Proliferating Pilar Tumor

Proliferating Pilar Tumor

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A proliferating pilar tumor (PPT) is a rare neoplasm arising from the isthmus region of the outer root sheath of the hair follicle. It is also commonly called a proliferating trichilemmal cyst or, less commonly, proliferating follicular-cystic neoplasm. It was first described by Wilson-Jones [1] as a proliferating epidermoid cyst in 1966. PPT was then distinguished from proliferating epidermoid cysts in 1995. [2] It occurs most commonly on the scalp in women older than 50 years. [3] Most tumors arise within a preexisting pilar cyst. Even though they usually are benign in nature, malignant transformation with local invasion and metastasis has been described. [4, 5, 6, 7, 8] A tentative stratification of PPTs into groups as benign, low-grade malignancy, and high-grade malignancy has been introduced. They may be inherited in an autosomal-dominant mode, linked to chromosome 3. See Pilar Cyst for more information.

A proliferating pilar tumor (PPT) usually arises in the setting of single or multiple pilar cysts. An asymptomatic nodule is often present for months to years before a rapid increase occurs in the size of the lesion. This is thought to signal neoplastic transformation of the cyst, with progression into a PPT. The underlying stimulus for this transformation is not known, but it has been hypothesized to be secondary to trauma, irritation, or chronic inflammation. In a review of 76 cases of PPTs seen in consultation from 1989-2000, Ye et al [9] proposed a distinction between benign and malignant variants. The authors also proposed histologic criteria that could predict behavior.

It is believed to be analogous to proliferating onycholemmal cyst, an entity that is even rarer in the literature as it pertains to the nail bed. [10]

A spectrum of transformation is hypothesized, which begins with a benign pilar cyst, proceeding to a proliferating pilar tumor (PPT), and then to a malignant PPT. The stimulus for changes in these lesions is currently unknown, although trauma, inflammation, and irritation may play roles.

Proliferating pilar tumor (PPT) are rare.

These tumors occur more commonly in females than in males.

PPTs are most common in older individuals aged 50-75 years, although they have been reported in individuals aged 20-30 years.

The prognosis is excellent with complete excision. Local recurrence and metastasis are extremely rare in benign cases. However, in proven malignant cases, metastasis has been suggested to occur in 30% of cases.

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Wilson-Jones E. Proliferating epidermoid cysts. Arch Dermatol. 1966. 94:11.

Sau P, Graham JH, Helwig EB. Proliferating epithelial cysts. Clinicopathological analysis of 96 cases. J Cutan Pathol. 1995 Oct. 22(5):394-406. [Medline].

López-Ríos F, Rodríguez-Peralto JL, Aguilar A, Hernández L, Gallego M. Proliferating trichilemmal cyst with focal invasion: report of a case and a review of the literature. Am J Dermatopathol. 2000 Apr. 22(2):183-7. [Medline].

Mehregan AH, Lee KC. Malignant proliferating trichilemmal tumors–report of three cases. J Dermatol Surg Oncol. 1987 Dec. 13(12):1339-42. [Medline].

Saida T, Oohara K, Hori Y, Tsuchiya S. Development of a malignant proliferating trichilemmal cyst in a patient with multiple trichilemmal cysts. Dermatologica. 1983. 166(4):203-8. [Medline].

Weiss J, Heine M, Grimmel M, Jung EG. Malignant proliferating trichilemmal cyst. J Am Acad Dermatol. 1995 May. 32(5 Pt 2):870-3. [Medline].

Ramaswamy AS, Manjunatha HK, Sunilkumar B, Arunkumar SP. Morphological spectrum of pilar cysts. N Am J Med Sci. 2013 Feb. 5(2):124-8. [Medline]. [Full Text].

Al-Saleh K, Hooda HS, El-Wakiel H, Safwat R, Bedair A, Eskaf W. Trichilemmal pilar tumor of the scalp: a case report. Gulf J Oncolog. 2012 Jul. 62-4. [Medline].

Ye J, Nappi O, Swanson PE, Patterson JW, Wick MR. Proliferating pilar tumors: a clinicopathologic study of 76 cases with a proposal for definition of benign and malignant variants. Am J Clin Pathol. 2004 Oct. 122(4):566-74. [Medline].

Alessi E, Zorzi F, Gianotti R, Parafioriti A. Malignant proliferating onycholemmal cyst. J Cutan Pathol. 1994 Apr. 21(2):183-8. [Medline].

Wang X, Yang J, Yang W. Multiple proliferating trichilemmal tumors in a middle-aged yellow man. Tumori. 2010 Mar-Apr. 96(2):349-51. [Medline].

Karaca S, Kulac M, Dilek FH, Polat C, Yilmaz S. Giant proliferating trichilemmal tumor of the gluteal region. Dermatol Surg. 2005 Dec. 31(12):1734-6. [Medline].

Avinoach I, Zirkin HJ, Glezerman M. Proliferating trichilemmal tumor of the vulva. Case report and review of the literature. Int J Gynecol Pathol. 1989. 8(2):163-8. [Medline].

Kang SJ, Wojno TH, Grossniklaus HE. Proliferating trichilemmal cyst of the eyelid. Am J Ophthalmol. 2007 Jun. 143(6):1065-7. [Medline].

Eskander A, Ghazarian D, Bray P, Dawson LA, Goldstein DP. Squamous cell carcinoma arising in a proliferating pilar (trichilemmal) cyst with nodal and distant metastases. J Otolaryngol Head Neck Surg. 2010 Oct. 39(5):E63-7. [Medline].

Lobo L, Amonkar AD, Dontamsetty VV. Malignant Proliferating Trichilemmal Tumour of the Scalp with Intra-Cranial Extension and Lung Metastasis-a Case Report. Indian J Surg. 2016 Dec. 78 (6):493-495. [Medline].

Trikudanathan G, Shaukat A, Bakman Y. Proliferating Pilar Tumor of Scalp Metastasizing to Pancreas: Diagnosis With Endoscopic Ultrasound-guided Fine-needle Aspiration. Clin Gastroenterol Hepatol. 2015 Nov. 13 (11):e164-5. [Medline].

Morgado B, Agostini P, Rivero A, Silva N. Extensive and ulcerated malignant proliferating trichilemmal (pilar) tumour, arising from multiple, large, degenerated trichilemmal (pilar) cysts. BMJ Case Rep. 2016 Feb 8. 2016:[Medline].

Chang SJ, Sims J, Murtagh FR, McCaffrey JC, Messina JL. Proliferating trichilemmal cysts of the scalp on CT. AJNR Am J Neuroradiol. 2006 Mar. 27(3):712-4. [Medline].

Gossner J, Larsen J. Incidental subcutaneous nodules on the scalp in patients undergoing CT of the brain; frequency, appearance, and differential diagnosis. Clin Radiol. 2010 May. 65(5):427-8. [Medline].

Leyendecker P, de Cambourg G, Mahé A, Imperiale A, Blondet C. 18F-FDG PET/CT Findings in a Patient With a Proliferating Trichilemmal Cyst. Clin Nucl Med. 2015 Jul. 40 (7):598-9. [Medline].

Trikudanathan G, Shaukat A, Bakman Y. Proliferating Pilar Tumor of Scalp Metastasizing to Pancreas: Diagnosis With Endoscopic Ultrasound-guided Fine-needle Aspiration. Clin Gastroenterol Hepatol. 2015 Apr 30. [Medline].

Shet T, Modi C. Nucleolar organizer regions (NORs) in simple and proliferating trichilemmal cysts (pilar cysts and pilar tumors). Indian J Pathol Microbiol. 2004 Oct. 47(4):469-73. [Medline].

González-Guerra E, Kutzner H, Rutten A, Requena L. Immunohistochemical study of calretinin in normal skin and cutaneous adnexal proliferations. Am J Dermatopathol. 2012 Jul. 34(5):491-505. [Medline].

Rangel-Gamboa L, Reyes-Castro M, Dominguez-Cherit J, Vega-Memije E. Proliferating trichilemmal cyst: the value of ki67 immunostaining. Int J Trichology. 2013 Jul. 5 (3):115-7. [Medline].

Chaichamnan K, Satayasoontorn K, Puttanupaab S, Attainsee A. Malignant proliferating trichilemmal tumors with CD34 expression. J Med Assoc Thai. 2010 Nov. 93 Suppl 6:S28-34. [Medline].

Masui Y, Komine M, Kadono T, Ishiura N, Maekawa T, Ihn H. Proliferating tricholemmal cystic carcinoma: a case containing differentiated and dedifferentiated parts. J Cutan Pathol. 2008 Oct. 35 Suppl 1:55-8. [Medline].

Alici O, Keles MK, Kurt A. A Rare Cutaneous Adnexal Tumor: Malignant Proliferating Trichilemmal Tumor. Case Rep Med. 2015. 2015:742920. [Medline].

Sutherland D, Roth K, Yu E. Malignant Proliferating Trichilemmal Tumor Treated with Radical Radiotherapy: A Case Report and Literature Review. Cureus. 2017 Jan 26. 9 (1):e999. [Medline].

Nyquist GG, Mumm C, Grau R, Crowson AN, Shurman DL, Benedetto P, et al. Malignant proliferating pilar tumors arising in KID syndrome: a report of two patients. Am J Med Genet A. 2007 Apr 1. 143(7):734-41. [Medline].

Marjon Vatanchi, MD Resident Physician, Department of Dermatology, State University of New York Downstate Medical Center

Marjon Vatanchi, MD is a member of the following medical societies: American Academy of Dermatology, American Society for Dermatologic Surgery, Dermatologic Society of Greater New York, Medical Dermatology Society, National Psoriasis Foundation, Society for Pediatric Dermatology, Texas Dermatological Society, Women’s Dermatologic Society

Disclosure: Nothing to disclose.

Raman K Madan, MD Resident Physician, Department of Dermatology, State University of New York Downstate Medical Center

Disclosure: Nothing to disclose.

Daniel Mark Siegel, MD, MS Clinical Professor of Dermatology, Department of Dermatology, State University of New York Downstate Medical Center

Daniel Mark Siegel, MD, MS is a member of the following medical societies: American Academy of Dermatology, American College of Mohs Surgery, American Association for Physician Leadership, American Society for Dermatologic Surgery, American Society for MOHS Surgery, International Society for Dermatologic Surgery

Disclosure: Nothing to disclose.

Rashid M Rashid, MD, PhD Director, Mosaic Clinic Hair Transplant Center of Houston

Rashid M Rashid, MD, PhD is a member of the following medical societies: American Academy of Dermatology, Texas Medical Association, Texas Dermatological Society, International Society of Hair Restoration Surgery, Council for Nail Disorders, Houston Dermatological Society

Disclosure: Nothing to disclose.

Richard P Vinson, MD Assistant Clinical Professor, Department of Dermatology, Texas Tech University Health Sciences Center, Paul L Foster School of Medicine; Consulting Staff, Mountain View Dermatology, PA

Richard P Vinson, MD is a member of the following medical societies: American Academy of Dermatology, Texas Medical Association, Association of Military Dermatologists, Texas Dermatological Society

Disclosure: Nothing to disclose.

John G Albertini, MD Private Practice, The Skin Surgery Center; Clinical Associate Professor (Volunteer), Department of Plastic and Reconstructive Surgery, Wake Forest University School of Medicine; President-Elect, American College of Mohs Surgery

John G Albertini, MD is a member of the following medical societies: American Academy of Dermatology, American College of Mohs Surgery

Disclosure: Serve(d) as a director, officer, partner, employee, advisor, consultant or trustee for: QualDerm Partners; Novascan<br/>Have a 5% or greater equity interest in: QualDerm Partners – North Carolina.

William D James, MD Paul R Gross Professor of Dermatology, Vice-Chairman, Residency Program Director, Department of Dermatology, University of Pennsylvania School of Medicine

William D James, MD is a member of the following medical societies: American Academy of Dermatology, Society for Investigative Dermatology

Disclosure: Received income in an amount equal to or greater than $250 from: Elsevier; WebMD.

R Stan Taylor, MD The JB Howell Professor in Melanoma Education and Detection, Departments of Dermatology and Plastic Surgery, Director, Skin Surgery and Oncology Clinic, University of Texas Southwestern Medical Center

R Stan Taylor, MD is a member of the following medical societies: American Academy of Dermatology, American College of Mohs Surgery, American Medical Association

Disclosure: Nothing to disclose.

Maria Christina Kessides, MD, MS Resident Physician, Department of Dermatology, State University of New York Downstate Medical Center

Disclosure: Nothing to disclose.

Amor Khachemoune, MD, CWS Mohs Micrographic Surgery, Dermatopathology, Department of Dermatology, State University of New York Downstate Medical Center; Consulting Staff, Department of Dermatology, Veterans Affairs Medical Center of Brooklyn

Amor Khachemoune, MD, CWS is a member of the following medical societies: American Academy of Dermatology, American Academy of Wound Management, American College of Mohs Micrographic Surgery and Cutaneous Oncology, American Medical Association, American Society for Dermatologic Surgery, and American Society for Laser Medicine and Surgery

Disclosure: Nothing to disclose.

Steve A McClain, MD Clinical Associate Professor, Department of Pathology, The School of Medicine at Stony Brook University Medical Center; Medical Director, Founder and Owner, McClain Laboratories LLC

Steve A McClain, MD is a member of the following medical societies: American Society for Clinical Pathology and College of American Pathologists

Disclosure: McClain Laboratories, LLC Ownership interest Management position

Kimberly Silvers, MD, PhD Staff Physician, Section of Dermatology, Guthrie Clinic

Kimberly Silvers, MD, PhD is a member of the following medical societies: American Academy of Dermatology and Phi Beta Kappa

Disclosure: Nothing to disclose.

Proliferating Pilar Tumor

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