scholarly journals A Unique Presentation of a Paraganglioma and Its Consequences

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A989-A990
Author(s):  
Kunal Shah ◽  
Remy Mimms ◽  
Jonathan Robert Anolik

Abstract Introduction: Paragangliomas (PGL) are extremely rare tumors arising from extra-adrenal neural crest cells with an incidence of 0.8 per 100,000 person-years. Sympathetic chain PGLs usually arise in the abdomen, with about 75% of them arising intra-abdominally [2]. On CT or MRI, PGLs are usually have homogenous enhancement or central areas of low attenuation. The consequences of PGLs are many, and one complication that is rarely noted is glycemic disturbances with catecholamine excess. Here, we present a case of a radiographically unique appearing PGL that resulted in dramatic improvement of diabetes after resection. Case: 41 year old male with a past medical history of diabetes, CKD and developmental delay presented with hyperglycemia. Patient’s home regimen of Glargine 15 units qHS and Humalog 5 units TID AC had run out a few days prior. Patient had Hemoglobin A1c of 11.8% mg/dL and a C-peptide of 0.4 ng/ml. Endocrine was initially consulted for glucose management. Hospital course was complicated by abdominal pain and CT imaging showed low attenuation splenic masses, retroperitoneal lymphadenopathy and a large, necrotic appearing mesenteric mass consistent with lymphoma. MRI reaffirmed mass with peripheral enhancement and central necrosis consistent with neoplasm. Patient underwent biopsy of lymphadenopathy, which came back benign and then went for biopsy of mass. As the needle entered the mass, the patient became acutely hypertensive and tachycardic. He received phentolamine with good response and labs were drawn for catecholamine producing tumor. Plasma norepinephrine level was 6048 pg/ml and a chromogranin level of 4788 ng/ml. An MIBG scan revealed focal uptake at the lesion without evidence of metastasis. In the subsequent weeks, he was alpha- and then beta-blocked and underwent resection of mass that was complicated by transient hypotension and hypoglycemia. Eventually, patient’s blood pressure was controlled and insulin requirements dropped precipitously to just requiring 4 units of glargine by discharge. Discussion: This patient had a mass on CT scan that appeared to be consistent with lymphoma as opposed to PGL that led to biopsy and subsequent catecholamine crisis. Fortunately, this was controlled and subsequent resection led to improvement in the patient’s glycemic control. It has been thought that glucose intolerance in PGL patients is due to impaired insulin release through desensitization of the beta-adrenergic receptor. Furthermore, Catecholamines, as counter-regulatory hormones, have been well documented to raise blood sugar on this basis as well. Fortunately for our patient, he survived an initial biopsy and post-operatively, was able to actually have an improvement in his glycemic control. It also is a reminder to always do a screen for PGL before doing a needle biopsy of a compatible abdominal mass.

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A390-A391
Author(s):  
Zahrah Alhammad ◽  
Abdulaziz Aljaman ◽  
Najah Younes Douba ◽  
Ali Saleh Alhamdan ◽  
Abdulrahman Aidh Alghamdi

Abstract Introduction: Ketone-prone diabetes (KPD) is an atypical type of diabetes with features from both Type 1 and 2 diabetes, which may lead to its misdiagnosis. Patients usually present with an attack of DKA, but after its resolution, Insulin can be discontinued gradually in most patients with the maintenance of sufficient glycemic control with oral hypoglycemic medications. Clinical Case: A 14 -year-old Saudi male student, presented to the emergency department of our tertiary hospital in November 2017 complaining of nausea and vomiting of two days duration proceeded by a one-week history of generalized fatigue, polydipsia, and polyuria. The patient suffered from long-standing obesity since childhood; otherwise, past medical history was unremarkable. Family history was positive for Type II diabetes in his mother at the age of 48. On examination, the patient was dehydrated, tachypneic, and obese with a BMI of 34 kg/m2 and has patches of acanthosis nigricans over the neck and both axillae. Laboratory investigations showed blood glucose of 455 mg/dL, metabolic acidosis with a pH of 7.22, HCO3 of 11, and a high anion gap of 17, with urine being strongly positive for ketones (++++), HbA1c was 11.6%, and lipid profile showed dyslipidemia. The patient was diagnosed with diabetes ketoacidosis (DKA) and managed with Fluid, Insulin, and potassium infusions according to the Hospital’s DKA protocol. DKA resolved, and the patient was discharged with the impression of ketone prone diabetes based on the presentation of DKA along with features of insulin resistance. Discharge medications were Insulin Glargine 30 units SC once daily, Metformin 500 mg twice daily, and Gliclazide 60 mg Once daily. He was educated on usage and titration of insulin dosage according to glucose readings and monitoring. In subsequent follow-ups as an outpatient, it was noticed that the insulin requirements were gradually declining until it was possible to stop it completely after around six months of follow-up. The patient was transitioned from insulin to GLP-1 agonist (Liraglutide), which helped to achieve significant weight loss. After about one year, Liraglutide (Victoza) was not available in the hospital. However, the patient was controlled on Metformin and Gliclazide. Consequently, the patient has been in remission with adequate glycemic control and had not developed another attack of DKA. Latest review in the clinic in April 2020 showed his BMI of 27.4 kg/m2 and HbA1C of 7.5%. Conclusion: Although it is a rare and atypical type of diabetes, Ketosis Prone diabetes should be kept in mind when a patient presents with DKA but has features of insulin resistance to avoid misdiagnosis and inappropriate management, with proper education to patients and their families on the importance to titrate insulin requirements.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Rebecca Unterborn ◽  
Bankim Bhatt

Abstract Introduction: Pheochromocytoma is a rare neuroendocrine tumor, afflicting around 0.8 out of 100,000 people per year [1]. The “classic triad” of headache, diaphoresis, and tachycardia is well documented in the literature. The “classic triad” in combination with hypertension has up to 91% sensitivity and 93.5% specificity for the diagnosis of pheochromocytoma [2]. Generalized sweating is part of the “classic triad”, however there is paucity of evidence in the literature describing diaphoresis as the only presenting symptom of pheochromocytoma.Case description: A 20-year-old male presented with five year history of worsening hyperhidrosis without any other associated symptoms. Vital signs were blood pressure of 138/82 mmHg and heart rate of 60 bpm. The remainder of the physical exam was normal. Laboratory testing showed 24 hour urine norepinephrine level of 1,002 ug/24 hours (normal: 0-135 ug/24hrs), plasma normetanephrine was 2,873 pg/mL (normal: 0-145 pg/mL) and plasma norepinephrine was 2,869 pg/mL (normal: 0-874 pg/mL). CT abdomen revealed a 4.0 x 3.1 x 4.3 cm left adrenal mass. After appropriate preoperative preparation, he successfully underwent a laparoscopic left adrenalectomy with complete resolution of diaphoresis. Pathology confirmed a completely resected pheochromocytoma.Discussion: This case reflects an atypical presentation of pheochromocytoma; a disease with high cardiovascular morbidity and mortality [3, 4]. The case demonstrates that the absence of hypertension or all three symptoms of the “classic triad” does not exclude the diagnosis of pheochromocytoma and any singular symptom of catecholamine excess warrants an evaluation for pheochromocytoma. This case report helps to establish the need to better quantify individual symptoms of patients with this disease in order to better understand the entire spectrum. Providers need to be aware of atypical presentation of this disease in order to decrease poor outcomes that can result from undiagnosed pheochromocytoma. 1. Beard, C.M., Sheps, S.G., Kurland, L.T., Carney, J.A., Lie, J.T., Occurence of pheochromocytoma in Rochester, Minnesota 1950-1979. Mayo Clinic proceedings, 1983.2. Bravo, E.L., Pheochromocytoma: New concepts and future trends. Kidney International, 1991. 40.3. Lenders, J.W.M., Duh, Q.Y., Young, W.F, et. al, Journal of Clinical Endocrinology and Metabolism. 2014.4. Zelinka, T., Petrak, O., Turkova, H., et. al, High Incidence of Cardiovascular Complications in Pheochromocytoma. Hormone and Metabolic Research, 2012.


2017 ◽  
Vol 1 (3) ◽  
pp. 161-164 ◽  
Author(s):  
Katherine Nolan ◽  
Reema Ishteiwy ◽  
John Alexis ◽  
Martin Zaiac ◽  
Anna Nichols

A 51-year-old female with a history of rheumatoid arthritis was admitted for progressive fevers, chills and malaise. Five weeks prior, she started minocycline for an RA exacerbation. Two weeks after starting minocycline she developed an abscess on her right ankle that was treated at an urgent care facility with ceftriaxone and trimethoprim-sulfamethoxazole. She had minimal improvement so was switched to clindamycin. She developed additional abscesses on her right ankle and right axilla and spiking fevers so she was treated with incision and drainage under general anesthesia. Routine blood work obtained prior to surgery revealed severe neutropenia (0.74 103/ul) and the patient was urgently referred to the emergency department.  Skin biopsy was obtained on admission and revealed ulceration, necrosis, acute and chronic inflammation, vasculitis with vascular thrombosis and rod-shaped bacteria in blood vessel walls and lumina consistent with ecthyma gangrenosum. The following day tissue and blood cultures confirmed the growth of Pseudomonas aureginosa. Bone-marrow biopsy showed decreased granulopoiesis and hematopoiesis, and a diagnosis of minocycline-induced agranulocytosis presenting as ecthyma gangrenosum was made.  The patient had dramatic improvement with appropriate antibiotic therapy, discontinuation of minocycline and initiation of filgrastrim. She has remained healthy without recurrence for 17 months.    


Circulation ◽  
2014 ◽  
Vol 130 (suppl_2) ◽  
Author(s):  
Partha Sardar ◽  
Ramez Nairooz ◽  
Saurav Chatterjee ◽  
Jacob A Udell ◽  
Dharam J Kumbhani ◽  
...  

Introduction: Hyperglycemia is associated with unfavorable prognosis in patients with acute coronary syndrome (ACS). Studies with intensive glycemic control in ACS patients have provided inconsistent results. A meta-analysis was performed to evaluate the effectiveness and safety of intensive glycemic control in patients with ACS. Methods: Search of PubMed, Cochrane CENTRAL, EMBASE, EBSCO, Web of Science and CINAHL databases from their inception through April 2014, identifying randomized controlled trials (RCTs) comparing the effects of intensive versus standard glucose management in patients with ACS. We calculated summary random-effect odds ratios (OR) and 95% confidence intervals (CI). Results: Results from 10 RCTs comprising 2,621 patients were analyzed. All-cause mortality between intensive versus standard glucose management groups did not differ significantly (OR 1.00, 95% CI 0.75-1.34). Similarly, no significant differences were observed between the comparator groups for the odds of cardiac mortality (OR 0.87, 95% CI, 0.67 to 1.12), recurrent myocardial infarction (OR 1.07, 95% CI, 0.76 to 1.52), or stroke (OR 1.20, 95% CI, 0.60 to 2.40). The risk of hypoglycemia (OR 5.95, 95% CI, 2.73 to 12.97; p<0.001) was significantly higher with intensive compared with standard glucose management. Conclusions: Intensive glucose control compared with standard care in ACS patients did not reduce mortality or morbidity, but significantly increased the risk of hypoglycemia. These data from prior clinical trials should be interpreted in the context of their significant methodological limitations.


2017 ◽  
Vol 20 (4) ◽  
pp. 335-339 ◽  
Author(s):  
Brent R Weil ◽  
Alyaa Al-Ibraheemi ◽  
Sara O Vargas ◽  
Shawn J Rangel

Autoamputation of the appendix has previously been reported in the literature, but it is likely an unusual event. We report a 2-year-old male child who had previously undergone laparotomy and bowel resection for necrotizing enterocolitis. Two years later a calcified intra-abdominal mass was identified on abdominal radiography and ultrasonography. Eventual laparotomy revealed a densely calcified mass within the transverse mesocolon. The mass was uneventfully resected. Pathologic evaluation showed appendiceal tissue, consistent with prior autoamputation of the vermiform appendix. Autoamputation of the appendix has not to our knowledge previously been associated with a calcified mass nor been associated with a history of necrotizing enterocolitis, and these factors distinguish this case as noteworthy.


2003 ◽  
Vol 40 (4) ◽  
pp. 251-255 ◽  
Author(s):  
Carlos Augusto Real Martinez ◽  
Rogério Tadeu Palma ◽  
Jaques Waisberg

BACKGROUND: Retroperitoneal lipoma is an extremely rare neoplasm. AIMS: The authors report a case of giant retroperitoneal lipoma in a 32-year-old white female, with a history of pain and an abdominal mass over a 2-year period. Total abdominal ultrasonography and barium enema showed a large mass located in the retroperitoneal space behind the ascending colon. Laparotomy showed a large encapsulated tumor measuring 20 x 13 x 10 cm and weighing 3.400 g. The histological study revealed a benign neoplasm of fatty cells. CONCLUSION: The patient remains well 17 years after surgery, without recurrentce of the disease.


2020 ◽  
Vol 8 (2) ◽  
pp. e001086 ◽  
Author(s):  
Salem Djebala ◽  
Julien Evrard ◽  
Nassim Moula ◽  
Arnaud Sartelet ◽  
Philippe Bossaert

A 19-month-old Belgian Blue heifer was referred to the Veterinary Clinic of Liege University. The heifer was 2 months pregnant by insemination and presented hyperthermia, anorexia and weight loss. Rectal palpation revealed a large, depressible abdominal mass. Diagnosis of parietal fibrinous peritonitis (PFP) was made by ultrasound, revealing a liquid and fibrin filled cavity attached to the abdominal wall. Blood analysis indicated an inflammatory status. Analysis of a PFP fluid sample confirmed bacterial contamination. Treatment, apart from fluids, antibiotics and non-steroidal anti-inflammatory drugs, consisted of surgical drainage and repeated flushing of the cavity. Complete resorption of the cavity was observed after 5 weeks. The heifer remained pregnant but died 7 months later, after elective caesarean section, due to generalised peritonitis. This is the first report of PFP in an animal without a history of laparotomy. The PFP may have been caused by an insemination-induced trauma.


Hypertension ◽  
2000 ◽  
Vol 36 (suppl_1) ◽  
pp. 725-725
Author(s):  
Kazuko Masuo ◽  
Hiroshi Mikami ◽  
Toshio Ogihara ◽  
Michael L Tuck

P180 This study was conducted to clarify the differences in mechanisms between weight reduction (WR) sensitive and insensitive BP reduction, and to evaluate the contribution of family history of obesity (FH) to WR-induced BP reduction. In 61 obese hypertensive men (HT, 28.1±0.9 kg/m2, 35±3 years, 171±6/106±5 mmHg) and 52 obese normotensive men (NT, 27.9±0.6 kg/m2, 34±4 years, 131±5/83±4 mmHg), BMI, BP, fasting plasma norepinephrine (NE), angiotensin II (Ang II), PRA, leptin, insulin were measured every 2 week for 24 weeks with weight loss program (low caloric diet 1000kcal, 7gNaCl + excercise≥1 hr/day). WR and WR sensitive BP reduction were defined as >10% reduction in BMI or mean BP at week 12. 64% of HT and 63% of NT succeeded in WR, and 59% of HT with WR (sensitive vs insensitive P<.05) and 70% of NT with WR (P<.01) were sensitive in BP reduction. When FH+ was defined as at least one parent was obese (BMI>27.0 kg/m2), prevalence of FH+ was higher in 86% of HT and 95% of NT who failed in WR, and higher in 94% of HT and 80% of NT with WR insensitive BP reduction. Only the subjects who succeeded in WR were analyzed in this study. At entry, BP, NE, Ang II, PRA and insulin were higher in HT than in NT, although BMI and leptin were similar. However, the parameters at entry were similar between WR sensitive and insensitive BP reduction in each NT and HT. The decrements (Δ) in BP, NE, Ang II, leptin,insulin were significantly greater in subjects with WR sensitive BP reduction than subjects with WR insensitive BP reduction regardless of BP status during the study, although ΔBMI was similar. Significant decreases in the parameters were noted in earlier period in subjects with WR sensitive BP reduction than in subjects with insensitive BP reduction, and in NT than in HT. In the 4 study groups regardless of BP status or WR induced BP reduction, the decrease in NE preceded BP decline, and the decreases in Ang II, insulin, leptin & PRA followed BP decline with WR. These results suggest that a family history of obesity appears to contribute closely to resistance in weight loss and also to WR insensitive BP reduction. Suppression on sympathetic overactivity is a major mechanism in WR induced BP reduction.


2019 ◽  
Vol 2019 (8) ◽  
Author(s):  
Aghyad K Danial ◽  
Ahmad Al-Mouakeh ◽  
Yaman K Danial ◽  
Ahmad A Nawlo ◽  
Ahmad Khalil ◽  
...  

Abstract Small bowel diaphragm disease is a rare complication related to non-steroidal anti-inflammatory drug (NSAID) use. It presents with non-specific symptoms such as vomiting, abdominal pain, subacute bowel obstruction and occasionally as an acute abdominal condition. We report a case of diaphragm disease in a 33-year-old female who presented with vomiting, constipation and abdominal pain started 5 days earlier. Physical examination revealed palpated abdominal mass. The patient’s past medical history was remarkable for NSAID use. The patient was managed by surgical resection of involved intestine and diagnosis was confirmed by histological examination. Although there are few published cases of diaphragm disease in the medical literature, we recommend that this disease should be considered as one of the differential diagnoses when assessing patients presenting with non-specific abdominal symptoms with remarkable past medical history of NSAID use.


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