a Müllerian heterotopia. Strong dynein positivity observed immunohistochemically in the apical portion of CCC lining cells suggests the integrity of the ciliary apparatus. There was a marked similarity between a CCC and normal salpingeal epithelium in the mode of staining for dynein, whereas the dynein reactivity of bronchial epithelial cells showed larger cytoplasmic aggregates of positive material in proximity to the nucleus. No immunohistochemical staining for estrogen or progesterone receptors was identified in our observation, although this finding could have supported the Müllerian histogenetic hypothesis. To the best of our knowledge, this is the first time that antidynein immunohistochemistry has been applied to paraffin-embedded samples from human surgical pathology.
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Affiliation(s)
- M Dini
- Department of Plastic Surgery, University of Florence, Italy
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Abstract
A 23-year-old Japanese woman with a cutaneous ciliated cyst on her right lower leg is reported. A subcutaneous cyst, measuring 2.5 cm in diameter with papillary projections into the lumen, was lined with ciliated cuboidal to columnar epithelia with partial stratification, histologically. These lining cells did not produce mucin. Immunohistochemically, the ciliated lining cells of the cyst were diffusely positive to epithelial membrane antigen and cytokeratin. In addition, positive immunoreaction with anti-desmin monoclonal antibody was observed in the body of the cilia. Less than 10% of the epithelial cells revealed positive immunoreaction to S-100 protein and estrogen receptor.
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Affiliation(s)
- H Yokozaki
- The First Department of Pathology, Hiroshima University School of Medicine, Japan.
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