INTRODUCTION

The 2020 World Health Organization Classification of Tumors of Soft Tissue and Bone[1] (Fourth Edition) defined glomus tumor as a mesenchymal tumor in normal glomus, with cells resembling modified smooth muscle cells. Glomus tumor may be classified as benign, intermediate glomangiomatosis (also known as diffuse glomus tumor), or malignant (ICD-O: 8711/0, ICD-O: 8711/1, and ICDO: 8711/3, respectively). Glomus tumors account for fewer than 2% of soft tissue tumors, and glomus hemangioma disease makes up 5% of glomus tumors.

Glomangiomatosis is very rare but occurs most often in young people, in the distal extremities. It is multiple, deep, and wide ranging and often causes pain. The lesions can recur after resection. The tumor shows diffuse growth, with an overall structure that is similar to angiomatosis, except that the vessel wall is surrounded by nests of glomus cells[2]. Although it may be diffuse or infiltrative and aggressive, it lacks the standard morphology of malignancy.

CASE PRESENTATION

Chief complaints

A 55-year-old female presented with idiopathic pain in the dorsum of the right foot and lateral leg, for more than 2 mo.

The treatment of glomangiomatosis is surgical resection. Although glomangiomatosis grows diffusely, conservative treatment is still advocated3. Local recurrence is related to the difficulty of resection. Zhou[3] retrospectively analyzed 11 cases of glomus tumors at home and abroad: 3 cases were not completely resected, 5 had positive margins at the first operation, 5 recurred after surgery, and 1 relapsed six times during 14 years. The patient described in the present article was followed for 1 year after resection, and no recurrence was found. Since the tumors in some cases were palliatively removed, glomangiomatosis should be regarded as a persistent disease rather than local recurrence. Therefore, complete resection of the mass is the most important prognostic factor. Due to the small number of known cases, the effect of adjuvant therapy is uncertain.

History of present illness

取轉(zhuǎn)接3次后的上述培養(yǎng)液1 mL適當(dāng)稀釋后分別涂布于MRS初篩平板和ATB初篩平板上，25℃培養(yǎng)48 h后，以菌落周圍有透明圈的菌落為目的菌進(jìn)行挑菌，革蘭氏染色，斜面保存陽性菌并進(jìn)行后續(xù)驗(yàn)證及保藏。

History of past illness

The patient had no significant medical history.

（2）益生素又稱微生態(tài)制劑或活菌制劑，是一類活性微生物添加劑，采用動(dòng)物腸道有益微生物經(jīng)發(fā)酵、純化、干燥精制的復(fù)合生物制劑。膽汁酸在動(dòng)物體內(nèi)最重要的功能是消化食物中的脂肪和脂溶性物質(zhì)，維持正常生長[2]。二者結(jié)合使用，可產(chǎn)生協(xié)同作用，具有促進(jìn)動(dòng)物生長、保肝利膽、鎮(zhèn)咳、平喘、消炎、抗過敏、抑菌等作用，尤其是對(duì)于仔豬，由于消化系統(tǒng)發(fā)育尚不完善，膽汁酸的合成不足，影響對(duì)飼料中脂質(zhì)的吸收利用，導(dǎo)致仔豬死亡，造成經(jīng)濟(jì)損失，而通過食療添加益生素和膽汁酸，可以促進(jìn)腸胃消化與吸收，降低各類腹瀉發(fā)生概率。

Personal and family history

The personal and family history did not reveal any problems.

Physical examination

Physical examination revealed pigmentation of about 6 cm × 6 cm on the right foot dorsum, with a clear boundary and pain upon pressure; local pressing pain on the lateral side of the right leg; limited ankle joint activity; good peripheral blood supply; and normal sensation. During surgery, many nodular masses around the ankle joint were observed, and the incision surface of the tumor was brown.

Laboratory examinations

The prothrombin and partial thromboplastin times were slightly low, and D-dimers were normal. The blood biochemistries and urinalysis were normal. The blood analysis, electrocardiogram, chest x-ray, and arterial blood gas were also normal.

Imaging examinations

Magnetic resonance imaging showed multiple nodules and soft tissue swelling around the right ankle, which was consistent with pigmented villonodular synovitis.

海島電力通信光纜運(yùn)維技術(shù)，應(yīng)以增強(qiáng)抗災(zāi)防災(zāi)能力為主要目的，立足于電網(wǎng)發(fā)展建設(shè)，既吸收陸地光纜巡視檢修的有益經(jīng)驗(yàn)，又要熟悉沿線海洋水文氣象條件，掌握海纜登陸點(diǎn)和周邊海域地質(zhì)地貌特點(diǎn)、路由區(qū)經(jīng)濟(jì)開發(fā)保護(hù)進(jìn)展以及運(yùn)維隊(duì)伍人員裝備狀況，多措并舉，實(shí)現(xiàn)電力通信資源的優(yōu)化配置和高效利用。

Pathological examination

Due to its rarity, only 15 clinicopathological analyses have been reported in the Chinese and English literature (Tables 1 and 2)[2-16]. In total, there have been 16 patients, of which 5 and 11 were women and men, respectively. The average and median ages were 32 years and 31.5 years. Eleven and 3 cases (68.75% and 18.75%) involved the lower and upper limbs. Other sites (25%) included the head and neck, chest wall, and paravertebral region. Unlike classic glomus tumor, none of these tumors were found under the nail but mostly in the hand, wrist, and foot, and the location was deep. The sizes of the lesions ranged from 1.5 to 22 cm. Therefore, glomangiomatosis is larger than glomus tumor, with extensive lesions and deep location, and all cases were infiltrative. In the present case, lesions were on the back of the right foot and the outer side of the calf and ranged from 0.8 to 2.5 cm in diameter. The deep location and clinical and imaging considerations led to a diagnosis of pigmented villous nodular synovitis.

Immunohistochemistry revealed diffuse immunoreactivity for smooth muscle actin (SMA, Figure 3), vimentin, type IV collagen, and Bcl-2. Caldesmon was weakly positive, CD34 was partially positive, and calponin was slightly positive. The Ki-67 value-added index was about 3%. Desmin, S-100, and HMB-45 were negative.

FINAL DIAGNOSIS

The pathological diagnosis was glomangiomatosis of the right ankle joint.

4Rs是指關(guān)聯(lián)（Relevancy）、反應(yīng)（Respond）、關(guān)系（Relation）、回報(bào)（Return）策略。4Rs既從企業(yè)的角度出發(fā)，又兼顧了消費(fèi)者的需求，還兼顧了競爭導(dǎo)向，是一個(gè)更符合新經(jīng)濟(jì)背景下、有利于企業(yè)和消費(fèi)者的營銷組合策略。

TREATMENT

Extended resection of the tumor.

PDCA循環(huán)理論認(rèn)為管理中的任何工作都可以分為四個(gè)階段：計(jì)劃階段（P），實(shí)施階段（D），檢查階段（C）和總結(jié)處理階段（A），這四個(gè)階段緊密銜接，缺一不可，構(gòu)成一個(gè)完整的系統(tǒng)。PDCA循環(huán)模式之所以能夠應(yīng)用于創(chuàng)建高校的優(yōu)良學(xué)風(fēng)，關(guān)鍵在于它的長效性、持續(xù)性、循環(huán)性和可改進(jìn)性等優(yōu)點(diǎn)。

OUTCOME AND FOLLOW-UP

After the extensive resection, no recurrence was found at the 2-year follow-up, and the prognosis was good.

DISCUSSION

The 2020 World Health Organization Classification of Tumors of Soft Tissue and Bone (Fourth Edition) defined glomus tumor as a mesenchymal tumor in normal glomus, with cells similar to modified smooth muscle cells. Glomus tumor may be classified as benign, intermediate glomangiomatosis (also known as diffuse glomus tumor), or malignant (ICD-O: 8711/0, ICD-O: 8711/1, and ICD-O: 8711/3, respectively). Glomus tumors account for fewer than 2% of soft tissue tumors, and glomus hemangioma disease makes up 5% of glomus tumors[1].

Glomangiomatosis is very rare but occurs most often in young people, in the distal extremities. It is multiple, deep, and wide ranging and often produces pain. The lesions can recur after resection. The tumor shows diffuse growth, with an overall structure that is similar to angiomatosis, except that the vessel wall is surrounded by nests of glomus cells[2]. Although it may be diffuse or infiltrative and aggressive, it lacks the standard morphology of malignancy.

Pathological examination revealed grayish-yellow irregular tissues, with a total volume of 7 cm × 6 cm × 1.5 cm, with multiple nodules within. The largest was 2.5 cm × 1.5 cm × 1.2 cm, and the smallest was 0.8 cm × 0.7 cm × 0.5 cm. The section was grayish-white and grayish-yellow and slightly tough (Figure 1). Under the microscope, the tumor cells in the fibrous connective tissue were nodular and lobulated, and the tumor cells grew around blood vessels. The blood vessels consisted of small arteries and veins, capillaries, and dilated veins showing diffuse hyperplasia (Figure 2A). With eosinophilic staining, the tumor cells appeared round or oval, uniform in size, with abundant cytoplasm and clear boundary. Necrosis and pleomorphism were absent. In the surrounding adipose tissue, a proliferation of oval cells adjacent to small vessels was observed, which was consistent with tumor cell morphology (Figure 2B). Mast cells were scattered in the stroma (Figure 2C).

Unlike classic glomus tumor, there have been no reports of symptoms related to glomangiomatosis in childhood, although the possibility cannot be ruled out. If perhaps it is present in childhood, symptoms do not develop until early adulthood. Pain is an obvious symptom of the disease[3,4].

Academic Promotion Program of Shandong First Medical University, No. 2019QL017.

Histologically, the overall structure of glomangiomatosis resembles diffuse angiomatosis, except that in the former, glomus cells surround blood vessels. Among the 16 reported cases, all showed a diffuse and infiltrative growth pattern. Skeletal muscle infiltration was observed in 5 cases, and extensive nerve and peripheral invasion was seen in 1 case. However, some authors suggest that the glomus cells and angiomatosis components in glomus hemangioma, often accompanied by adipose tissue, are a sign of mesenchymal proliferation and not simple glomus cell proliferation[3,10]. Further study is necessary to determine whether adipose is one of its components or whether it infiltrates into the adipose tissue.

Fan[9] reported a case of glomus angiomatosis in a 33-year-old man who showed multiple glomus angiomatosis on the medial right foot and right lower leg. The patient developed repeated pain in the right ankle during 2 years, worsening in the week before presentation. Immunohistochemistry was positive for SMA and negative for CD34. Fitzhugh[10] reported a 33-year-old man with a mass on the distal right calf. Immunohistochemistry showed that the tumor cells were diffusely and strongly positive for SMA, and the vascular components were strongly positive for CD31 and CD34. In the current case, the tumor cells are diffusely and strongly positive for SMA, and negative for CD31 and CD34, which is consistent with the immunohistochemical expression of the above cases.

Masson proposed in 1924 that glomus angiomatosis may be caused by the overgrowth of normal globular cells[17]. Glomus tumor is a kind of interstitial tumor composed of variant smooth muscle cells similar to normal blood vessels. Glomus cells are located around the wall of the small bulb arteriovenous anastomosis. They are a variant of smooth muscle cells. Pericytes include vascular glomus cells. Pericytes were first discovered by Rouget in 1873 and further described by Zimmerman in 1923[3]. Most glomus hemangioma clinically manifests as small benign tumors, most often found in the dermis of the hand, especially in the subungual area, and distributed throughout the body. Most globular tumors have occurred at all ages.

Glomangiomatosis is a rare type of glomus tumor. Its overall structure resembles diffuse hemangioma, but the vessel wall surrounds the nest bulb cells. Combined with histological morphology and immunohistochemical staining of SMA and collagen IV, a diagnosis of glomus hemangioma is not difficult. The disease may be differentiated from other suspects by the characteristic features of each. For example, epithelioid angiomyolipoma is composed of blood vessels, smooth muscle, and fat. The tumor cells immunohistochemically show the presence of HMB45 and Melan A[10]. In paraganglioma, the tumor cells are arranged in an organ-like structure, and the stroma is rich in capillaries. Synaptophysin and chromogranin A can be observed in the main cells, while Sertoli cells show S-100 protein but not SMA[1]. The cells in malignant glomus tumor are heteromorphic, and the mitosis is pathological. Angiomatosis is composed of blood vessels of different sizes, but globular angiomatosis is surrounded by spheroid cells around the blood vessel wall, while the former is not surrounded by spheroid cells.

Studies of the molecular genetics of glomangiomatosis mainly focus on multiple familial cases. These studies show an autosomal dominant hereditary disease, caused by an inactivation mutation of the gene that encodes glomulin in the short arm of chromosome 1. It is possible that there is a family history of glomus angiomatosis, because genetic studies have revealed the truncation of the globulin gene on chromosome 1p21-22, and four germline mutations have been found[17]. However, there are also cases reported in which there was no familial genetic predisposition.

一般存在于項(xiàng)目前期階段不完整，施工中形成的更多的矛盾，使施工方疲于應(yīng)付各種局部的矛盾，削弱了能源管理的質(zhì)量。還有就是負(fù)責(zé)人考慮到自己利益或者別的因素而對(duì)設(shè)計(jì)和監(jiān)管工作進(jìn)行過多的干擾，也會(huì)對(duì)工程的進(jìn)展以及質(zhì)量產(chǎn)生一些影響。

There was no obvious cause for pain on the back of the right foot and the outside of the lower leg for more than 2 mo. When the pain was severe, it affected movement of the affected limb.

CONCLUSION

Glomangiomatosis is very rare but occurs most often in young people, in the distal extremities. Its overall structure resembles diffuse hemangioma, but the vessel wall surrounds the nest bulb cells. Multiple nodules in glomangiomatosis are even rarer. The treatment of glomangiomatosis is surgical resection.

FOOTNOTES

Zhang SP was the patient’s doctor in charge, was responsible for collecting the medical history, and contributed to the revision of the manuscript; Wu RC was responsible for collecting the medical history, reviewing the literature, drafting the paper, and contributing to the revising of the manuscript; Gao YH, Zhang XY, and Sun WW revised the manuscript; All authors issued final approval for the submitted version.

賀州市要充分利用粵桂縣域經(jīng)濟(jì)產(chǎn)業(yè)合作示范區(qū)（信都工業(yè)區(qū)）、旺高工業(yè)園區(qū)（千億碳酸鈣產(chǎn)業(yè)園）等產(chǎn)業(yè)平臺(tái)。探索一區(qū)多園的運(yùn)作模式，鼓勵(lì)與外地資本合作發(fā)展“飛地”園區(qū)，引導(dǎo)建立產(chǎn)業(yè)集群；推進(jìn)體制創(chuàng)新、管理創(chuàng)新，建立健全快速?zèng)Q策、高效服務(wù)體系；給予更加優(yōu)惠的政策，在園區(qū)做到地價(jià)低于周邊市、地方稅收在企業(yè)建成后的前幾年全返、市級(jí)及以下收費(fèi)全免（零收費(fèi)）；同時(shí)加強(qiáng)生態(tài)建設(shè)，營造更富吸引力的投資載體。

Written informed consent was obtained from the patient for publication of this report.

模糊語言作為自然語言的一個(gè)必不可少的部分，是普遍存在的語言現(xiàn)象。在書面語和口語中，我們都能發(fā)現(xiàn)模糊語言的存在。模糊限制語（Hedges）是模糊語言的一個(gè)重要分支，眾多語言學(xué)家從各種角度對(duì)其進(jìn)行了詳盡的研究。美國著名語言學(xué)家拉可夫（George Lakoff）（1972：195）對(duì)模糊限制語給出了定義，它是指“一些有意把事物弄得更加模糊或更不模糊的詞語（Wordswhosejob it ismakethingsfuzzier or lessfuzzier）”。

The authors declare that there is no conflict of interest

The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).

把多軸系統(tǒng)簡化成單軸系，把阻轉(zhuǎn)轉(zhuǎn)矩Mf折算至變頻電動(dòng)機(jī)軸上為Mr，此時(shí)把Mr看作等效的阻轉(zhuǎn)轉(zhuǎn)矩。將計(jì)算軸作為計(jì)算基準(zhǔn)點(diǎn)，以確保系統(tǒng)的功率傳輸關(guān)系和機(jī)械總動(dòng)能一定為折算原則，將系統(tǒng)轉(zhuǎn)動(dòng)慣量化為兩部分：變頻電機(jī)端轉(zhuǎn)動(dòng)慣量的和標(biāo)記為JⅠ；靠低速軸側(cè)的轉(zhuǎn)動(dòng)慣量之和記作JⅡ[4]，如圖2(b)所示。

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China

3.4.2 直流電源自身具有完善的蓄電池管理功能，可對(duì)蓄電池進(jìn)行充放電管理，能有效延長蓄電池使用壽命，減少站點(diǎn)蓄電池投資和維護(hù)費(fèi)用。

Ruo-Chen Wu 0000-0003-4751-9814; Ying-Hua Gao 0000-0002-4896-0014; Wen-Wen Sun 0000-0002-5424-7223; Xiang-Yun Zhang 0000-0002-4861-5376; Shu-Peng Zhang 0000-0003-0237-5159.

Ma YJ

Filipodia

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