RESUMEN
Introduction: Lichen planus is a relatively common inflammatory condition of the nails, skin, and mucosal surfaces. Oesophageal involvement of lichen planus is thought to be very rare, mainly described in case reports, but is associated with a high risk of oesophageal stenosis as well as squamous cell carcinoma. No evidence-based treatment recommendations exist, with the majority of described treatment regimens involving systemic immunosuppression. Case Report: In this case report, we describe a novel approach in treating oesophageal lichen planus in a patient with budesonide orodispersible tablets, a treatment normally reserved for eosinophilic oesophagitis. The patient achieved complete relief of dysphagia, with a follow-up oesophagogastroduodenoscopy 2 months after treatment commencement being macroscopically and microscopically free of inflammatory activity. This case report is to our knowledge the first to report this treatment regimen in oesophageal lichen planus. Conclusion: We consider a trial of budesonide orodispersible tablets a reasonable initial management as it's a local therapy specific to the oesophagus with a more benign side effect profile than systemic immunosuppression, but further studies need to be undertaken to corroborate our findings. Also, based on the severity and malignant potential of oesophageal lichen planus, we suggest that physicians be liberal in ordering oesophagogastroduodenoscopy with biopsy taking as part of the workup of dysphagia in a patient with known lichen planus.
RESUMEN
Background: Hydrophilic polymer gel coatings are used on different intravascular devices to prevent vasospasm and thrombosis. However, it may become dislodged from these devices, leading to ischaemic complications in various organs including the skin, kidneys, brain, heart or lungs. Hydrophilic polymer embolisation (HPE) is a rare complication following endovascular procedures that is currently not fully recognised. The current knowledge of this phenomenon is based on reports consisting of histologic evidence of foreign polymers in the affected organ. Case description: A 76-year-old male with a history of hypertension, type 2 diabetes, renal cell carcinoma and chronic kidney disease underwent endovascular stenting of the superficial femoral artery due to critical limb ischaemia of the right foot. The patient had an acute kidney injury following the procedure. Upon examining the legs, there were tender non-blanching macular lesions on the right lower limb. A skin biopsy of the lesion was performed and showed hydrophilic polymer embolisation. Unfortunately, a few weeks later the patient was readmitted due to a worsening of the right foot wound situation, which required below-knee amputation. Conclusion: HPE is a rarely reported complication after endovascular interventions, with the potential to embolise to multiple organs. By observing skin manifestations, it is possible to aid the early detection of ischaemic events in other organs and identify their underlying causes. Generally speaking, the course is benign and self-limiting when the skin is involved, but may be more sinister especially when other organs (e.g. brain) are involved. LEARNING POINTS: Meticulous skin examination is an essential part of the diagnostic armamentarium of an internist, aiding in formulating a differential diagnosis based on such findings.Hydrophilic polymer embolisation (HPE) is a rarely reported complication after endovascular procedures.Given the potentially attenuated histological findings of HPE, history details of recent endovascular intervention are key to the diagnosis.
RESUMEN
Immunoglobulin G4-related disease (IgG4-RD), which was initially identified as a type of autoimmune pancreatitis around the year 2000, is now widely acknowledged to be a systemic sickness. Based on both general and organ-specific criteria, alongside laboratory measurements of IgG4-subtype, the diagnosis is made. The diagnosis requires, however, a heightened index of suspicion, especially given the nonspecific clinical presentation. In addition to this, the symptoms may be "disseminated" in time and the multitude of organ-system involvement may seem initially unrelated. Furthermore, IgG4 levels may be falsely normal especially during the first presentation of IgG4-RD. We report a case of a 33-year-old male who was referred by his general practitioner (GP) to the fast access nephrology clinic due to elevated creatinine and fatigue, which was found after the patient had undergone some investigations at the GP office. He had history of atopic dermatitis and a prior admission for acute pancreatitis of unknown cause and recent bilateral anterior uveitis treated with steroid eyedrops. His urinalysis showed one to two granular casts per high-power field (HPF), and his creatinine was 262 µmol/L (previously normal). Three main differential diagnoses were considered given the patient's history: sarcoidosis, tubulointerstitial nephritis with uveitis (TINU) and IgG4-related disorder. Investigations were undertaken in that regard showing elevated serum IgG4 levels (2.7 times upper-limit of normal). Renal biopsy demonstrated tubulointerstitial nephritis (TIN) with 30 IgG4-positive plasma cells per HPF. Given the patient's presentation over time, a diagnosis of IgG4-TIN was considered. The patient was treated with high-dose steroids and has shown signs of improvement of both his renal and ocular problems. The uniqueness of the case is reflected through the fact that IgG4-renal disease is usually diagnosed in patients with an already established manifestation of another organ, whilst in our patient the renal involvement led to establishing IgG4-RD. It is also important to note that, in spite of initially negative serum IgG4 levels, the diagnosis still needs to be considered especially if multisystem involvement is present (as in this case).
