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.

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.

Intraosseous angiolipoma of the mandible.

INTRODUCTION: The angiolipoma is an uncommon histologic variant of lipomas, accounts for 5-17% of lipomas. Intraosseous lipomas of the jaws are extremely rare. The aim of the present article is to report and discuss another case of intramandibular angiolipoma. CASE REPORT: A 66-year-old man was referred to the Dentistry Department for the diagnosis and treatment of a lesion in the mandible. Past medical history included HIV positivity. The patient was asymptomatic. A CT scan revealed the incidental finding of a well-defined radiolucent image in the right body of the mandible, in the edentulous first molar / second premolar region, with sclerotic margins, and in contact with the mandibular canal, that was preserved. A conservative enucleation under local anesthesia was decided, together with the patient. Histopathological findings were consistent with the diagnosis of angiolipoma. DISCUSSION: Clinically, most intraosseous lipomas are asymptomatic, including the present case. Histological findings are essential for the diagnosis of intraosseous angiolipoma: mature neoplastic adipocytes streaked with numerous interspersed vascular spaces / blood vessels surrounded by mast cells and filled with red cells and several fibrous microthrombi are characteristics of angiolipomas. CONCLUSIONS: The diagnosis of intraosseous angiolipomas of the jaws may be difficult due to their rarity and it requires a histopathological examination.

Esophageal Angiolipoma:Report of One Case and Literature Review / 中国医学科学院学报

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.

Thoracic Spinal Angiolipomas: A Systematic Review.

BACKGROUND: Spinal angiolipomas (SAs) are rare, benign tumors, representing 0.0004 to 1.2% of angiolipomas, usually located at the extradural and posterior thoracic level, with multimetameric extension. METHODS: A systematic literature review was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. The authors searched on PubMed and Scopus databases for published articles with the Mesh term "spinal angiolipoma" and pertinent associations. Language restriction to English papers was applied. The authors also reported three emblematic cases of patients who underwent surgical resection of spindle-shaped (type IA) and dumbbell-shaped (type II) SAs between 2014 and 2020. RESULTS: Of the 256 retrieved articles, 33 were included in the meta-analysis. These 33 studies, together with our 3 reported cases, included a total of 60 patients, 36 females (60%) and 24 males (40%), with a mean age of 53.12 ± 12.82 years (range: 12-77 years).T5 was the most represented level (22 patients). Usually, the localization of SA was extradural, with 53 patients suffering from spindle-shaped type IA SA (88.3%) and 7 patients from dumbbell-shaped type II SA (11.6%). Almost all patients underwent laminectomy (78.3%) and presented a full recovery of motor deficits (85%). Gross total removal (GTR) was performed in 93.3% of patients. The mean follow-up was of 22.71 ± 21.45 months (range: 2-80 months). There was no documented recurrence at follow-up magnetic resonance imaging in any of the patients. CONCLUSIONS: SAs are rare, benign tumors with a great vascular component that presents a favorable outcome. GTR is the gold standard treatment and usually an adjuvant therapy is not required. Even infiltrative lesions, which are more complex, can be treated successfully with a good prognosis. Dumbbell-shaped SAs must be differentiated from schwannomas and meningiomas, and require different surgical techniques, given the profuse bleeding associated with the attempt of debulking. En block resection is the key to treat these common benign tumors with acceptable blood loss.

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.

Intestinal Angiolipofibroma: Clinicopathologic Characteristics of 11 Cases.

Mesenchymal lesions of the gastrointestinal tract are generally uncommon compared with epithelial derived entities. Angiolipofibroma describes a rare gastrointestinal tract mesenchymal lesion composed of varied amounts of adipose tissue, fibrous tissue, along with admixed blood vessels. Descriptions of this entity are limited to few case reports describing a total of 5 lesions. Angiolipofibroma represents a benign entity that may uncommonly present as a mass lesion concerning for malignancy. The etiology is unclear; however, these may represent a reactive or hamartomatous process. In this article, we sought to further describe this entity and present an additional 11 cases.

Sacral Extradural Angiolipoma Associated with Tight Filum Terminale and Spina Bifida Coexisting with Spinal Arteriovenous Fistula.

BACKGROUND: Spinal arteriovenous fistula (AVF) may rarely associate with spinal dysraphism, that is, tethered spinal cord and spinal intradural lipoma. Spinal extradural angiolipoma coexisting with spinal AVF has not been reported in the literature. We reported an extremely rare case of sacral angiolipoma associated with tight filum terminale and sacral spina bifida coexisting with spinal AVF within this tumor. CASE DESCRIPTION: A 55-year-old women presented with progressive myelopathy for 10 months. She had a painless, slow-growing mass at her left buttock since birth. Magnetic resonance imaging of the lumbosacral spine showed an extradural mass at the level of S3-S4, extending from the spinal canal through the spina bifida to the subcutaneous fat of the left buttock. There was a low conus medullaris at S2. Magnetic resonance imaging of the thoracic spine disclosed venous congestion with tortuous intradural flow voids along both ventral and dorsal surfaces of the spinal cord. Magnetic resonance angiography and spinal angiography revealed a hypervascular mass at the sacral level and associated arteriovenous shunt with cranial drainage into an enlarged medullary vein. Due to an infected pressure sore on the mass, endovascular treatment was initially performed with minimal recovery. Six months after complete healing of her infected pressure ulcer, the patient underwent surgical removal of extradural mass containing the AVF, and subsequent release of the tight filum. Histologic findings were consistent with angiolipoma. CONCLUSIONS: Sacral extradural angiolipoma in the present case may be congenital in origin with development of an acquired spinal AVF within the tumor.

Sudden Aggravated Radicular Pain Caused by Hemorrhagic Spinal Angiolipomas After Back Massage.

BACKGROUND: Spinal angiolipomas (SALs) are benign tumors that usually present a slow progressive spinal cord or radicular compression. Acute myelopathy or acute aggravated radicular syndrome are exceedingly rare. CASE DESCRIPTION: The authors report an original case with sudden aggravated radicular pain caused by hemorrhagic SALs. A 54-year-old woman presented with a 2-month history of mild back pain, and the pain was significantly aggravated after a therapeutic back massage. Neurologic examination showed pain and hyperalgesia between T4 and T7 dermatome, from back to front, just like a band. Spinal magnetic resonance imaging (MRI) showed a dorsally located epidural lesion (T4-T6) and a small intratumoral hemorrhage at the lower part of the tumor. A bilateral T4-T6 laminectomy was performed to achieve total excision of the tumors. Histologic examination showed that the tumors were composed of mature adipose tissue and vascular tissue as angiolipomas. The postoperative course was uneventful with complete neurologic recovery 4 days after the surgery. MRI at 1-year follow-up indicated no recurrence. CONCLUSIONS: SALs are unusual benign tumors that are composed of mature fatty tissue and abnormal blood vessels; sudden aggravated spinal cord or radicular compression syndrome is rare. MRI is the best choice in the diagnosis of SALs. Surgery may be performed in different ways depending on the type of SALs, and the prognosis is generally good.

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.

Angiolipomas espinales epidurales: presentación de 7 casos y revisión de la literatura / Spinal extradural angiolipomas: 7 new cases and review of the literatura

Introducción: Presentamos nuestra experiencia en el manejo diagnóstico y terapéutico de los angiolipomas espinales. Métodos: Hemos realizado una revisión en nuestra base de datos quirúrgica correspondiente al diagnóstico patológico de angiolipoma, recogiendo las variables epidemiológicas, clínicas, diagnósticas, terapéuticas y de seguimiento de cada caso. Para la valoración funcional de los pacientes se utilizó la escala de mielopatía modificada de la Asociación Japonesa de Ortopedia (EAJO). Resultados: Obtuvimos un total de 7 casos, con edades comprendidas entre los 42 y 63 años; 4 eran mujeres. Todas las lesiones se localizaron en el espacio epidural torácico, con extensión extrarraquídea en 2 casos. La presentación más frecuente fue la combinación de dolor y mielopatía progresiva, con una puntuación entre 5 y 9 en la EAJO. Los hallazgos radiológicos fueron variables, claramente influenciados por la proporción de los componentes tumorales, identificándose un caso de variante celular, uno de predominio angiomatoso y otro mixoide. Cuatro estudios evidenciaban signos de afectación ósea. Tras el tratamiento quirúrgico se registraron puntuaciones óptimas en la EAJO, salvo en un caso (puntuación de 10 en la EAJO), sin evidenciarse recurrencias tras un periodo de seguimiento que varió entre uno y 5 años. Conclusiones: Los angiolipomas epidurales pueden dar lugar a manifestaciones clínicas y radiológicas muy variables. Su tratamiento quirúrgico debe diseñarse de acuerdo a la extensión de la tumoración, incluyendo en ocasiones un procedimiento reconstructivo espinal, y se traduce en resultados excelentes con independencia de la situación clínica previa. La posibilidad de recurrencia es baja, incluso en caso de resección incompleta

Introduction: We present our experience with the diagnosis and treatment of spinal angiolipomas (SAL). Methods: We used our database of surgical patients to gather epidemiological, clinical, diagnostic, treatment, and follow-up data from confirmed cases of SAL. Patients’ functional status was evaluated using the modified Japanese Orthopaedic Association (mJOA) score for thoracic myelopathy. Results: We included 7 patients with ages ranging between 42 and 63 years; 4 were women. Lesions in all patients were located in the thoracic epidural space; in 2 cases, lesions extended outside the spinal canal. The most frequent form of presentation was pain combined with progressive myelopathy; patients scored 5 to 9 points on the mJOA scale. Radiological findings varied; this variability clearly depended on the proportion of tumour components (cellular variant in one case, predominantly angiomatous in another, and myxoid in yet another case). Four cases displayed radiological signs of bone involvement. Surgery resulted in excellent mJOA scores except in one case (mJOA score of 10); no recurrences were detected during follow-up (periods ranging from 1 to 5 years). Conclusions: Extradural SAL can present with highly variable radiological and clinical features. Surgery must be tailored to each individual situation according to tumour extension; in some cases, it may include spinal reconstruction. Surgery results in excellent outcomes regardless of preoperative functional status and it is associated with low recurrence rates, even in case of incomplete resection

Giant angiolipoma of the back complicated with ulceration and sepsis: unusual presentation of a rare benign tumour.

A 63-year-old woman presented to the emergency unit with a huge (40 × 35 × 10 cm) oval-shaped pedunculated growth in the back with superficial ulceration in its lower aspect, associated with signs of sepsis. After adequate resuscitation, the patient was transferred to the operation theatre where the tumour was completely excised and the resultant wound was closed in primary manner using polypropylene sutures with suction drains inserted in the wound bed. The patient was discharged home after two days and was followed-up for two months. No wound complications were recorded on follow-up. Angiolipoma is a benign variant of lipoma that, despite what has been reported in the literature, may affect any age and any region of the body. Complications of angiolipoma may include ulceration, sepsis, gangrene and possible systemic inflammatory response syndrome which may pose a threat to the patient's life, hence should be managed promptly after adequate patient preparation.

Spinal angiolipomas in pregnancy: Natural history and surgical treatment.

Spinal angiolipomas (SALs) are rare extradural tumors frequently located at the level of the thoracic spine and they are associated with spinal cord compromise that might result in severe myelopathy. While the first macroscopic description dates 1890, histologically these tumors where not described as angiolipomas until 1986 by Haddad et al. Occurrence in pregnancy is even more infrequent. Since their first macroscopic description, spinal angiolipomas were reported anecdotally in pregnant women. We present a case of spinal angiolipoma in pregnancy with confirmed histologic diagnosis. In the present paper, we reviewed the literature regarding spinal angiolipomas in order to characterize their clinical manifestation, natural history, radiologic and histologic appearance. We add also a further case of spinal angiolipoma in a pregnant woman. Finally, we provide suggestions for the management of such rare tumors in pregnancy.

Spinal extradural angiolipomas: 7 new cases and review of the literature. / Angiolipomas espinales epidurales: presentación de 7 casos y revisión de la literatura.

INTRODUCTION: We present our experience with the diagnosis and treatment of spinal angiolipomas (SAL). METHODS: We used our database of surgical patients to gather epidemiological, clinical, diagnostic, treatment, and follow-up data from confirmed cases of SAL. Patients' functional status was evaluated using the modified Japanese Orthopaedic Association (mJOA) score for thoracic myelopathy. RESULTS: We included 7 patients with ages ranging between 42 and 63 years; 4 were women. Lesions in all patients were located in the thoracic epidural space; in 2 cases, lesions extended outside the spinal canal. The most frequent form of presentation was pain combined with progressive myelopathy; patients scored 5 to 9 points on the mJOA scale. Radiological findings varied; this variability clearly depended on the proportion of tumour components (cellular variant in one case, predominantly angiomatous in another, and myxoid in yet another case). Four cases displayed radiological signs of bone involvement. Surgery resulted in excellent mJOA scores except in one case (mJOA score of 10); no recurrences were detected during follow-up (periods ranging from 1 to 5 years). CONCLUSIONS: Extradural SAL can present with highly variable radiological and clinical features. Surgery must be tailored to each individual situation according to tumour extension; in some cases, it may include spinal reconstruction. Surgery results in excellent outcomes regardless of preoperative functional status and it is associated with low recurrence rates, even in case of incomplete resection.

PET/CT and MR Features of Infiltrating Spinal Angiolipoma.

Spinal angiolipomas are benign tumors predominantly found in the epidural space. Infiltrating spinal angiolipoma is an extremely rare type, which tends to invade the surrounding tissue and may mimic an aggressive tumor. Herein, we reported the case of a 64-year-old woman with a thoracic angiolipoma involving the T9 vertebral body and its appendix. The lesion showed long T1 and T2 signal intensity on MRI and slightly increased FDG uptake on PET/CT. After surgery, the pathologic examination demonstrated features consistent with angiolipoma, and the patient was free of symptoms.