Objective: We present a case of a 45-year-old woman with history of Graves disease treated with thyroidectomy who developed panhypopituitarism due to granulomatous hypophysitis.Methods: The details of the case presentation, evaluation, diagnosis, and treatment are reviewed. On routine follow-up visits for thyroid condition, the patient complained of symptoms of hyperthyroidism, despite being on a previously adequate dose of levothyroxine. Work-up led to the diagnosis of hypophysitis.Results: The patient responded to corticosteroid therapy, but 5 months later, magnetic resonance imaging of the brain showed recurrence of the disease. Eventually, the patient underwent partial transsphenoidal hypophysectomy, later developed recurrence of disease, and had total hypophysectomy. The postoperative pathology report was consistent of granulomatous hypophysitis.Conclusion: Granulomatous hypophysitis is an uncommon inflammatory disease affecting the pituitary. It is commonly misdiagnosed as a macroadenoma because of its clinical presentation. (AACE Clinical Case Rep. 2018;4:e329-e333) Abbreviations: FT4 = free thyroxine; GH = granulomatous hypophysitis; LH = lymphocytic hypophysitis; MRI = magnetic resonance imaging; TSH = thyroid-stimulating hormone CASE REPORTHypophysitis is a rare inflammatory process of the pituitary gland that mimics pituitary macroadenomas. It can be idiopathic or manifest as a part of a systemic autoimmune disease. Granulomatous hypophysitis (GH) and lymphocytic hypophysitis (LH) are the most common subtypes reported in literature (1-3). We present a case of a 45-year-old female with past medical history of hypothyroidism after total thyroidectomy for Graves disease and surgical history of hysterectomy. On presentation, she complained of hyperthyroid symptoms (e.g., fatigue, excess sweating, hot flashes, presyncope, weight loss, and headaches), despite being on a previously adequate levothyroxine dose. Two months prior, her thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were normal. Her physical exam was unremarkable except for a surgically absent thyroid. Laboratory evaluation revealed TSH <0.006 mU/L (normal range, 0.34 to 5.6 mU/L) and FT4 of 1.1 ng/dL (normal range, 0.58 to 1.64 ng/dL); thus, her levothyroxine dose was decreased. Despite this, she continued to have symptoms and low TSH. Secondary hypothyroidism was suspected, as TSH was undetectable and FT4 0.47 ng/dL off levothyroxine. Severe headache, insomnia, and worsening fatigue developed, so imaging was done and magnetic resonance imaging (MRI) revealed a large bilobed enhancing pituitary mass with suprasellar extension and thickening of the pituitary stalk (Fig. 1). Laboratory results showed low am cortisol, estrogen, follicle-stimulating hormone (FSH), insulin-like growth factor See accompanying article, p. e350.
Introduction: Hemolytic uremic syndrome (HUS) is a thrombotic microangiopathy defined by hemolytic anemia, thrombocytopenia, and acute kidney injury. HUS can be categorized as typical in the presence of Shiga toxin-producing Escherichia coli, or atypical (aHUS). aHUS is caused by dysregulation of complement pathways from inherited or acquired abnormalities in complement proteins, or secondary to systemic disease. aHUS is exceptionally rare, with an incidence of 1 in 1,000,000. aHUS secondary to pancreatitis has been reported in few cases in literature, and the underlying pathogenic mechanism has yet to be elucidated. Pancreatitis has also been described as a rare extrarenal manifestation of aHUS. It is difficult to differentiate between the two, but genetic evaluation can provide some insight. We present a rare case of secondary aHUS from pancreatitis with negative genetic workup. Case Description/Methods: A 35-year-old male with no medical history was brought in by ambulance after being found unresponsive. Initial labs revealed a glucose of 1375mg/dL, Creatinine 2.25mg/dL, lipase 1636U/L, hemoglobin 19.8g/dL, sodium 162mmol/L, and platelets 388x10 9 /L. CT abdomen showed signs of pancreatitis. Two days later, his platelets dropped to 65x10 9 /L. Further lab workup included low haptoglobin, elevated bilirubin, and schistocytes on peripheral smear. Coombs' test was negative. He developed anasarca and oliguria with creatinine increase to 11.7mg/dL and hemodialysis was initiated. Treatment with pulse dose steroids and plasmapheresis was started. ADAMTS13 levels returned normal, and Shiga toxin was negative. He was then started on eculizumab and evaluated for genetic complement mutations which returned negative, supporting secondary aHUS. Eculizumab was continued with return of renal function. Discussion: Direct damage on the vascular endothelium from drugs, autoimmune diseases, infections, and cancer are well-known causes of secondary aHUS. aHUS secondary to pancreatitis has been described few times in literature. The underlying mechanism in the setting of acute pancreatitis remains elusive but may be related to release of activated enzymes causing endothelial damage and subsequent activation of proinflammatory cascades, leading to complement dysregulation and end-organ damage. The introduction of eculizumab, a C5 complement inhibitor, is now considered first line therapy and can prevent multiorgan damage. This case aims to provide more information in the diagnosis and management of this rare disease.
Figure 1. a. Early filling of dilated mesenteric veins (bottom arrow) with adjacent bowel wall edema due to venous congestion (top arrow) b & c. inferior mesenteric artery (arrows) adjacent to illdefined area of contrast (dashed arrow) thought to represent arterio-venous fistula d. view from sigmoidoscopy showing ischemic colitis in the sigmoid colon.
Merkel cell carcinoma (MCC) is a rare, rapidly growing, and aggressive dermatological neoplasm. It is commonly reported in Caucasian ethnicities, and almost 50% of the patients have a concomitant malignancy and are on immunosuppressive chemotherapy. Here, we present a 79-year-old woman with a history of relapsed Stage II, grade III follicular lymphoma, receiving maintenance rituximab infusions. She presented with a raised erythematous papule on her left cheek. An excisional biopsy of the lesion confirmed a diagnosis of Merkel cell carcinoma. After which, she underwent a wider excision with 1-2 cm margins. PET scan did not reveal any FDG-avid uptake lesions that would be concerning for metastatic disease. However, she underwent a sentinel lymph node biopsy which was also negative. Thus, the diagnosis was finalized as Stage I (T1 N0 M0) MCC. There are only two reported cases in literature about the significant progression of Merkel cell carcinoma in patients who coincidentally were receiving rituximab as a part of treatment for another disease. This raises questions for future investigation and research on whether there is a direct association between rituximab use specifically and the rapid growth of MCC.
e16212 Background: Portal vein thrombosis (PVT) occurs in 10-40% of patients with hepatocellular carcinoma (HCC). Advanced imaging modalities, such as MRI, can differentiate between bland PVT and tumor thrombus, and help determine the need for anticoagulation (AC). Our study aims to assess the use of AC in patients with HCC and concomitant PVT. Methods: We conducted a retrospective observational study at eight hospitals in a community healthcare system in southeast Michigan. Patients with a combined diagnosis of HCC and PVT were identified between August 2016 and September 2022. Demographics, clinical characteristics, use of AC, type of thrombus, and median survival were collected through chart review. Results: A total of 122 patients were diagnosed with HCC and PVT (17 patients were lost to follow-up). The median age at diagnosis was 72.7 years and 77.1% of patients were male. Majority (66.6%) of patients were white. Underlying cirrhosis was present in 85.7% of patients, of which 41% was due to HCV infection. Around half of the patients had a Child-Pugh Class-B at diagnosis. AC was started in 46% of patients, with apixaban being the most commonly used agent (56%). Imaging results in the AC group confirmed that 52% of patients had a tumor thrombus. Of all the patients started on AC, 25% had bleeding complications. Overall, death occurred in 52.1% of patients in the AC group compared to 57.9% of patients who did not receive AC. Median survival was 6 months in both groups (p=0.7). Using cox-proportional hazards model, there was no difference observed in overall survival between patients who received AC and patients who did not (HR 0.91, 95% CI 0.54-1.53, p=0.72). Conclusions: In this retrospective review of patients with HCC and PVT, no difference in survival was observed between patients who received AC and patients who did not receive AC. Further randomized prospective data are needed to guide management in this high-risk patient group. [Table: see text]
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