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World Journal of Clinical Cases Oct 2023Aggressive angiomyolipoma is an extremely rare benign mesenchymal tumor that was originally described as a locally recurrent mucinous spindle cell tumour. Aggressive...
BACKGROUND
Aggressive angiomyolipoma is an extremely rare benign mesenchymal tumor that was originally described as a locally recurrent mucinous spindle cell tumour. Aggressive angiomyolipoma originates from myofibroblasts, vascular smooth muscle cells, or fibroblasts, and displays various phenotypes of myofibroblasts and abnormal muscle arteries. Aggressive angiomyolipoma was first identified in 1983 and fewer than 50 male patients have been reported to date. It is an extremely rare mesenchymal tumour and often confused with other diseases. Patients with epididymal aggressive angiomyolipoma lack typical symptoms, most of which occur incidentally, although some patients may experience mild pain, discomfort, and swelling. Pain may be exacerbated by pressure from the mass.
CASE SUMMARY
A 66-year-old male was admitted to the hospital on January 14, 2022 with chief complaint of swelling in the left scrotum for one year. There was no apparent cause for the swelling. The patient did not consult with any doctor or receive any treatment for the swelling. The enlarged scrotum increased in size gradually until it reached approximately the size of a goose egg, and was accompanied by discomfort and swelling of the left cavity of the scrotum. The patient had no history of any testicular trauma, infection, or urinary tract infection. The patient urinated freely, 1-2 times at night, without urgency, dysuria (painful urination), or haematuria. There was no significant family history of malignancy. The patient underwent excision of the enlarged tumour and the left epididymis under general anaesthesia on January 18, 2022. Twelve months of follow-up revealed no recurrence. The patient was satisfied with the treatment.
CONCLUSION
Aggressive angiomyolipoma is extremely rare clinically and often confused with other diseases. The pathogenesis of aggressive angiomyolipoma is unclear and the clinical presentation is mostly a painless enlarged mass. The diagnosis of aggressive angiomyolipoma requires a combination of medical history, preoperative imaging such as computed tomography and magnetic resonance imaging, cytological examination and preoperative and postoperative pathological biopsy. The preferred treatment is surgery, with the possibility of a new alternative treatment option after hormonal therapy. Aggressive angiomyolipoma should be considered in the differential diagnosis of parametrial tumors of the male genital area that present as clinically significant masses. The high recurrence rate of aggressive angiomyolipoma may be related to incomplete tumor resection, and patients with aggressive angiomyolipoma are advised to undergo annual postoperative follow-up and imaging for recurrence.
PubMed: 37946787
DOI: 10.12998/wjcc.v11.i29.7214 -
PloS One 2023Excitatory feedback from muscle spindles, and inhibitory feedback from Golgi tendon organs and recurrent inhibitory circuits are widely distributed within the spinal...
Excitatory feedback from muscle spindles, and inhibitory feedback from Golgi tendon organs and recurrent inhibitory circuits are widely distributed within the spinal cord to modulate activity between human lower limb muscles. Heteronymous feedback is most commonly studied in humans by stimulating peripheral nerves, but the unique effect of non-spindle heteronymous feedback is difficult to determine due to the lower threshold of excitatory spindle axons. A few studies suggest stimulation of the muscle belly preferentially elicits non-spindle heteronymous feedback. However, there remains a lack of consensus on the differential effect of nerve and muscle stimulation onto the H-reflex, and the relation of the heteronymous effects onto H-reflex compared to that onto ongoing EMG has not been determined. In this cross-sectional study, we compared excitatory and inhibitory effects from femoral nerve and quadriceps muscle belly stimulation onto soleus H-reflex size in 15 able-bodied participants and in a subset also compared heteronymous effects onto ongoing soleus EMG at 10% and 20% max. Femoral nerve stimulation elicited greater excitation of the H-reflex compared to quadriceps stimulation. The differential effect was also observed onto ongoing soleus EMG at 20% max but not 10%. Femoral nerve and quadriceps stimulation elicited similar inhibition of the soleus H-reflexes, and these results were better associated with soleus EMG at 20%. The results support surface quadriceps muscles stimulation as a method to preferentially study heteronymous inhibition at least in healthy adults. The primary benefit of using muscle stimulation is expected to be in persons with abnormal, prolonged heteronymous excitation. These data further suggest heteronymous feedback should be evaluated with H-reflex or onto ongoing EMG of at least 20% max to identify group differences or modulation of heteronymous feedback in response to treatment or task.
Topics: Adult; Humans; H-Reflex; Quadriceps Muscle; Femoral Nerve; Feedback; Cross-Sectional Studies; Muscle, Skeletal; Electric Stimulation; Electromyography
PubMed: 37578948
DOI: 10.1371/journal.pone.0290078 -
Veterinary Clinical Pathology Dec 2023A five-year-old male English Bulldog was presented with a firm, well-circumscribed, 1 cm in diameter cutaneous mass on the left flank. Fine-needle aspiration (FNA)...
A five-year-old male English Bulldog was presented with a firm, well-circumscribed, 1 cm in diameter cutaneous mass on the left flank. Fine-needle aspiration (FNA) biopsy samples were collected for cytologic analysis. Cytology revealed a highly cellular sample consisting of spindle cells, numerous bundles of thick, glassy eosinophilic material (hyalinized collagen), and inflammatory cells. Spindle cells showed moderate anisocytosis and anisokaryosis, had oval nuclei with coarsely stippled chromatin, 1-3 prominent round nucleoli, and moderate amounts of wispy cytoplasm. Cells were occasionally associated with an eosinophilic extracellular matrix. Binucleated and trinucleated spindle cells were often noted. Low numbers of macrophages, small lymphocytes, and individual well-granulated mast cells were also present. The lesion was excised and submitted for histopathologic examination, revealing a well-delineated, nonencapsulated mass composed of hyalinized collagen fibers separated by spindle-shaped mesenchymal cells in the deep dermis and subcutis. Mild anisocytosis and anisokaryosis and less than one mitosis per 10 × high power fields were present. Excision of the mass was complete. The findings were consistent with a keloidal fibroma, a rare benign variant of fibroma. Neoplastic cells showed positive immunoreactivity for vimentin, and a small-to-moderate number of tumor cells showed positive immunoreactivity for α-smooth muscle actin. This is the first cytologic description of a keloidal fibroma correlated with histopathologic findings and immunolabeling. In cases where keloidal neoplasia is suspected, and since moderate cellular atypia can be present on cytologic examination even in cases of keloidal fibroma, histopathologic examination is necessary to differentiate between keloidal fibroma and keloidal fibrosarcoma.
Topics: Male; Dogs; Animals; Fibroma; Keloid; Subcutaneous Tissue; Biopsy, Fine-Needle; Collagen; Diagnosis, Differential; Dog Diseases
PubMed: 37661191
DOI: 10.1111/vcp.13282 -
Head and Neck Pathology Sep 2023Low-grade myofibroblastic sarcoma (LGMS) represents an atypical tumor composed of myofibroblasts with a variety of histological patterns and with a high tendency to... (Review)
Review
Low-grade myofibroblastic sarcoma (LGMS) represents an atypical tumor composed of myofibroblasts with a variety of histological patterns and with a high tendency to local recurrence and a low probability of distant metastases. LGMS has predilection for the head and neck regions, especially the oral cavity. This study aimed to report 13 new cases of LGMS arising in the oral and maxillofacial region. This study included LGMS cases from five oral and maxillofacial pathology laboratories in four different countries (Brazil, Peru, Guatemala, and South Africa). Their clinical, radiographic, histopathological, and immunohistochemical findings were evaluated. In this current international case series, most patients were females with a mean age of 38.7 years, and commonly presenting a nodular lesion in maxilla. Microscopically, all cases showed a neoplasm formed by oval to spindle cells in a fibrous stroma with myxoid and dense areas, some atypical mitoses, and prominent nucleoli. The immunohistochemical panel showed positivity for smooth muscle actin (12 of 13 cases), HHF35 (2 of 4 cases), β-catenin (3 of 5 cases), desmin (3 of 11 cases), and Ki-67 (range from 5 to 50%). H-caldesmon was negative for all cases. The diagnosis of LGMS was confirmed in all cases. LGMS shows predominance in young adults, with a slight predilection for the female sex, and maxillary region. LGMS should be a differential diagnosis of myofibroblastic lesions that show a proliferation of spindle cells in a fibrous stroma with myxoid and dense areas and some atypical mitoses, supporting the diagnosis with a complementary immunohistochemical study. Complete surgical excision with clear margins is the treatment of choice. However, long-term follow-up information is required before definitive conclusions can be drawn regarding the incidence of recurrence and the possibility of metastasis.
Topics: Humans; Female; Adult; Male; Fibrosarcoma; Myofibroblasts; Head; Diagnosis, Differential; Brazil
PubMed: 37540486
DOI: 10.1007/s12105-023-01577-3 -
Zhonghua Bing Li Xue Za Zhi = Chinese... Jul 2023To investigate the clinicopathological features, diagnosis and differential diagnosis of breast myofibroblastoma. The clinicopathological data and prognostic...
To investigate the clinicopathological features, diagnosis and differential diagnosis of breast myofibroblastoma. The clinicopathological data and prognostic information of 15 patients with breast myofibroblastoma diagnosed at the Department of Pathology of the First Affiliated Hospital of Zhengzhou University, Zhengzhou, China from 2014 to 2022 were collected. Their clinical characteristics, histological subtypes, immunophenotypes and molecular characteristics were analyzed. There were 12 female and 3 male patients, ranging in age from 18 to 78 years, with a median and average age of 52 years. There were 6 cases in the left breast and 9 cases in the right breast, including 12 cases in outer upper quadrant, 2 cases in inner upper quadrant and 1 case in outer lower quadrant. Most of the cases showed a well-defined nodule grossly, including pushing growth under the microscope in 13 cases, being completely separated from the surrounding breast tissue in 1 case, and infiltrating growth in 1 case. Among them, 12 cases were classic subtype and composed of occasional spindle cells with varying intervals of collagen fiber bundles; eight cases had a small amount of fat; one case had focal cartilage differentiation; one case was epithelioid subtype, in which epithelioid tumor cells were scattered in single filing or small clusters; one case was schwannoma-like subtype, and the tumor cells were arranged in a significant palisade shape, resembling schwannoma, and one case was invasive leiomyoma-like subtype, in which the tumor cells had eosinophilic cytoplasm and were arranged in bundles, and infiltrating into the surrounding mammary lobules like leiomyoma. Immunohistochemical studies showed that the tumor cells expressed desmin (14/15) and CD34 (14/15), as well as ER (15/15) and PR (15/15). Three cases with histologic subtypes of epithelioid subtype, schwannoma-like subtype and infiltrating leiomyoma-like subtype showed RB1 negative immunohistochemistry. Then FISH was performed to detect RB1/13q14 gene deletion, and identified RB1 gene deletion in all three cases. Fifteen cases were followed up for 2-100 months, and no recurrence was noted. Myofibroblastoma is a rare benign mesenchymal tumor of the breast. In addition to the classic type, there are many histological variants, among which the epithelioid subtype is easily confused with invasive lobular carcinoma. The schwannoma-like subtype is similar to schwannoma, while the invasive subtype is easily misdiagnosed as fibromatosis-like or spindle cell metaplastic carcinoma. Therefore, it is important to recognize the various histological subtypes and clinicopathological features of the tumor for making correct pathological diagnosis and rational clinical treatment.
Topics: Female; Humans; Male; Antigens, CD34; Biomarkers, Tumor; Leiomyoma; Neoplasms, Muscle Tissue; Neurilemmoma; Adolescent; Young Adult; Adult; Middle Aged; Aged
PubMed: 37408398
DOI: 10.3760/cma.j.cn112151-20221228-01075 -
Stomatologiia 2024The article describes a clinical case of a benign tumor from smooth muscle cells - piloleiomyoma. The incidence of leiomyoma in the skin is 3-5% of all leiomyomas. A...
The article describes a clinical case of a benign tumor from smooth muscle cells - piloleiomyoma. The incidence of leiomyoma in the skin is 3-5% of all leiomyomas. A 27-year-old patient applied to a medical institution with complaints about an intradermal formation in the ear region that occurred repeatedly within 5 months after surgical treatment. After the first surgical intervention, the patient was consulted in various medical organizations, where the following diagnoses were made: «nodular fasciitis», «smooth muscle tumor without signs of malignancy» and «non-epithelial spindle cell neoplasm». According to ultrasound examination, the formation with dimensions of 11×9×5 mm reached the mastoid process of the temporal bone and was characterized by increased internal blood flow. After surgical removal of the neoplasm, taking into account the difficulties of differential diagnosis, an immunohistochemical study was conducted. An accumulation of smooth muscle cells was detected in the surface layers of the dermis under the epidermis by the immunohistochemical study with the use of the marker SMA. A study on CD34 protein revealed a high density of blood capillaries and the absence of its expression in smooth muscle cells. The proliferative index (Ki-67) and mitotic activity (PHH-3) of cells was also studied. The index of proliferative activity was less than 2%, mitoses were isolated. Thus, the results of immunohistochemical study proved the conclusion of piloleiomyoma.
Topics: Humans; Adult; Leiomyoma; Skin Neoplasms; Fasciitis
PubMed: 38372608
DOI: 10.17116/stomat202410301155 -
Journal of Bodywork and Movement... Jul 2024This study aimed to determine whether massage pressure on the target muscles (biceps brachii muscle [BB] and the medial head of the gastrocnemius muscle [MG]) is related...
INTRODUCTION
This study aimed to determine whether massage pressure on the target muscles (biceps brachii muscle [BB] and the medial head of the gastrocnemius muscle [MG]) is related to the massage effect (reducing muscle stiffness).
METHOD
Nine healthy participants participated in this study. A physiotherapist massaged the upper arms and lower legs of participants on a rigid desk in a laboratory. Massage was delivered for 10 min with a 3-min rest. The shear modulus (i.e., the muscle stiffness), assessed by shear wave elastography, was measured at various time points (before [PRE], immediately after [POST], and 5 [POST-5], 10, 15, and 20 min after the massage). The massage pressure data (N) were obtained only during massage by force plate sensors.
RESULTS
The BB shear modulus was significantly reduced POST massage. The MG shear modulus significantly reduced POST massage and remained clearly reduced until POST-5. There was a negative correlation between the total massage pressure and the % change in the shear modulus in both muscles.
DISCUSSION
Since the spindle (BB) and pennate (MG) muscles have structural differences, our results suggest that these differences may affect the pattern of changes in the shear modulus in response to massage.
CONCLUSION
Massage pressure is related to the massage effect (reducing muscle stiffness), and its relationships to POST are not related to the differences in the morphologies of the spindle (BB) and pennate muscles (MG). However, differences in the morphologies of the spindle and pennate muscles may cause differences in the duration of the massage effects.
Topics: Humans; Massage; Muscle, Skeletal; Male; Adult; Female; Young Adult; Pressure; Elasticity Imaging Techniques
PubMed: 38876651
DOI: 10.1016/j.jbmt.2024.03.025 -
Journal of Oral Pathology & Medicine :... May 2024Myofibromas are rare benign neoplasms composed of myoid cells and myofibroblasts. This study aimed to systematically review case reports and a series of myofibromas (MF)... (Review)
Review
BACKGROUND
Myofibromas are rare benign neoplasms composed of myoid cells and myofibroblasts. This study aimed to systematically review case reports and a series of myofibromas (MF) and myofibromatosis (MFT) occurring in the oral and maxillofacial regions in order to describe their main clinicopathological features.
METHODS
This systematic review was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Electronic searches were conducted in 2023 in four databases: MEDLINE/PubMed, Web of Science, Scopus, and EMBASE. A manual search and a search in the grey literature were also conducted. The lesions were classified as MF or MFT according to their original report.
RESULTS
A total of 169 cases were included in this systematic review. Men were slightly more affected, with a painless nodule. When occurring in soft tissue, MF usually developed in the gingiva (mean age:29.23 ± 21.93 years) and when it was intra-osseous, it occurred more frequently in the posterior mandible (mean age:14.33 ± 15.62 years). MFT occurred mainly in the mandible and was predominantly described as well-circumscribed masses of spindle cells organized in fascicles with a prominent vascular activity in a hemangiopericytoma-like pattern. The lesions were mainly positive for smooth muscle actin and vimentin immunomarkers. Surgical excision was the treatment of choice in the majority of cases and recurrence was observed in only three cases.
CONCLUSION
MF and MFT affect more men, with an indolent clinical course. Intra-osseous tumors and MFT seem to occur more frequently in younger individuals. These lesions seem to have a good prognosis and low recurrence.
PubMed: 38711183
DOI: 10.1111/jop.13537 -
Modern Pathology : An Official Journal... Feb 2024RAD51B-rearranged sarcomas are rare neoplasms that exhibit a heterogeneous morphology. To date, 6 cases have been reported, all involving the uterus, including 4...
RAD51B-rearranged sarcomas are rare neoplasms that exhibit a heterogeneous morphology. To date, 6 cases have been reported, all involving the uterus, including 4 perivascular epithelioid cell tumors (PEComas) and 2 leiomyosarcomas (LMS). In this study, we describe the morphologic, immunohistochemical, and molecular features of 8 additional sarcomas with RAD51B rearrangement, including the first extrauterine example. All patients were women with a median age of 57 years at presentation. Seven tumors originated in the uterus, and one in the lower extremity soft tissue, with a median tumor size of 12 cm. Histologically, 4 tumors showed predominantly spindle cell morphology with eosinophilic fibrillary cytoplasm, with or without nuclear pleomorphism, whereas 2 tumors exhibited pleomorphic epithelioid cells, featuring clear to eosinophilic, granular cytoplasm. Two neoplasms exhibited undifferentiated cytomorphology, including one with uniform small blue round cells. All tumors showed high-grade cytologic atypia and high mitotic activity (median: 30/10 high-power fields), whereas coagulative necrosis was noted in 6 cases and lymphovascular invasion in 2. By immunohistochemistry, 2 showed myoid and melanocytic markers in keeping with PEComa, whereas 4 cases were only positive for smooth muscle markers consistent with LMS (including 3 myxoid). The remaining 2 cases had a nonspecific immunoprofile. Five cases tested by targeted RNA sequencing (Archer FusionPlex, Illumina TruSight) showed different fusion partners (HMGA2, PDDC1, and CEP170). RAD51B rearrangements were identified by FISH in the remaining 3 cases. Targeted DNA sequencing in 2 cases was negative for TSC gene alterations. Clinical outcome, available in 5 patients (median follow-up, 19 months), revealed 3 local recurrences, 2 lung metastases, and 4 deaths due to disease. Our results expand the spectrum of sarcomas with RAD51B fusions, demonstrating variable clinical presentations, morphologic spectrum, and fusion partners. These tumors have a predilection for a uterine location, with either LMS, PEComa, or undifferentiated phenotypes, and are associated with an aggressive clinical course.
Topics: Humans; Female; Middle Aged; Male; Biomarkers, Tumor; Sarcoma; Leiomyosarcoma; Perivascular Epithelioid Cell Neoplasms; Soft Tissue Neoplasms; Phenotype; DNA-Binding Proteins
PubMed: 38141829
DOI: 10.1016/j.modpat.2023.100402