Tuesday, February 23, 2021

Compressive cervicothoracic pseudomeningocele as a rare manifestation of idiopathic intrathecal hypotension after past trauma: a review

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Eur Spine J. 2021 Feb 23. doi: 10.1007/s00586-021-06766-8. Online ahead of print.

ABSTRACT

PURPOSE: Spontaneous intracranial or intrathecal hypotension (SIH) is an underdiagnosed phenomenon predominantly presenting with low cerebrospinal fluid (CSF) pressure and postural headache in the setting of CSF leak. Extrathecal CSF collections causing compression of the spinal cord or nerve roots present an even rarer subset of this disease. We aim to describe this pathology in a comprehensive manner while illustrating with a case of our own.

METHODS: We present a literature review on spinal idiopathic pseudomeningoceles and their neurological implications illustrated with a case of an anterior compressive pseudomeningocele between C2 and D7. Further investigations through a myelography and myelo-CT were able to postulate a CSF leak through a discogenic osteophytic microspur at the level C5-C6.

RESULTS: Spinal manifestations are uncommon i n cases of idiopathic or spontaneous CSF leak, occurring in about 6% of patients, but myelopathy and radiculopathy involving all spinal segments do occur. In contrast to the cranial complaints, the spinal manifestations usually are not positional and are caused by mass effect from an extradural CSF collection.

CONCLUSION: The utility of multiple imaging modalities such as dynamic myelography and the use of epidural blood patches and fibrin glue polymers should be explored, and surgery is an option if the symptoms persist despite other measures.

PMID:33620574 | DOI:10.1007/s00586-021- 06766-8

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Corneal perforation with tyrosine kinase inhibitor chemotherapy: REGORAFENIB

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J Fr Ophtalmol. 2021 Feb 18:S0181-5512(21)00095-4. doi: 10.1016/j.jfo.2020.06.053. Online ahead of print.

ABSTRACT

INTRODUCTION: Tyrosine kinase inhibitors (TKIs) are active in a variety of metastatic cancers. They have a good general tolerance with mainly hepatic and dermatological side effects. Rarely, ophthalmologic side effects may occur: eyelash abnormalities, eyelids abnormalities, disorders of the ocular surface with ocular dryness or even corneal erosions that can even lead to perforation. Regorafenib is a new oral multi-targeted tyrosine kinase inhibitor that inhibits multiple protein kinases, including those involved in tumor angiogenesis, oncogenesis and tumor microenvironment.

CASE DESCRIPTION: We describe, to the best of our knowledge, the first case of complicated bilateral ulcers of corneal perforation in a patient under REGORAFENIB.

OBSERVATION: 20-year-old patient with metastatic chondrosarcomas of the pelvis, man dible and thorax received chemotherapy with REGORAFENIB. A few weeks after initiation of treatment, he experienced an increased dry eye syndrome associated with bilateral corneal ulcers complicated by perforation. Despite discontinuation of chemotherapy and maximal medical and surgical treatment (iterative amniotic membrane grafts and corneal transplantation), the progression was unfavorable.

DISCUSSION: This is the first known case of corneal perforation under REGORAFENIB. The pathophysiology is multifactorial. On the one hand, this chemotherapy targets angiogenesis (VEGFR), oncogenesis (KIT, RET, RAF1, BRAF) and the tumor microenvironment (PDGFR, FGFR). On the other hand, other triggers are added, namely mixed dry eye syndrome, hypovitaminosis A (anorexia), the neurotrophic component, as well as the toxicity of chemotherapy via tears.

CONCLUSION: First described case of corneal perforation under REGORAFENIB, non-regressive at the end of chemotherapy, and despite medica l and surgical treatments. Ophthalmologic surveillance is therefore necessary for patients under chemotherapy with tyrosine kinase inhibitors, as serious ocular complications, especially corneal ones, may occur.

PMID:33612324 | DOI:10.1016/j.jfo.2020.06.053

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Association of thyrolipoma and thyrolipomatosis: A case report

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Ann Pathol. 2021 Jan 21:S0242-6498(20)30216-9. doi: 10.1016/j.annpat.2020.08.005. Online ahead of print.

ABSTRACT

We report a rare case of thyrolipoma-thyrolipomatosis' association. A 48-year-old patient, with a past history of high blood pressure and chronic renal failure secondary to AA amyloidosis, presented with a midline neck swelling increasing in size since one year. Ultrasound showed a multinodular goiter, the worst nodule was left-lobar and categorized as EU-TIRADS 4. Grossly, cut section of the thyroïd specimen showed a yellow-tan left-lobar nodule measuring 2.5cm, the surrounding tissue had a diffuse yellow-brown appearance with scattered whitish areas. Histopathologic examination revealed that the nodule corresponded to a follicular adenoma.Thyroid fat infiltration was noticed both within the nodule and in the neighboring parenchyma. Thus, the diagnosis of an association thyrolipoma-thyrolipomatosis was rendered. The association of thyrolipoma-thyrolipomatosis is a very rare occurrence of unclear pathogenesis. To the best of our knowledge, this report describes the second case in the literature. The diagnosis is retained on the basis of certain histological features, revealing mature adipose tissue within thyroid gland as a combined nodular and diffuse pattern.

PMID:33487459 | DOI:10.1016/j.annpat.2020.08.005

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A thyroid-like low-grade nasopharyngeal papillary adenocarcinoma

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Ann Pathol. 2021 Jan 22:S0242-6498(20)30217-0. doi: 10.1016/j.annpat.2020.08.006. Online ahead of print.

ABSTRACT

Thyroid-like low-grade nasopharyngeal papillary adenocarcinoma is a rare histopathological entity. Patients usually complain of nasal obstruction and epistaxis. Diagnosis is confirmed on endonasal biopsy using immunohistochemical studies. Surgery is the treatment of choice and this pathology exhibits no metastasizes nor recurrence after treatment.

PMID:33494952 | DOI:10.1016/j.annpat.2020.08.006

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Revision Surgery for Idiopathic Macular Hole after Failed Primary Vitrectomy

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J Ophthalmol. 2021 Jan 7;2021:8832538. doi: 10.1155/2021/8832538. eCollection 2021.

ABSTRACT

PURPOSE: To investigate the anatomical and functional outcomes of revision surgery after failed primary surgery for idiopathic macular hole (MH).

METHODS: All consecutive patients with MH were identified from a cohort of patients operated between 2014 and 2018 at the CHU de Québec-Université Laval (Québec). The clinical and anatomical features of patients with unclosed MH after primary surgery were retrospectively collected. Our primary outcome was MH nonclosure rate after revision surgery. Our secondary outcomes were best-corrected visual acuity (BCVA) with ETDRS scale and MH size of eyes with revision surgery preoperatively and at 3 and 12 months after revision surgery.

RESULTS: In our cohort of 1085 eyes, 926 eyes met inclusion criteria and were analyzed in the study. We identified 22 eyes with failed primary surgery (2.4%), of which 20 underwent revision surgery. We had no bilateral MH in these 22 eyes. The nonclosure rate of MH after revision surgery was 15%. The mean final BCVA for closed MH after revision surgery was 55 ± 19 letters. Compared to the initial presentation, the mean change in visual acuity (VA) for closed MH was +4 ± 31 letters and +16 ± 17 letters at 3 and 12 months after the revision surgery, respectively. At initial presentation, patients with failed primary surgery had a baseline MH size of 665 ± 226 μm. The mean MH size after failed primary surgery was 607 ± 162 μm and 546 ± 156 μm for the three unclosed MHs one month after revision surgery.

CONCLUSION: The success rate of revision surgery in eyes with unclosed MH is 85%. After successful revision surgery, eyes demonstrated an improvement in VA and closure of the MH.

PMID:33520301 | PMC:PMC7817234 | DOI:10.1155/2021/8832538

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Oropharyngeal squamous cell carcinoma: p16/p53 immunohistochemistry as a strong predictor of HPV tumor status

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Histopathology. 2021 Feb 9. doi: 10.1111/his.14350. Online ahead of print.

ABSTRACT

AIMS: Oropharyngeal squamous cell carcinomas (OPSCC) related to human papillomavirus (HPV) infection have a better prognosis than those without HPV infection. Although p16INK4a overexpression is used as a surrogate marker for HPV infection, 5%-20% of p16-positive OPSCC are described as being unrelated to HPV infection, with worse overall survival compared to OPSCC-related HPV. There is, therefore, a risk of undertreating a proportion of OPSCC patients falsely considered as HPV-driven because of p16 positivity. TP53 mutations are highly prevalent in OPSCC driven by mutagens in tobacco and alcohol. We describe herein a combined p16/p53 algorithm to predict HPV tumor status in OPSCC.

METHODS AND RESULTS: 110 OPSCC were identified in the database of the pathology department and were studied using p16 and p53 immunohistochemistry. For p16-positive or p16-negative/wild-type patterns-p53 (WT-p53) cases (n = 63), DNA in-situ hybridization for high-risk HPV was performed and if negative, the HPV status was controlled by HPV DNA PCR (n=19). A significant association between TP53 mutation and pattern of p53 expression was found (WT-p53, 7/16, P<0.001). The p16-positive/WT-p53 was significantly associated with HPV+ tumor status (p16-positive/WT-p53, 50/110, P<0.001). Interestingly, a subset of p16-positive OPSCC was unrelated to HPV (13.5%, 8/59) and showed mutant-type staining of p53 expression.

CONCLUSIONS: The p16 protein immunopositivity in conjunction with the mutant-type pattern of p53 staining helped to reclassify a subset of p16-positive OPSCC as OPSCC-unrelated HPV. This approach could be routinely applied by pathologists involved in the management of OPSCC, because of their potential therapeutic implications.

PMID:33560536 | DOI:10.1111/his.14350

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Matrix metalloproteinases and their inhibitors in Fuchs endothelial corneal dystrophy

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Exp Eye Res. 2021 Feb 19:108500. doi: 10.1016/j.exer.2021.108500. Online ahead of print.

ABSTRACT

Fuchs endothelial dystrophy (FECD) is characterized by a progressive loss of corneal endothelial cells (CECs) and an abnormal accumulation of extracellular matrix in Descemet's membrane leading to increased thickness and formation of excrescences called guttae. Extracellular matrix homeostasis is modulated by an equilibrium between matrix metalloproteinases (MMPs) and their endogenous tissue inhibitors (TIMPs). This study aimed to investigate MMPs and TIMPs profile in FECD, taking into account cell morphology. Populations of FECD and healthy CECs were cultured and their conditioned media collected for analysis. The presence of proteases in the conditioned media was studied using a semi-quantitative proteome profiler array, and MMPs levels were assessed using quantitative assays (ELISA and quantitative antibody array). MMP activity was determined by zymography and fluorometry. The expression pattern of the membrane type 1-MMP (MT1-MMP, also known as MMP-14) was examined by immunofluorescence on ex vivo FECD and healthy explants of CECs attached to Descemet's membrane. Finally, MMPs and TIMPs protein expression was compared to gene expression obtained from previously collected data. FECD and healthy CEC populations generated cultures of endothelial, intermediate, and fibroblastic-like morphology. Various MMPs (MMP-1, -2, -3, -7, -8, -9, -10, and -12) and TIMPs (TIMP-1 to -4) were detected in both FECD and healthy CECs culture supernatants. Quantitative assays revealed a decrease in MMP-2 and MMP-10 among FECD samples. Both these MMPs can degrade the main extracellular matrix components forming guttae (fibronectin, laminin, collagen IV). Moreover, MMPs/TIMPs ratio was also decreased among FECD cell populations. Activity assays showed greater MMPs/Pro-MMPs proportions for MMP-2 and MMP-10 in FECD cell populations, although over all activities were similar. Moreover, the analysis according to cell morphology revealed among healthy CECs, both increased (MMP-3 and -13) and decreased (MMP-1, -9, -10, and -12) MMPs proteins along with increased MMPs activity (MMP-2, -3, -9, and -10) in the fibroblastic-like subgroup when compared to the endothelial subgroup. However, FECD CECs did not show similar behaviors between the different morphology subgroups. Immunostaining of MT1-MMP on ex vivo FECD and healthy explants revealed a redistribution of MT1-MMP around guttae in FECD explants. At the transcriptional level, no statistically significant differences were detected, but cultured FECD cells had a 12.2-fold increase in MMP1 and a 4.7-fold increase in TIMP3. These results collectively indicate different, and perhaps pathological, MMPs and TIMPs profile in FECD CECs compared to healthy CECs. This is an important finding suggesting the implication of MMPs and TIMPs in FECD pathophysiology.

PMID:33617849 | DOI:10.1016/j.exer.2021.108500

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Duplication of the external jugular vein: a language barrier of database search in classic anatomical studies

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Surg Radiol Anat. 2021 Feb 23. doi: 10.1007/s00276-021-02717-6. Online ahead of print.

ABSTRACT

OBJECTIVE: Many anatomical variations of the superficial veins of the head and neck have been reported throughout the literature. Accordingly, anatomists and surgeons must have a comprehensive understanding of these variations to avoid confusion. Duplication of the external jugular vein (EJV) is occasionally observed during routine cadaveric dissections; however, this variation se ems to be reported less often than actual experience suggests. Therefore, to gain a better understanding of its anatomical and clinical implications, an analysis of the available data should be available. Thus, in this article, we reviewed the current available literature for studies reporting duplication of the EJV.

METHODS: We conducted a search using PubMed and Google Scholar with the following keywords: "duplication of the external jugular vein," "division of the external jugular vein," and "fenestration of the external jugular vein," "double external jugular vein," and "doubled external jugular vein." As a case illustration, we also describe a case of a duplicated EJV found during a right neck dissection of a female cadaver.

RESULTS: Twenty sides across sixteen different studies were analyzed including the present case. All studies were published between 2009 and 2020. EJV division patterns were classified as either duplication, fenestration, fenestration followed b y duplication, or double fenestrations.

CONCLUSIONS: We have reviewed the literature regarding cases documenting duplication/fenestration of the EJV. As it is often difficult to find recent studies that report on classic anatomical variations, therefore, revisiting older articles and textbooks is necessary for achieving a "comprehensive" review, especially across different languages.

PMID:33620594 | DOI:10.1007/s00276-021-02717-6

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Monday, February 22, 2021

Combined Transverse Cordotomy‐ Anteromedial Arytenoidectomy for Isolated Glottic Stenosis

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Objectives/Hypothesis

Glottic stenosis is a discrete cause of airway compromise. We aimed to determine the surgical outcomes of transverse cordotomy with anteromedial arytenoidectomy (TCAMA), performed in the setting of isolated glottic stenosis resulting from two discrete etiologies: bilateral vocal fold paralysis (BVFP) and posterior glottic stenosis (PGS).

Study Design

Retrospective, analytic cohort study.

Methods

Twenty‐six patients with isolated glottic stenosis were treated with TCAMA between 2006 and 2019. A retrospective analysis determined decannulation rates and intervals, voice outcomes, swallowing outcomes, and reoperation rates postoperatively. Outcomes between the two etiologic cohorts were compared.

Results

Of the 26 patients, 16/26 patients were diagnosed with PGS and 10/26 with BVFP. Eighteen patients required tracheotomies during their clinical course (11/16 PGS, and 7/10 BVFP), and 100% were ultimately decannulated. The PGS cohort required two‐sided interventions more frequently than the BVFP cohort (45.5% vs. 0%, P = .066). Trach‐dependent PGS patients required a longer time to achieve decannulation than BVFP patients by a factor of 2.38, although the difference was not statistically significant (102.3 days vs. 42.9 days, respectively, P = .113). Patients demonstrated a significant change in maximum phonation time but no statistically significant differences with preoperative versus postoperative voice outcomes like voice‐related quality of life. All patients ultimately returned to their baseline swallow function postoperatively.

Conclusion

TCAMA is an effective treatment for surgical rehabilitation of glottic stenosis caused by both BVFP and PGS. Patient‐reported outcomes of postoperative vocal function remain consistent following surgical intervention. Additional, prospective studies with greater power are warranted to validate the contrasting outcomes observed when applying this discrete surgical technique across two distinct diagnostic cohorts in this retrospective study.

Level of Evidence

4. Laryngoscope, 2021

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Cricopharyngeal Myotomy in Inclusion Body Myositis: Comparison of Endoscopic and Transcervical Approaches

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Objective

Inclusion body myositis (IBM) is a progressive inflammatory myopathy with dysphagia as a debilitating sequalae. Otolaryngologists are consulted for surgical candidacy when there are findings of cricopharyngeal dysfunction. We aim to compare transcervical cricopharyngeal myotomy (TCPM) versus endoscopic cricopharyngeal myotomy (ECPM) in the IBM population with particular focus on objective swallow study outcomes, complications, and recurrence rates.

Methods

A retrospective cohort study was performed on IBM patients who underwent TCPM or ECPM (1981–2020) in the Department of Otolaryngology at a tertiary academic center with a high volume IBM referral base. Videofluoroscopic swallow studies, Eating Assessment Tool (EAT‐10), Reflux Symptom Index (RSI), and Functional Outcome Swallowing Scale (FOSS) were collected at preoperative and follow‐up visits. Baseline patient characteristics, intraoperative data, and postoperative course were recorded.

Results

Forty‐one patients were identified (18 TCPM; 23 ECPM). There was no significant difference in the recurrence rates, complications, hospitalization length, operative time, or return to preoperative diet between approaches. For the 12 patients (11 ECPM; 1 TCPM) that had subjective swallow data, there was a statistically significant difference in the pre and postoperative scores for EAT‐10, RSI, and FOSS (P < .05). There was a statistically significant improvement in the degree of narrowing between pre and postoperative imaging for both approaches (P < .05).

Conclusion

Both TCPM and ECPM are safe approaches for the management of dysphagia in patients with IBM with objective evidence of cricopharyngeal dysfunction. Cricopharyngeal myotomy is a durable technique that has demonstrated improved subjective and objective outcomes in this patient population.

Level of Evidence

3. Laryngoscope, 2021

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Long‐term neck and shoulder function among survivors of oropharyngeal squamous cell carcinoma treated with chemoradiation as assessed with the neck dissection impairment index

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Abstract

Background

Of interest is the long‐term neck and shoulder impairment of patients treated with primary chemoradiotherapy (CRT). This is important for counseling patients regarding treatment decisions when discussing primary CRT.

Methods

A cross‐sectional study to identify factors that contribute to neck and shoulder dysfunction in patients treated with primary CRT. We utilized the neck dissection impairment index (NDII). Eighty‐seven patients treated between 2003 and 2010, who were free of disease, responded; 24 of these 87 underwent post‐CRT neck dissection. Mean interval since completion of CRT was over 5 years (62.7 months). Mean age, 63.5 years, male:female 75:12.

Results

Mean NDII score was 87.4 (SD 22.1, range 5–100). Multiple linear regression revealed worse NDII scores for patients with larger pre‐CRT gross tumor nodal volume (GTVnodal), controlled for age, sex, body mass index (BMI), and the presence of neck dissection (p = 0.02). There were significant associations with increasing GTVnodal and "low" scores for components of the NDII that assessed neck pain (p = 0.02), neck stiffness (p = 0.01), lifting heavy objects (p = 0.02), reaching overhead (p = 0.02), and ability to do work (p = 0.02). Physical therapy (PT) was evaluated as an "anchor" but it was prescribed "as needed." Regression revealed participation in PT was associated with higher GTVnodal, lower BMI, presence of neck dissection, and female sex (p = 0.00007).

Conclusion

GTVnodal was an independent predictor of neck and shoulder impairment. High GTVnodal was associated with increased pain and stiffness, and increased difficulty lifting heavy objects, reaching overhead, overall ability to perform work‐related tasks and was associated with participation in post‐treatment PT.

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