Angiolipoma associated with antiretroviral switch therapy: a case report.

BACKGROUND: Angiolipomas have been well described in patients with HIV exposed to protease inhibitors with possible resolution after switching to non-nucleoside reverse transcriptase inhibitor-based regimens. Resolution of symptoms have occurred with switches to non-nucleoside reverse transcriptase inhibitor (NNRTI)-based regimens; however, little is known regarding the development of angiolipomas when switching from NNRTI- to modern, integrase strand transfer inhibitor-based regimens. We describe a patient who underwent switch therapy from tenofovir disoproxil fumarate/emtricitabine/efavirenz (TDF/FTC/EFV) to tenofovir alafenamide/FTC/bictegravir (TAF/FTC/BIC) who later developed angiolipomas. CASE PRESENTATION: A 55-year-old male had been on TDF/FTC/EFV for 8 years before switching to TAF/FTC/BIC. Nineteen months after antiretroviral switch, the patient presented with multiple lesions in the upper extremities and abdomen. Diagnostic biopsies revealed non-encapsulated angiolipomas and HHV-8 and non-alcoholic fatty liver disease was ruled out. New lesions continued to appear 29 months after ART switch, after which now lesions appeared and prior lesions remained stable with no increase in size noted. No surgical intervention or change in antiretroviral therapy was needed. CONCLUSIONS: Angiogenesis may have been suppressed with TDF/FTC/EFV treatment, however when switched to TAF/FTC/BIC, promoted the growth of angiolipomas. Clinicians should be aware of the impact of switching to modern ART therapies resulting in possible adipogenesis.

Angiolipoma an Uncommon Breast Mass in Men: A Case Series.

Angiolipomas are uncommon benign masses of the breast which are rarely described in the male breast. They do not have a typical mammographic appearance and can present with concerning features such as microcalcifications or irregular borders. Ultrasound is helpful in evaluating these masses most commonly appearing as oval, circumscribed, and hyperechoic. Clinical, radiological, and pathological information needs to be carefully evaluated as angiolipomas can be confused with malignant pathology. Three cases of angiolipomas of the male breast are reported in this study with mammographic, sonographic, and pathologic correlation.

[Esophageal Angiolipoma:Report of One Case and Literature Review].

Esophageal angiolipoma is a rare disease with unspecific clinical manifestations.This paper reported a case of esophageal angiolipoma confirmed by upper gastrointestinal endoscopy and summarized the clinical manifestations,endoscopic and pathological features,treatment and prognosis of the patients by reviewing the relevant literature,aiming to provide references for clinical diagnosis and treatment of this disease in the future.

Frequent activating PIK3CA mutations in sporadic angiolipoma.

BACKGROUND: Angiolipoma (AL) is considered as a lipoma variant that is characterized by the combination of mature adipocytes and capillary blood vessels diffusely distributed within the tumor. With the exception of recurrent PRKD2 mutations of uncertain pathogenetic significance, the genetic abnormalities of ALs are unknown, in the absence of any of the specific chromosomal aberrations described in other lipoma variants. METHODS: Formalin-fixed and paraffin-embedded blocks of 13 conventional ALs and 5 cellular ALs from 17 individuals were retrieved and analyzed for mutations in exons 9 and 20 of PIK3CA by polymerase chain reaction and Sanger sequencing. RESULTS: Activating PIK3CA mutations were identified in 14 tumors (78%). All PIK3CA-mutated samples carried the same exon 9 mutation, c.1634A>C (p.E545A). No mutation was detected in exon 20 of PIK3CA. No significant difference between PIK3CA-mutated and wild-type samples appeared to exist based on age, gender, and location of the tumor. All 5 cellular ALs carried the p.E545A PIK3CA mutation. CONCLUSION: The high frequency of the p.E545A PIK3CA mutation in both conventional and cellular ALs suggests that activation of the PI3K/AKT pathway plays a key role in AL pathogenesis and reinforces the concept that cellular AL should be regarded as a variant of AL.

Solitary noninfiltrating angiolipoma on the finger, an unusual localization.

Angiolipoma, a subtype of lipoma, is a benign adypocytic soft tissue tumor composed of mature adipose tissue and small vascular proliferations. This entity makes up 5-17% of all lipomas. The diagnosis is made by clinical and pathological examination, ultrasonography, and/or magnetic resonance imaging (MRI). It is generally an encapsulated tumor (noninfiltrative), but rarely has an infiltrative form. Angiolipoma mostly occurs on the trunk and extremities with male predominance. The forearm is the most frequent location for angiolipomas. It is very rarely seen on the fingers. Herein, we report a patient with solitary noninfiltrating angiolipoma on a finger. The patient was treated with surgical excision and no recurrence has been noted over one year of observation.

Frequent low-level mutations of protein kinase D2 in angiolipoma.

Tumours displaying differentiation towards normal fat constitute the most common subgroup of soft tissue neoplasms. A series of such tumours was investigated by whole-exome sequencing followed by targeted ultra-deep sequencing. Eighty per cent of angiolipomas, but not any other tumour type, displayed mutations in the protein kinase D2 (PRKD2) gene, typically in the part encoding the catalytic domain. The absence of other aberrations at the chromosome or RNA level suggests that PRKD2 mutations are critical for angiolipoma development. Consistently, the mutated PRKD2 alleles were present at low (3-15%) frequencies, indicating that only a subset of the tumour cells is affected. Indeed, by sequencing mature fat cells and other cells separately, the former typically showed the highest mutation frequencies. Thus, we hypothesize that altered PRKD2 signalling in the adipocytic cells drives tumourigenesis and, in agreement with its pivotal role in angiogenesis, induces the vessel formation that is characteristic for angiolipoma. Copyright © 2016 Pathological Society of Great Britain and Ireland. Published by John Wiley & Sons, Ltd.

Infiltrating angiolipoma of the foot: magnetic resonance imaging features and review of the literature.

Angiolipoma is a benign soft tissue tumor with two subtypes: non-infiltrating and infiltrating. Although histologically benign, infiltrating angiolipoma can invade surrounding structures. The foot is a very rare location for angiolipoma, with only four cases reported in the English literature, including one infiltrating type. Here, we report a very rare case of infiltrating angiolipoma of the foot with magnetic resonance imaging (MRI) and ultrasonography (US) findings. A 7-year-old boy presented with a slowly growing foot mass. MRI showed an unencapsulated mass involving the third web space extending to the foot dorsum and sole. The mass was isointense to subcutaneous fat and was mixed with internal T1 low-signal-intensity enhancing areas. On US, we observed a heterogeneously hypoechoic mass with internal vascularity. Imaging and clinical features of angiolipoma and the radiologic differential diagnoses of a fat-containing mass in the pediatric foot are reviewed here. When there is an ill-defined foot mass with a fat component and variable enhancing portions in a child, infiltrating angiolipoma should be included in the differential diagnosis along with other fat-containing tumors.

Lipomatous tumors of the breast: A contemporary review.

Breast tumors with lipomatous or liposarcomatous components are infrequently encountered, but can be a source of diagnostic difficulty if the context of the fatty differentiation is not recognized. Among the true adipocytic tumors, lipoma is the most common lipomatous tumor arising in the breast. Several mammary spindle cell tumors may show adipocytic differentiation, including fibroepithelial tumors and myofibroblastoma. Liposarcomatous components most often arise in malignant phyllodes tumors, as opposed to primary liposarcomas of the breast which are believed to be uncommon. This article will review the spectrum fat-containing tumors of the breast with an emphasis on differential diagnosis and insights from recent molecular studies.

Rapamycin and rapalogs for tuberous sclerosis complex.

BACKGROUND: Previous studies have shown potential benefits of rapamycin or rapalogs for treating people with tuberous sclerosis complex. Although everolimus (a rapalog) is currently approved by the FDA (U.S. Food and Drug Administration) and the EMA (European Medicines Agency) for tuberous sclerosis complex-associated renal angiomyolipoma and subependymal giant cell astrocytoma, applications for other manifestations of tuberous sclerosis complex have not yet been established. A systematic review is necessary to establish the clinical value of rapamycin or rapalogs for various manifestations in tuberous sclerosis complex. OBJECTIVES: To determine the effectiveness of rapamycin or rapalogs in people with tuberous sclerosis complex for decreasing tumour size and other manifestations and to assess the safety of rapamycin or rapalogs in relation to their adverse effects. SEARCH METHODS: Relevant studies were identified by authors from the Cochrane Central Register of Controlled Trials (CENTRAL), Ovid MEDLINE, and clinicaltrials.gov. Relevant resources were also searched by the authors, such as conference proceedings and abstract books of conferences, from e.g. the Tuberous Sclerosis Complex International Research Conferences, other tuberous sclerosis complex-related conferences and the Human Genome Meeting. We did not restrict the searches by language as long as English translations were available for non-English reports.Date of the last searches: 14 March 2016. SELECTION CRITERIA: Randomized or quasi-randomized studies of rapamycin or rapalogs in people with tuberous sclerosis complex. DATA COLLECTION AND ANALYSIS: Data were independently extracted by two authors using standard acquisition forms. The data collection was verified by one author. The risk of bias of each study was independently assessed by two authors and verified by one author. MAIN RESULTS: Three placebo-controlled studies with a total of 263 participants (age range 0.8 to 61 years old, 122 males and 141 females, with variable lengths of study duration) were included in the review. We found high-quality evidence except for response to skin lesions which was judged to be low quality due to the risk of attrition bias. Overall, there are 175 participants in the treatment arm (rapamycin or everolimus) and 88 in the placebo arm. Participants all had tuberous sclerosis complex as proven by consensus diagnostic criteria as a minimum. The quality in the description of the study methods was mixed, although we assessed most domains as having a low risk of bias. Blinding of treatment arms was successfully carried out in all of the studies. However, two studies did not report allocation concealment. Two of the included studies were funded by Novartis Pharmaceuticals.Two studies (235 participants) used oral (systemic) administration of everolimus (rapalog). These studies reported response to tumour size in terms of the number of individuals with a reduction in the total volume of tumours to 50% or more relative to baseline. Significantly more participants in the treatment arm (two studies, 162 participants, high quality evidence) achieved a 50% reduction in renal angiomyolipoma size, risk ratio 24.69 (95% confidence interval 3.51 to 173.41) (P = 0.001). For the sub-ependymal giant cell astrocytoma, our analysis of one study (117 participants, high quality evidence) showed significantly more participants in the treatment arm achieved a 50% reduction in tumour size, risk ratio 27.85 (95% confidence interval 1.74 to 444.82) (P = 0.02). The proportion of participants who showed a skin response from the two included studies analysed was significantly increased in the treatment arms, risk ratio 5.78 (95% confidence interval 2.30 to 14.52) (P = 0.0002) (two studies, 224 participants, high quality evidence). In one study (117 participants), the median change of seizure frequency was -2.9 in 24 hours (95% confidence interval -4.0 to -1.0) in the treatment group versus -4.1 in 24 hour (95% confidence interval -10.9 to 5.8) in the placebo group. In one study, one out of 79 participants in the treatment group versus three of 39 in placebo group had increased blood creatinine levels, while the median percentage change of forced expiratory volume at one second in the treatment arm was -1% compared to -4% in the placebo arm. In one study (117 participants, high quality evidence), we found that those participants who received treatment had a similar risk of experiencing adverse events compared to those who did not, risk ratio 1.07 (95% confidence interval 0.96 - 1.20) (P = 0.24). However, as seen from two studies (235 participants, high quality evidence), the treatment itself led to significantly more adverse events resulting in withdrawal, interruption of treatment, or reduction in dose level, risk ratio 3.14 (95% confidence interval 1.82 to 5.42) (P < 0.0001).One study (28 participants) used topical (skin) administration of rapamycin. This study reported response to skin lesions in terms of participants' perception towards their skin appearance following the treatment. There was a tendency of an improvement in the participants' perception of their skin appearance, although not significant, risk ratio 1.81 (95% confidence interval 0.80 to 4.06, low quality evidence) (P = 0.15). This study reported that there were no serious adverse events related to the study product and there was no detectable systemic absorption of the rapamycin during the study period. AUTHORS' CONCLUSIONS: We found evidence that oral everolimus significantly increased the proportion of people who achieved a 50% reduction in the size of sub-ependymal giant cell astrocytoma and renal angiomyolipoma. Although we were unable to ascertain the relationship between the reported adverse events and the treatment, participants who received treatment had a similar risk of experiencing adverse events as compared to those who did not receive treatment. Nevertheless, the treatment itself significantly increased the risk of having dose reduction, interruption or withdrawal. This supports ongoing clinical applications of oral everolimus for renal angiomyolipoma and subependymal giant cell astrocytoma. Although oral everolimus showed beneficial effect on skin lesions, topical rapamycin only showed a non-significant tendency of improvement. Efficacy on skin lesions should be further established in future research. The beneficial effects of rapamycin or rapalogs on tuberous sclerosis complex should be further studied on other manifestations of the condition.

Angiolipoma of the Orbit: A Rare Tumor in an Unusual Location.

Despite their relatively frequent occurrence on the trunk and extremities, lipomas rarely present in the orbit. Rarer still are variants of lipoma such as fibrolipoma, myxoid lipoma, and angiolipoma. The authors report a 66-year-old woman who presented with a large, slowly growing tumor of the forehead and orbit. The case presentation conforms to the tenets of the Declaration of Helsinki and is HIPAA compliant. Clinical and radiographic evidence suggested a lipomatous type of tumor, and excisional biopsy revealed adipose proliferation with numerous small vessels and fibrin thrombi consistent with angiolipoma. The tumor was completely excised without ophthalmic sequelae or recurrence in 6 months of follow up. To the authors' knowledge, this tumor represents only the second reported case of this type of highly vascularized lipomatous lesion within the orbit. This case is a novel entity in the differential diagnosis of orbital tumors and demonstrates the value of total excision in such cases.

Imaging diagnosis--computed tomographic, surgical, and histopathologic characteristics of an infiltrative angiolipoma in a dog.

A 6-year-old female spayed Shetland Sheepdog presented for evaluation of a subcutaneous mass over the right prescapular region. The mass had been cytologically diagnosed as a lipoma by the referring veterinarian 20 months prior, but had grown significantly and was very firm. CT scan of the mass was suggestive of neoplasia; however, the tissue of origin could not be determined. Histopathologic evaluation diagnosed infiltrative angiolipoma, and marginal resection of the tumor was performed. Infiltrative angiolipomas are benign but locally aggressive neoplasms uncommonly reported in veterinary medicine. This report correlates the clinical, CT, and histopathologic characteristics of an infiltrative angiolipoma.

[Adipocytic tumors]. / Tumeurs adipeuses.

Adipocytic tumors are the most common mesenchymal neoplasms, liposarcoma accounting for approximately 20% of soft tissue sarcomas. The differential diagnosis between benign and malignant tumors is often problematic and represents a significant proportion of consultation cases. The goal of this article is to review liposarcoma subtypes, the main benign adipocytic neoplasms: lipoblastoma, hibernoma, spindle/pleomorphic cell lipoma, chondroid lipoma, as well as non adipocytic neoplasms with a lipomatous component such as lipomatous solitary fibrous tumor, emphasizing on practical differential diagnosis issues, and immunohistochemical and molecular tools allowing their resolution.

Management of infiltrating spinal epidural angiolipoma.

Angiolipomas of the spine are rare benign tumors commonly presenting with compressive myelopathy. The present report describes a case of spinal angiolipoma with thoracic mediastinal extension in a 50-year-old woman. She presented with a long-standing history of mid-back pain with progressive lower extremities weakness. An MRI showed a heterogeneously enhancing mass located in the posterior epidural space of the thoracic spine with mediastinal extension. Histopathological examination demonstrated features consistent with spinal angiolipoma. This report emphasizes the diagnosis and therapeutic management options of infiltrating spinal angiolipomas.

Spinal angiolipoma: etiology, imaging findings, classification, treatment, and prognosis.

PURPOSE: To summarise our experience treating patients with spinal angiolipomas (SAs) and to evaluate factors relating to its prognosis. METHODS: We retrospectively reviewed the records of patients diagnosed with SAs who received surgical treatment from January 2001 to February 2013. RESULTS: Twenty-one patients were described. We divide SAs into two types: "intraspinal" and "dumbbell-shaped". The former were further subclassified as "with lipomatosis" and "without lipomatosis". Overweight people are more likely to get the "with lipomatosis" type which needs different surgical strategy and/or a diet therapy to get better outcomes. CONCLUSION: Diagnosis of SAs should be made with reference to clinical, radiological, and pathological findings. Application of different methods is needed to treat SAs.

Spinal angiolipoma: experience of twelve patients and literature.

PURPOSE: To summarize our experience in treating patients with spinal angiolipomas. MATERIALS AND METHODS: We conducted a retrospective review of the case records of patients with spinal angiolipoma who underwent surgery between March 1992 and March 2013. RESULTS: Of the 12 patients seen during the study period, 10 patients had total removal of the lesion and 2 patients had sub-total removal. The postoperative course was uneventful, and the outcomes were good in patients who underwent total excision of the lesion. No patient received adjuvant radiation even in the patients with sub-total excision. CONCLUSION: In patients with spinal angiolipoma, total excision should be achieved and is associated with good outcomes.

[Sonographic appearance of benign subcutaneous nodules--including color Doppler sonography].

OBJECTIVE: Color Doppler sonography is used to distinguish malignant from benign subcutaneous nodules because many malignant tumors show an increased number of vessels. But benign lesions, such as inflammatory epidermal cysts, pilomatricomas, palmoplantar fibromatoses show vascular flow signals. We analyze diagnostic factors for subcutaneous nodules on the basis of ultrasonographic appearance including color Doppler sonography. METHODS: We evaluated with sonography 79 subcutaneous nodules in histopathologically proven cases. RESULTS AND DISCUSSION: The diagnostic findings for 28 epidermal cysts were increased posterior echo(sensitivity 77%, specificity 90%). Vascular flow signals were seen in 7 cases(25%). The signals were seen from periphery to adjacent zone of the mass. They were accompanied by inflammatory granulation tissues histologically. The percentage of blood flow may be related to the percentage of "ruptured" epidermal cysts. Pilomatricomas (n = 9) had internal echogenic foci or Doppler flow signals, and no posterior enhancement (sensitivity 89%, specificity 78%). Seven pilomatricomas showed Doppler flow signals in the mass. The percentage of flow signals is explained by the degree of internal echogenic foci because remarkable calcium deposits producing posterior acoustic shadowing prevent us from flow signals. Palmoplantar fibromatoses (n = 5) were ill-defined margins and showed internal vascularity with no posterior enhancement (sensitivity 80%, specificity 94%). CONCLUSION: More benign lesions showed vascular flow signals than reported previously. We have to diagnose subcutaneous nodules referring to not only color Doppler but also gray scale sonography.

Angiofibrolipoma of the retromolar pad region: case report.

Angiofibrolipoma is a rare histopathological variant of lipoma, characterized by mature adipocytes, blood vessels and dense collagenous tissue. It is seldom seen in the oral and maxillofacial region. Diagnosis of angiofibrolipoma is only possible based on its histopathological features. This report presents the case of a 63-year-old male patient with the complaint of a polyp-like mass, felt from the left retromolar pad region. The mass was found as a small prominent lesion that had grown gradually for 1.5 years. Our differential diagnosis was irritation fibroma and fibrous pyogenic granuloma. This report also includes a comprehensive reference to previously reported data, as found through PubMed and Google Search, which revealed this type of case rarely has been documented.

Demographic and clinical data of patients with spinal epidural angiolipomas.

Spinal epidural angiolipomas are rare, benign, mesenchymal tumors. It remains unclear whether spinal epidural angiolipomas are genuinely rare or merely underreported. Herein, we assessed the demographic and clinical characteristics of patients with spinal epidural angiolipoma. We collected data from patients with spinal epidural angiolipoma from three sources. First, we retrospectively analyzed data from patients diagnosed with spinal epidural angiolipoma in our hospital between January 1, 2014, and December 31, 2023. Second, we performed a literature review of studies retrieved from PubMed. Third, we retrieved detailed data of patients with spinal angiolipoma from the Surveillance, Epidemiology, and End Results (SEER) database. We conducted a descriptive analysis to investigate the demographic and clinical characteristics of patients with spinal epidural angiolipoma. At our institution, three patients were diagnosed with spinal epidural angiolipoma. Additionally, we identified 116 patients from the literature review and 15 patients from the SEER database. We reviewed the treatment history and imaging features of the three patients from our institution. The descriptive analysis of the data collected from the literature review was consistent with previous reports. For example, 63.0% of lesions were located at the thoracic level. 31.9% of these lesions involved two vertebral bodies, while 75.6% involved 2-4 vertebral bodies. The most common symptoms experienced by patients were back pain, paraparesis, and numbness in the legs. Surgery was the primary treatment option for most patients, and complete tumor resection was achieved in the majority of patients. The male:female ratio was 1:1.4, the median age at diagnosis for the patients from the literature was 49 years old, and the median follow-up was 24 months. Notably, most of the reports came from Asia and there were few reports from Africa. The findings from the SEER database indicated a male:female ratio of 2:1. The peak incidence, which is typically reported in the fifth decade of life, was not observed. We presented three cases of spinal epidural angiolipoma and supplemented our findings with a literature review and population-based analysis according to the SEER database for the United States population. We believe that our research will enhance clinicians' comprehension of this uncommon tumor.

Cellular angiolipoma: a clinicopathological and immunohistochemical study of 12 cases.

Cellular angiolipoma is a rare variant of angiolipoma. To date, only a few reports have been published in the literature. In this report, we analyzed a series of 12 cases to further evaluate this entity. Seven patients were men and 5 were women, aged from 23 to 62 years (average, 39 years). Clinically, the lesions occurred predominantly in the subcutaneous tissue of the extremities and trunk wall, usually as part of multiple small tender nodules. In 5 cases, cellular angioloipoma coexisted with the conventional angiolipoma. Histologically, the tumor differed from its conventional counterpart by containing densely cellular areas that occupied more than 90% of the tumors. By comparison, adipocytic component accounted only for a minimal portion (<10%). CD31 and CD34 immunostainings highlighted the rich vasculature in the cellular areas. In conclusion, the clinical and morphological overlaps between the cellular angiolipoma and its conventional counterpart indicate that they lie within the same spectrum of a single entity.