INTRODUCTION: Henoch-Schonlein Purpura (HSP) is an IgA mediated systemic, small and medium vessel vasculitis that typically presents with palpable purpura, arthralgias, abdominal pain and hematuria. Gastrointestinal manifestations occur in about 20% of patients prior to skin involvement. We report a case of a 51-year-old woman that presented with HSP duodenitis. CASE DESCRIPTION/METHODS: A 51-year-old female presented with a several week history of nausea, vomiting and epigastric abdominal pain. In addition, she had been experiencing dysphagia, arthralgias, fever and, later on, a palpable, petechial and purpuric rash on her face, ears and all extremities. She had no previous medical history. Physical exam showed an extensive palpable, ecchymotic and purpuric rash involving her arms, face, right ear and purpura on plantar surface of her feet (See Figure 1). Her wrist, elbow, knee and ankle joints were tender to palpation, warm and swollen. She had epigastric tenderness to palpation with no rebound or guarding. Laboratory workup was notable for a white blood cell count of 18.32 M/μL (4.20–5.60 M/μL). Computed tomography of the abdomen showed bowel wall thickening at the junction of duodenum and jejunum with fat stranding in the central mesentery. Biopsy of the skin showed features of leukocytoclastic vasculitis with associated perivascular necrotizing inflammation and hemorrhage (See Figure 2). EGD showed diffuse circumferential erythema with boggy mucosa as well as friability with touch bleeding consistent with ischemic type changes from distal bulb to the second portion of the duodenum (See Figure 3). Biopsies of the duodenum showed evidence of intramucosal hemorrhage, a mixed inflammatory infiltrate of neutrophils, lymphocytes and some plasma cells within the lamina propria. There were also fragments of advanced necrosis with evidence of small vessel thrombosis and vasculitis. Her abdominal pain and oral intake improved after treatment with 1 g methylprednisone for three days. The patient was discharged from the hospital with an oral steroid taper. DISCUSSION: HSP is rare in adults with roughly 20-fold fewer cases than in children. The range of endoscopic findings includes gastritis, ulceration, duodenitis, and submucosal hemorrhage with the second portion of the duodenum being involved more than the duodenal bulb. The end capillaries of small intestinal villi are particularly susceptible to ischemic injury. HSP is a self-limiting disease and majority of the patients improve spontaneously.
Background: Immune checkpoint inhibitors have changed the therapeutic milieu for patients with metastatic melanoma. However, their use may promote autoimmunity in virtually any organ in the body due to the blockade of intrinsic immune down regulators such as cytotoxic T-lymphocyte antigen- 4 (CTLA-4), programmed cell death 1 (PD1) or its ligand (PDL1). Immune mediated adverse neurological events are rare with these agents, however, and are seen in < 1% of treated patients. We report a patient with immune checkpoint inhibitor associated autoimmune encephalitis, with complete clinical recovery after treatment.Case Report: A 49-year-old female with metastatic melanoma currently on nivolumab therapy but recently on ipilimumab/nivolumab combined therapy presented with a new headache. She also reported associated confusion, loss of balance, personality changes and language difficulty. Magnetic resonance imaging of the brain did not reveal any evidence of metastasis, infarct, meningitis, or encephalitis. Lumbar puncture revealed an elevated protein level and pleocytosis, with a normal glucose level. She was started on empiric glucocorticoid therapy with a presumptive diagnosis of immune checkpoint inhibitor associated autoimmune encephalitis. She improved considerably by day 3 of treatment with complete resolution of neurological symptoms by day 5.Conclusion: Immune checkpoint inhibitors are increasingly important in cancer immunotherapy as they can cause sustained remissions in patients with metastatic melanoma and other malignancies. Because these drugs block immune-regulatory targets, they can lead to enhanced activation of immune system resulting in immune-related adverse events. Autoimmune encephalitis is a rare immune-related adverse event associated with immune checkpoint inhibitors. The incidence of autoimmune encephalitis is higher with combination or sequential CTLA-4 (ipilimumab) and PD1(nivolumab) inhibitor therapy than with monotherapy. With more widespread use of immunotherapy, it is important for clinicians to be aware of this rare and reversible cause of encephalitis. Early recognition and prompt initiation of immunosuppressive therapy with glucocorticoids is essential to enhance neurological recovery.
Olmesartan, an angiotensin II receptor blocker, is widely used for the treatment of hypertension. An association of olmesartan use with a reversible, sprue-like enteropathy has recently been recognized. There are also rare reports of olmesartan use and the combination of sprue-like enteropathy and collagenous colitis. We report a case of an elderly female who presented with both severe collagenous sprue and collagenous colitis associated with the use of olmesartan, with complete clinical and histologic recovery after discontinuation of the drug.
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