scholarly journals Combined Intestinal Adenomas/Microcarcinoids

2019 ◽  
Vol 13 (3) ◽  
pp. 410-417
Author(s):  
Hiromi Tamura ◽  
Hiroka Ando ◽  
Reiko Doi ◽  
Shiro Adachi

The combined colonic adenoma/microcarcinoid tumor is a rare intestinal neoplasm featuring intermingled adenomatous and carcinoid components. A few case reports and small case series have suggested that this entity exhibits an indolent clinical course. Here, we report two cases with these tumors, and describe the morphological features and clinical follow-up. A 61-year-old male and 78-year-old male presented with heme-positive stools at their medical checkups. Colonoscopy revealed masses in the colons; we performed endoscopic mucosal resection. Both lesions featured low-grade adenomas and low-grade neuroendocrine tumors. We diagnosed combined colonic adenomas/microcarcinoids. The clinical courses of both patients were benign at follow-up at 2.5 and 6 years. Awareness of this rare colonic tumor should prevent potential diagnostic pitfalls and may help clarify the natural history of these tumors and their possible relationships with composite glandular/carcinoid tumors.

2021 ◽  
Author(s):  
Huy Gia Vuong ◽  
Truong P.x. Nguyen ◽  
Hanh T.t. Ngo ◽  
Lewis Hassell ◽  
Kennichi Kakudo

Malignant thyroid teratoma (MTT) is a very rare thyroid malignancy. These neoplasms have been reported only in case reports and small-sized case series so far. In this study, we searched for MTTs in the Surveillance, Epidemiology, and End Result (SEER) program during 1975-2016. Subsequently, we incorporated the SEER data with published MTT cases in the literature to analyze the characteristics and prognostic factors of MTTs. Integrated data were analyzed using Chi-square or Fisher’s exact test for categorical covariates, and t-test or Mann-Whitney test for continuous variables. We included 28 studies with 36 MTT cases and found additional 8 cases from the SEER program for final analyses. Our results showed that MTT is typically seen in adult females. These neoplasms were associated with an aggressive clinical course with high rates of extrathyroidal extension (80%) and nodal involvement (62%). During follow-up, the development of recurrence and metastases were common (42% and 46%, respectively), and one-third of patients died at the last follow-up. Large tumor size (p = 0.022) and the presence of metastases during follow-up (p = 0.008) were associated with a higher mortality rate. In conclusion, our study demonstrated the characteristic features of MTT patients and outlined some parameters associated with a negative outcome which could help clinicians better predict the clinical course of these neoplasms.


Neurosurgery ◽  
2019 ◽  
Vol 85 (6) ◽  
pp. E975-E991 ◽  
Author(s):  
Stephen Shelby Burks ◽  
Ross C Puffer ◽  
Iahn Cajigas ◽  
David Valdivia ◽  
Andrew E Rosenberg ◽  
...  

Abstract BACKGROUND Synovial sarcoma of the nerve is a rare entity with several cases and case series reported in the literature. Despite an improved understanding of the biology, the clinical course is difficult to predict. OBJECTIVE To compile a series of patients with synovial sarcoma of the peripheral nerve (SSPN) and assess clinical and pathological factors and their contribution to survival and recurrence. METHODS Cases from 2 institutions collected in patients undergoing surgical intervention for SSPN. Systematic review including PubMed and Scopus databases were searched for related articles published from 1970 to December 2018. Eligibility criteria: (1) case reports or case series reporting on SSPN, (2) clinical course and/or pathological features of the tumor reported, and (3) articles published in English. RESULTS From patients treated at our institutions (13) the average follow-up period was 3.2 yr. Tumor recurrence was seen in 4 cases and death in 3. Systematic review of the literature yielded 44 additional cases with an average follow-up period of 3.6 yr. From pooled data, there were 10 recurrences and 7 deaths (20% and 14%, respectively). Adjuvant treatment used in 62.5% of cases. Immunohistochemical markers used in diagnosis varied widely; the most common are the following: Epithelial membrane antigen (EMA), cytokeratin, vimentin, cluster of differentiation (CD34), and transducin-like enhancer of split 1 (TLE1). Statistical analysis illustrated tumor size and use of chemotherapy to be negative predictors of survival. No other factors, clinically or from pathologist review, were correlated with recurrence or survival. CONCLUSION By combining cases from our institution with historical data and performing statistical analysis we show correlation between tumor size and death.


Author(s):  
Morena J ◽  
◽  
Antimisiaris M ◽  
Singh D ◽  
◽  
...  

Objective: We present the distinct clinical and laboratory findings in two cases of Ehrlichia meningoencephalitis, along with one suspected case. Background: The number of cases of Ehrlichia chaffeensis reported to the CDC has more than doubled from 2007-2017. A PubMed literature search using the words “Ehrlichiosis and meningoencephalitis” revealed five case reports with neurologic manifestations. Design/Methods: This is a retrospective observational study. Two elderly patients presented with encephalopathy, fever, transaminitis, thrombocytopenia, a positive E. chaffeensis Polymerase Chain Reaction (PCR) in Serum, and Cerebrospinal Fluid (CSF) with a lymphocytic or neutrophilic pleocytosis and elevated protein. One patient had similar symptoms and a positive E. chaffeensis PCR, but lumbar puncture was unable to be performed due to severe thrombocytopenia. They presented in May or June. Doxycycline was started within 2-3 days after presentation to the hospital. Follow up five months later revealed all patients were close, or back to baseline. Results and Conclusions: Suspicion of Ehrlichia meningoencephalitis should be raised in elderly patients presenting with fever and encephalopathy in the summer season with history of tick bite or residence in wooded areas. Thrombocytopenia and transaminitis should raise further suspicion. CSF studies typically show a lymphocytic pleocytosis and elevated protein. PCR technique allows for direct detection of pathogen-specific DNA and is the preferred method of detection during the acute phase of illness. Prompt treatment with doxycycline results in good outcomes. Doxycycline is not included in the typical meningitis regimen, therefore, this disease is important to quickly identify as delay in Doxycycline can result in worse outcomes.


Author(s):  
Mohammad M. Al-Qattan ◽  
Nada G. AlQadri ◽  
Ghada AlHayaza

Abstract Introduction Herpetic whitlows in infants are rare. Previous authors only reported individual case reports. We present a case series of six infants. Materials and Methods This is a retrospective study of six cases of herpetic whitlows in infants seen by the senior author (MMA) over the past 23 years (1995–2017 inclusive). The following data were collected: age, sex, digit involved in the hand, mode of transmission, time of presentation to the author, clinical appearance, presence of secondary bacterial infection, presence of other lesions outside the hand, method of diagnosis, treatment, and outcome. Results All six infants initially presented with classic multiple vesicles of the digital pulp. In all cases, there was a history of active herpes labialis in the mother. Incision and drainage or deroofing of the vesicles (for diagnostic purposes) resulted in secondary bacterial infection. Conclusion The current report is the first series in the literature on herpetic whitlows in infants. We stress on the mode of transmission (from the mother) and establishing the diagnosis clinically. In these cases, no need for obtaining viral cultures or polymerase chain reaction; and no medications are required. Once the vesicles are disrupted, secondary bacterial infection is frequent and a combination of oral acyclovir and intravenous antibiotics will be required.


2019 ◽  
Vol 24 (5) ◽  
pp. 558-571 ◽  
Author(s):  
Kartik Bhatia ◽  
Hans Kortman ◽  
Christopher Blair ◽  
Geoffrey Parker ◽  
David Brunacci ◽  
...  

OBJECTIVEThe role of mechanical thrombectomy in pediatric acute ischemic stroke is uncertain, despite extensive evidence of benefit in adults. The existing literature consists of several recent small single-arm cohort studies, as well as multiple prior small case series and case reports. Published reports of pediatric cases have increased markedly since 2015, after the publication of the positive trials in adults. The recent AHA/ASA Scientific Statement on this issue was informed predominantly by pre-2015 case reports and identified several knowledge gaps, including how young a child may undergo thrombectomy. A repeat systematic review and meta-analysis is warranted to help guide therapeutic decisions and address gaps in knowledge.METHODSUsing PRISMA-IPD guidelines, the authors performed a systematic review of the literature from 1999 to April 2019 and individual patient data meta-analysis, with 2 independent reviewers. An additional series of 3 cases in adolescent males from one of the authors’ centers was also included. The primary outcomes were the rate of good long-term (mRS score 0–2 at final follow-up) and short-term (reduction in NIHSS score by ≥ 8 points or NIHSS score 0–1 at up to 24 hours post-thrombectomy) neurological outcomes following mechanical thrombectomy for acute ischemic stroke in patients < 18 years of age. The secondary outcome was the rate of successful angiographic recanalization (mTICI score 2b/3).RESULTSThe authors’ review yielded 113 cases of mechanical thrombectomy in 110 pediatric patients. Although complete follow-up data are not available for all patients, 87 of 96 (90.6%) had good long-term neurological outcomes (mRS score 0–2), 55 of 79 (69.6%) had good short-term neurological outcomes, and 86 of 98 (87.8%) had successful angiographic recanalization (mTICI score 2b/3). Death occurred in 2 patients and symptomatic intracranial hemorrhage in 1 patient. Sixteen published thrombectomy cases were identified in children < 5 years of age.CONCLUSIONSMechanical thrombectomy may be considered for acute ischemic stroke due to large vessel occlusion (ICA terminus, M1, basilar artery) in patients aged 1–18 years (Level C evidence; Class IIb recommendation). The existing evidence base is likely affected by selection and publication bias. A prospective multinational registry is recommended as the next investigative step.


2018 ◽  
Vol 64 (6) ◽  
pp. 708-715
Author(s):  
Natalya Severskaya ◽  
Andrey Rodichev ◽  
Aleksey Ilin ◽  
Dmitriy Semin ◽  
Pavel Isaev ◽  
...  

Struma ovarii is a rare variant of the mature ovarian teratoma composed of more than 50% thyroid tissue. Thyroid type carcinoma can occur in 5% of struma ovarii. Given the rarity of this pathology, as well as the different clinical course, approaches to the treatment of this disease are controversial. The proposed approaches to treatment vary from ovarian resection to total hysterectomy with bilateral salpingo-oophorectomy and adjuvant therapy. We present here 6 case reports of thyroid type carcinoma in struma ovarii and outcome of patients treated in our clinic. All patients had pelvic surgery of different extent, followed by thyroidectomy and radioiodine therapy. The incidence of metastasis is 67% (4/6), 2 - intraperitoneal metas-tases, 2 - bone metastases. Among patients with metastases, 2 have reached a complete response, one with a good response continues treatment, one had progression. The follow-up period is 1 to 15 years (median 4 years). One patient with follicular carcinoma died of progression 8 years after diagnosis. The remaining patients are alive.


2019 ◽  
Vol 25 (1) ◽  
Author(s):  
Danielle Whiting ◽  
Ian Rudd ◽  
Amit Goel ◽  
Seshadri Sriprasad ◽  
Sanjeev Madaan

Abstract Background Angiomyolipomas are rare mesenchymal tumours arising from the perivascular epithelioid cells consisting of variable amounts of adipose, thick-walled blood vessels and smooth muscle cells. These benign tumours commonly occur in the kidney with only a few case reports of adrenal angiomyolipomas which have the potential to reach a large size and haemorrhage. Case presentation A 45-year-old lady presented with a 3-week history of right loin pain, nausea and vomiting. A CT scan revealed a right adrenal angiomyolipoma measuring 6.3 × 6.8 cm with associated haemorrhage. The lesion was successfully treated with right open adrenalectomy, and histology confirmed the diagnosis of adrenal angiomyolipoma. The patient remained well with no evidence of recurrence at the 36-month follow-up. Conclusion Adrenal angiomyolipomas are rare benign tumours that have the ability to reach a large size and potential to bleed. Here, we report the second case of spontaneous haemorrhage in an adrenal angiomyolipoma, which was successfully treated with open adrenalectomy.


Author(s):  
S. Harvey ◽  
S. Ryan ◽  
A. Tarrant ◽  
M. King ◽  
B. Hayes

BACKGROUND: Damage to the basal ganglia and thalamus (BGT) can be caused by multiple perinatal factors and may be associated with movement disorders, cognitive delay and visual difficulties. Changes in BGT structure, seen as echogenicity on ultrasound, are difficult to objectively quantify. The aetiology, clinical relevance and developmental outcomes of BGT echogenicity are poorly understood. We aimed to gain a better understanding of the natural history of BGT echogenicity in a preterm population. METHODS: Retrospective review of clinical course, neuroimaging and development in infants born <32weeks gestation over 5 years with evidence of BGT echogenicity. RESULTS: BGT echogenicity was reported in 18/650 infants (2.7%). Echogenicity appeared at a median of 8 days (2–45 days) and resolved on pre-discharge ultrasound in 50%. Thirteen infants had a term corrected MRI brain with abnormal BGT signal seen in 3 infants (23%). All 3 infants had persisting echogenicity on discharge ultrasound. No infant with echogenicity resolution on ultrasound had changes on term MRI. 14 infants had developmental progress available at 1 year corrected. Abnormal development was reported in four children of whom one had BGT changes on term MRI. Two children with persistent BGT changes but an otherwise normal MRI had reported normal neurodevelopment. CONCLUSION: BGT echogenicity is relatively common on routine ultrasound and resolves in the majority of infants by term corrected. This review suggests that at term corrected, normal cranial ultrasound may obviate the need for MRI where no other concerns exist. BGT echogenicity did not appear to independently influence neurodevelopment.


2019 ◽  
Vol 98 (5) ◽  
pp. 291-294 ◽  
Author(s):  
Saudamini J. Lele ◽  
Mickie Hamiter ◽  
Torrey Louise Fourrier ◽  
Cherie-Ann Nathan

Sialendoscopy has emerged as a safe, effective and minimally invasive technique for management of obstructive and inflammatory salivary gland disease. The aim of our study was to analyze outcomes of sialendoscopy and steroid irrigation in patients with sialadenitis without sialoliths. We performed a retrospective analysis of patients who underwent interventional sialendoscopy with steroid irrigation from 2013 to 2016, for the treatment of sialadenitis without sialolithiasis. Twenty-two patients underwent interventional sialendoscopy with ductal dilation and steroid irrigation for the treatment of sialadenitis without any evidence of sialolithiasis. Conservative measures had failed in all. Eleven patients had symptoms arising from the parotid gland, 4 patients had symptoms arising from the submandibular gland, while 6 patients had symptoms in both parotid and submandibular glands. One patient complained of only xerostomia without glandular symptoms. The mean age of the study group which included 1 male and 21 females was 44.6 years (range: 3-86 years). Four patients had autoimmune disease, while 7 patients had a history of radioactive iodine therapy. No identifiable cause for sialadenitis was found in the remaining 11 patients. The mean follow-up period was 378.9 days (range: 16-1143 days). All patients underwent sialendoscopy with ductal dilation and steroid irrigation. Twelve patients showed a complete response and 9 patients had a partial response, while 1 patient reported no response. Only 3 patients required repeat sialendoscopy. The combination of sialendoscopy with ductal dilation and steroid irrigation is a safe and effective treatment option for patients with sialadenitis without sialoliths refractory to conservative measures. Prospective studies with a larger case series are needed to establish its role as a definitive treatment option.


Dermatology ◽  
2021 ◽  
pp. 1-7
Author(s):  
Jurr Boer

<b><i>Background:</i></b> Patients with hidradenitis suppurativa (HS) are still often disappointed with the current treatments offered and there is a clear demand for more effective options. Since the late 1990s there has been a revival in the use of radiotherapy (RT) for different benign diseases, including HS. During the past 20 years one case series and some scattered case reports have described promising results of RT. <b><i>Objectives:</i></b> To evaluate the long-term efficacy of RT in early-stage HS. <b><i>Methods:</i></b> A postal survey-based long-term follow-up with simple factual questions of partly retrospective and partly contemporary characteristics was performed. Sixty-four patients (96 axillae), diagnosed with mild to moderate HS were irradiated with a orthovoltage unit with 100 kV, 3 mm Al or 200 kV, 0.5 Cu filtering, respectively. Four to six biweekly fractional doses ranging from 0.75 to 1 Gy up to a total dose of 6 Gy in one series, and in chronic cases followed by four daily fractions of 2 Gy up to a total dose of 14 Gy, were given. Late treatment toxicity and the rate of remission of the disease were evaluated. <b><i>Results:</i></b> The overall response rate of the survey was 64.1% with 40.6% (26/64) valid, complete questionnaires. In total, 40 axillae were irradiated in these 26 patients. After a median follow-up of 40 years (range 32–52) complete remission of the lesions occurred in 34 of the 40 sites (85%). None of the 26 patients with 40 irradiated sites reported adverse effects at the time of the survey. <b><i>Conclusions:</i></b> RT appears to be an effective treatment for early and mild HS in the majority of patients. In this case series, no side effects were reported after a median follow-up period of 40 years.


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