Pilar neurocristic hamartoma: its relationship to blue nevus and equine melanotic disease.
Abstract: A unique pigmented lesion, judged to be a hamartoma of neural crest origin, occurring in a female patient, is compared with equine melanotic disease, The characteristic perifollicular arrangement of pigment-laden spindle cells is remarkably similar in both. Previously described patch- and plaque-like blue nevi in humans are also closely related. Light and ultrastructural features showed differentiation toward both nevus cells and Schwann cells, and it is proposed that the lesion be termed pilar neurocristic hamartoma.
Publication Date: 1982-08-01 PubMed ID: 7103529DOI: 10.1001/archderm.118.8.592Google Scholar: Lookup
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Summary
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The research report investigates a unique pigmented lesion of neural crest origin, termed as pilar neurocristic hamartoma, that occurred in a female patient and reveals its similarity to equine melanotic disease and human blue nevi, evident from the arrangement of pigment-loaded spindle cells around the hair follicle.
Elaboration of The Research Paper
- The study focuses on a rare pigmented lesion, identified as a hamartoma, which is a benign, focal malformation that resembles a neoplasm in the tissue of its origin. This particular hamartoma originated from neural crest cells, a group of cells unique to vertebrates and crucial for their development.
- This neural crest-originated hamartoma was found in a female patient and the division of the particular lesion displays remarkable similarities with equine melanotic disease, a type of skin disease primarily observed in horses that results in dark, thickened, and wrinkled skin.
- The researchers found a characteristic arrangement of pigment-laden (color filled) spindle cells, a type of cell usually found in the smooth muscle tissue, around the hair follicle. This layout was extraordinarily identical in both the patient’s lesion and the equine melanotic disease.
- The paper also suggests a close relation of the identified hamartoma with previously explained patch and plaque-like blue nevi in human beings. Blue nevus is a type of mole that is usually blue in color.
- Inspection under the light and through ultrastructural imaging showed features differentiating towards both nevus cells, cells constituting a type of birthmark or mole, and Schwann cells, cells that produce a protective myelin sheath covering the nerve fibers in the peripheral nervous system.
- Based on these observations and findings, the researchers propose that the studied lesion be termed as “pilar neurocristic hamartoma”. The term ‘pilar’ suggests relation to hair follicle (from Latin – ‘Pilus’ means hair).
Implications of the Findings
- The research provides insight into a rare skin lesion of neural crest origin and its similarity to other established skin disorders helps the scientific and medical community to better understand the characteristics of such lesions.
- This study can be instrumental in formulating innovative and targeted strategies to manage and treat such pigmented lesions.
Cite This Article
APA
Tuthill RJ, Clark WH, Levene A.
(1982).
Pilar neurocristic hamartoma: its relationship to blue nevus and equine melanotic disease.
Arch Dermatol, 118(8), 592-596.
https://doi.org/10.1001/archderm.118.8.592 Publication
Researcher Affiliations
MeSH Terms
- Adolescent
- Animals
- Female
- Hair Diseases / embryology
- Hair Diseases / pathology
- Hamartoma / embryology
- Hamartoma / pathology
- Hamartoma / ultrastructure
- Horse Diseases / pathology
- Horses
- Humans
- Melanosis / pathology
- Melanosis / veterinary
- Neural Crest
- Nevus, Pigmented / pathology
- Skin Neoplasms / embryology
- Skin Neoplasms / pathology
- Skin Neoplasms / ultrastructure
Grant Funding
- CA25298 / NCI NIH HHS
- CA25874 / NCI NIH HHS
- CA26655 / NCI NIH HHS
Citations
This article has been cited 8 times.- Mariki H, Mwamanenge N, Mkony M, Manji KP. Neurocristic cutaneous hamartoma of the scalp with disseminated melanocytic nevi. BMJ Case Rep 2023 Feb 14;16(2).
- Peter D, Kuryan P, Gupta AK, Thomas M. Dermoscopy of Cutaneous Neurocristic Hamartoma and Report of its Rare Clinical Presentation. Dermatol Pract Concept 2022 Jul;12(3):e2022138.
- Tarasen A, Carlson JA, Leonard MK, Merlino G, Kaetzel D, Slominski AT. Pigmented Epithelioid Melanocytoma (PEM)/Animal Type Melanoma (ATM): Quest for an Origin. Report of One Unusual Case Indicating Follicular Origin and Another Arising in an Intradermal Nevus. Int J Mol Sci 2017 Aug 15;18(8).
- Turel MK, Chacko G, Raja A, Scheithauer BW. Neurocristic cutaneous hamartoma of the scalp. J Pediatr Neurosci 2012 Sep;7(3):181-4.
- Kim SK, Kim YC. Neurocristic cutaneous hamartoma of the scalp. Ann Dermatol 2009 Nov;21(4):396-8.
- El-Rayes D, Wilson K, Maguiness S, Miller D, Cazzato G, Giubellino A. Congenital Melanocytic Nevus with Neurocristic Cutaneous Hamartoma: A Case Report. Dermatopathology (Basel) 2025 Apr 10;12(2).
- Tahir M, Samman A, Shalin S, Knowles K, Herrera GA, Liles JS, Phung T. Malignant Cutaneous Neurocristic Hamartoma With Features of Melanoma: A Rare Entity. Cureus 2024 Sep;16(9):e69594.
- Cantisani C, Paolino G, Di Guardo A, Gomes V, Carugno A, Greco ME, Musolff N, Azzella G, Rossi G, Soda G, Longo C, Pellacani G. Diagnostic Imaging of Agminated Blue Lesions and Blue Lesions with Satellitosis: Case Series with a Concise Review of the Current Literature. J Clin Med 2024 Feb 3;13(3).
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