Journal of medical cases最新文献

{"title":"Multiple Complications of Crohn's Disease and the Need for Early and Continuous Multidisciplinary Undertaking.","authors":"David Dogahe, Maxime Taghavi, Edouard Cubilier, Said Sanoussi, Ruth Duttman, Joelle Nortier, Maria do Carmo Filomena Mesquita","doi":"10.14740/jmc4154","DOIUrl":"https://doi.org/10.14740/jmc4154","url":null,"abstract":"<p><p>Crohn's disease is an inflammatory disease that typically affects the bowels but can also have many different extraintestinal manifestations. One of those complications is immunoglobulin A nephropathy (IgAN), which is one of the most encountered renal lesions in the setting of Crohn's disease. Another point of focus for Crohn's patients is the risk of cancer, with a higher risk of colorectal cancer but also extraintestinal neoplasia such as hepatobiliary, hematological, and urinary tract neoplasia. We present the case of a young patient suffering from long-term Crohn's disease and subsequent IgAN leading to end-stage kidney disease and hemodialysis. The patient was diagnosed young and had undergone multiple surgeries and different treatments in various countries. He then presented in our center already with advanced chronic renal failure from IgAN that was unknown due to poor multidisciplinary follow-up. Shortly after starting hemodialysis, he developed a large abdominal mass, first thought to result from Crohn's-related fistula. This mass turned out to be a urachal adenocarcinoma, a rare type of bladder cancer with an especially poor prognosis. It is not known whether this type of cancer is associated with either Crohn's disease or IgAN, and no such association has been previously described. The treatment of urachal cancer usually relies on surgery, with the addition of chemotherapy in some cases. Unfortunately for our patient, his case was already so advanced at the moment of diagnosis that he was excluded from curative treatment and quickly passed away thereafter. This case illustrates many important aspects of the rigorous follow-up that is needed for Crohn's patients, with regular check-ups, screening investigations, and the need for multidisciplinary evaluation. Furthermore, it describes the development of a rare type of cancer in the setting of Crohn's disease and IgAN, with no prior established link between these different pathologies.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 11","pages":"356-361"},"PeriodicalIF":0.0,"publicationDate":"2023-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10681768/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138465304","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Epidural-Like Effect of a Continuous Right-Sided Erector Spinae Plane Blockade for Complicated Pediatric Abdominal Surgery.","authors":"Neil Doshi, Vipin Bansal, Emmanuel Alalade","doi":"10.14740/jmc4161","DOIUrl":"https://doi.org/10.14740/jmc4161","url":null,"abstract":"<p><p>The erector spinae plane block (ESPB) is increasingly gaining popularity in pediatric anesthesiology as it provides an alternative to neuraxial anesthesia in those with relative and absolute contraindications. Recent studies show craniocaudal spread in cadavers and multi-level spread impacting neural structures in live subjects. We present a case report of a pediatric patient with a history of abdominal surgeries, contraindication to neuraxial anesthesia, and thoracic vertebrae fractures. Bilateral ESPB catheters were initially placed but the left catheter was accidentally dislodged. Each ESPB catheter was initially programmed to flow at rate of 2 cc/h of ropivacaine 0.1% for a max combined rate of 4 cc/h. Once the left ESPB catheter was dislodged, the right ESPB catheter was programmed to flow at 4 cc/h which surprisingly continued to provide adequate bilateral analgesia for the patient without the need for additional narcotics. In cases where a unilateral ESPB catheter is the only option due to catheter displacement or contamination, administering a higher volume of local anesthetic may still yield satisfactory pain relief for managing postoperative discomfort following abdominal surgery.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 11","pages":"351-355"},"PeriodicalIF":0.0,"publicationDate":"2023-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10681764/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138465303","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Percutaneous Intracardiac Mass Extraction in High Surgical-Risk Patients.","authors":"Mariam Riad, Mustafeez Ur Rahman, Rajasekhar Mulyala, Nadia Sayyed, Danielle Bayer, Bassam Omar","doi":"10.14740/jmc4150","DOIUrl":"10.14740/jmc4150","url":null,"abstract":"<p><p>Large intracardiac masses including tumors, thrombi, and vegetations result in detrimental embolic or obstructive sequelae and present a management dilemma. Open heart surgery, the traditional approach, may not be an option for many patients with a prohibitive surgical risk due to multiple comorbidities. Recently, percutaneous options have emerged with reported success in extracting such intracardiac masses. A 42-year-old female with history of advanced primary sclerosing cholangitis with decompensated liver cirrhosis causing ascites and variceal bleed presented to the emergency department with fatigue, subjective fevers, chills and melena. Laboratory results revealed neutrophil-predominant leukocytosis and normocytic anemia, and blood cultures were positive for <i>Candida albicans</i>. Electrocardiography showed sinus tachycardia. Chest X-ray was unremarkable. She underwent packed red blood cell transfusion and esophageal banding for variceal bleeding. Transthoracic echocardiogram revealed normal left ventricular ejection fraction and no wall motion abnormalities. A right atrial mobile mass measuring approximately 1.0 × 3.0 cm was noted. Multidisciplinary heart team discussion concluded that while the mass posed a high embolic risk, the patient had a prohibitive risk for surgical intervention. Successful percutaneous removal of the mass using Penumbra system device (Penumbra Incorporated, Alameda, CA) was accomplished. This case report details the procedure and outcomes, as well as presents a literature review.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 11","pages":"362-368"},"PeriodicalIF":0.0,"publicationDate":"2023-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10681765/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138465305","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Cervical Radiculopathy Presenting as Ischemic Stroke After Carotid Artery Stent Placement.","authors":"Itzhak Brook, Bilaal Sirdar, Andrew Stemer","doi":"10.14740/jmc4162","DOIUrl":"https://doi.org/10.14740/jmc4162","url":null,"abstract":"<p><p>Abrupt loss of focal brain function is the main characteristic of the beginning of ischemic stroke. However, individuals with disorders other than stroke can also present with similar features. These conditions include seizure disorders, migraine, central nervous system abscess or tumor, head trauma, subdural hematoma, cerebral venous thrombosis, viral encephalitis, conversion reaction, hypertensive encephalopathy, multiple sclerosis, and spinal cord disorder. An 82-year-old man presented with a sudden onset of numbness in his left forearm in the distribution of C6 and C7 spinal nerves, 2 days after undergoing endarterectomy and stent placement in his right carotid artery because of stenosis. He was diagnosed with hypo-pharyngeal squamous cell carcinoma (T1, L0, M0) 17 years earlier (2006) which was treated with 70 Gy intensity-modulated radiotherapy (IMRT). The patient underwent stent insertion into his left carotid artery 3.5 years earlier because of 80% carotid artery stenosis. He was initially suspected to have an ischemic stroke. However, computed tomography angiography of the head and neck did not show stenosis or occlusion of the major intracranial arteries and no aneurysms were identified. It showed interval stenting of the cervical portion of the right carotid artery and stable appearance of left carotid artery stent. Both carotid artery stents and the vertebral arteries were patent. The cervical spine showed bilateral moderate to severe foramen stenosis in C3-C4 and C5-C6, and moderate to severe stenosis in the right C2-C3 and left C4-C5. His symptoms subsided after performing neck extension exercises. This is the first report of a patient whose cervical radiculopathy symptoms were suspected to be caused by ischemic stroke. The recent angioplasty and stent placement in the right carotid artery made the association more likely and had to be excluded. Clinicians should be aware that cervical radiculopathy could present as ischemic stroke. It is therefore important that disorders that cause symptoms similar to ischemic stroke are also considered in these individuals.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 11","pages":"387-392"},"PeriodicalIF":0.0,"publicationDate":"2023-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10681767/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138465301","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Chronic Pancreatitis-Induced Thrombosis of Celiac and Superior Mesenteric Artery.","authors":"Harshavardhan Sanekommu,&nbsp;Sobaan Taj,&nbsp;Rida Mah Noor,&nbsp;Muhammad Umair Akmal,&nbsp;Claudia Ramirez,&nbsp;Pranav Shah,&nbsp;Mohammad Hossain,&nbsp;Asif Arif","doi":"10.14740/jmc4112","DOIUrl":"10.14740/jmc4112","url":null,"abstract":"<p><p>Every year, nearly 60,000 hospitalizations occur in the United States due to chronic pancreatitis (CP). CP can cause severe chronic abdominal pain, pancreatic insufficiency, and increased risk of pancreatic cancer. While venous thrombotic complications are common, arterial thrombotic events are rarely reported in CP. This report describes a case of a 43-year-old female who presented with severe worsening abdominal pain due to CP. Diagnostic imaging disclosed thrombosis of superior mesenteric artery (SMA) and celiac artery (CA) with acute bowel wall changes reflecting ischemic changes, resulting in acute-on-chronic mesenteric ischemia. Endovascular stent placement relieved the ischemia with the resolution of pain. Arterial thrombosis should be considered as a diagnostic possibility when patients with CP present with a significant change in symptoms. Importantly, the case demonstrates that endovascular treatment with stent placement can relieve ischemia and resolve symptoms in patients with CP.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 9-10","pages":"322-326"},"PeriodicalIF":0.0,"publicationDate":"2023-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/50/d6/jmc-14-322.PMC10586330.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49695860","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Synchronous Left Ventricular and Endocranial Mass.","authors":"Cornelia Tsokkou,&nbsp;Andreas Mitsis,&nbsp;Evi Christodoulou,&nbsp;Panayiotis Avraamides,&nbsp;Stefanos Sakellaropoulos","doi":"10.14740/jmc4153","DOIUrl":"10.14740/jmc4153","url":null,"abstract":"<p><p>Myocardial cysts represent a miscellaneous and infrequent spectrum of conditions, with each of them coming from a different etiological background. Congenital myocardial cysts, neoplasia, cysts of infectious origin (bacterial, viral, or parasitic), and cardiac pathologies that may fake cystic content are all encompassed in this group. Although most patients are asymptomatic, some may occasionally present with obstruction, valvular dysfunction, or heart failure. Even more uncommon is the coexistence of a myocardial cyst with other extracardiac locations causing extracardiac symptoms. In this direction, the coexistence of a myocardial and endocranial cyst is extremely rare and can cause symptomatology from the affected organs (e.g., seizures). Cardiac investigation in this context is mainly dependent on non-invasive diagnostic modalities, and laboratory procedures. In this case report, we present a 26-year-old Congolese male admitted with dyspnea and epileptic seizures. Echocardiography revealed left ventricular and both mitral and tricuspid valve dysfunction and the presence of two myocardial cysts, while brain computed tomography showed an additional frontal cystic lesion. A precise diagnostic workup with a combination of non-invasive imaging, laboratory results, and epidemiology data assisted the diagnosis and guided the most suitable therapeutic choice.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 9-10","pages":"332-338"},"PeriodicalIF":0.0,"publicationDate":"2023-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/bd/b8/jmc-14-332.PMC10586335.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49695863","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Acute Cranial Nerve VI Palsy Following Prolonged Prone Positioning in an Adolescent With Neurofibromatosis Type 1: A Rare Complication of Spinal Surgery in the Prone Position.","authors":"Christian Mpody,&nbsp;Vanessa Olbrecht,&nbsp;Joseph D Tobias","doi":"10.14740/jmc4145","DOIUrl":"10.14740/jmc4145","url":null,"abstract":"The abducens or sixth cranial nerve provides motor innervation to the lateral rectus muscle, which abducts the ipsilateral eye with secondary innervation of the contralateral medial rectus muscle to allow for coordinated movement of both eyes. Various acute and chronic pathologic conditions, most importantly pontine infarctions and increased intracranial pressure, can result in acute sixth cranial nerve palsies. We report the uncommon occurrence of acute abducens nerve palsy following spinal fusion surgery in an 18-year-old male patient with a history of multiple neurological and orthopedic conditions. Postoperatively, the patient presented with symptoms that included left diplopia with restricted upward and downward gaze, indicative of abducens nerve palsy. The anatomy of the sixth cranial nerve is discussed, potential etiologies of sixth nerve palsy presented, and a proposed diagnostic workup reviewed. Our report emphasizes the need for comprehensive exploration of ocular symptoms following spinal surgery, given the various potential etiologies of sixth nerve palsy.","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 9-10","pages":"317-321"},"PeriodicalIF":0.0,"publicationDate":"2023-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/24/72/jmc-14-317.PMC10586333.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49695859","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A Double Hit to Ubiquitination Leading to a New Diagnosis of VEXAS Syndrome.","authors":"Helen Pozdniakova,&nbsp;Apurva Vedire,&nbsp;Anand Kadakia,&nbsp;Steven Imburgio,&nbsp;Ravneet Bajwa,&nbsp;Varsha Gupta,&nbsp;Ruchi Bhatt,&nbsp;Mohammad A Hossain","doi":"10.14740/jmc4127","DOIUrl":"10.14740/jmc4127","url":null,"abstract":"<p><p>VEXAS (vacuoles, E1 enzyme, X-linked, auto-inflammatory, somatic) syndrome is a newly defined illness that bridges hematology, oncology, and rheumatology. Its pathophysiology originates in a mutation in the <i>UBA1</i> gene that leads to a defect in ubiquitination resulting in a severe systemic inflammatory syndrome. It is associated with significant morbidity and mortality; however, data are scarce due to limited cases described in the literature. Here we describe a case of a male in his 60s who was referred to hematology-oncology due to progressive dyspnea, poor oral intake, and weight loss. He was diagnosed with relapsing polychondritis 2 years prior; however, his symptoms did not improve despite treatment. He was ultimately diagnosed with VEXAS syndrome with a mutation in <i>UBA1</i> (ubiquitin-like modifier activating enzyme 1) and a concurrent <i>SQSTM1</i> mutation. In addition, the coexistence of two mutations in the ubiquitination pathway in the same patient has not been reported to date. This patient and the treatment course were compared to pre-existing literature to increase awareness and improve the medical management of VEXAS syndrome.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 9-10","pages":"327-331"},"PeriodicalIF":0.0,"publicationDate":"2023-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/63/4f/jmc-14-327.PMC10586336.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49695858","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Pancreatic Vasoactive Intestinal Peptide-Producing Tumor as a Rare Cause of Acute Diarrhea and Severe Hypokalemia.","authors":"Vasilios Giampatzis,&nbsp;Christina Kotsiari,&nbsp;Prodromos Bostantzis,&nbsp;Alexandra Chrisoulidou,&nbsp;Aimilia Fotiadou,&nbsp;Soultana Loti,&nbsp;Stefanos Papantoniou,&nbsp;Persefoni Papadopoulou","doi":"10.14740/jmc4141","DOIUrl":"10.14740/jmc4141","url":null,"abstract":"<p><p>Pancreatic vasoactive intestinal peptide-producing tumor (VIPoma) is a rare functional neuroendocrine tumor most commonly presenting with watery diarrhea and electrolyte abnormalities that include hypokalemia, hypercalcemia and metabolic acidosis. This type of tumor has usually insidious clinical behavior that is characterized by chronic secretory diarrhea, lasting usually from months to years before diagnosis, not responsive to usual medical or dietary treatment approaches. Given the resemblance of VIPoma with other more common causes of chronic watery diarrhea, the final diagnosis is often delayed and the tumors are usually large and metastatic at the time of detection. Our case of pancreatic VIPoma demonstrates an unusual clinical course for this type of tumor with acute refractory diarrhea and rapid deterioration of patient's clinical and biochemical status that required emergent in-hospital diagnosis and treatment. Our patient is a 45-year-old woman who presented with abrupt, watery diarrhea during the past 24 h before admission accompanied with severe hypokalemia as well as hyponatremia, hyperglycemia and hypercalcemia. Despite aggressive management with fluid administration and electrolyte replenishment, no significant improvement in patient's symptoms and electrolyte imbalance was observed. After exclusion of other causes of acute diarrhea from the medical history and the laboratory tests, the clinical suspicion of a functional neuroendocrine tumor was raised. After the establishment of final diagnosis of pancreatic VIPoma with biochemical tests and magnetic resonance imaging (MRI), somatostatin analogues were prescribed and the patient underwent distal pancreatectomy and splenectomy with no signs of lymph node and splenic metastases. Few days after the surgical resection of the tumor, the patient readmitted to our hospital with tarry stools and severe anemia. The abdominal computed tomography (CT) revealed a retroperitoneal cystic lesion. The gastrointestinal bleeding gradually recessed after endoscopic hemostasis of duodenal ulcer lesions whereas the cystic lesion (postoperative lymphocele) was successfully drained under CT-guidance before discharge. After almost 10 years postoperatively, the patient is still asymptomatic with no signs of relapse or metastasis of the disease in the periodic laboratory and imaging follow-up. In conclusion, pancreatic VIPoma can sometimes manifest symptoms of abrupt onset and rapid progression that require high clinical suspicion, appropriate diagnostic workup and aggressive management.</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 9-10","pages":"307-316"},"PeriodicalIF":0.0,"publicationDate":"2023-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/0f/f1/jmc-14-307.PMC10586331.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49695862","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A Case Report of a Challenging Disease: Immunoglobulin G4-Related Disease With Acute Kideny Injury.","authors":"Mostafa Mohrag,&nbsp;Mohammed Abdulrasak,&nbsp;Mohammed Binsalman,&nbsp;Majid Darraj","doi":"10.14740/jmc4159","DOIUrl":"10.14740/jmc4159","url":null,"abstract":"<p><p>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).</p>","PeriodicalId":101328,"journal":{"name":"Journal of medical cases","volume":"14 9-10","pages":"339-343"},"PeriodicalIF":0.0,"publicationDate":"2023-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/3f/d5/jmc-14-339.PMC10586334.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49695857","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}