Case Reports in MedicinePub Date : 2026-06-28eCollection Date: 2026-01-01DOI: 10.1155/carm/2788624
Husam Hamshary, Mosub Qatu, Fathi Milhem, Radi Kukhun, Zaid Sawaftah, Mohammad Bdair, Abdelfattah M Dahmas, Qutaiba Ja'far A Mahmoud, Omar Naseef, Mohammed Ibrahim Abu Kamesh
{"title":"Incidental Tentorial Cerebelli Hypoplasia: A Case Report and Comprehensive Literature Review of Its Clinical and Imaging Characteristics.","authors":"Husam Hamshary, Mosub Qatu, Fathi Milhem, Radi Kukhun, Zaid Sawaftah, Mohammad Bdair, Abdelfattah M Dahmas, Qutaiba Ja'far A Mahmoud, Omar Naseef, Mohammed Ibrahim Abu Kamesh","doi":"10.1155/carm/2788624","DOIUrl":"10.1155/carm/2788624","url":null,"abstract":"<p><strong>Background: </strong>Tentorial hypoplasia is a rare congenital condition characterized by underdevelopment or absence of the tentorium cerebelli, a dural structure separating the cerebrum from the cerebellum. It is often detected incidentally on computed tomography (CT) or magnetic resonance imaging (MRI) and may be associated with conditions such as Dandy-Walker syndrome and Chiari malformation, although many cases remain asymptomatic. Imaging plays an essential role in identifying associated abnormalities when present.</p><p><strong>Case presentations: </strong>Case 1: A 16-year-old male presented with a left subdural hematoma following a road traffic accident. Neuroimaging incidentally revealed tentorial hypoplasia with brain herniation into the quadrigeminal cistern, though he remained asymptomatic. Case 2: A 3-year-old child with metastatic neuroblastoma had incidentally discovered tentorial hypoplasia with brain herniation on CT and MRI, with no neurological deficits.</p><p><strong>Conclusion: </strong>Tentorial hypoplasia is often an incidental finding with a favorable prognosis, though early imaging is essential for identifying associated central nervous system (CNS) malformations and guiding management.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"2788624"},"PeriodicalIF":1.0,"publicationDate":"2026-06-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13310512/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148343932","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":"Seizure as a Rare Initial Manifestation of Empty Sella Turcica Associated With Idiopathic Intracranial Hypertension: A Case Report.","authors":"Karim Lakhdar, Zineb Moudafia, Mohamed Amine Elhasnaoui, Ounci Essaad, Rajae Alkouh, Siham Rachidi Alaoui, Smael Labib","doi":"10.1155/carm/4244868","DOIUrl":"10.1155/carm/4244868","url":null,"abstract":"<p><p>Empty sella turcica (EST) is a radiological condition frequently associated with idiopathic intracranial hypertension (IIH), typically presenting with headache and visual disturbances. Seizures are rarely reported in this context. We report the case of a 41-year-old woman presenting with new-onset generalized tonic-clonic seizures preceded by a 15-day history of severe headache. Neurological examination was unremarkable, while fundoscopy revealed bilateral papilledema. Brain imaging demonstrated an empty sella, and lumbar puncture confirmed elevated intracranial pressure (32 cm H<sub>2</sub>O) with normal cerebrospinal fluid composition, consistent with IIH. Endocrine evaluation showed isolated thyrotropic insufficiency. The patient was treated with acetazolamide and topiramate, leading to rapid clinical improvement, normalization of intracranial pressure, and no recurrence of seizures during follow-up. This case highlights that seizures may represent an unusual initial manifestation of IIH associated with EST and underscores the importance of considering intracranial hypertension in the diagnostic workup of unexplained seizures.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"4244868"},"PeriodicalIF":1.0,"publicationDate":"2026-06-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13309902/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148343980","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}
Case Reports in MedicinePub Date : 2026-06-24eCollection Date: 2026-01-01DOI: 10.1155/carm/3863128
Mehrgan Shahryari, Boshra Shams, Helge Herthum, Pablo Gottheil, Jeremias Heider, Josef Käs, Eberhard Siebert, Thomas Picht, Ingolf Sack
{"title":"Stiffness and Tissue Viscosity in a Cerebral Neoplasm Measured by Preoperative Multifrequency Magnetic Resonance Elastography (MRE) Guide the Differential Diagnosis of Brain Tumors by Ruling Out Glioma.","authors":"Mehrgan Shahryari, Boshra Shams, Helge Herthum, Pablo Gottheil, Jeremias Heider, Josef Käs, Eberhard Siebert, Thomas Picht, Ingolf Sack","doi":"10.1155/carm/3863128","DOIUrl":"10.1155/carm/3863128","url":null,"abstract":"<p><p>Here, we report the case of a 65-year-old man who presented to the emergency department with paresthesia, psychomotor retardation, and dysarthria. Contrast-enhanced brain MRI revealed a solitary, contrast-enhancing mass in the right head of the caudate nucleus with extensive perifocal edema, raising a primary differential diagnosis of glioblastoma versus metastasis, among other possibilities. Prior to surgical removal of the suspected glioblastoma, the patient underwent additional diffusion tensor imaging and research MRI, including multifrequency magnetic resonance elastography (MRE) to generate mechanical property maps of stiffness and tissue viscosity and to potentially differentiate between glioma, brain metastases, and benign masses based on tissue biomechanical properties. MRE revealed abnormally high values of stiffness and viscosity-a mechanical signature known to be associated with brain metastases or meningioma, but not glioblastoma, which typically exhibits soft tissue properties with reduced viscous dissipation. Histopathologic evaluation confirmed MRE by revealing a distant metastasis of squamous cell carcinoma, the primary tumor of which was detected by PET-CT within the right hilus of the lung. This case demonstrates the diagnostic value of the biomechanical properties of brain tumors and raises the prospect of applying MRE as an easy-to-use MRI contrast that can rule out glioma when the suspicious lesion exhibits stiff-viscous material properties.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"3863128"},"PeriodicalIF":1.0,"publicationDate":"2026-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13292019/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148327040","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}
Case Reports in MedicinePub Date : 2026-06-24eCollection Date: 2026-01-01DOI: 10.1155/carm/8542341
Abbigail Niewchas, Brandon Baccari, Christopher Stewart, Suporn Sukpraprut-Braaten, Donald G Smith
{"title":"Guillain-Barré Syndrome Emerging From Syringomyelia Management: A Case Report.","authors":"Abbigail Niewchas, Brandon Baccari, Christopher Stewart, Suporn Sukpraprut-Braaten, Donald G Smith","doi":"10.1155/carm/8542341","DOIUrl":"10.1155/carm/8542341","url":null,"abstract":"<p><p>The coexistence of Guillain-Barré syndrome with a spinal cord syrinx is extremely rare. This report presents the emergency admission of a 57-year-old female presenting with an abrupt and progressive loss of motor function and abnormal sensation in her lower extremities. MRI of the cervical spine revealed a syrinx extending from C2 to C7. Neurology referred the case to neurosurgery, who assessed the patient and recommended nonoperative and supportive treatment given the size of the syrinx and the patient's status as a poor surgical candidate. She was subsequently discharged to a local extended care facility (ECF) with a diagnosis of syringomyelia. Despite physical therapy and rehabilitation, her symptoms progressed to include loss of motor function in bilateral lower extremities, worsening paresthesia in bilateral upper and lower extremities, and progressive dyspnea. Nerve conduction studies performed during neurologic evaluation at the ECF demonstrated a sensorimotor demyelinating polyneuropathy, and she was urgently transferred to the emergency department, where Guillain-Barré syndrome was confirmed. The patient developed respiratory failure requiring mechanical ventilation and was treated with intravenous immunoglobulin. Although she experienced transient stabilization, her clinical course was complicated by progressive functional decline. This case highlights how a cervical syrinx may obscure the initial presentation of Guillain-Barré syndrome, contributing to delayed treatment, and demonstrates the importance of maintaining a broad differential diagnosis in patients with atypical or overlapping neurologic findings.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"8542341"},"PeriodicalIF":1.0,"publicationDate":"2026-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13293096/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148327009","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":"Co-Occurrence of <i>SCN9A</i> and <i>PRRT2</i> Variants in a Patient With Paroxysmal Extreme Pain Disorder, Contradictory Analgesia, and Intractable Paroxysmal Non-Kinesigenic Dyskinesia.","authors":"Miki Ikeda, Aritomo Kawashima, Kaori Kodama, Ryo Sato, Saki Uneoka, Yu Katata, Yukimune Okubo, Wakaba Endo, Takehiko Inui, Noriko Togashi, Chika Mizuno, Hisao Yaoita, Naoya Saijo, Jun Takayama, Gen Tamiya, Atsuo Kikuchi, Shigeo Kure, Kazuhiro Haginoya","doi":"10.1155/carm/5942985","DOIUrl":"10.1155/carm/5942985","url":null,"abstract":"<p><p>Gain-of-function mutations in <i>SCN9A</i>, encoding the voltage-dependent Nav1.7 sodium channel, cause three autosomal-dominant disorders associated with severe pain: primary erythromelalgia, paroxysmal extreme pain disorder (PEPD), and small fiber neuropathy. On the other hand, biallelic loss-of-function mutations have been linked to impaired pain perception. Notably, the coexistence of both hyperalgesia and hypoalgesia within the same patient harboring the I234T variant has been reported in three independent patients to date. We report a 7-year-old girl harboring co-occurring <i>SCN9A</i> (I234T) and <i>PRRT2</i> variants who presented with paroxysmal extreme pain disorder, contradictory analgesia, sensitivity to heat, and intractable head-drop attacks. Based on the genetic and clinical analyses, she was diagnosed as having PEPD and <i>PRRT2</i>-related paroxysmal dyskinesia. The intractable head-drop attacks were considered as paroxysmal non-kinesigenic dyskinesia. In addition, she exhibited easy fatigability and hypotonia. Taken together with her cold, cyanotic feet, these findings suggest that she may have also had small fiber neuropathy.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"5942985"},"PeriodicalIF":1.0,"publicationDate":"2026-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13287326/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148315942","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":"Cutaneous Focal Mucinosis Successfully Treated With Superpulsed Carbon Dioxide Laser.","authors":"Maryam Hekmat, Arezou Hosseini, Babak Shirazi Yeganeh, Negin Fazelzadeh Haghighi","doi":"10.1155/carm/8484405","DOIUrl":"10.1155/carm/8484405","url":null,"abstract":"<p><p>Cutaneous focal mucinosis (CFM) is a rare dermatological condition characterized by localized mucin deposition in the dermis, presenting as solitary or multiple asymptomatic papules or nodules. Treatment options are limited, with spontaneous resolution occurring in some cases, while others persist or require intervention. Systemic therapies, such as isotretinoin, have variable efficacy, and invasive approaches may not suit all patients. We report a 19-year-old male case with persistent bilateral papular lesions in the beard area, diagnosed as CFM via biopsy. Due to the patient's reluctance to use systemic medication, superpulsed carbon dioxide (CO<sub>2</sub>) laser therapy was employed, resulting in significant lesion resolution without adverse effects. At 6-month follow-up, the outcome remained favorable, suggesting laser therapy as a promising, noninvasive alternative for localized CFM. This study suggests that superpulsed CO<sub>2</sub> laser therapy could be a valuable treatment for CFM, especially for patients who prefer nonsystemic or less invasive options. Further studies are warranted to confirm superpulsed CO<sub>2</sub> laser therapy efficacy and long-term outcomes in managing this uncommon condition.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"8484405"},"PeriodicalIF":1.0,"publicationDate":"2026-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13284545/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148307278","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}
Case Reports in MedicinePub Date : 2026-06-22eCollection Date: 2026-01-01DOI: 10.1155/carm/2688113
Sandra Stankovic M, Marija Jankovic Ratkovic, Danica Mihajlović, Milica Ignjatović, Milica Jakovljević, Vesna Cvetković, Dušan Miljković, Jelena Vučić, Danijela Jovančić Petković, Tatjana Stanković
{"title":"Infant Nephrogenic Diabetes Insipidus: Challenges Leading to Delayed Management.","authors":"Sandra Stankovic M, Marija Jankovic Ratkovic, Danica Mihajlović, Milica Ignjatović, Milica Jakovljević, Vesna Cvetković, Dušan Miljković, Jelena Vučić, Danijela Jovančić Petković, Tatjana Stanković","doi":"10.1155/carm/2688113","DOIUrl":"10.1155/carm/2688113","url":null,"abstract":"<p><strong>Background: </strong>Nephrogenic diabetes insipidus (NDI) is a rare, potentially life-threatening renal tubular disorder characterized by a decreased ability to concentrate urine due to antidiuretic hormone (ADH) resistance, leading to significant free water loss. The congenital form, most often caused by X-linked mutations in the AVPR2 gene, typically presents in early infancy. Due to its rarity and nonspecific clinical features in young children, such as failure to thrive, vomiting, and excessive fluid intake, diagnostic delays are common, especially in centers with limited prior experience in managing NDI.</p><p><strong>Case presentation: </strong>We report a case of a 17-month-old male infant presenting with severe hypernatremia, dehydration, vomiting, and failure to thrive. Initial clinical findings, a partial response to desmopressin, and absent neurohypophysis signaling on MRI suggested central diabetes insipidus (CDI), leading to a delay in the correct diagnosis. However, persistent polyuria, despite escalating desmopressin doses, prompted reconsideration. Genetic testing confirmed a pathogenic AVPR2 mutation (c.541C > T, p. Arg181Cys), establishing the diagnosis of congenital NDI. Therapeutic management included thiazide diuretics, NSAIDs, and dietary sodium restriction, resulting in slow and gradual clinical stabilization.</p><p><strong>Conclusion: </strong>This case highlights several diagnostic challenges, such as misinterpreting partial desmopressin response and MRI results. It emphasizes the importance of systematic diagnostic algorithms for infants with polyuria and early genetic testing when clinical presentations lack clarity. We aim to share our clinical experience and the pitfalls encountered while managing a patient with NDI and contribute to earlier and more effective recognition and treatment of this rare condition, preventing the development of life-threatening complications.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"2688113"},"PeriodicalIF":1.0,"publicationDate":"2026-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13287331/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148315906","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}
Case Reports in MedicinePub Date : 2026-06-19eCollection Date: 2026-01-01DOI: 10.1155/carm/2936999
Patricia Capdevila, Elisa Gómez, Cristobal Carrasco, Carlos Escrivá, Emilio Soria, Josep M Esteve, Daniel Vidal, Alba Ortiz, Javier Caballero, Jose Gomez-Codina
{"title":"Diffuse Large B-Cell Lymphoma Arising in Postpneumonectomy Cavities: Report of Two Cases and Literature Review.","authors":"Patricia Capdevila, Elisa Gómez, Cristobal Carrasco, Carlos Escrivá, Emilio Soria, Josep M Esteve, Daniel Vidal, Alba Ortiz, Javier Caballero, Jose Gomez-Codina","doi":"10.1155/carm/2936999","DOIUrl":"10.1155/carm/2936999","url":null,"abstract":"<p><p>Diffuse large B-cell lymphoma (DLBCL) most commonly arises in nodal regions but may also present extranodally. We report two cases of large B-cell lymphoma developing in postpneumonectomy cavities more than a decade after surgery for primary lung carcinoma. In both patients, no history of systemic immunodeficiency, organ transplantation, or chronic immunosuppressive therapy was present, and HIV, hepatitis B, and hepatitis C serologies were negative. In Case 1, the lymphoma showed an activated postgerminal center phenotype and was negative for EBER and HHV-8. In Case 2, the tumor was CD20-negative and EBER-positive, with pathological features of inflammation-associated large B-cell lymphoma. PET-CT was used for staging and response assessment in both patients, and both achieved complete metabolic remission after curative-intent chemotherapy. These cases underscore a rare clinical entity that differs pathophysiologically from pyothorax-associated lymphoma (PAL) or primary effusion lymphoma (PEL). They highlight the postpneumonectomy cavity as a rare but potentially permissive microenvironment for large B-cell lymphomagenesis and underscore the biological heterogeneity of these presentations.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"2936999"},"PeriodicalIF":1.0,"publicationDate":"2026-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13280979/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148293553","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}
Case Reports in MedicinePub Date : 2026-06-13eCollection Date: 2026-01-01DOI: 10.1155/carm/9942449
Sepideh Aarabi
{"title":"From Fiber to Obstruction: A Case of Small Bowel Obstruction Due to Phytobezoar.","authors":"Sepideh Aarabi","doi":"10.1155/carm/9942449","DOIUrl":"10.1155/carm/9942449","url":null,"abstract":"<p><strong>Introduction: </strong>Phytobezoars, a rare cause of small bowel obstruction (SBO), account for 0.4%-4% of cases and are most commonly associated with altered gastric motility from prior gastric surgery or systemic conditions like diabetes mellitus and hypothyroidism. Phytobezoars form from indigestible plant fibers that resist enzymatic breakdown, and their incidence may increase in populations with high-fiber diets. Due to their nonspecific clinical presentation, prompt diagnosis requires a combination of clinical suspicion and imaging studies, particularly computed tomography (CT), by which characteristic intraluminal masses are identified.</p><p><strong>Case report: </strong>A case is presented of a 48-year-old female with a history of partial gastrectomy for peptic ulcer disease who presented with abdominal pain, nausea, vomiting, and distension. Tenderness and hyperactive bowel sounds consistent with mechanical obstruction were found on examination. A mottled gas-containing mass in the jejunum, suggestive of a phytobezoar, was identified by CT imaging. Laparotomy was performed, and an enterotomy was carried out to extract the bezoar. Postoperative recovery was uneventful, and dietary counseling was provided to prevent recurrence.</p><p><strong>Conclusion: </strong>Phytobezoars are an uncommon but important cause of SBO, particularly in patients with risk factors such as prior gastric surgery or high-fiber diets. Awareness of this condition is crucial for timely diagnosis and management. Surgical intervention remains the mainstay of treatment for complete obstructions, while postoperative dietary counseling and management of underlying conditions are essential to prevent recurrence. The need for heightened clinical awareness to minimize complications associated with phytobezoar-induced SBO is underscored by this case.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"9942449"},"PeriodicalIF":1.0,"publicationDate":"2026-06-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13263701/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148249354","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}
Case Reports in MedicinePub Date : 2026-06-13eCollection Date: 2026-01-01DOI: 10.1155/carm/6883361
Reem Shihab, May Shihab, Hala O Abdallah, Suha Nori, Obay Mashaqi
{"title":"Severe Diabetic Ketoacidosis With Refractory Hypernatremia due to Hypokalemia-Induced Arginine Vasopressin Resistance: A Case Report and Literature Review.","authors":"Reem Shihab, May Shihab, Hala O Abdallah, Suha Nori, Obay Mashaqi","doi":"10.1155/carm/6883361","DOIUrl":"10.1155/carm/6883361","url":null,"abstract":"<p><p>Diabetic ketoacidosis (DKA) is a life-threatening complication of diabetes characterized by hyperglycemia, high-anion-gap metabolic acidosis, and ketonemia. Hypernatremia in DKA is uncommon and typically reflects disproportionate free water loss. Arginine vasopressin resistance (AVP-R), previously known as nephrogenic diabetes insipidus, may occur in the setting of hypokalemia, leading to polyuria, hypotonic urine, and worsening hypernatremia. We report the case of a 14-year-old previously healthy female who presented with new-onset severe DKA complicated by profound acidosis, hyperglycemia, hypokalemia, and hypernatremia. Despite standard DKA management, she developed persistent polyuria and rising serum sodium levels. Urine studies demonstrated dilute urine with inappropriately elevated pH, raising suspicion for hypokalemia-induced AVP-R. Management included aggressive potassium repletion, careful adjustment of intravenous fluids, and adjunctive desmopressin therapy. This approach resulted in normalization of electrolyte abnormalities, resolution of polyuria, and full clinical recovery. This case highlights the importance of recognizing hypokalemia-induced AVP-R as a reversible cause of refractory hypernatremia in DKA and underscores the need for individualized fluid and electrolyte management beyond standard treatment protocols.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2026 ","pages":"6883361"},"PeriodicalIF":1.0,"publicationDate":"2026-06-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13263702/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148249342","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}