Transformation from Sinonasal Seromucinous Hamartoma to Adenocarcinoma: A Case Report

ORL ◽  
2021 ◽  
pp. 1-3
Author(s):  
David A. Rengifo ◽  
Varun V. Varadarajan ◽  
Jinping Lai ◽  
Jeb M. Justice
Keyword(s):  
Malignant Transformation ◽  
Middle Turbinate ◽  
Sinus Surgery ◽  
Low Grade ◽  
Complete Excision ◽  
Nasal Mass ◽  
Respiratory Epithelial Adenomatoid Hamartoma ◽  
Seromucinous Hamartoma ◽  
First Case ◽  
Risk Of Malignancy

<b><i>Objectives:</i></b> Sinonasal hamartomas are benign neoplasms composed of disorganized mature tissue elements. Epithelial variants include respiratory epithelial adenomatoid hamartoma (REAH) and seromucinous hamartoma (SMH). Malignant transformation of REAH is rarely reported; however, the malignant transformation of SMH to adenocarcinoma has not been described. We report the first case of a transformation from SMH to adenocarcinoma. <b><i>Methods:</i></b> The medical records of a patient presenting with sinonasal SMH with malignant transformation to adenocarcinoma were reviewed. The NCBI database was queried for the literature regarding SMH and malignant transformation of sinonasal hamartomas. <b><i>Results:</i></b> A 39-year-old man presented with a left nasal mass, nasal obstruction, and epistaxis. Computed tomography and magnetic resonance imaging demonstrated a nonaggressive and heterogeneous left nasal mass with involvement of the middle turbinate and posterior ethmoid sinuses. He underwent endoscopic sinus surgery with complete excision of the mass. Pathology revealed SMH with focal areas of transition to low-grade adenocarcinoma characterized by stromal invasion but no bony, perineural, or lymphovascular invasion. Adjuvant treatment was not recommended. Literature review revealed no reported cases of malignant transformation of SMH. <b><i>Conclusion:</i></b> We report the first case of malignant transformation of SMH. Patients with SMH must be counseled that there is an extremely rare and potentially unrecognized risk of malignancy that may influence treatment and postoperative monitoring.

scholarly journals Case Report: Response to Intra-arterial Cisplatin and Concurrent Radiotherapy Followed by Salvage Surgery in a Patient With Advanced Primary Sinonasal Low-Grade Non-intestinal Adenocarcinoma

2020 ◽  
Vol 7 ◽  
Author(s):  
Hirohiko Tachino ◽  
Hiromasa Takakura ◽  
Hideo Shojaku ◽  
Michiro Fujisaka ◽  
Katsuichi Akaogi ◽  
...  
Keyword(s):  
Case Report ◽  
Salvage Surgery ◽  
Histopathological Examination ◽  
Locally Advanced ◽  
Middle Turbinate ◽  
Lymphocytic Infiltration ◽  
Low Grade ◽  
Fractionated Radiotherapy ◽  
First Case ◽  
Positron Emission

Background: The clinical usefulness of concurrent chemoradiotherapy before surgery in the treatment of primary, locally advanced sinonasal low-grade, non-intestinal type adenocarcinoma (LG non-ITAC) is unclear.Methods: We present the first case report of the efficacy of super-selective intra-arterial cisplatin (CDDP) infusion concurrent with conventional fractionated radiotherapy (RT) for LG non-ITAC in a Japanese patient.Results: A white, rugged-marginal mass that was histopathologically diagnosed as LG non-ITAC occupied the right nasal cavity. Based on the imaging findings, including computed tomography, magnetic resonance imaging, and positron emission tomography, the tumor was diagnosed as T4aN0M0, stage IVa. After treatment, the nasal tumor disappeared leaving only a small bulge in the medial wall of the middle turbinate. The patient also underwent right transnasal ethmoidectomy performed as salvage surgery. A histopathological examination revealed that the lesion was replaced by granulation tissue with lymphocytic infiltration and hemosiderin-laden macrophages, and no viable tumor cells remained. In the seven years after treatment, the patient has not experienced any local recurrence or regional or distant metastasis.Conclusions: Super-selective intra-arterial CDDP infusion concurrent with conventional fractionated RT followed by salvage surgery might be useful for the management of sinonasal LG non-ITAC.

Nasal craniopharyngioma: case report and literature review

2011 ◽  
Vol 125 (5) ◽  
pp. 517-519 ◽  
Author(s):  
J C Magill ◽  
M S Ferguson ◽  
A Sandison ◽  
P M Clarke
Keyword(s):  
Case Report ◽  
Adjuvant Radiotherapy ◽  
English Language ◽  
Middle Turbinate ◽  
Craniofacial Resection ◽  
Complete Excision ◽  
First Case ◽  
History Of ◽  
One Year ◽  
Midfacial Degloving

AbstractObjective:We report the first case in the English language literature of an adamantinomatous infrasellar craniopharyngioma, and we describe our management strategy.Case report:A 46-year-old woman presented with a six-month history of left-sided nasal obstruction and epistaxis. Rhinological examination revealed a left-sided, polypoidal lesion lying medial to the middle turbinate. An urgent examination under anaesthesia was organised; biopsies were considered characteristic of craniopharyngioma. Magnetic resonance imaging and computed tomography demonstrated a well defined, heterogeneous, infrasellar mass centred in the midline, extending anteriorly into the left nasal cavity and posteriorly encasing both internal carotid arteries. Craniofacial resection was performed via a midfacial degloving approach, with adjuvant radiotherapy. The patient was disease-free one year post-operatively.Conclusion:Craniopharyngiomas should be considered in the differential diagnosis of a unilateral nasal polyp. Although technically benign, they are locally aggressive. Therefore, we recommend complete excision with adjuvant radiotherapy if margins are involved or close.

scholarly journals Rare Sinonasal Non-intestinal Type Adenocarcinoma of Right Nasal Septum

2019 ◽  
Vol 3 (1) ◽  
pp. 43
Author(s):  
Adam Mohamad ◽  
Baharudin Abdullah ◽  
Seoparjoo Azmel Mohd Isa ◽  
Norhafizah Abdul Rahim
Keyword(s):  
Nasal Septum ◽  
Young Male ◽  
Intestinal Type ◽  
Low Grade ◽  
Anatomic Structure ◽  
Respiratory Epithelial Adenomatoid Hamartoma ◽  
Seromucinous Hamartoma ◽  
Obstructive Sleep ◽  
Sinonasal Adenocarcinoma ◽  
The Right

Sinonasal tract is a complex anatomic structure with many possible diagnoses. Common diagnoses that usually encountered from the biopsied samples are inflammatory polyps and papillomas as well as squamous cell carcinoma. Another entity which most poorly understood is low grade glandular or tubular proliferations which includes reactive lesions, respiratory epithelial adenomatoid hamartoma, seromucinous hamartoma, and low grade sinonasal adenocarcinoma. We describe a case of a young male with right nasal septum mass which found incidentally upon flexible nasopharyngolaryngoscopic examination, who was referred earlier for possible obstructive sleep apnea. After excision of the tumour was done, it was confirmed to be low grade sinonasal adenocarcinoma non-intestinal type. This case highlights the rarity of the entitiy of low grade sinonasal adenocarcinoma nonintestinal type involving the right nasal septum; and how its subtle finding can be mistaken to be a clinically benign condition.International Journal of Human and Health Sciences Vol. 03 No. 01 January’19. Page : 43-46

scholarly journals Upbeat vertical nystagmus after brain stem cavernoma resection: a rare case of nucleus intercalatus/nucleus of roller injury

2020 ◽  
Vol 267 (10) ◽  
pp. 2865-2870
Author(s):  
Torstein R. Meling ◽  
Aria Nouri ◽  
Adrien May ◽  
Nils Guinand ◽  
Maria Isabel Vargas ◽  
...  
Keyword(s):  
Rare Case ◽  
Vascular Malformations ◽  
Clinical Manifestations ◽  
Neurological Injury ◽  
Medial Longitudinal Fasciculus ◽  
Blurred Vision ◽  
Complete Excision ◽  
First Case ◽  
Vertical Nystagmus ◽  
Upbeat Nystagmus

Abstract Introduction CNS cavernomas are a type of raspberry-shaped vascular malformations that are typically asymptomatic, but can result in haemorrhage, neurological injury, and seizures. Here, we present a rare case of a brainstem cavernoma that was surgically resected whereafter an upbeat nystagmus presented postoperatively. Case report A 42-year old man presented with sudden-onset nausea, vomiting, vertigo, blurred vision, marked imbalance and difficulty swallowing. Neurological evaluation showed bilateral ataxia, generalized hyperreflexia with left-sided predominance, predominantly horizontal gaze evoked nystagmus on right and left gaze, slight left labial asymmetry, uvula deviation to the right, and tongue deviation to the left. MRI demonstrated a 13-mm cavernoma with haemorrhage and oedema in the medulla oblongata. Surgery was performed via a minimal-invasive, midline approach. Complete excision was confirmed on postoperative MRI. The patient recovered well and became almost neurologically intact. However, he complained of mainly vertical oscillopsia. The videonystagmography revealed a new-onset spontaneous upbeat nystagmus in all gaze directions, not suppressed by fixation. An injury of the rarely described intercalatus nucleus/nucleus of Roller is thought to be the cause. Conclusion Upbeat nystagmus can be related to several lesions of the brainstem, including the medial longitudinal fasciculus, the pons, and the dorsal medulla. To our knowledge, this is the first case of an iatrogenic lesion of the nucleus intercalatus/nucleus of Roller resulting in an upbeat vertical nystagmus. For neurologists, it is important to be aware of the function of this nucleus for assessment of clinical manifestations due to lesions within this region.

scholarly journals Microbiome and PCOS: State-of-Art and Future Aspects

2021 ◽  
Vol 22 (4) ◽  
pp. 2048
Author(s):  
Pierluigi Giampaolino ◽  
Virginia Foreste ◽  
Claudia Di Filippo ◽  
Alessandra Gallo ◽  
Antonio Mercorio ◽  
...  
Keyword(s):  
Insulin Resistance ◽  
Bile Acid ◽  
Peptide Yy ◽  
Fecal Microbiota Transplantation ◽  
Polycystic Ovary ◽  
Current Evidence ◽  
Low Grade ◽  
Fecal Transplant ◽  
Secondary Bile Acid ◽  
First Case

Polycystic ovary syndrome (PCOS) is a complex and heterogeneous endocrine disease. The hypothesis that alterations in the microbiome are involved in the genesis of PCOS has been postulated. Aim of this review is to summarize the available literature data about the relationship between microbiome and PCOS. A search on PubMed and Medline databases was performed from inception to November 20Most of evidence has focused on the connection of intestinal bacteria with sex hormones and insulin-resistance: while in the first case, a relationship with hyperandrogenism has been described, although it is still unclear, in the second one, chronic low-grade inflammation by activating the immune system, with increased production of proinflammatory cytokines which interfere with insulin receptor function, causing IR (Insulin Resistance)/hyperinsulinemia has been described, as well as the role of gastrointestinal hormones like Ghrelin and peptide YY (PYY), bile acids, interleukin-22 and Bacteroides vulgatus have been highlighted. The lower genital tract microbiome would be affected by changes in PCOS patients too. The therapeutic opportunities include probiotic, prebiotics and synbiotics, as well as fecal microbiota transplantation and the use of IL-22, to date only in animal models, as a possible future drug. Current evidence has shown the involvement of the gut microbiome in PCOS, seen how humanized mice receiving a fecal transplant from women with PCOS develop ovarian dysfunction, immune changes and insulin resistance and how it is capable of disrupting the secondary bile acid biosynthesis. A future therapeutic approach for PCOS may involve the human administration of IL-22 and bile acid glycodeoxycholic acid.

scholarly journals Molecular Landscape for Malignant Transformation in Diffuse Astrocytoma

2021 ◽  
Author(s):  
Thara Tunthanathip ◽  
Surasak Sangkhathat ◽  
Kanet Kanjanapradit
Keyword(s):  
Malignant Transformation ◽  
Extent Of Resection ◽  
Low Grade ◽  
Diffuse Astrocytoma ◽  
Continuous Variables ◽  
Isocitrate Dehydrogenase 1 ◽  
Molecular Alteration ◽  
Exact Test ◽  
Molecular Abnormalities ◽  
Biological Variables

Abstract Background Malignant transformation (MT) of low-grade gliomas changes dramatically the natural history to poor prognosis. Currently, factors associated with MT of gliomas have been inconclusive, in particular, diffuse astrocytoma (DA). Objective The present study aims to explore the molecular abnormalities related to MT in the same patients with different MT stages. Methods Twelve specimens from five DA patients with MT were genotyped using next-generation sequencing (NGS) to identify somatic variants in different stages of MT. We used cross-tabulated categorical biological variables and compared the mean of continuous variables to assess for association with MT. Results Ten samples succussed to perform NGS from one male and four females, with ages ranging from 28 to 58 years. The extent of resection was commonly a partial resection following postoperative temozolomide with radiotherapy in 25% of cases. For molecular findings, poly-T-nucleotide insertion in isocitrate dehydrogenase 1 (IDH1) was significantly related to MT as a dose–response relationship (Mann–Whitney's U test, p = 0.02). Also, mutations of KMT2C and GGT1 were frequently found in the present cohort, but those did not significantly differ between the two groups using Fisher's exact test. Conclusion In summary, we have identified a novel relationship between poly-T insertion polymorphisms that established the pathogenesis of MT in DA. A further study should be performed to confirm the molecular alteration with more patients.

Scytalidium dimidiatum associated invasive fungal sinusitis in an immunocompetent patient

2014 ◽  
Vol 128 (11) ◽  
pp. 1018-1021 ◽  
Author(s):  
A Hariri ◽  
N Choudhury ◽  
H A Saleh
Keyword(s):  
Corticosteroid Treatment ◽  
Fruit Trees ◽  
Immunocompetent Patient ◽  
Human Infection ◽  
Functional Endoscopic Sinus Surgery ◽  
Sinus Surgery ◽  
Fungal Sinusitis ◽  
Endemic Region ◽  
Invasive Fungal Sinusitis ◽  
First Case

AbstractBackground:Scytalidium dimidiatum is a soil and plant pathogen that frequently affects fruit trees, but can also cause human infection. There are only two reported cases of invasive fungal sinusitis involving this rare micro-organism.Objective:This paper reports the first case of invasive fungal sinusitis caused by Scytalidium dimidiatum occurring in a young immunocompetent patient from a non-endemic region, and discusses potential sources of exposure and relevance of local factors.Method:Case report.Results:The patient was treated successfully with a combination of functional endoscopic sinus surgery, and antifungal and corticosteroid treatment.Conclusion:This paper describes the first reported case of invasive fungal sinusitis secondary to Scytalidium dimidiatum in a young immunocompetent patient from a non-endemic region. Importance is placed on following a systematic process of investigation and management, and adhering to well-defined basic surgical principles.

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