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JAAD Case Rep 2020 Jun 18;6(6):479-481. Epub 2020 Apr 18.

Department of Dermatology, University of Texas Southwestern Medical Center, Dallas, Texas.

Turk J Med Sci 2020 Apr 14. Epub 2020 Apr 14.

Background And Aim: Cutaneous adnexal tumors (CAT) are rare tumors originating from the adnexal epithelial parts of the skin. Due to its clinical and histopathological characteristics comparable with other diseases, clinicians and pathologists experience difficulties in its diagnosis. We aimed to reveal the clinical and histopathological characteristics of the retrospectively screened cases and to compare the pre-diagnoses and histopathological diagnoses of clinicians. Read More

Indian J Dermatol 2020 Mar-Apr;65(2):151-153

Department of Dermatology, Rutgers-New Jersey Medical School, Newark, New Jersey, USA.

A 52-year-old female presented with an asymptomatic cyst-like lesion on her right labia majora present for several years. Excision revealed a 3-mm cystic lesion containing complex networks of thin fronds of clear apocrine cells and basaloid cells, features characteristic of hidradenoma papilliferum (HP). In contrast to the medical literature, the lesion was connected to the overlying epithelium by a straight duct, a feature characteristic of apocrine glands. Read More

Ann Dermatol Venereol 2020 May 19;147(5):334-339. Epub 2020 Feb 19.

Clinique dermatopathologique, hôpitaux universitaires de Strasbourg, 67000 Strasbourg, France.

Introduction: Trichoadenoma is a very rare follicular tumour with a remarkable histopathological appearance. In this article we present a series of 12 cases of trichoadenoma, as well as the anatomoclinical and dermatoscopic findings in a typical case. We discuss these findings in the light of an extensive literature research. Read More

Lin Chung Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 2020 Jan;34(1):83-85

The trichilemmal carcinoma is a rare tumor that usually occurs on sun-exposed skin, especially on the face, scalp, neck and back of hands, mainly in elderly subjects but commonly between the 4th and 9th decades of life. We report a case of giant trichilemmal carcinoma. A 65-year-old man presented with a 5-year history of a slowly developing mass arising from his right retroauricular region, with local destruction of the auricle. Read More

J Dermatol 2020 May 20;47(5):e197-e198. Epub 2020 Feb 20.

Department of Dermatology, Gunma University Graduate School of Medicine, Maebashi, Japan.

Acta Dermatovenerol Croat 2019 Dec;27(4):282-284

Vladimír Bartoš, MD, PhD, MPH, Deapartment of Pathology Faculty Hospital in Žilina, Vojtecha Spanyola 43, 012 07 Žilina, Slovakia;

Dear Editor, I read an interesting recent article by Karimzadeh et al. (1) in an earlier issue of your journal, who provided a comprehensive review addressing a relatively rare benign tumor originating from the hair follicles - trichoepithelioma (TE). They rightly claimed that trichoepithelioma can be divided into the following 3 subgroups: a) multiple familiar TE, b) solitary non-hereditary TE, and c) desmoplastic trichoepithelioma (DTE). Read More

Clin Exp Dermatol 2020 Jun 13;45(4):508-511. Epub 2019 Dec 13.

Departments of, Department of, Dermatology and Venereology, Faculty of Medicine, Trakya University, Edirne, Turkey.

J Community Genet 2019 Dec 3. Epub 2019 Dec 3.

Post-graduate Program in Genetics and Molecular Biology, Department of Biosciences, Universidade Federal do Rio Grande do Sul, Porto Alegre, RS, 91501-970, Brazil.

Germline mutations in the cylindromatosis gene (CYLD) are associated with a rare autosomal dominant disease known as CYLD cutaneous syndrome (CCS). Patients present multiple neoplasms originating from skin appendages. Here, we investigated the main clinical and molecular features of a large family with CCS having lived in a small Brazilian town for 6 generations, making its prevalence significantly high. Read More

Int J Dermatol 2020 Mar 28;59(3):321-325. Epub 2019 Nov 28.

Fundación Piel Sana Academia Española de Dermatología, Madrid, Spain.

Background: The use of Mohs micrographic surgery (MMS) for rare cutaneous tumors is poorly defined. We aim to describe the demographics, tumor presentation and topography, surgery characteristics and complications of MMS for rare cutaneous tumors in a national registry.

Methods: Prospective cohort study of patients treated with MMS in Spain between July 2013 and June 2018. Read More

J Small Anim Pract 2019 Nov 18. Epub 2019 Nov 18.

Department of Veterinary Pathology, Graduate School of Agricultural and Life Science, The University of Tokyo, Tokyo, 1138657, Japan.

This retrospective study of a series of 18 cases aimed to describe the clinical and pathological findings of oral tumours in rabbits, as there have been few reports detailing spontaneous oral tumours in this species. A total of 13 different tumour types were diagnosed: squamous cell carcinoma (three), ameloblastoma (two), fibrosarcoma (two), osteosarcoma (two), cementoma (one), complex odontoma (one), giant cell epulis (one), sarcoma (one), chondrosarcoma (one), trichoepithelioma (one), papilloma (one), malignant melanoma (one) and basal cell carcinoma (one). Odontogenic tumours were relatively common in this study as compared to the oral tumours typically identified in dogs and cats. Read More

J Cutan Pathol 2020 Mar 3;47(3):207-218. Epub 2019 Dec 3.

Department of Medicine, Mackay Medical College, New Taipei City, Taiwan.

Background: Basaloid tumors arising from seborrheic keratosis (SK) that resembled basal cell carcinoma (BCC) were infrequently observed in our patients. They also exhibited morphologic features similar to those of trichoblastoma or basaloid follicular hamartoma.

Methods: We retrospectively collected cases of SK with basaloid tumors from 2001 to 2017. Read More

Cureus 2019 Aug 16;11(8):e5401. Epub 2019 Aug 16.

Dermatology, University of California San Diego, La Jolla, USA.

Differentiating between trichoepithelioma and basal cell carcinoma (BCC) is sometimes diagnostically challenging. We present a case of a 61-year-old male with a BCC arising within a trichoepithelioma, which is rarely reported in the literature. Clinical and histological diagnosis of trichoepithelioma is sometimes complicated by its many similarities to BCC. Read More

Ann Dermatol Venereol 2019 Nov 7;146(11):760-763. Epub 2019 Oct 7.

Hôpitaux universitaires de Strasbourg, BP 426, 1, place de l'Hôpital, 67091 Strasbourg cedex, France.

Am J Dermatopathol 2020 Feb;42(2):122-124

Department of Dermatology, Instituto Valenciano de Oncología, Valencia, Spain.

Solid carcinoma, probably the solid variant of microcystic adnexal carcinoma, is an apocrine adnexal tumor first described in 1998. The authors report an additional new case of the tumor at an unusual localization. A 78-year-old man presented with an asymptomatic firm plaque on his right thigh that had been present for 15 years. Read More

Am J Dermatopathol 2020 Feb;42(2):103-110

Laboratoire Central d'Anatomie Pathologique, Hôpital L. Pasteur, University of Nice, France.

This is a report of a previously undescribed type of onychomatricoma (OM) with an unusual clinical presentation as a thickened free edge of the nail plate without discernible cavities and distinguished histologically from the ordinary OM by 3 features: (1) the lack of cavitation at the proximal border of the nail plate and the small sizes of the cavities at the free edge of the distal nail plate; (2) a papillated epithelial hyperplasia pattern very different from the digitate pattern of the ordinary OM; and (3) a special pattern of matrical keratinization with pseudohorn cysts that mirror closely those found in onychocytic matricoma (OCM). Furthermore, the sex ratio and sites of the lesion seem different than those of conventional OM with the caveat that the numbers in this series are small. A practical approach to the diagnosis of onychogenic tumor mainly involves consideration of tumors that clinically present as localized longitudinal pachyonychia including melanoma and Bowen disease. Read More

Histopathology 2020 Jan 13;76(2):318-324. Epub 2019 Nov 13.

Department of Pathology, Fukuoka University School of Medicine and Hospital, Fukuoka, Japan.

Aims: Laminin (Ln)-γ 2, one of the chains of Ln-332, is a marker of invasive tumours and is frequently expressed as a monomer in malignant tumours. Desmoplastic trichoepithelioma (DTE), some types of basal cell carcinoma (BCC) (infiltrating and morphoeic BCC) and microcystic adnexal carcinoma (MAC) belong to a group of tumours known as sclerosing adnexal neoplasms (SAN) that are frequently difficult to differentiate and often require immunohistochemistry for diagnosis. The aim of this study was to assess the usefulness of Ln-γ 2 expression in the differential diagnosis of DTE, infiltrating/morphoeic BCC, MAC and syringoma. Read More

BMC Infect Dis 2019 Aug 9;19(1):707. Epub 2019 Aug 9.

Department of Dermatology, Nanfang Hospital, Southern Medical University, Guangzhou, 510515, China.

Background: Talaromyces marneffei is a thermally dimorphic fungus endemic in south-east Asia. It predominantly occurs in both immunocompromised and immunosuppressed patients and can be fatal if diagnosis and treatment are delayed. The clinical manifestations of T. Read More

J Biol Chem 2019 09 31;294(38):14135-14148. Epub 2019 Jul 31.

Division of Cell-Free Sciences, Proteo-Science Center (PROS), 3 Bunkyo-cho, Matsuyama, Ehime 790-8577, Japan

The tumor suppressor CYLD is a deubiquitinating enzyme that suppresses polyubiquitin-dependent signaling pathways, including the proinflammatory and cell growth-promoting NF-κB pathway. Missense mutations in the gene are present in individuals with syndromes such as multiple familial trichoepithelioma (MFT), but the pathogenic roles of these mutations remain unclear. Recent studies have shown that CYLD interacts with a RING finger domain protein, mind bomb homologue 2 (MIB2), in the regulation of NOTCH signaling. Read More

Ophthalmologe 2020 Jan;117(1):73-77

Zentrum für Augenheilkunde, Universität zu Köln, Medizinische Fakultät und Uniklinik Köln, Kerpener Str. 62, 50924, Köln, Deutschland.

Background: A space-occupying lesion of the eyelid in young adults is often a sign of an inflammation, a trauma or a benign neoplasm. The aim of this case report is to demonstrate a rare basal cell carcinoma, which presumably already arose in adolescence without further high-risk factors.

Methods: A 28-year-old male patient presented for a second opinion on a painless swelling of the right lower eyelid. Read More

JAMA Dermatol 2019 Jul 10. Epub 2019 Jul 10.

Department of Dermatology, University of São Paulo Medical School, São Paulo, Brazil.

Indian J Pathol Microbiol 2019 Apr-Jun;62(2):313-318

Department of Pathology, Government Medical College, Kottayam, Kerala, India.

Brooke-Spiegler syndrome (BSS) is a rare autosomal dominant disease characterized by the development of multiple cutaneous adnexal neoplasms namely cylindroma, trichoepithelioma and spiradenoma. The neoplasms associated with this syndrome are generally benign, but rarely they may undergo malignant transformation. A 63-year-old male presented with an ulcerated nodular lesion over glabella and multiple asymptomatic nodular lesions over face, scalp, chest and limbs. Read More

Am J Dermatopathol 2019 Nov;41(11):810-818

CellCOM-SB Research Group Department, Institute for Biomedical Research of A Coruña (INIBIC), University of A Coruña (UDC), A Coruña, Spain.

Introduction: Benign cutaneous tumors with follicular differentiation are alleged to differentiate toward parts of the hair follicle. Connexin 43 (Cx43) is a gap junction protein, the tumoral role of which has been investigated in several types of tumors.

Objective: To study the pattern of expression of Cx43 in benign cutaneous tumors with follicular differentiation and to compare it with that shown by their alleged anatomical counterparts of the hair follicle. Read More

J Dermatol 2019 May 27;46(5):413-417. Epub 2019 Feb 27.

Department of Dermatology, Air Force General Hospital, Chinese People's Liberation Army, Beijing, China.

Marie Unna hereditary hypotrichosis (MUHH) and multiple familial trichoepithelioma (MFT) are both autosomal dominant disorders. Recently, certain genes (HR and EPS8L3) have been found to be responsible for MUHH, while CYLD has been demonstrated to be the main pathogenic gene in MFT patients. However, there exist a number of CYLD mutation-negative MFT cases, for which the causative gene has been unknown. Read More

J Cytol 2018 Oct-Dec;35(4):204-207

Department of Pathology, ESI Hospital, New Delhi, India.

Background: Skin adnexal neoplasms are relatively rare and thus uncommonly encountered in routine pathology practice.

Aims: The present study was conducted with the aim to diagnose various skin adnexal tumors on fine-needle aspiration cytology (FNAC) followed by confirmation on histopathology and immunohistochemistry of the excised tumor.

Materials And Methods: A total of 11 cases of superficial nodular swellings of the skin were studied over a period of 2 years from 2015 to 2016. Read More

J Craniofac Surg 2019 May/Jun;30(3):e197-e199

Department of Plastic and Reconstructive Surgery, Uijeongbu St Mary's Hospital, College of Medicine, the Catholic University of Korea, Gyeonggi-do, Korea.

Trichoepithelioma is a benign tumor which can be easily confused with basal cell carcinoma. Since both tumors are histologically similar, it is important to distinguish them correctly as the courses for treatment are very different. Basal cell carcinoma is malignant; therefore, it requires total surgical excision, including a wide healthy margin. Read More

Am J Dermatopathol 2019 Oct;41(10):778-780

Department of Plastic, Reconstructive and Aesthetic Surgery, Bülent Ecevit University Health Practice and Research Center, Zonguldak, Turkey.

Indian J Ophthalmol 2018 Oct;66(10):1506-1508

Department of Orbit and Oculoplasty, Aravind Eye Hospital and Post-graduate Institute of Ophthalmology, Pondicherry, India.

Benign eyelid tumors derived from hair follicles are rare and frequently misdiagnosed as basal-cell carcinoma when evaluation is based on clinical evidence alone. The discrepancy between the clinical aspect and the histological diagnosis can be as high as 30-40%, even for experienced clinicians. Unfortunately, this masquerade is not only clinical but also histological. Read More

Int J Surg Case Rep 2018 12;51:277-281. Epub 2018 Sep 12.

Department of Plastic and Reconstructive Surgery, University Hospital of Foggia, Foggia, Italy.

Introduction: The authors present a "four-step" integrated surgical protocol to treat a rare case of multiple giant eccrine spiradenoma (ES) of the head and neck in a young patient.

Presentation Of Case: An 18-year-old female patient presented with multiple swellings in the head and neck regions. The patient had a severe psychological trauma with a negative impact on her social life. Read More

Medicine (Baltimore) 2018 Sep;97(38):e12252

Department of Plastic and Reconstructive Surgery, Chonnam National University Medical School, Gwangju, Korea.

Rationale: Trichoepithelioma (TE) is a rare benign skin tumor that originates from the hair follicle epithelium. Although skin lesions arising from scar tissues are mostly malignant, the development of a benign tumor such as TE is a rare event.

Patient Concerns: A 28-year-old male patient who had a scar on the left cheek which arose 10 years ago because of a chemical burn visited our hospital. Read More

Eplasty 2018 23;18:ic14. Epub 2018 Jul 23.

Department of Plastic Surgery, Morsani College of Medicine, Tampa, Fla.

S D Med 2018 Apr;71(4):164-166

Department of Pathology, University of South Dakota, Sanford School of Medicine, Sioux Falls, South Dakota.

Primary cutaneous osteosarcoma is an exceedingly rare malignant mesenchymal neoplasm of the skin which produces bone, osteoid, or chondroid material and does not involve the underlying bone. The most common site for extraskeletal osteosarcoma is in the deep soft tissues of the thigh, upper extremities, and retroperitoneum; however, it may occur anywhere in the body. Involvement of the skin is rare and when it does occur it is more commonly due to metastatic disease rather than a primary malignancy. Read More

Acta Dermatovenerol Croat 2018 Jun;26(2):162-165

Amin Karimzadeh, MD, Department of Dermatology, Faculty of Medicine, Jahrom University of Medical Sciences, Motahari Blvd, PO Box: 74148-46199, Jahrom, Iran;

Trichoepithelioma is an uncommon benign adnexal neoplasm. It can present as a solitary non-familial or multiple familial form. Trichoepithelioma usually develops in early childhood or puberty. Read More

Arch Dermatol Res 2018 Sep 4;310(7):599-606. Epub 2018 Jul 4.

Department of Dermatology, Venereology and Allergology, University Medical Center Göttingen, Robert-Koch-Str. 40, 37075, Göttingen, Germany.

Mutations in the tumor suppressor gene CYLD underlie phenotypically heterogeneous hereditary tumor disorders of the skin appendages. These diseases are inherited autosomal dominantly and include Brooke-Spiegler syndrome (BSS; OMIM 605041), familial cylindromatosis (FC; OMIM 132700) and multiple familial trichoepithelioma (MFT; OMIM 601606). Clinically, cylindromas, trichoepitheliomas and spiradenomas can be found in affected individuals. Read More

Virchows Arch 2018 Aug 22;473(2):241-246. Epub 2018 Jun 22.

Department of Dermatology, Paracelsus Medical University Nuremberg, Prof.-Ernst Nathan Straße 1, 90419, Nürnberg, Germany.

Multiple familial trichoepitheliomas (MFT) is an autosomal dominantly inherited disease characterized by multiple skin appendage tumors. We describe a patient showing a continuous spectrum of follicular differentiated neoplasms including classical trichoepitheliomas but also infiltrative growing and finally metastasizing malignant follicular differentiated tumors. Germline mutation analysis revealed a nonsense mutation in the cylindromatosis (CYLD) gene. Read More

Case Rep Dermatol 2018 May-Aug;10(2):133-137. Epub 2018 May 18.

Dermatology Clinic, University Hospital Zurich, Zurich, Switzerland.

Basal cell carcinoma (BCC) is the most common cancer in humans worldwide. Many highly specific dermoscopic criteria for BCC are well established in the literature. On the contrary, other malignant or benign skin tumors may mimic BCC by exhibiting similar or even the same dermoscopic features and therefore obscuring the diagnosis of BCC in certain situations. Read More

J Cutan Pathol 2018 Nov 13;45(11):807-816. Epub 2018 Jul 13.

Dermatology Department, Faculty of Medicine, Zagazig University, Zagazig, Egypt.

Background: The distinction of trichoepithelioma from basal cell carcinoma in small superficial biopsies is important but often challenging. This has inspired many scientists to test the validity of immunohistochemical markers in the differential diagnosis.

Objectives: To develop an immunohistochemical protocol that helps in differentiation between both trichoepithelioma (TE) and basal cell carcinoma (BCC) with higher sensitivity and specificity. Read More

J Dtsch Dermatol Ges 2018 Jun;16(6):719-728

Department of Dermatology, Venereology and Allergology, University Medical Center, Würzburg, Germany.

Background And Objectives: The histological differential diagnosis between benign trichogenic skin tumors and basal cell carcinoma may be challenging. We therefore set out to investigate whether expression of CD10, a cell surface protein with neutral endopeptidase activity, might be a suitable marker for the histological differential diagnosis of trichogenic skin tumors.

Patients And Methods: We immunohistochemically analyzed 119 cases of benign trichogenic skin tumors and basal cell carcinoma. Read More

Indian Dermatol Online J 2018 May-Jun;9(3):222-223

Department of Dermatology, Venereology, and Leprology, AIIMS, Rishikesh, Uttarakhand, India.

Acta Med Port 2018 Mar 29;31(3):180. Epub 2018 Mar 29.

Serviço de Dermatovenereologia. Hospital de Vila Franca de Xira. Vila Franca de Xira, Portugal.

Am J Dermatopathol 2018 Aug;40(8):575-579

Department of Pathology, Section of Dermatopathology, University of Virginia Health System, Charlottesville, VA.

Sclerosing squamous cell carcinoma (SCC), also known as "desmoplastic" SCC, is a rare subtype of cutaneous malignancy. This variant is clinically significant because it is associated with an increased risk of local recurrence and metastasis. We herein present 16 examples of sclerotic SCC of the skin in 8 men and 3 women, with a median age of 66 years. Read More

Appl Immunohistochem Mol Morphol 2019 09;27(8):584-588

Department of Pathology and Laboratory Medicine, University of Calgary, Calgary, AB, Canada.

Ber-EP4 has been the traditional immunostain used for the detection of basaloid skin tumors. Recently, MOC-31 has shown be superior to Ber-EP4 in the detection of basosquamous basal cell carcinoma (BCC) and many centers are now using both Ber-EP4 and MOC-31 antibodies together to detect these lesions. The objective of this study was to compare the utility of using both Ber-EP4 and MOC-31 immunostains in the detection of basaloid skin tumors and to better characterize the previously unknown staining properties of MOC-31 in cutaneous lesions. Read More

Ann Pathol 2018 Jun 2;38(3):198-201. Epub 2018 Apr 2.

Service de dermatologie, CHU de Caen, avenue côte-de-nacre, 14033 Caen, France.

We report a case of microcystic adnexal carcinoma in a 80-year-old woman. This is a rare malignant adnexal cutaneous tumor with glandular and follicular differentiation, rare, often asymptomatic, with late diagnosis and slow growth, locally aggressive and rarely metastatic. The distinction with other benign and malignant skin tumors is difficult. Read More

Int J Trichology 2017 Oct-Dec;9(4):181-183

Department of Pathology, Clínica Las Condes, Santiago, Chile.

Trichofolliculoma (TF) is a hamartomatous hair follicle-related tumor, clinically described as a dome-shaped papule with a central pore crossed by one or more silky white hairs. Histologically, it described as a cystic cavity containing keratinous debris, hair shaft fragments, and numerous hair follicles arising from its linings. Collision or compound tumors are a coexistence of two or more identifiable tumors in the same lesion. Read More

Arch Pathol Lab Med 2017 Nov;141(11):1490-1502

From the Departments of Pathology (Drs Stanoszek, Wang, and Harms) and Dermatology (Dr Harms), University of Michigan Medical School, Ann Arbor. Drs Stanoszek and Wang contributed equally.

Context: - Basal cell carcinoma (BCC) is the most common human malignant neoplasm and is a frequently encountered diagnosis in dermatopathology. Although BCC may be locally destructive, it rarely metastasizes. Many diagnostic entities display morphologic and immunophenotypic overlap with BCC, including nonneoplastic processes, such as follicular induction over dermatofibroma; benign follicular tumors, such as trichoblastoma, trichoepithelioma, or basaloid follicular hamartoma; and malignant tumors, such as sebaceous carcinoma or Merkel cell carcinoma. Read More

Am J Dermatopathol 2018 Dec;40(12):924-926

Dermatopathology Laboratory, Dermatologikum, Hamburg, Germany.

Induction of follicular germinative structures above a dermatofibroma (DF) is a common finding. Rarely, induction of a trichoblastoma in a DF has been observed. Here, we report the case of a desmoplastic trichoepithelioma induced by a DF. Read More

Indian Dermatol Online J 2017 Sep-Oct;8(5):358-360

Department of Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi, India.

Trichoepithelioma is a benign tumor of follicular unit. It has been rarely described as the cause of leonine facies. We are presenting a classical case of multiple familial trichoepitheliomas (MFTs) with characteristic histopathological features leading to leonine facies. Read More

Clin Exp Dermatol 2017 Dec 4;42(8):906-909. Epub 2017 Oct 4.

Department of Dermatology, Turkiye Cumhuriyeti Saglik Bakanligi Okmeydani Egitim ve Arastirma Hastanesi, Istanbul, Turkey.

Brooke-Spiegler syndrome (BSS) is a rare, autosomal dominant disorder characterized by multiple adnexal tumours, especially trichoepitheliomas, cylindromas and occasionally spiradenomas. These lesions usually begin to appear in the second or third decade of life. Malignant transformation of pre-existing tumours may occur. Read More

Clin Exp Dermatol 2018 Jan 20;43(1):108-109. Epub 2017 Sep 20.

Department of Dermatology, Chung-Ang University College of Medicine, Seoul, South Korea.