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1.
Dermatopathology (Basel) ; 10(3): 193-200, 2023 Jun 25.
Article in English | MEDLINE | ID: mdl-37489451

ABSTRACT

Folliculosebaceous cystic hamartoma is a cutaneous malformation composed of a cystic folliculosebaceous structure associated with mesenchymal elements, generally consisting of fibrous stroma, adipocytes and small vascular channels. We report the case of a 55-year-old female patient with a cutaneous nodule of the right nasal wing. Microscopically, the lesion showed a dilated hair follicle with multiple sebaceous glands, surrounded by a mesenchymal component composed of fibromyxoid stroma, spindle cells, mature-appearing adipocytes and collagen bundles, resembling spindle cell lipoma, associated with an additional neural component, consisting of small nerve bundles. In folliculosebaceous cystic hamartoma, the association of spindle cell lipomatous and neural components has not previously reported.

2.
Dermatopathology (Basel) ; 11(1): 19-25, 2023 Dec 31.
Article in English | MEDLINE | ID: mdl-38247726

ABSTRACT

Folliculosebaceous cystic hamartoma (FSCH) is a rare and benign form of cutaneous hamartomas. These skin lesions often lead to clinical and histopathological misdiagnosis due to their similarities to cutaneous lesions with overproduction of clustered sebaceous glands. Clinically, the lesions often present as solitary, skin-colored, pedunculated warts to cauliflower-like, exophytic papules and nodules, usually with a diameter ranging 0.5-1.5 cm that rarely exceed 2 cm in size. Only a small number of giant variants are reported in the literature with a diameter in the range of 5-23 cm. The vast majority of the lesions appear in the central face and show a striking predilection for the nose, ears, and scalp, but also emerge on the nipples, extremities, and genitals. Histologically, the epithelial components of folliculosebaceous cystic hamartoma comprise dilated infundibular cystic proliferation with surrounding mesenchymal components, which commonly include fibroplasia and vascular and adipose tissue proliferation. These histological characteristics were coined by Kimura and colleagues (1991). To the best of our knowledge, our case represents the biggest variant of giant folliculosebaceous cystic hamartoma.

3.
Dermatopathology (Basel) ; 9(3): 277-281, 2022 Aug 01.
Article in English | MEDLINE | ID: mdl-35997349

ABSTRACT

Folliculosebaceous cystic hamartoma (FSCH) is an uncommon hamartoma that usually presents on the central face area of adults as an asymptomatic, solitary dome-shaped or pedunculated papule. We report a case of a 35-year-old female who presented with six-months history of skin lesions on her labia majora. Histological findings included cystically dilated hair follicles with branching epithelial strands and interconnecting sebaceous gland consistent with the diagnosis of FSCH. The genital variant of FSCH was first described in 1998 and since then only six cases have been reported in the literature. We aim to increase awareness of this rare presentation due to the significant psychological implications and the risk of misdiagnosis.

4.
Indian J Dermatol ; 67(5): 625, 2022.
Article in English | MEDLINE | ID: mdl-36865871

ABSTRACT

Sebaceous trichofolliculoma is a very uncommon skin tumour, the tumour occurred on the nose and consisted of a solitary lesion. Sebaceous trichofolliculoma occurring in patients of the scrotum is very rare; only one case has been reported to date. The patient has many small soft nodules on the scrotum for several years, then the number and the size of the nodules increased. Histological examination showed many large cystic cavities open to the skin surface and numerous sebaceous glands connected to the cavity. Plastic surgery excision or necessary skin grafting is planned for the patient until maturity is attained.

5.
Indian J Dermatol ; 66(6): 674-676, 2021.
Article in English | MEDLINE | ID: mdl-35283505

ABSTRACT

Folliculosebaceous cystic hamartoma (FSCH) is a rare cutaneous hamartoma that consists of follicular, sebaceous, and mesenchymal elements. These lesions are mostly seen on the central face and scalp of adults and mostly present as single 0.5-1.5 cm papules or exophytic nodules. A 17-year-old female patient presented at our clinic with the complaint of swelling, pain and discharge in front of the left ear, which had been recurrent since childhood. The lesion was clinically suspected to be an infected preauriculer sinus cyst. After the medical treatment, the patient was operated on with a pre-diagnosis of preauricular sinus cyst. Histopathological findings revealed FSCH. To the best of our knowledge, this is the first reported case of FSCH originating from the preauricular region, located subcutaneously.

6.
Indian J Dermatol ; 64(6): 490-492, 2019.
Article in English | MEDLINE | ID: mdl-31896850

ABSTRACT

Hamartomatous conditions of skin are comprised of variable composition of normal skin elements. They can present as nodular masses. They can be mistaken for neoplastic skin lesions. Folliculosebaceous cystic hamartoma is a rare skin condition seen in adults, commonly on head, neck and face region. Congenital variant is rare. Only few case reports have been described in literature occurring on sites other than face. Definitive diagnosis can be given only on histopathology. We report a case presenting as thigh mass in a 1 year old female child clinically diagnosed as papilloma of skin.

7.
J Dermatol ; 44(9): 1050-1054, 2017 Sep.
Article in English | MEDLINE | ID: mdl-28370423

ABSTRACT

Some authors have reported that the secondary hair follicles in trichofolliculomas (TF) undergo regressive changes and are subsequently replaced by the developed sebaceous elements, and that folliculosebaceous cystic hamartoma (FSCH) is a TF at a very late stage. In the present study, we revaluated the histopathological features of 40 TF lesions, focusing on their chronological changes. The results of the present study indicate that while the secondary follicles in the TF exhibited the hair cycle, the normal hair cycle was out of control, and tertiary hair follicles randomly developed from the involuting secondary follicles. The repeated development of hair follicles in this disordered hair cycle caused the development of chains of several continuous hair follicles in late-stage TF. In the TF lesions, no features indicating the replacement of the regressing secondary hair follicles by any sebaceous elements were observed, thereby suggesting that FSCH is not a very-late-stage TF.


Subject(s)
Follicular Cyst/pathology , Hair Follicle/pathology , Hamartoma/pathology , Neoplasms, Basal Cell/pathology , Sebaceous Glands/pathology , Skin Neoplasms/pathology , Adolescent , Adult , Aged , Aged, 80 and over , Child , Female , Humans , Male , Middle Aged , Neoplasm Staging , Young Adult
8.
Article in Korean | WPRIM (Western Pacific) | ID: wpr-32963

ABSTRACT

PURPOSE: Folliculosebaceous cystic hamartoma is a rare cutaneous hamartoma consisting of dilated folliculosebaceous units invested in mesenchymal elements. There is no report of folliculosebaceous cystic hamartoma case occurred in the eyelid. We report here on this case along with a review of the relevant literature. CASE SUMMARY: 72-year-old female visited for the complaint of a mass in right upper eyelid. The mass was 1.9 × 1.2 cm sized and palpated in the subcutaneous level of right upper eyelid. The mass was not tender and had hardness like rubber. It was covered by skin without adhesion but fixed on the upper tarsal plate. Turning the eyelid inside out, it was found that the upper tarsal plate was penetrated by the mass. There was no specific finding except both cataract by other ophthalmic examination. The paranasal sinus computed tomography finding was well demarcated 0.9 cm sized mass with calcification. The excisional biopsy was performed for diagnosis and treatment. In pathologic finding, various sized normal sebaceous lobules were connected with the dilated follicles through the sebaceous canal and formed infundibular structure. There were sclerosing collagen, adipose cells and vessels between follicles and sebaceous lobules. So it was compatible with folliculosebaceous cystic hamartoma. 18 months later, there was no recurrence and wound was clear. CONCLUSIONS: Folliculosebaceous cystic hamartoma of the eye lid is rare disease, and differential diagnosis is necessary in patient with mass of eyelid.


Subject(s)
Aged , Female , Humans , Biopsy , Cataract , Collagen , Diagnosis , Diagnosis, Differential , Eyelids , Hamartoma , Hardness , Rare Diseases , Recurrence , Rubber , Skin , Wounds and Injuries
10.
Dermatol Pract Concept ; 4(3): 47-9, 2014 Jul.
Article in English | MEDLINE | ID: mdl-25126458

ABSTRACT

A 29-year-old Japanese man presented to our institution with a nodule on his nose that had increased in size since childhood. Physical examination indicated the presence of an elastic, firm, pedunculated red nodule measuring 15 mm in size. Dermatoscopic examination of the nodule indicated a yellowish-white network, yellowish-orange dots/globules at its center, and a pinkish-white structureless peripheral area. Histopathological examination of an excisional biopsy specimen showed a dilated infundibulocystic structure with sebaceous lobules proliferating radially, surrounded by fibrous stroma. Moreover, mature adipocytes and small vessels were noted in the stroma. Based on these histopatho-logic findings, the patient was diagnosed with folliculosebaceous cystic hamartoma.

11.
Ann Dermatol ; 23(Suppl 2): S185-7, 2011 Oct.
Article in English | MEDLINE | ID: mdl-22148046

ABSTRACT

Folliculosebaceous cystic hamartoma (FSCH) is a rare cutaneous hamartoma composed of dilated folliculosebaceous units and mesenchymal elements. It presents as a papule or nodule usually on the face and scalp, rarely on the genital or trunk area. Histologically, FSCH shares several similar features to sebaceous trichofolliculoma. We report one case of FSCH misdiagnosed as a neurofibroma. He was a 38-year-old man with a neurofibromatosis type I and a nodule on his left earlobe was excised under the impression of neurofibroma. Pathological examination revealed FSCH. Although FSCH is clinically not distinctive, awareness of the lesion is important to differentiate papulonodular or cyst-like cutaneous lesions.

12.
Annals of Dermatology ; : S185-S187, 2011.
Article in English | WPRIM (Western Pacific) | ID: wpr-200938

ABSTRACT

Folliculosebaceous cystic hamartoma (FSCH) is a rare cutaneous hamartoma composed of dilated folliculosebaceous units and mesenchymal elements. It presents as a papule or nodule usually on the face and scalp, rarely on the genital or trunk area. Histologically, FSCH shares several similar features to sebaceous trichofolliculoma. We report one case of FSCH misdiagnosed as a neurofibroma. He was a 38-year-old man with a neurofibromatosis type I and a nodule on his left earlobe was excised under the impression of neurofibroma. Pathological examination revealed FSCH. Although FSCH is clinically not distinctive, awareness of the lesion is important to differentiate papulonodular or cyst-like cutaneous lesions.


Subject(s)
Adult , Humans , Follicular Cyst , Hamartoma , Neoplasms, Basal Cell , Neurofibroma , Neurofibromatoses , Neurofibromatosis 1 , Scalp , Skin Neoplasms
13.
Ann Dermatol ; 21(1): 63-5, 2009 Feb.
Article in English | MEDLINE | ID: mdl-20548860

ABSTRACT

Clinically, folliculosebaceous cystic hamartoma (FSCH) lacks distinct features, but it has been reported as an asymptomatic, 1- to 3-cm, dome-shaped nodule on the face. Histopathologically, FSCH is characterized by adnexal and folliculosebaceous cystic proliferation with various mesenchymal changes. This case presented an unusually large, small-fist-sized mass in the right mandibular area that was accompanied by intermittent itching. Histopathologic findings demonstrated appropriate features of FSCH. We present an interesting case of giant FSCH in a 48-year-old female.

14.
Annals of Dermatology ; : 63-65, 2009.
Article in English | WPRIM (Western Pacific) | ID: wpr-103062

ABSTRACT

Clinically, folliculosebaceous cystic hamartoma (FSCH) lacks distinct features, but it has been reported as an asymptomatic, 1- to 3-cm, dome-shaped nodule on the face. Histopathologically, FSCH is characterized by adnexal and folliculosebaceous cystic proliferation with various mesenchymal changes. This case presented an unusually large, small-fist-sized mass in the right mandibular area that was accompanied by intermittent itching. Histopathologic findings demonstrated appropriate features of FSCH. We present an interesting case of giant FSCH in a 48-year-old female.


Subject(s)
Female , Humans , Middle Aged , Hamartoma , Pruritus
15.
Can J Plast Surg ; 15(4): 231-3, 2007.
Article in English | MEDLINE | ID: mdl-19554184

ABSTRACT

BACKGROUND: Folliculosebaceous cystic hamartomas (FSCH) are a recently recognized cutaneous hamartoma comprised of follicular, sebaceous and mesenchymal elements. Only nine cases have been previously reported. The authors present a case of FSCH on the lower extremity in addition to a literature review. OBJECTIVE: To further characterize the clinical, histological and immunohistological features of FSCH. METHODS: The clinical, histological and immunohistological features of FSCH were evaluated and the literature reviewed. RESULTS: This lesion uncommonly presents as exophytic papules or nodules on the leg, and most commonly presents on the central part of the face. Clinical differential diagnoses included nevus, neurofibroma and adnexal tumour. The diagnostic microscopic features of infundibular cystic structures with attached sebaceous glands and characteristic stroma were present in all lesions. Many of the stromal spindle cells were CD34-positive. CONCLUSION: The FSCH is a recently recognized hamartoma comprised of follicular, sebaceous and mesenchymal elements. Although reported only rarely, its incidence is likely much higher. In the present patient, the FSCH was located in femoral region, which is very rare.

16.
Annals of Dermatology ; : 13-15, 2007.
Article in English | WPRIM (Western Pacific) | ID: wpr-120766

ABSTRACT

Folliculosebaceous cystic hamartoma (FSCH) is a recently-recognized cutaneous hamar- toma composed of follicular, sebaceous and mesenchymal elements. We describe an unusual case of FSCH in a 61-year-old male, who had a relatively large, 3x2.5cm sized, smooth subcutaneous nodule on the occipital area of the scalp, an uncommon location for FSCH.


Subject(s)
Humans , Male , Middle Aged , Hamartoma , Scalp
17.
Article in Korean | WPRIM (Western Pacific) | ID: wpr-86142

ABSTRACT

Folliculosebaceous cystic hamartoma is a distinctive skin malformation, which presents as a solitary, smooth- surfaced, skin-colored papule or nodule. This hamartoma usually occurs on the central part of the face, particularly on the nose. In 1998, Bolognia et al. described a genital variant of folliculosebaceous cystic hamartoma, which has not been reported to date in Korea. We herein report a rare case of folliculosebaceous cystic hamartoma which occurred on the labia majora of a 28-year-old woman. Histopathologically, multiple, dilated, follicular, cystic structures were observed with numerous sebaceous lobules arising from its wall. There was an excess of fibrous components around these structures, which included small venules, adipocytes and neural tissue.


Subject(s)
Adult , Female , Humans , Adipocytes , Hamartoma , Korea , Nose , Skin , Venules
18.
Annals of Dermatology ; : 170-172, 2007.
Article in English | WPRIM (Western Pacific) | ID: wpr-20438

ABSTRACT

Folliculosebaceous cystic hamartoma is a rare cutaneous hamartoma comprised of follicular, sebaceous, and mesenchymal elements. It usually presents as an exophytic papule or nodule on the central part of the face. The histological findings are intradermal cystic structures lined by infundibular epithelium, numerous sebaceous lobules radiating from cystic structures and a surrounding stroma composed of mesenchymal changes, including variable proportions of fibrous, adipose, vascular and neural tissues. We herein report a case of folliculosebaceous cystic hamartoma in a 73-year-old man who presented with a skin-colored, dome-shaped papule on the right nasal ala.


Subject(s)
Aged , Humans , Epithelium , Hamartoma
19.
Article in Korean | WPRIM (Western Pacific) | ID: wpr-136869

ABSTRACT

Folliculosebaceous cystic hamartoma is a rare cutaneous hamartoma comprising follicular, sebaceous, and mesenchymal elements. This tumor usually presents during adulthood as an exophytic papule or nodule on the scalp or central area of the face. Histopathologic features are central cystic structures lined with infundibular epithelium, numerous sebaceous gland lobules radiating from cystic structures, and surrounding stroma composed of fibrillary bundles of collagen, small vessels, neural tissue and occasionally mucin deposits. We herein report a case of folliculosebaceous cystic hamartoma in a 21-year-old man who presented with a skin-colored, dome-shaped papule on the scalp.


Subject(s)
Humans , Young Adult , Collagen , Epithelium , Hamartoma , Mucins , Scalp , Sebaceous Glands
20.
Article in Korean | WPRIM (Western Pacific) | ID: wpr-136864

ABSTRACT

Folliculosebaceous cystic hamartoma is a rare cutaneous hamartoma comprising follicular, sebaceous, and mesenchymal elements. This tumor usually presents during adulthood as an exophytic papule or nodule on the scalp or central area of the face. Histopathologic features are central cystic structures lined with infundibular epithelium, numerous sebaceous gland lobules radiating from cystic structures, and surrounding stroma composed of fibrillary bundles of collagen, small vessels, neural tissue and occasionally mucin deposits. We herein report a case of folliculosebaceous cystic hamartoma in a 21-year-old man who presented with a skin-colored, dome-shaped papule on the scalp.


Subject(s)
Humans , Young Adult , Collagen , Epithelium , Hamartoma , Mucins , Scalp , Sebaceous Glands
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