scholarly journals Giant Parotid Pleomorphic Adenoma with Atypical Histological Presentation and Long-Term Recurrence-Free Follow-Up after Surgery: A Case Report and Review of the Literature

2020 ◽  
Vol 2020 ◽  
pp. 1-18
Author(s):  
Mohammed AlKindi ◽  
Sundar Ramalingam ◽  
Lujain Abdulmajeed Hakeem ◽  
Manal A. AlSheddi

Salivary gland tumors (SGT) comprise 3% of all head and neck tumors, are mostly benign, and arise frequently in the parotid gland. Pleomorphic adenoma (PA) is the commonest SGT, representing 60-70% of all benign parotid tumors. Clinically, parotid PA presents as irregular, lobulated, asymptomatic, slow-growing preauricular mass, involving both superficial and deep lobes, and could grow to gigantic proportions. Histologically, PA has epithelial and mesenchymal elements in chondromyxoid matrix and is managed surgically. Based on a review of 43 cases reported in English literature since 1995, giant parotid PA is reported as large as 35 cm (diameter) and 7.3 kg (resected weight). Although rare, 10 cases of malignant transformation were reported in the review. Surgical management included extracapsular dissection (ECD), superficial parotidectomy, and total parotidectomy for benign tumors, and adjuvant radiation or chemotherapy for malignant tumors. We further present the case of a 36-year-old healthy male with slow-growing and asymptomatic giant parotid PA, of 4-year duration. The patient presented with firm, lobulated preauricular swelling, provisionally diagnosed as PA based on radiographic and cytological findings. The tumor was resected through ECD, and the patient had uneventful postoperative recovery and a 7-year recurrence-free follow-up period. Histological examination revealed epimyoepithelial proliferation punctuated by chondromyxoid areas, with extensive squamous metaplasia and keratin cysts. To the best of knowledge from indexed literature, giant parotid PA is rarely reported in Saudi Arabia. In addition to its rarity, this case is reported for its benign nature despite atypical histological presentation, successful surgical management without complications, and long-term recurrence-free follow-up. Based on this report, clinicians must be aware of atypical histological presentations associated with PA and plan suitable surgical management and follow-up to avoid morbidity. Nevertheless, attempts must be made to diagnose and manage these lesions at an early stage and before they reach gigantic proportions.

BMC Urology ◽  
2020 ◽  
Vol 20 (1) ◽  
Author(s):  
Athanasios Zachariou ◽  
Maria Filiponi ◽  
Fotios Dimitriadis ◽  
Aris Kaltsas ◽  
Nikolaos Sofikitis

Abstract Background Bladder leiomyomas are rare and benign tumors of the bladder. They account for 0.43% of all bladder tumors, and only 250 cases have been reported in English literature. Based on the size and localization of the lesion, their symptoms vary considerably. Women seem to be more affected, and obstructive symptoms predominate. Surgical treatment is almost always highly effective, leaving a low recurrence rate. Case presentation We present a clinical case of a 52-year old man with macroscopic hematuria and obstructive lower urinary tract symptoms due to a large bladder trigone leiomyoma. CT and MRI showed a well-defined large bladder leiomyoma and cystoscopy established the initial findings. The patient underwent successful transurethral resection of the lesion, and pathology findings confirmed the diagnosis. Conclusions This case report demonstrates that transurethral resection of a large bladder trigone leiomyoma is a feasible and successful procedure. Long term follow-up proves that there is neither scarring distortion of the bladder trigone area nor damage in the ureteral orifices, even though there was a thorough removal of the trigone wall.


Open Heart ◽  
2021 ◽  
Vol 8 (1) ◽  
pp. e001440
Author(s):  
Shameer Khubber ◽  
Rajdeep Chana ◽  
Chandramohan Meenakshisundaram ◽  
Kamal Dhaliwal ◽  
Mohomed Gad ◽  
...  

BackgroundCoronary artery aneurysms (CAAs) are increasingly diagnosed on coronary angiography; however, controversies persist regarding their optimal management. In the present study, we analysed the long-term outcomes of patients with CAAs following three different management strategies.MethodsWe performed a retrospective review of patient records with documented CAA diagnosis between 2000 and 2005. Patients were divided into three groups: medical management versus percutaneous coronary intervention (PCI) versus coronary artery bypass grafting (CABG). We analysed the rate of major cardiovascular and cerebrovascular events (MACCEs) over a period of 10 years.ResultsWe identified 458 patients with CAAs (mean age 78±10.5 years, 74.5% men) who received medical therapy (N=230) or underwent PCI (N=52) or CABG (N=176). The incidence of CAAs was 0.7% of the total catheterisation reports. The left anterior descending was the most common coronary artery involved (38%). The median follow-up time was 62 months. The total number of MACCE during follow-up was 155 (33.8%); 91 (39.6%) in the medical management group vs 46 (26.1%) in the CABG group vs 18 (34.6%) in the PCI group (p=0.02). Kaplan-Meier survival analysis showed that CABG was associated with better MACCE-free survival (p log-rank=0.03) than medical management. These results were confirmed on univariate Cox regression, but not multivariate regression (OR 0.773 (0.526 to 1.136); p=0.19). Both Kaplan-Meier survival and regression analyses showed that dual antiplatelet therapy (DAPT) and anticoagulation were not associated with significant improvement in MACCE rates.ConclusionOur analysis showed similar long-term MACCE risks in patients with CAA undergoing medical, percutaneous and surgical management. Further, DAPT and anticoagulation were not associated with significant benefits in terms of MACCE rates. These results should be interpreted with caution considering the small size and potential for selection bias and should be confirmed in large, randomised trials.


Author(s):  
Marco Pavanello ◽  
Pietro Fiaschi ◽  
Andrea Accogli ◽  
Mariasavina Severino ◽  
Domenico Tortora ◽  
...  

AbstractMorning glory disc anomaly is a congenital abnormality of the optic disc and peripapillary retina reported as an isolated condition or associated with various anomalies, including basal encephaloceles and moyamoya vasculopathy. However, the co-occurrence of these three entities is extremely rare and the pathogenesis is still poorly understood. Moreover, data on the surgical management and long-term follow-up of the intracranial anomalies are scarce. Here, we describe the case of a 11-year-old boy with morning glory disc anomaly, transsphenoidal cephalocele, and moyamoya vasculopathy, who underwent bilateral indirect revascularization with encephalo-duro-myo-arterio-pericranio-synangiosis at the age of 2 years, and endoscopic repair of the transsphenoidal cephalocele at the age of 6 years. A rare missense variant (c.1081T>C,p.Tyr361His) was found in OFD1, a gene responsible for a X-linked ciliopathy, the oral-facial-digital syndrome type 1 (OFD1; OMIM 311200). This case expands the complex phenotype of OFD1 syndrome and suggests a possible involvement of OFD1 gene and Shh pathway in the pathogenesis of these anomalies.


2004 ◽  
Vol 130 (6) ◽  
pp. 327-333 ◽  
Author(s):  
Hans Geinitz ◽  
Frank B. Zimmermann ◽  
Reinhard Thamm ◽  
Monika Keller ◽  
Raymonde Busch ◽  
...  

2021 ◽  
Vol 15 (1) ◽  
Author(s):  
Ozer Birge ◽  
Mehmet Sait Bakır ◽  
Ceyda Karadag ◽  
Zivar Eldarova ◽  
Tayup Simsek

Abstract Background Hidradenoma papilliferum is a rare benign neoplasm arising from apocrine glands. It occurs commonly on the anogenital region of middle-aged women. It usually presents as a slow growing, solitary asymptomatic, skin colored or red nodule less than 1 cm in diameter. Case presentation The case is a 38-year-old, white woman who presented with a painful nodule occurring within a month in the himenal region of the posterior vaginal introitus. The nodule was excisied and the histology revealed a hidradenoma papilliferum. The diagnosis and treatment of hidradenoma papilliferum is possible with surgical removal and histopathological evaluation of nodules. Conclusion When an adult woman presents with a noduler lesion in the anogenital area, sexually transmitted diseases and other benign and malignant vulvar lesions, as well as malignant transformation is very rare but,should be kept in mind; however because it has been reported and long-term clinical follow-up is suggested


2021 ◽  
pp. 1-11
Author(s):  
Hamid Reza Niknejad ◽  
Melissa Frederickx ◽  
Emiel Salaets ◽  
Jurgen Lemiere ◽  
Lieven Lagae ◽  
...  

<b><i>Introduction:</i></b> Tectal plate gliomas (TPG) constitute a distinct entity of benign tumors of the brain stem which show an indolent clinical course. Adequate treatment of secondary hydrocephalus is undoubtedly a major factor in the outcome. However, little is known about to what degree the tumor itself determines the long-term outcome of these patients. <b><i>Methods:</i></b> We retrospectively analyzed and compared the clinical and radiological data of 16 pediatric TPG patients with data of 12 pediatric idiopathic aqueductal stenosis (IAS) patients treated in our center from 1988 to 2018. For both groups, we assessed the long-term outcome in terms of hydrocephalus management, and for the TPG group, we assessed tumor growth during follow-up. In a separate prospective part of the study, we performed a neuropsychological evaluation in a subgroup of patients using a standardized testing battery, covering intelligence, learning, memory, executive functions, and an inventory on depression. <b><i>Results:</i></b> In the TPG group, the mean clinical and radiological follow-up was 84 and 70 months, respectively. On average, the maximum diameter of the tumor increased by 11% (<i>p</i> = 0.031) and the estimated tumor volume with 35% (<i>p</i> = 0.026) on radiological follow-up. The fronto-occipital horn ratio (FOHR) decreased by 23% on average after treatment. In the IAS group, the mean clinical and radiological follow-up was 117 and 85 months, respectively. In this group, the FOHR decreased by 21% on average. Neurocognitive testing revealed significant higher scores in the TPG group on global intelligence (TPG = 109, IAS = 85.5, <i>U</i> = 3, <i>p</i> &#x3c; 0.01, <i>z</i> = −2.71), performance (TPG= 100, IAS = 85, <i>U</i> = 7, <i>p</i> = 0.03, <i>z</i> = −2.2), and verbal intelligence (TPG = 122, IAS = 91.5, <i>U</i> = 2, <i>p</i> &#x3c; 0.00, <i>z</i> = −2.87) as well as working memory (TPG = 109.5, IAS = 77, <i>U</i> = 0.5, <i>p</i> = 0.01, <i>z</i> = −2.46). <b><i>Conclusion:</i></b> Our results suggest that the long-term outcome in TPG patients is acceptable and that cognition is substantially better preserved than in patients with IAS. This puts the idea of a significant contribution of the tumoral mass to disease outcome on the long term in question. Adequate and prompt management of hydrocephalus is the most important factor in long-term cognitive outcome.


2020 ◽  
pp. 014556132098457
Author(s):  
Tae Seong Eo ◽  
Jeong Hae Kie ◽  
Hyun Seung Choi ◽  
Junhui Jeong

A myopericytoma in the auricle is rare. If an auricle contains a large, firm, red-brown mass, excision should be considered because the mass may be a myopericytoma. After excision, histopathological and immunohistochemical diagnoses are essential to rule out malignancy. Long-term follow-up is required because the tumor is slow-growing.


2021 ◽  
Vol 80 (Suppl 1) ◽  
pp. 1306.1-1306
Author(s):  
P. Tremaskina ◽  
E. Loginova ◽  
T. Korotaeva ◽  
S. Glukhova ◽  
A. Lila

Background:The concept of treat to target (T2T) in psoriatic arthritis (PsA) has been established recently and already shown its benefits [1]. But the long-term outcomes of the T2T have not been studied yet.Objectives:To study 5 years (yrs) follow-up of PsA patients (pts) treated according to T2T strategy at the early stage.Methods:35 (M/F–17/18) PsA pts fulfilling CASPAR criteria, who were treated according to T2T strategy at the early stage (PsA duration≤2 yrs) within 24 months (mos) were analyzed. At the time of evaluation mean age is 42.7±11.2 yrs, median (Me) PsA duration 72 [60;95] mos, psoriasis duration 120 [88;180] mos. All pts underwent standard clinical examinations of PsA before started T2T therapy and at follow-up. Within 24 mos of T2T strategy all pts were taking Methotrexate (MTX) monotherapy in increasing dose up to 25 mg/wk and 18 out of 35 (51%) pts received MTX in combination with iTNF. When T2T study was stopped all pts were treated according to standard care with NSAIDs, bDMARDs, MTX, tsDMARDs based on PsA activity and physician decision. The number of pts achieved minimal disease activity (MDA, 5 of 7) and remission by DAPSA (≤4)/low disease activity (LDA)≤14) at the 24 mos of T2T strategy and at 5 yrs follow-up were calculated. The results are presented in the form of mean values, median, upper and lower quartiles.Results:Me duration of follow-up is 68 [53.5;81.5] mos. At 24 mos Me DAPSA 3.48 [0.45;21.76], remission by DAPSA (REM-DAPSA) were seen in 20 out of 35 (57%) pts, LDA-DAPSA in 4 (12%) pts, moderate activity (MoA) by DAPSA in 6 (17%) pts and high disease activity by DAPSA (HDA-DAPSA) in 5 (14%) pts. MDA was noted in 21 out of 35 (60%) pts. At 5 yrs Me DAPSA 7.4 [2.22;13.87], REM-DAPSA was noted in 12 (34%) pts, LDA-DAPSA in 14 (40%), MoA-DAPSA in 5 (14%), HDA-DAPSA in 4 (12%) pts. MDA was observed in 17 of 35 pts (49%). Among 20 pts who had REM-DAPSA at 24 mos only 6 pts (30%) remained in remission at 5 yrs follow-up and 12 out of 21 pts (57.14%) remained in MDA status.Conclusion:In early PsA pts remission and MDA are achievable goal of T2T strategy. But most pts lost remission/MDA after this strategy was changed to a standard care, despite being in remission/MDA status before change of therapy. Further investigations of the long-term outcomes of T2T strategy in PsA, including radiographic outcomes are needed.References:[1]Coates LC, Moverley AR, McParland L, et al. Lancet 2015; 386: 2489–98.Disclosure of Interests:None declared.


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