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PubMed2001

Localized Birt-Hogg-Dubé syndrome with prominent perivascular fibromas.

The autosomal dominant Birt-Hogg-Dubé syndrome is a cutaneo-intestinal condition that manifests on the skin in the form of multiple, skin-colored small papules that, histologically, prove to be mantleomas (fibrofolliculomas and trichodiscomas). These cutaneous lesions usually appear in the region of the head, neck, and upper part of the trunk. To date, only a single report in the literature describes the localized occurrence of this syndrome. We now describe a localized form of the Birt-Hogg-Dubé syndrome in a man with multiple mantleomas that were confined to the left half of the face, and which, in part, were arranged in the form of plaques. Another striking finding in this patient was a conspicuous vascular component in the lesions, characterized by a pronounced, well-demarcated fibrosis in the region of cutaneous blood vessel proliferations. Because perivascular fibromas have already been observed in other patients with Birt-Hogg-Dubé-syndrome, the perivascular fibroma, with fibrofolliculoma and trichodiscoma, must be included within this syndrome's spectrum of skin changes.

Adult↗
PubMed2005

Exogenous trauma simulating perifollicular fibromas.

The patient is a 63-year-old woman who presented to her dermatologist for the removal of two nevi near her mouth. Histologic examination revealed melanocytic nevi showing maturation with dermal descent. Within the dermis, adjacent to these nevoid cells, were multiple large hair follicles that were surrounded by dense fibrosis. The fibrous sheaths contained thickened, dense collagen bundles and were well circumscribed, resembling perifollicular fibromas. Further discussion with the patient and the dermatologist revealed that the patient had "plucked" hair from these nevi. We, therefore, believe that the perifollicular fibrosis that we observed is secondary to trauma. We present this case to remind all that post-traumatic events can simulate perifollicular fibromas and that the erroneous diagnosis of such could lead to the erroneous diagnosis of Birt-Hoggs Dubé syndrome.

Diagnosis, Differential↗
PubMed2001

Gardner-associated fibromas (GAF) in young patients: a distinct fibrous lesion that identifies unsuspected Gardner syndrome and risk for fibromatosis.

Gardner syndrome (GS), caused by mutations in the adenomatous polyposis coli (APC) gene, is characterized by polyposis coli, osteomas, and various soft-tissue tumors. If undetected or untreated, virtually all patients develop colonic carcinoma at a young age. Early detection, while essential, can be difficult because of attenuated phenotypes or spontaneous mutations. We present the clinicopathologic features of 11 identical fibromatous lesions that we have termed Gardner-associated fibroma (GAF), which not only appear to be a part of the spectrum of lesions associated with GS but, in some cases, represent the sentinel event leading to its detection. The GAFs occurred in 11 patients (5 boys and 6 girls; age range, 3 months-14 years), were solitary (n = 7) or multiple (n = 4), and occurred in the superficial and deep soft tissues of the paraspinal region (n = 7), back (n = 3), face (n = 2), scalp (n = 2), chest wall (n = 2), thigh (n = 1), neck (n = 1), and flank (n = 1). Histologically, GAFs resemble nuchal-type fibromas (NFs), consisting of thick, haphazardly arranged collagen bundles between which are found occasional bland fibroblasts, and having margins that frequently engulf surrounding structures including adjacent fat, muscle and nerves. After surgical excision, four patients developed recurrences that were classic desmoid fibromatoses (DFs). In one patient with multiple GAFs, one lesion had the features of GAF and DF in the absence of surgical trauma. A family history of GS or polyposis (n = 6) or DF (n = 1) was known at the time of surgery in seven patients. In three patients, the diagnosis of GAF resulted in the diagnosis of unsuspected APC in older family members, with the detection of an occult colonic adenocarcinoma in one parent. In the family of the remaining patient, no stigmata of GS were present. Genetic analysis of this child was performed to investigate the presence of a spontaneous (new) mutation; however, no abnormalities were detected. The significance of GAF is that it serves as a sentinel event for identifying GS kindreds, including those with a high risk for the development of DF, and it may potentially identify children with spontaneous mutations of the APC gene. Because NFs and GAFs resemble one another, we suggest that a subset of NF occurring in multiple sites, unusual locations, or children may be GAF.

Adenomatous Polyposis Coli↗
PubMed2006

Cellular fibromas of the ovary: a study of 75 cases including 40 mitotically active tumors emphasizing their distinction from fibrosarcoma.

Cellular fibroblastic tumors of the ovary are currently classified as either cellular fibroma (CF) or fibrosarcoma. The former are characterized by bland nuclei, 3 or fewer mitotic figures per 10 high-power fields (MFs/10 HPFs), and a low malignant potential, whereas fibrosarcomas usually have severe nuclear atypia, > or = 4 MFs/10 HPFs, and an aggressive clinical course. The prognosis of cellular fibromatous tumors with > or = 4 MFs/10 HPFs and low-grade cytology is not established and it is the purpose of this study to investigate that aspect. It has been our anecdotal experience that otherwise typical CFs with > or = 4 MFs/10 HPFs usually have a benign clinical course, suggesting that such tumors should be regarded as "mitotically active cellular fibroma" (MACF) rather than fibrosarcoma. Seventy-five cellular fibromatous neoplasms were analyzed to determine their clinicopathologic features and the appropriateness of "MACF" as a designation for otherwise typical CFs with > or = 4 MFs/10 HPFs. The mean age of patients with CF (n = 35, 0 to 3 MFs/10 HPFs) and MACF (n = 40, > or = 4 MFs/10 HPFs) was 51 and 41 years, respectively. Patients most commonly presented with symptoms related to a pelvic mass. All tumors were unilateral. The mean tumor size of CFs was 8.0 cm and 9.4 cm for MACFs. The majority of the tumors were solid; approximately one-third of them had a cystic component. Ovarian surface adhesions, involvement of the ovarian surface, or both, was present in 6% of CFs and 10% of MACFs. Eleven percent of CFs and 13% of MACFs were associated with extraovarian involvement. All tumors consisted of cellular, intersecting bundles of spindle cells with bland nuclear features. The mean highest mitotic count for MACFs was 6.7 MFs/10 HPFs (range 4 to 19 MFs/10 HPFs). Follow-up of 3 months to 12 years (mean 4.75 y) was available in 18 of the 40 patients with MACFs and was uneventful in all cases. We conclude that cellular fibromatous neoplasms with bland cytology and elevated mitotic counts are associated with favorable patient outcome and should be diagnosed as MACF rather than fibrosarcoma, which usually have moderate to severe atypia and elevated mitotic rates. As prior observations have shown that even typical CFs can occasionally recur locally, particularly if they are associated with rupture or adherence, long-term follow-up for patients with CFs and MACFs is appropriate.

Adult↗
PubMed1999

Juvenile ossifying fibroma of the midface.

Juvenile ossifying fibroma is an unusual maxillofacial fibro-osseous lesion characterized by cell-rich osteoid strands. A 7-year-old girl presenting with a massive juvenile ossifying fibroma of the maxilla resulting in facial deformity, orbital displacement, and extension into the anterior skull base is discussed. The importance of combining clinical, radiographic, and histopathological findings for the diagnosis and management of the tumor is discussed.

Child↗
PubMed2004

Extensive neglected psammomatoid ossifying fibroma with craniofacial deformity.

The psammomatoid ossifying fibroma is a rare and aggressive lesion that usually arises from the sinonasal region. The lesion is well circumscribed with the pathognomonic feature of concentric or laminated spherical ossicles that appear similar to psammoma bodies. The authors report a case of an extensive and neglected psammomatoid ossifying fibroma in a 10-year-old West African girl. She had a 5-year history of a slowly growing tumor, which at presentation was 15 cm in diameter and had resulted in left craniofacial deformity as well as left visual impairment. Due to the large tumor, the surgical resection plan necessitated a cranio-facial neurosurgical approach in combination with autogenous bone grafts for cranofacial reconstruction, and allogenous demineralized bone grafts for donor site coverage. In addition, preoperative embolization was utilized to reduce tumor vascularity. However, this embolization resulted in reduction of pericranial flap vascularity, leading to flap ischemia, infection and subsequent removal of necrotic autogenous bone graft. Ultimately, the objectives of large tumor resection and acceptable aesthetic outcome were met after controlling an episode of postoperative infection.

Bone Transplantation↗
PubMed1991

Desmoplastic fibroma in the thoracic spine.

Desmoplastic fibroma of the spine is an extremely rare, benign tumor. So far, only 7 cases have been reported in the world. We have treated 1 case of desmoplastic fibroma in the thoracic spine. Due to insufficient removal of the tumor, local recurrence was observed, and a second surgery posteriorly and anteriorly was performed for complete removal 8 years following the first operation. After resection of the tumor, Luque instrumentation was used to reconstruct the destroyed spine. At present, 7 years after the second operation, there are no signs or symptoms. Therefore, complete resection of the tumor is most important in the treatment, and rigid spinal instrumentation and bone grafts must be used after removing the tumor in spine cases.

Adult↗
PubMed2000

Thoracic epidural desmoplastic fibroma.

Desmoplastic fibroma is a relatively uncommon tumor and rarely involves the spine. The authors describe a 20-year-old woman with a thoracic epidural desmoplastic fibroma treated by complete resection and posterior spinal fusion. Four years after surgery, neither the tumor nor clinical symptoms have recurred. Thus, complete resection is considered necessary to treat this tumor.

Adult↗
PubMed1983

Computed tomography of ossifying fibroma of the tibia.

Two ossifying fibromas of the tibia were studied by computed tomography, which showed that the lesions were composed predominantly of high density tissue and that they were located in the cortex. The tissue density indicated that the tumors were diffusely ossified or calcified. The intracortical site is typical of ossifying fibroma, but it is unusual in other lesions of similar appearance.

Adult↗
PubMedDate not supplied

Meigs syndrome and ovarian fibroma: CT findings.

We present a case of ovarian fibroma with associated Meigs syndrome, Even though the CT findings of ovarian fibroma have been reported previously, our case, with associated meigs syndrome, demonstrated a broader spectrum of CT findings.

Female↗
PubMed1981

Morphological and histochemical studies on the collagen in neurinomas, neurofibromas, and fibromas.

The collagen in neurinomas, neurofibromas was studied by optical and electron microscopy. Neurinomas were shown to be composed mainly of thin argyrophilic, weakly birefringent fibers formed by thin collagen fibrils. Fibromas presented only thick, non-argyrophilic, strongly birefringent fibers composed of thick collagen fibrils. Neurofibromas showed populations of the two above-mentioned types of fibers occupying different areas of their parenchyma. These results suggest that the collagen of neurinomas is mainly of type III, while fibromas present collagen type I, and neurofibromas are composed of both types of collagen.

Collagen↗
PubMed2000

An intrasylvian "fibroma" in a child with cystic fibrosis: case report.

OBJECTIVE AND IMPORTANCE: Intracranial fibrous tumors are uncommon during childhood. An unusual case of benign intrasylvian "fibroma" that has remained clinically and radiographically stable more than 3 years after a subtotal resection is described. CLINICAL PRESENTATION: A 9-year-old girl with cystic fibrosis presented with new-onset focal seizures referable to a large calcified left sylvian fissure mass. INTERVENTION: An open biopsy with subtotal resection of the lesion revealed a benign process characterized by exuberant fibrocollagenous tissue intermeshed with chronic inflammatory cells and foreign body giant cells, encompassing islands of gliotic brain tissue. Immunohistochemical analysis showed staining for epithelial membrane antigen and reticulin within some of the spindle cells, although the majority were nonreactive. The majority of tumor cells exhibited staining for laminin; CD34 staining was absent. Ultrastructural studies were also suggestive of a fibroblastic rather than a meningothelial origin of the lesion, with elongated cells separated by abundant extracellular collagen. Although dense adherence of the mass to the pial surface and the middle cerebral artery vessels precluded a complete resection, the patient remains seizure-free without anticonvulsant therapy more than 3 years postoperatively with no evidence of growth of the lesion. CONCLUSION: The lesion in this patient bears morphological similarity to a rare group of tumors referred to as "intracerebral fibromas," although a variety of other rare mesenchymal neoplasms were also considered within the differential diagnosis. However, the absence of any definite neoplastic features, the finding of chronic inflammatory changes, and the lack of growth of the residual tumor during an extended follow-up interval indicate that the mass may represent either an extremely indolent neoplasm or a nonneoplastic process. The differential diagnosis of intracranial fibrous tumors is contrasted with that of the reported case.

Brain Neoplasms↗
PubMed1999

Desmoplastic fibroma of the zygoma.

Desmoplastic fibroma is an exceedingly rare primary bone tumor. Previous reports have not included the presentation of this tumor associated with the zygoma. This report describes the unusual presentation and surgical management of a desmoplastic fibroma of the zygoma.

Bone Neoplasms↗
PubMed1995

Desmoplastic fibroma: a role for radiotherapy?

Desmoplastic fibroma is a rare, locally aggressive, benign tumor that is considered the skeletal counterpart of the desmoid tumor of soft tissues. Although the treatment of choice of desmoplastic fibroma is surgical excision, radiation therapy should be considered when surgery is not a viable option.

Adult↗
PubMed2005

CASE REPORTS: intraarticular fibroma of the tendon sheath in the knee.

We report a rare case of fibroma of the tendon sheath in the knee. To our knowledge, only three previous cases of fibroma of the tendon sheath in the knee have been reported in the English literature. In our case, T1-weighted magnetic resonance imaging revealed a low intensity signal, T2-weighted magnetic resonance imaging showed a mixed area of high and low signal intensity, and gadolinium-diethylene triaminepentaacetic acid-enhanced magnetic resonance imaging showed faint enhancement at the periphery of the lesion. Magnetic resonance imaging therefore was valid for detection of this intraarticular lesion. The lesion, which was located between the posterior capsule of the left knee and the posterior cruciate ligament, was excised arthroscopically.

Adult↗
PubMed2004

Unguioblastoma and unguioblastic fibroma--an expanded spectrum of onychomatricoma.

Onychomatricoma is a rare tumor that appears to originate from cells of the nail matrix. Three cases of onychomatricoma that met Perrin et al.'s1 histologic criteria of onychomatricoma are described. However, using a single term to classify all three tumors ignores the apparent microscopic differences that exist among them. To demonstrate better the spectrum of so-called onychomatricoma and properly acknowledge the noticeable disparity among our cases, a series of terms is proposed. This terminology is based on the histologic spectrum of epithelial-stromal ratio of stromal cellularity and of extent nuclear pleomorphism. Use of such criteria has a precedent in the classification of follicular and odontogenic fibroepithelial neoplasms. This new nomenclature includes "unguioblastoma" for tumors with a predominant epithelial component and "unguioblastic fibroma" for tumors where a cellular stroma is more prominent and characteristic. The term "atypical unguioblastic fibroma" is used to describe a third rare neoplasm, in which the cellular stroma shows nuclear pleomorphism and atypia with an increase of mitotic activity.

Adult↗
PubMed1991

Sclerotic fibromas of the skin.

We report a case of a 34-year-old Japanese woman with a fibrous tumor on the left palm that fulfilled the criteria of sclerotic fibroma of the skin. Immunohistochemically, the tumor cells were positive for vimentin, but negative for S-100 protein and neuron-specific enolase. Ultrastructurally, the cross-striations of collagen microfibrils were less prominent. Our case supports the concept that sclerotic fibroma of the skin is a distinctive lesion which may represent a hamartoma of the skin.

Adult↗