Acquired smooth muscle hamartoma with sebaceous component.

BACKGROUND: Acquired smooth muscle hamartoma (ASMH) is a rare benign lesion characterized clinically by hyperpigmented plaques with hypertrichosis and some follicular papules. The main histologic finding is the presence of disorganized smooth muscle bundles in the dermis. Only 25 cases of ASMH have been reported in the literature. CLINICAL CASE: We present the case of an 18-year-old male who reported a pigmented area and increased hair growth on the left hemifacial with one year of evolution. Clinically, a plaque was observed in the preauricular region and on the left cheek with a linear Blaschkoid path, consisting of hyperpigmentation, hypertrichosis, and some papular lesions, with negative pseudo-Darier sign. Histological analysis showed an increase in the number of smooth muscle bundles in the middle and deep dermis surrounding abundant sebaceous glands and numerous hair follicles in different stages of evolution. CONCLUSIONS: The sebaceous component in this lesion was prominent. Therefore, we considered this lesion part of a spectrum where the acquired smooth muscle hamartoma and folliculosebaceous cystic hamartoma are found at the extremes. This case would fall in the middle of the range, as it combines both histological features.

INTRODUCCIÓN: El hamartoma de músculo liso adquirido (HMLA) es una lesión benigna adquirida, poco frecuente, caracterizada clínicamente por presentar placas hiperpigmentadas, con hipertricosis y algunas pápulas foliculares. El principal hallazgo histológico es la presencia de abundantes haces de músculo liso desorganizados en la dermis. Se han reportado solo 25 casos de HMLA en la literatura. CASO CLÍNICO: Se presenta el caso de un paciente de sexo masculino de 18 años que refirió una zona pigmentada y el aumento de vello en la hemicara izquierda con un año de evolución. Clínicamente se observó una placa en la región preauricular y mejilla izquierda con trayecto lineal blaschkoide, constituida por hiperpigmentación, hipertricosis y algunas lesiones papulares, con signo pseudo-Darier negativo. Histológicamente se encontró un aumento en el número de haces de músculo liso en la dermis media y profunda rodeando abundantes glándulas sebáceas, así como numerosos folículos pilosos en diferentes estadios de evolución. CONCLUSIONES: El componente sebáceo en esta lesión fue muy marcado, por lo que se considera que forma parte de un espectro donde en los extremos se encuentran el hamartoma de músculo liso adquirido y el hamartoma quístico folículo sebáceo. El presente caso se encontraría en medio, ya que combina ambas características histológicas.

A conscious minimally invasive approach to remove follicular cysts and infundibular keratinizing acanthomas in five dogs.

Follicular cysts and infundibular keratinizing acanthomas are common benign cutaneous lesions in dogs. Current treatment options include surgical excision under general anaesthesia, cryotherapy, carbon dioxide laser and retinoids, each with potential disadvantages. We describe a conscious, minimally invasive, surgical excision procedure with high success rate and no complications in five dogs.

Les kystes folliculaires et les acanthomes kératinisant infundibulaires sont des lésions cutanées bénignes fréquentes chez le chien. Les options de traitement actuelles comprennent l'exérèse chirurgicale sous anesthésie générale, la cryothérapie, le laser au dioxyde de carbone et les rétinoïdes, chacun présentant des inconvénients potentiels. Nous décrivons une procédure d'exérèse chirurgicale consciente, peu invasive, avec un taux de réussite élevé et aucune complication chez cinq chiens.

Los quistes foliculares y los acantomas queratinizantes infundibulares son lesiones cutáneas benignas comunes en perros. Las opciones de tratamiento actuales incluyen la escisión quirúrgica bajo anestesia general, crioterapia, láser de dióxido de carbono y retinoides, cada uno con posibles desventajas. Describimos un procedimiento de escisión quirúrgica consciente, mínimamente invasivo, con una alta tasa de éxito y sin complicaciones en cinco perros.

Cistos foliculares e acantomas infundibulares queratinizantes são lesões cutâneas benignas em cães. As opções terapêuticas existentes atualmente são excisão cirúrgica sob anestesia geral, crioterapia, laser de dióxido de carbono e retinoides, cada um com desvantagens potenciais. Nós descrevemos aqui um procedimento de excisão cirúrgica minimamente invasivo, consciente, com um grande potencial de sucesso e sem complicações em cinco cães.

In vivo reflectance confocal microscopic findings in a case of trichofolliculoma.

Trichofolliculoma is a rare follicular hamartoma whose dermoscopic features have been scarcely reported. On the other hand, reflectance confocal microscopy features have not been described yet. In the present study, the authors report reflectance confocal microscopy features in a case of trichofolliculoma as squamous hyperplasia forming irregular finger-like protrusions around the hair follicle and papillomatous hyperplasia of the hair follicle epithelium, which correlated with histopathology. This case suggests that reflectance confocal microscopy may help incorrect in vivo diagnosis of trichofolliculoma in cases difficult to diagnose by morphology and dermoscopy.

Foreign body reaction to ruptured follicular cysts in dogs.

BACKGROUND: Cutaneous cysts are common in dogs, and surgical resection is the recommended treatment. However, additional therapy may be required for ruptured follicular cysts with severe cutaneous complications. CASE PRESENTATION: A 3-year-old neutered male Samoyed was presented with multifocal masses on the forelimbs. A 5-year-old neutered female Maltese was also presented with multiple masses and ruptured lesions, which were ulcerative and painful, around the parotid and submandibular glands. The lesions were examined cytologically. In addition, bacterial and fungal cultures and histopathologic examination were performed. Cutaneous multifocal nodules in the Samoyed could not be diagnosed via cytological examination or bacterial/fungal culture. Histopathology revealed numerous follicular cysts with multiple pyogranulomas of various sizes, some of which contained central keratin debris. In the Maltese, cytologic examination revealed central keratins or enucleated ghost cells in the intact cysts and few keratinized squamous cells mixed with neutrophils, mucus and metachromatic cells in the ruptured cysts. Histopathologic examination revealed severely dilated follicular cysts. Oral steroid and cyclosporine therapy resulted in marked improvement in the aseptic pyogranulomas after 2 weeks in formal case and combined with a surgery for residual cysts in latter case. CONCLUSIONS: We have reported two canine cases of ruptured follicular cysts causing foreign body-like aseptic pyogranulomas around cutaneous tissues and their successful management with pharmacological therapy and surgery.

Solitary asymptomatic lobular perianal nodule on an infant.

Folliculosebaceous cystic hamartoma (FSCH) is a benign lesion that presents as a solitary papule or nodule that typically affects the face and scalp of adults. A few reports have observed a congenital presentation. We present an infant boy exhibiting a tumor with overlap features between an FSCH and fibrofolliculoma in the perianal region that was first noticed at 6 months of age. The histological examination showed a hamartomatous benign proliferation of hair follicles and disordered sebaceous glands, which is consistent with the infundibular structures and radiating sebaceous glands that are typically observed in previously reported cases of FSCH. Sebaceous differentiation is a hallmark feature of FSCH. Folliculosebaceous cystic hamartoma is believed to be a late-stage form of trichofolliculoma (TF). Another consideration is that FSCH and TF are two distinct entities set apart by their degree of sebaceous or follicular differentiation and development of the mesenchymal stroma.

Hyperreactio Luteinalis (Multiple Luteinized Follicle Cysts): A Report of 10 Cases.

Hyperreactio luteinalis is a rare entity arising in pregnancy and in the setting of gestational trophoblastic diseases (ie choriocarcinoma, molar pregnancy) that presents with, typically, bilateral ovarian enlargement due to numerous follicle cysts. While the phenomenon is benign and spontaneously regresses following delivery or treatment, a specimen may be seen in pathology when oophorectomy or cystectomy is performed to exclude malignancy or to manage acute complications such as torsion. Such resections may exhibit overlapping microscopic features with cystic granulosa cell tumors. We thus reviewed 10 cases of hyperreactio luteinalis in the setting of pregnancy, the largest pathologic cohort to date, to highlight notable features of this disorder. Patients ranged from 22 to 30 yr old. Most patients (n=6) presented at time of cesarean section with incidentally discovered ovarian masses. Three patients presented in the postpartum period, and 1 underwent surgery at 28 wk gestation due to the finding of a unilateral ovarian mass. The ovaries ranged from 8.5 to 29 cm and were multicystic and bilateral in 8 of the cases. Histologic examination demonstrated multiple, variably sized cystic follicles lined by a granulosa cell layer of varying thickness and theca cells with marked eosinophilic cytoplasm. Stromal edema was often prominent, with theca cells occasionally noted in nests, cords, and as single cells in foci of edema. Mitoses were generally seen more often in the granulosa cell layer (mean=2.6 per high power fields) compared with the theca cell layer (mean=1 per 10 high power fields). This series documents the key features of hyperreactio luteinalis that differentiate it from the other benign mass forming lesions encountered in pregnancy, most notably large solitary follicle cyst of pregnancy and puerperium, as well as cystic granulosa cell tumors, especially the juvenile variant, which may also present during pregnancy. Of particular use in differentiating them from juvenile granulosa cell tumor is the absence of pale or vacuolated cytoplasm and solid growth of granulosa cells in cases of hyperreactio luteinalis.

Congenital trichofolliculoma: a very rare presentation.

Trichofolliculoma is an uncommon hair follicle hamartoma. It usually appears during adulthood on the face or scalp as a single, asymptomatic, skin-colored papule/nodule with small protruding hairs. Histopathological features are diagnostic. Very rare congenital cases have been reported. Herein, we report a congenital trichofolliculoma in a 15-year-old girl.

Giant follicular cyst with maxillary sinus and pterygomaxillary space extension.

BACKGROUND: Dentigerous, follicular, cysts are developmental odontogenic cysts of reduced adamantine epithelium origin, associated with an impacted tooth. They are the second most frequent cystic pathology after inflammatory cysts. Although the frequency of dentigerous cysts associated with upper third molars is far lower than the mandibular ones, their complications are nothing but ordinary. Most of the times asymptomatic, being discovered during routine x-ray examinations, maxillary follicular cysts can grow to important size, altering the position of adjacent teeth, producing osteolysis of the nearby bone structures, as well as infectious complications, antral and orbital pathology. CASE REPORT: The present manuscript describes the clinical, radiological, pathological and therapeutical aspects of a giant follicular cyst with antral and pterygomaxillary extension, associated with an impacted upper third molar. CONCLUSION: The most efficient treatment of maxillary follicular cysts consists in the surgical removal of the lesion along with the involved tooth. Complete excision is mandatory to prevent recurrence. Extension into adjacent structures might complicate the surgery Preoperative evaluation and rigorous planning are essential, especially in large-sized lesions. Pathological examination is outmost importance in order to exclude aggressive transformations. KEY WORDS: Dentigerous follicular cyst, Maxillary sinus extension, Pterygomaxillary space.

Trichoblastic infundibular cyst should be renamed infundibular cyst with unique papillary projections.

Trichoblastic infundibular cyst (TBIC) was previously reported as a unique keratinous cystic lesion, which was characterized by the papillary projections of follicular germinative-like cells emanating from the cyst wall. Here, we report three additional cases of this cyst and discuss the pathogenesis of this unique entity. In all cases, a unilocular cyst contained keratin, and the cyst wall was composed of squamous epithelium. A number of cords and papillary projections emanated from the basal layer of the cyst wall. They were composed of cells with large nuclei and scant cytoplasm arranged in a peripheral palisade. Immunohistochemically, anti-cytokeratin 15, anti-cytokeratin 20, and anti-epithelial cell adhesion molecule antibodies were negative. Thus, these cells resembled follicular germinative cells or sebaceous mantle morphologically, but we failed to prove the differentiation immunohistochemically. The cyst was surrounded by fibrotic stroma and inflammatory cells, suggesting previous rupture of the cyst. We speculate that the cells of the projections possibly differentiate into the mantle rather than follicular germinative cells, even though we could not provide sufficient immunohistochemical evidence. We also suggest that they may be induced by special reaction to fibrohistiocytic stroma surrounding the infundibular cyst. Therefore, TBIC should be renamed infundibular cyst with unique papillary projections.

Eruptive vellus hair cysts in identical triplets with dermoscopic findings.

Eruptive vellus hair cysts (EVHCs) are asymptomatic, follicular, flesh-colored to hyperpigmented papules that are typically located on the chest and extremities with an unclear inheritance pattern and pathogenesis. We report a case of EVHCs in 4-year-old identical triplet girls. Our aim is to present another rare case of autosomal-dominant inheritance of EVHCs and to emphasize the utility of dermoscopy as a diagnostic aid for EVHCs that may be misdiagnosed clinically.

Ovarian lesions in 44 rabbits (Oryctolagus cuniculus).

The clinical and pathological records of 44 domestic, female rabbits with an age ranging from 6-124 months (median age: 63.5 month) were assessed retrospectively for ovarian lesions. Included were all rabbits that underwent an ovariohysterectomy with a subsequent pathological examination of the genital tract between March 1997 and June 2016. Pathological examination revealed ovarian lesions in 12 of the 44 rabbits including follicular cysts (n=7), cystic rete ovarii (n=3), widespread ovarian necrosis with dystrophic calcification (n=2), ovarian adenoma (n=1). Clinical examination including radiographs only suggested ovarian disorders in two cases of ovarian necrosis with dystrophic calcification and in two cases of cystic rete ovarii. Clinical significance was only conclusive in a case of cystic rete ovarii.

Co-occurrence of steatocystoma multiplex, eruptive vellus hair cysts, and trichofolliculomas.

An association between steatocystoma multiplex (SCM) and eruptive vellus hair cysts (EVHCs) has been recognized. Steatocystoma multiplex and EVHC have similar clinical features but distinctive histologic features. Rare cases of co-occurrence of these conditions have been known to occur on the trunk and the forehead. We report a rare case of the simultaneous occurrence of SCM, EVHC, and trichofolliculomas localized to the forehead.

Inflammatory follicular cysts associated to necrotic primary teeth.

BACKGROUND: Cysts are pathological cavities lined by epithelium and supported by connective tissue, containing fluid or semi-fluid substances and presenting developmental or inflammatory origins. CASE REPORT: This study aimed to evaluate a case series of inflammatory follicular cysts in children to clarify their nomenclature, diagnosis, clinical implications and treatment outcomes. Prevalence, aetio-pathogenesis, clinical, radiographic and microscopic findings, treatments and sequelae of this condition were assessed by using the records of patients treated between 2000 and 2015. Data were analysed and presented descriptively. FOLLOW-UP: This was performed periodically for 2 years to monitor lesion regression and complete eruption of impacted permanent teeth. All of the involved permanent teeth erupted naturally. Twelve cases of asymptomatic lesions with volumetric bone expansion were identified through radiographic findings or delayed tooth eruption in patients aged between 8 and 14 years. After establishing the diagnosis of inflammatory follicular cyst, necrotic primary teeth extraction was followed by decompression of the cystic lesions. CONCLUSIONS: The investigated cases were consistent with inflammatory follicular cyst diagnoses. The proposed treatment was considered to be appropriate, that was revealed by the maintenance of the permanent teeth involved during the patients follow-up.