New-onset primary adrenal insufficiency in pregnancy associated with a unilateral adrenal infarction: a case report.

Summary: New-onset primary adrenal insufficiency is rare in pregnancy. The symptoms of adrenal insufficiency such as nausea, vomiting and dizziness may be attributed to the pregnancy itself, which can lead to a delay in the diagnosis. The presence of hypotension, hypoglycemia or hyperkalemia should raise the suspicion for adrenal insufficiency. We report the case of a 25-year-old woman who presented with tachycardia, left flank pain and vomiting at 36 weeks' gestation. She was found to have primary adrenal insufficiency and started on hydrocortisone and fludrocortisone with resolution of the vomiting and tachycardia. MRI of the abdomen revealed an acute nonhemorrhagic infarct of the left adrenal gland. The contralateral adrenal gland was normal. Autoimmune and infectious etiologies of primary adrenal insufficiency were ruled out and the adrenal insufficiency was attributed to the unilateral adrenal infarction. Adrenal insufficiency persisted after delivery and then resolved at approximately 16 months post partum. This case highlights the need to test women with unilateral adrenal infarction in pregnancy for the presence of primary adrenal insufficiency. Learning points: Adrenal insufficiency should be considered when a pregnant woman develops nausea, vomiting and dizziness in association with hypotension or hypoglycemia. Hypovolemic hyponatremia related to vomiting can occur in pregnancy, but the failure to correct hyponatremia despite adequate IV hydration should raise the suspicion for adrenal insufficiency. Adrenal infarction should be in the differential diagnosis for unilateral flank pain in pregnancy. Other common etiologies for flank pain in pregnancy include nephrolithiasis, pyelonephritis and acute cholecystitis. Unilateral adrenal infarction in pregnancy can lead to the development of primary adrenal insufficiency. Following delivery, these patients need to be monitored for the resolution of the adrenal insufficiency.

Ectopic Adrenocorticotropic Hormone-Producing Metastatic Gastrinoma: A Case Report.

Pancreatic neuroendocrine tumors secreting gastrin and adrenocorticotropic hormone (ACTH) are rare. The presentation of the cases can be varied, making the diagnosis challenging and often delayed. Here, we present a patient who presented with severe hypokalemia and was found to have ectopic Cushing's syndrome. An abdominal CT scan showed a pancreatic lesion with metastatic liver disease. A biopsy of the liver lesion confirmed a metastatic neuroendocrine tumor. The final diagnosis was ectopic ACTH-producing metastatic gastrinoma. Twenty-four-hour urinary cortisol was significantly elevated at 9,790 mcg/24 hours. The excess hormonal secretion was successfully treated with ketoconazole and somatostatin analogs. She was further started on chemotherapy with capecitabine plus temozolomide, which has become the preferred chemotherapy treatment after the results of the recently completed trial. She also received Y90 therapy for metastatic liver disease. The prognosis of metastatic pancreatic neuroendocrine tumors is poor. Multidisciplinary combined therapies can help control disease and improve prognosis. We present an 18-month-long patient follow-up and a literature review of ectopic ACTH-producing metastatic gastrinomas.

A New Diagnosis of Autoimmune Addison's Disease in an 80-Year-Old Female.

Hyponatremia is a common cause of hospitalization in older adults. Addison's disease (AD), an uncommon cause of hyponatremia, is primary adrenal insufficiency (AI) caused by autoimmune adrenalitis, infections, adrenal hemorrhage, or infiltration. Autoimmune adrenalitis is an uncommon cause of AD after age 60 years. We report the case of an 80-year-old female with steroid-controlled asthma, who was diagnosed with primary AI after presenting with weakness, nausea, vomiting, and hyponatremia two weeks after a urinary tract infection (UTI). Further testing revealed a positive 21 hydroxylase antibody confirming autoimmune AD. The patient has done well on hydrocortisone and fludrocortisone daily with occasional need for stress dosing during infections. AD should be considered as a cause of hyponatremia in hospitalized elderly patients. Non-specificity of symptoms of AD makes the diagnosis difficult in this age group, often causing delays in the appropriate management. Our patient's autoimmune AD was masked by intermittent steroid use over the years. During the COVID-19 pandemic, masking and social distancing decreased her steroid needs and unmasked AD after the UTI. We highlight the importance of considering AD as a cause of hyponatremia and autoimmune AD as a cause of AI even in the elderly.

Diabetic Ketoacidosis in a Patient With Type I Diabetes Treated With a Closed-Loop Sensor-Augmented Insulin Infusion System.

Background/Objective: Closed-loop insulin infusion systems (CLSs) such as Tandem t:slim with Control-IQ (t:slim CIQ) improve glycemic control and decrease diabetic ketoacidosis (DKA) risk in type 1 diabetes mellitus (T1DM). We report a case of CLS failure, likely from tirzepatide-induced volume depletion, leading to DKA. Case Report: A 36-year-old woman with T1DM on t:slim CIQ CLS was prescribed tirzepatide for weight loss. Three months later, 4 days after the last tirzepatide injection, she presented with worsening nausea, vomiting, 50-lbs weight loss, minimal oral intake for 3 days, and positive urine ketone result. Her heart rate was 137 beats/min and respiratory rate was 35 breaths/min, and she had Kussmaul breathing, with dry oral mucosa indicating volume depletion. Laboratory examination showed a fingerstick glucose level of 289 mg/dL, serum glucose level of 322 mg/dL, bicarbonate level of 12 mmol/L, and anion gap of 21 mmol/L confirming high-anion-gap metabolic acidosis, suggesting DKA. A concurrent continuous glucose monitor (CGM) reading was 40 mg/dL. The CLS and CGM were removed. DKA resolved within 72 hours (serum glucose level of 143 mg/dL, anion gap of 8 mmol/L, bicarbonate level of 24 mmol/L) on intravenous insulin and fluids. The CLS and CGM were restarted with good glycemic control. Tirzepatide was discontinued to avoid future episodes of volume depletion. Discussion: Volume depletion affects interstitial fluid glucose levels due to compensatory mechanisms. This may result in CLS failure due to CGM dependence on interstitial glucose measurements, precipitating DKA. Conclusion: Patients on CLS therapy should be cautioned against CLS failure in volume-depleted states with interstitial glucose-level changes. A back-up plan with multiple daily insulin injections should be discussed.

A Case Report of Rare Synchronous Esophageal Malignancies With Dissimilar Histology: Squamous Cell Carcinoma and Small Cell Carcinoma.

The presence of synchronous primary malignancies is a rare phenomenon reported in the literature. Most synchronous malignancies reported include carcinomas and adenocarcinomas of the gastrointestinal tract, head and neck cancers, thyroid and breast cancers. Among the neuroendocrine tumors, carcinoid tumors in the duodenum or the esophagus are most commonly reported with other primary malignancies. We report the case of a 56-year-old male with tobacco use disorder, presenting with dysphagia and weight loss for six months, who was thought to have multicentric squamous cell carcinoma of the esophagus. In actuality, he was diagnosed with synchronous metastatic neuroendocrine tumor (NET) favoring small cell carcinoma and squamous cell carcinoma of the esophagus. The patient responded well to minimally invasive thoracoscopic esophagectomy with regional lymphadenectomy followed by chemotherapy and radiation therapy. We have not been able to find a literature referencing the presence of synchronous small cell carcinoma and squamous cell carcinoma of the esophagus, making our case unique.

Factors Affecting the Job Satisfaction of U.S. Adult Endocrinologists: A Nationwide Survey.

OBJECTIVE: Despite the increased demand and worsening burnout among U.S. endocrinologists, there is a paucity of data on job satisfaction and associated factors. This study examines the factors associated with job satisfaction among a nationally representative sample of U.S. endocrinologists. METHODS: We conducted a cross-sectional survey of 1700 U.S. adult endocrinologists on the Facebook group "Endocrinologists." The survey was conducted over 4 weeks using an anonymous online questionnaire. The 45-question survey assessed job and salary satisfaction scores on a 5-point Likert scale along with multiple job-related variables. Univariate and multivariate analyses were conducted to identify the factors affecting job satisfaction. RESULTS: Out of 1700, 654 adult endocrinologists (504 women and 139 men) completed the survey. The mean job satisfaction score was 3.72 ± 0.86, with 67.5% having high job satisfaction. Comparatively, 339 (52.1%) had high salary satisfaction. There was a statistically significant relationship between the job and salary satisfaction scores (P < .01). Factors significantly associated with the job satisfaction score (P < .05) included the practice region, gender, number of medical assistants per endocrinologist, self-performance of thyroid ultrasound, and number of patients in the hospital per week. Multivariate analysis showed that full-time employment, along with high salary satisfaction, seeing fewer new patients per day, performing thyroid ultrasounds, and fewer patients in the hospital were associated with the highest job satisfaction. CONCLUSION: This study found about one-third of endocrinologists to have lower job satisfaction and identified multiple modifiable factors associated with endocrinologists' job satisfaction. Interventions focused on these potentially modifiable factors may improve job satisfaction among U.S. endocrinologists.

Use of Lithium in Hyperthyroidism Secondary to Graves' Disease: A Case Report.

BACKGROUND The therapeutic approach to Graves' disease (GD) comprises thionamides, radioiodine ablation, or surgery as first-line therapy, and cholestyramine and oral iodine as second-line therapies. The role of lithium (Li) in GD as a primary or adjunctive therapy remains contentious. We present a case of GD managed by Li therapy with oral iodine solution. CASE REPORT A 26-year-old man, admitted with acute blast crisis secondary to chronic myeloid leukemia (CML), reported palpitations, 40-lb weight loss, heat intolerance, and fatigue. An examination revealed sinus tachycardia, elevated body temperature, and thyromegaly. Laboratory evaluation confirmed hyperthyroidism (TSH <0.005 mcIU/l, FT4 5.57 ng/dl, TT3 629 ng/dl) secondary to GD (TRAb >40 IU/l, TSIg 178%). Thionamides and surgery were contraindicated due to pancytopenia from a blast crisis. Inability to maintain post-radiation precautions precluded use of RAI. Cholestyramine was attempted and discontinued due to nausea. We introduced oral Li carbonate with oral iodine, which the patient tolerated. Thyroid functions improved with therapy (TSH 0.007 mcIU/l, FT4 0.82 ng/dl, TT3 122 ng/dl) with stable Li level (0.5-0.8 mmol/l). CONCLUSIONS Li inhibits iodine uptake through interference with sodium-iodide symporter and tyrosine iodination, thyroglobulin structure changes, peripheral deiodinase blockage, and preventing TSH and TSIg stimulation. Our case shows that a low therapeutic level of Li, in combination with oral iodine, can suppress thyroid overactivity without adverse effects. We suggest that low-dose Li carbonate is a safe and effective adjunctive antithyroid medication to be considered if primary therapies for hyperthyroidism are unavailable.

The Unlikely Suspect: A Case Report of New-Onset Hyperthyroidism Due to Graves' Disease in an 89-Year-Old Gentleman and Review of Literature.

Thyroid dysfunction in the elderly commonly manifests as hypothyroidism. With advancing age, toxic nodules are the more common cause of hyperthyroidism as compared to Graves' disease. Due to the lack of classical symptoms of hyperthyroidism in the elderly, the diagnosis can often be delayed. Previously, an 82-year-old gentleman with oropharyngeal dysphagia due to Graves' disease was the oldest reported case with atypical symptoms. We report a case of an 89-year-old gentleman with no prior history of thyroid disease, who presented with non-specific gastrointestinal symptoms that ultimately led to the diagnosis of hyperthyroidism secondary to Graves' disease. We also review the available literature regarding the pathophysiology, clinical presentation, and management of hyperthyroidism and Graves' disease in the elderly.

An Unusual Presentation of Lichen Planus.

Lichen planus is a chronic papulosquamous eruption of the skin, scalp, nails, and mucous membranes. "Pruritic, purple, polygonal, planar, papules, plaques" are the traditional six "P's" of lichen planus. We describe an unusual case of lichen planus presenting as cellulitis. A 64-year-old lady with a past medical history of pyoderma gangrenosum, inclusion body myositis, and chronic kidney disease presented with a two-week history of swelling, erythema, tenderness, hyperkeratotic plaques, and blisters on the medial aspect of both thighs. She had a previous history of pyoderma gangrenosum exacerbations with similar presentations; however, current lesions were different from prior presentations. We considered the differential diagnoses of bacterial cellulitis versus pyoderma gangrenosum exacerbation. Due to the difference in these lesions from previous episodes, the patient was empirically treated for bacterial cellulitis with intravenous cefepime and linezolid. The infectious diseases team was consulted and valacyclovir was added to cover for possible herpes infection, with no improvement in symptomatology. Dermatology was then consulted, and a clinical diagnosis of psoriasiform dermatitis was made. A skin biopsy was obtained and the patient was started on prednisone. There was an immediate improvement in the papules within 24 hours. The papules cleared, leaving behind violaceous flat plaques, clinically diagnosed as lichen planus. The affected area was shrinking as compared to previous examinations. The skin biopsy was reported as chronic psoriasiform dermatitis with the main differential of lichen planus. The patient was discharged home on a tapering dose of oral prednisone, topical clobetasol, and oral moxifloxacin. This case demonstrates the importance of familiarity with rare clinical subtypes as a suspicion for lichen planus. The vesiculobullous subtype of lichen planus, as seen in this patient, tends to present as blisters and cellulitis from infection of the bullae. Treatment of the infection alone is not enough and steroids are essential. This knowledge helps change management, allows for earlier improvement and better patient outcomes.

Small Bowel Obstruction Due to Metastatic Urachal Adenocarcinoma: A Rare Presentation.

Urachal adenocarcinoma is a rare but highly malignant epithelial cancer that accounts for <1% of all bladder malignancies and commonly presents with hematuria. We report a case of metastatic urachal adenocarcinoma presenting as bowel obstruction. A 54-year-old male patient with a history of alcohol abuse presented to the emergency with acute-onset, diffuse, cramping abdominal pain, worst in the epigastrium and lasting one day. Abdominal examination revealed moderate guarding and generalized tenderness with hypoactive bowel sounds. Imaging confirmed an evolving small bowel obstruction and a urachal remnant with a superimposed mass lesion. The patient underwent an exploratory laparotomy and a high-grade small bowel obstruction due to the mass was identified. An intraoperative frozen section identified adenocarcinoma. A biopsy of the urachal mass confirmed urachal adenocarcinoma. The final diagnosis was moderately differentiated urachal adenocarcinoma. The tumor was deemed unresectable due to the involvement of multiple loops of the small bowel and the mesentery of the small and large bowels. Systemic chemotherapy with 5-fluorouracil (5-FU), leucovorin, and oxaliplatin (modified FOLFOX-6) was initiated. Our patient did not report any prior urinary symptoms or recurrent abdominal pain, which are the common symptoms that urachal adenocarcinoma presents with. Bowel obstruction is a rare presentation of urachal adenocarcinoma since the spread of the disease to the viscera occurs much later in the course. This case report highlights a rare presentation of an even rarer malignancy.

Outcomes of Frontalis Sling Versus Levator Resection in Patients With Monocular Elevation Deficiency Associated Ptosis.

PURPOSE: To compare outcomes of frontalis sling (FS) silicone and levator resection (LR) in ptosis associated with monocular elevation deficiency. METHODS: Retrospective interventional comparative case series of FS and LR in monocular elevation deficiency associated ptosis. Favorable outcome was defined as difference in margin reflex distance 1 of ≤1 mm between the 2 eyes in unilateral cases and margin reflex distance 1 of 4 mm in bilateral cases at last follow-up visit. RESULTS: One hundred four eyes of 95 patients were included. Median age at surgery was 14 years, and the mean follow-up period was 19.75 ± 34.55 months. Ptosis was severe in 91 (87.5%) patients. Associated Marcus Gunn jaw-winking (MGJW) phenomenon was seen in 43 (42%) patients. Frontalis sling was performed in 76 (73%) and LR in 28 (27%). Mean pre- and postoperative margin reflex distance 1 were -1.27 ± 2.17 mm and 2.18 ± 1.49 mm (p < 0.0001). The mean improvement in margin reflex distance 1 was significantly more with FS (4.46 ± 2.19) compared with LR (1.85 + 2.5) (p < 0.0001). There were no cases of exposure keratopathy requiring reversal of surgery in either group. The number of resurgeries required was 42 (55%) in the FS group and 10 (36%) (p = 0.08) in the LR group. Favorable outcome was seen in 54 (71%) in FS group and 16 (57%) (p = 0.17) in LR group. CONCLUSIONS: When compared with levator resection, frontalis suspension with silicone gives a better eyelid elevation but has greater regression requiring more resurgeries. In spite of a poor Bells phenomenon, exposure keratopathy is not a concern.

Endophthalmitis and Concurrent or Delayed-Onset Rhegmatogenous Retinal Detachment Managed With Pars Plana Vitrectomy, Intravitreal Antibiotics, and Silicone Oil.

BACKGROUND AND OBJECTIVE: The current study describes the treatment outcomes in patients with endophthalmitis and concurrent or delayed-onset retinal detachment managed with pars plana vitrectomy, intravitreal antibiotics, and silicone oil. PATIENTS AND METHODS: In this noncomparative, retrospective case series, the medical records of patients diagnosed with endophthalmitis and retinal detachment from January 1991 through December 2014 at a tertiary eye care center in South India were reviewed. All patients received silicone oil for the management of retinal detachment either concurrently or during follow-up treatment. RESULTS: A total of 93 patients were included in the current study. Retinal detachment was diagnosed at presentation in 20 of 93 patients (21.5%) (concurrent group: Group 1) and during follow-up in the remaining 73 of 93 patients (78.5%) (delayed-onset group: Group 2). In Group 1, the initial treatment consisted of vitrectomy, intravitreal antibiotics, and silicone oil injection in 19 of 20 patients. In Group 2, patients did not receive silicone oil during initial treatment but underwent silicone oil injection during subsequent surgery for repair of retinal detachment. Rates of complete retinal reattachment and visual acuity of 20/400 or better were 73.7% and 30.0%, respectively, in Group 1 and 98.5% and 39.7%, respectively, in Group 2. The median visual acuity at last follow-up in 44 eyes undergoing silicone oil removal was 20/100 (logMAR 0.7), whereas in the remaining 49 eyes that did not undergo silicone oil removal, median visual acuity was 20/2000 (logMAR 2.0). CONCLUSION: In these patients with endophthalmitis with concurrent or delayed-onset retinal detachment, the use of silicone oil can be a useful adjunct. [Ophthalmic Surg Lasers Imaging Retina. 2017;48:546-551.].

Clinical presentations, risk factors and outcomes of ceftazidime-resistant Gram-negative endophthalmitis.

BACKGROUND: To describe the clinical features and outcomes of patients diagnosed with ceftazidime-resistant Gram-negative endophthalmitis and the role of intravitreal imipenem in these cases. DESIGN: Retrospective consecutive interventional case series at a tertiary eye care centre in South India. PARTICIPANTS: Consecutive cases of ceftazidime-resistant Gram-negative endophthalmitis from April 2010 to December 2014. Fifty-six cases diagnosed during this time period were included. METHODS: All cases were managed with vitreous biopsy/vitrectomy, microscopy and undiluted vitreous culture, antimicrobial susceptibility of bacterial isolates and received intravitreal antibiotics. MAIN OUTCOME MEASURES: Anatomic and visual outcome of these cases, antimicrobial susceptibility pattern of intravitreal imipenem and outcome of cases injected with it. RESULTS: Commonest presentation was acute endophthalmitis following cataract surgery (27 eyes, 48.21%). Pseudomonas aeruginosa was isolated in 33 eyes (58.93%; 95% CI 46.05-71.81%). Nineteen eyes (34%; 95% CI 21.59-46.41%) developed phthisis; 14 eyes (25%; 95% CI 13.66-36.34%) had vision <20/200; 17 eyes (30.35%; 95% CI 18.31-42.39%) eyes had an ambulatory vision >20/200 (logMAR 1); 6 eyes (10.71%; 95% CI 2.61-18.81%) had a reading vision >20/40 (logMAR 0.3). Trend was towards better anatomic (72.73% vs. 40%) (P = 0.05) and visual improvement in the imipenem group (logMAR 3.94 + 0.21 to 2.43 + 1.4; P = 0.002), as compared with non-imipenem group (logMAR 2.99 + 1.3 to 2.55 + 1.4; P = 0.13). CONCLUSIONS: Outcome of ceftazidime-resistant Gram-negative endophthalmitis is poor. P. aeruginosa is the commonest isolated organism. All cases were sensitive to imipenem. There was a trend towards better anatomic outcome in imipenem-treated eyes.

Endophthalmitis After Pars Plana Vitrectomy: Clinical Features, Risk Factors, and Management Outcomes.

PURPOSE: To report the clinical presentations, risk factors, and outcomes of endophthalmitis after pars plana vitrectomy at 4 tertiary eye care centers of an institute in South India. DESIGN: A retrospective case series. METHODS: The records of 38,591 patients undergoing vitrectomy were reviewed using the coding assigned by the medical records department. Consecutive cases diagnosed as endophthalmitis after pars plana vitrectomy (PPV) between 1990 and 2014 for various indications were analyzed. RESULTS: The clinical incidence of postvitrectomy endophthalmitis was 0.052%, and culture-positive incidence of postvitrectomy endophthalmitis was 0.031%. Twelve cases (60%) were culture positive. Mean presenting vision was 2.16 ± 1.51 logMAR (Snellen equivalent 20/2890). Seventeen eyes had received sutureless vitreous surgery (15 cases 23G, 2 cases 25G) and 3 eyes had received 20G suture-assisted vitreous surgery (P < 0.0001). The odds of developing endophthalmitis in sutureless versus sutured vitrectomy were 25.14 [95% confidence interval (CI), 7.37-85.84] (P < 0.0001) and those of developing endophthalmitis in sutureless surgery versus sutured with final tamponade of Ringer lactate (RL) were 19.53 (95% CI, 5.37-71.03) (P < 0.0001). In sutureless surgeries, the odds of developing endophthalmitis in RL tamponaded eyes versus non-RL ones was 4.39 (95% CI, 1.67-11.56) (P = 0.002). Mean interval between vitreous surgery and endophthalmitis was 4 ± 6.89 days; median, 1.5 days. Mean postoperative vision was 1.7 ± 1.36 logMAR (Snellen equivalent 20/1002) (P = 0.31). CONCLUSIONS: Endophthalmitis after vitrectomy is an acute presentation. Sutureless surgery, especially with aqueous tamponade, has a higher risk. The visual outcome is relatively poor.