Evidence for the role of oral microbiota in tongue cancers is limited despite early-onset and unexplained etiology. We assessed oral microbiota profiles in saliva among participants with tongue cancers in the oral cavity compared to cancer-free controls. We conducted a case-control study in a tertiary care cancer hospital in Chennai, South India. Cases (N=31) were newly diagnosed, pre-treatment, biopsy-confirmed, early-stage (<4 cm size) tongue cancers; further classified as early-onset (diagnosed at 20-49 years (N=12)) or late-onset cancers (diagnosed ≥50 years (N=19)), for secondary exploratory analyses. Cancer-free controls (N=31) were unrelated, and frequency matched to the cases on age and sex. Bacterial characterization of saliva was performed using 16S rRNA sequencing. Analysis (n=175 genera) included linear regression for α-diversity, constrained analysis of principal coordinates, permutational multivariable analysis of variance for β-diversity, and analysis of compositions of microbiomes with bias correction for taxa abundance. We also examined differences in established bacterial pathogens selected a priori. The median age of participants was 52 years and a third were women. While there was no difference in α-diversity, β-diversity showed distinct clustering of bacterial genera in controls, early-onset, and late-onset cases. In overall and early-onset cases (vs. controls), genera Eikenella and Campylobacter showae were enriched. Further, in early-onset cases (vs. controls), genera Alloprevotella, Comamonas, Aggregatibacter were enriched, Oribacterium was depleted, and W5053 had differentially absent patterns. Beyond the established pathogens, this study offers important leads for future etiological investigations, particularly in exploratory analyses of early-onset tongue cancers in the oral cavity.
We are aimed at describing the clinical course and management of a rare case of phakomatosis pigmentovascularis (PPV Type IIb) presenting with bilateral glaucoma, complicated by bilateral Urrets-Zavalia syndrome (UZS) following MicroPulse transscleral cyclophotocoagulation (MP-TSCPC). A 6-year-old girl with PPV Type IIb and syndromic glaucoma underwent MP-TSCPC as an initial surgical intervention for uncontrolled intraocular pressure (IOP). Postoperatively, the patient developed bilateral fixed dilated pupils consistent with UZS. Due to persistent IOP elevation and the complication of UZS, bilateral deep sclerectomy (DS) was performed, resulting in adequate IOP control. (10-mmHg OD, 17-mmHg OS) and gradual improvement in pupillary function. No further complications were observed. The case was classified under PPV Type II based on the coexistence of vascular and pigmentary anomalies. A review of the literature confirms the rarity of such a constellation of findings and the sparsity of reported bilateral UZS following MP-TSCPC. This case highlights that PPV Type IIb requires careful examination because of the risk of associated glaucoma. UZS should be considered as a possible complication after MP-TSCPC, especially when both eyes are treated. In complex cases with vascular involvement, DS can provide effective and safe IOP control.
Cutaneous metastases from breast cancer (CMBC), including those involving the scalp, are typically observed in advanced stages of the disease and are relatively uncommon, particularly in hormone receptor-positive subtypes. We present the case of a 68-year-old woman with breast cancer metastatic to the skin, characterized by multiple firm, hyperpigmented, painless nodules distributed over the body, including the scalp. The patient has been treated with a CDK4/6 inhibitor (ribociclib) in combination with endocrine therapy (ET) (letrozole) since April 2024. During follow-up, all subcutaneous nodular lesions showed significant regression. By April 2026, skin color had returned to normal in most previously affected areas, and the majority of nodules had resolved. The scalp lesion completely disappeared, with full hair regrowth at the affected site. Treatment of CMBC with CDK4/6 inhibitor-based therapy has been scarcely reported, with most cases describing ET alone. This case highlights the potential role of CDK4/6 inhibitors and documents a notable response in the scalp, skin, and subcutaneous tissue, which has not been widely described.
Extracranial metastasis of glioblastoma (GBM) is an extremely rare phenomenon and is associated with a poor prognosis. Here, we present the case of a 55-year-old woman who developed pleural metastasis of GBM, manifesting non-specific breathing difficulties during treatment. This case highlights the importance of integrating multiple diagnostic approaches, as only the comparison of histopathological examination results with the patient's clinical history - right temporal lobe GBM - enabled the final diagnosis of GBM metastasis. Next-generation sequencing was performed as part of the diagnostic workup and revealed a p.G266R TP53 mutation. Additionally, imaging revealed an extensive heterogeneous lesion at the site of the previously resected GBM, which proved to be an inoperable gliosarcoma (World Health Organization grade 4). This rare presentation of GBM with distant pleural metastasis, combined with the identification of an uncommon TP53 mutation, represents a unique diagnostic and clinical challenge and underscores the novelty of this case.
Giant endometrial polyps (EPs) are rare in postmenopausal women without hormonal exposure. This study aimed to summarize their clinical presentation and management. Fifteen postmenopausal women with primary giant EPs were included: one case from our center and fourteen additional cases were retrieved from Pubmed, Web of science and Embase databases. The fifteen patients ranged in age from 55 to 70 years, with polyp diameters ranging from 4.0 to 12.0 cm. The most common symptom was postmenopausal bleeding (PMB). Surgical intervention was performed in all cases: six patients underwent hysteroscopic polypectomy, eight received total abdominal hysterectomy with bilateral salpingo-oophorectomy (TAH-BSO) and one underwent dilation and curettage D&C. Most polyps (11/15) were benign on histology and exhibited cystic dilatation. Giant EPs in postmenopausal women are rare but predominantly benign, especially in the absence of hormonal exposure. Transvaginal sonography (TVS) is the first-line modality for initial evaluation. Diagnostic hysteroscopy with complete polypectomy remains the gold standard, enabling accurate histopathological assessment and avoiding unnecessary hysterectomy.
Pneumatosis cystoides intestinalis (PCI) is a rare manifestation of pneumatosis intestinalis (PI), characterized by intramural gas cysts within the gastrointestinal tract. While frequently benign and many times idiopathic, PCI can also be associated with underlying critical conditions. Radiographically, benign cases of PCI can mimic life-threatening abdominal pathology, creating diagnostic uncertainty and challenging operative decision-making. We report a case of a 71-year-old male with a three-year history of progressive abdominal bloating, early satiety, postprandial pain and significant weight loss who presented acutely with worsening abdominal symptoms following a mechanical fall. Computed tomography (CT) revealed extensive small bowel pneumatosis with associated pneumoperitoneum. Despite concerning imaging findings, the patient lacked peritoneal signs, laboratory abnormalities, or evidence of mesenteric ischemia on computed tomography angiography (CTA). Given persistent symptoms and worsening radiologic appearance, an exploratory laparotomy was pursued. Intraoperative findings demonstrated a segment of dilated, atonic small bowel containing diffuse intramural air cysts without evidence of ischemia, perforation, obstruction, or purulent peritonitis. Small bowel resection with primary anastomosis was performed. The patient recovered uneventfully and experienced complete resolution of chronic gastrointestinal symptoms at postoperative follow-up. This case highlights the importance of integrating clinical presentation with laboratory and radiologic findings when evaluating pneumatosis intestinalis. Although small bowel involvement is often associated with pathologic etiologies, PCI may present as an acute exacerbation of chronic disease. Selective surgical intervention can be both diagnostic and therapeutic in symptomatic patients.
Ovarian torsion is defined as the twisting of the ovary at its pedicle, which can result in significant vascular and lymphatic compromise. This condition is a rare gynecological emergency, warranting prompt recognition and diagnosis. This report describes an 11-year-old girl who presented with a two-day history of left iliac fossa pain and associated vomiting. Ultrasound revealed a 3-cm simple ovarian cyst on the left ovary, with preserved blood flow but evidence of ovarian edema, raising suspicion for ovarian torsion. The patient underwent a diagnostic laparoscopy, which confirmed left ovarian torsion, and a detorsion was performed. Nine weeks later, the patient re-presented with similar symptoms, and intraoperative findings again confirmed left ovarian torsion. At this presentation, left ovarian detorsion and cystectomy were performed. This case underscores the diagnostic challenges posed by the nonspecific presentation of ovarian torsion and highlights the importance of investigating predisposing factors in recurrent cases.
Sjogren's syndrome (SS) is an autoimmune disorder characterized by inflammation of exocrine glands, often presenting with symptoms such as dry eyes and mouth. Although less common, renal involvement can lead to serious complications like hypokalemic paralysis. We report a case of a 28-year-old female with no prior medical history, who initially presented with fever, myalgia, and arthralgia. Her condition later progressed to hypokalemia and metabolic acidosis. Further evaluation confirmed Type 1 renal tubular acidosis (RTA) secondary to SS. Treatment with potassium supplementation, steroids, and bicarbonate resolved the symptoms. This case highlights the importance of considering renal involvement in SS patients and underscores the need for prompt intervention to prevent severe complications.
The arc of Bühler (AOB) is a rare embryonic anastomosis between the celiac artery and the superior mesenteric artery, which may become a crucial collateral pathway in cases of celiac axis stenosis. We report a challenging case of a 70-year-old woman presenting with hemorrhagic shock owing to a bleeding branch of an aneurysmal and highly tortuous AOB, with median arcuate ligament compression of the celiac trunk. Endovascular treatment was performed successfully by selective embolization of the bleeding branch with the sacrifice of the AOB using a Penumbra Occlusion Device and packing coils, preserving celiac-mesenteric circulation via the pancreaticoduodenal arcade. This case highlights the importance of recognizing vascular variants and adapting embolization strategies to complex anatomy in emergency settings, reinforcing the pivotal role of interventional radiology in managing visceral aneurysms and acute abdominal hemorrhage.
We report a case of foetal-onset biventricular noncompaction with an MYH7 variant. Prenatal fetal hydrops and right heart failure resolved after birth, but progressive left heart failure developed at 5 months. This transition reflects how postnatal hemodynamic changes influence the biventricular noncompaction phenotype, necessitating vigilant monitoring for left heart failure in foetal-onset biventricular noncompaction cases with MYH7 variants.
Psoriasis frequently coexists with other immune-mediated conditions such as arthritis, alopecia areata, vitiligo, and hidradenitis suppurativa, many of which remain challenging to manage. To date, there have been rarely reported cases of treating psoriasis combined with lichen planus. We report a case of coexisting psoriasis and lichen planus in a female patient. Despite initial efficacy with acitretin and topical mometasone, the patient self-discontinued treatment over safety concerns, prompting a relapse. Upadacitinib was subsequently initiated and yielded marked improvement within 3 months. By discussing the disease interplay and treatment rationale, this report seeks to inform clinical decision-making.
Displacement of dental implants into the maxillary sinus is an uncommon yet clinically significant complication in implant dentistry. Migration toward the maxillary sinus ostium is rare and presents unique management challenges. This case report describes a 75-year-old woman who experienced intraoperative implant displacement into the left maxillary sinus during full-arch rehabilitation. Initially positioned at the sinus floor, the implant was later found at the ostium on cone-beam computed tomography one week after the incident, likely due to patient head movement. The patient reported mild discomfort but showed no signs of sinusitis or oroantral communication. Declining endoscopic surgery, she opted for a less invasive intraoral approach under local anesthesia. A custom-modified dental explorer with a notched tip was designed to securely engage the implant threads. A lateral sinus window was created, and indirect visualization was achieved using rhodium-coated mirrors. The implant was successfully retrieved through the intraoral route, and the sinus entry was sealed using a Bichat fat pad graft. Postoperative management included antibiotics, nasal decongestants, and analgesics. Healing progressed uneventfully, with no complications observed at 10-day, one-month, and three-month follow-ups. This case underscores the potential for dynamic implant migration within the sinus and demonstrates the effectiveness of customized instrumentation and early intervention in achieving successful, minimally invasive retrieval without the need for endoscopic techniques.
Mesenteric cyst is a rare benign cystic lesion of childhood that has a range of presentations varying from asymptomatic to acute abdomen. Hence, it is often an incidental finding or missed in diagnosis. Very few cases with complications have been reported. The underlying etiology still remains unknown despite multiple theories. Definitive diagnosis requires histopathological confirmation. We present a case of a 17-month-old girl with a large symptomatic hemorrhagic cyst who underwent complete excision of the cyst.
A 52-year-old gentleman presenting with tachycardia was found to have a large pericardial effusion and cardiac tamponade. This case was on a background of long-standing myelofibrosis managed with ruxolitinib, which was recently withheld for an orthopaedic procedure. He was diagnosed with presumed ruxolitinib discontinuation syndrome (RDS), resulting in a large pericardial effusion due to an increased inflammatory response. He was managed with a pericardial window and prompt recommencement of ruxolitinib and steroids. This case highlights the importance and increasing prevalence of RDS with the serious consequence of cardiac tamponade. Symptoms of RDS should be recognised and managed with recommencement of a JAK inhibitor and steroids.
Right aortic arch encompasses multiple anatomical variants, among which the subtype involving an aberrant left subclavian artery is most frequently encountered. By contrast, a right aortic arch with an anomalous origin of the left brachiocephalic artery arising from the main pulmonary artery constitutes an exceptionally rare congenital vascular anomaly and is most often identified incidentally. A rare case of a right aortic arch accompanied by an isolated left innominate artery is described. The anomaly was initially identified during the fetal period, subsequently confirmed after birth, and monitored over time to inform decision-making regarding the most appropriate timing of surgical intervention. Despite its extreme rarity, this congenital anomaly is being recognized with increasing frequency due to technological advances and heightened awareness of fetal echocardiography. In the postnatal setting, the integration of computed tomography angiography with three-dimensional reconstruction facilitates more accurate and timely diagnosis. This multimodal approach supports early identification and management, thereby potentially reducing the risk of irreversible injury.
Paroxysmal kinesigenic dyskinesia (PKD) is a rare movement disorder characterized by brief dystonic or dyskinetic attacks precipitated by sudden voluntary movement or arousal. Pregnancy complicated by PKD is extremely rare, and evidence guiding perinatal decision-making, especially regarding delivery mode and anesthetic management remains scarce. We report a term pregnancy in a woman with long-standing PKD managed through a multidisciplinary team involving neurology, obstetrics, anesthesiology, and neonatology. Following individualized optimization of antiseizure medications (ASMs) and intensive antenatal surveillance, the patient showed moderate symptomatic improvement during pregnancy. Given the high risk of pain, stress, or movement-triggered dystonic attacks with transient loss of voluntary motor control during labor, elective cesarean delivery was planned. Combined spinal-epidural anesthesia (CSEA) was performed to provide effective analgesia while minimizing arousal and sensory triggers. The procedure was uneventful, with no perioperative dystonic episodes. A healthy neonate was delivered with no congenital anomalies. This case demonstrates that favorable maternal and neonatal outcomes can be achieved in pregnancies complicated by PKD through individualized antiseizure medication management, multidisciplinary care, and trigger-oriented perinatal planning. The structured clinical decision-making pathway proposed in this study may provide a practical framework for managing this rare and challenging condition.
Fluid retention due to tyrosine kinase inhibitor administration might lead to pericardial effusion and right heart failure. Here, we present the case of a 63-year-old man with subacute cardiac tamponade complicated with heart failure, acute kidney injury, and generalized edema due to the bosutinib treatment.
Pancreatic adenocarcinoma typically presents late, most often with nonspecific symptoms such as weight loss, jaundice, and abdominal pain. Presentation with prolonged fever and weakness mimicking an infectious etiology is exceedingly rare. We report a case of a 54-year-old man with no known comorbidities who presented with persistent fever, cough, and weakness of 20 days' duration. Initial investigations suggested hepatic abscesses on ultrasonography. However, further evaluation with a contrast-enhanced computed tomography scan revealed a pancreatic uncinate process mass with multiple hepatic and skeletal metastases, suggestive of advanced pancreatic malignancy. Positron-emission tomography computed tomography confirmed the presence of a pancreatic mass with widespread metastatic lesions. Increased serum cancer antigen (CA) 19-9 and CA-125 levels also supported the diagnosis of metastatic pancreatic adenocarcinoma. This case highlights the diagnostic challenge of pancreatic carcinoma presenting atypically as fever with incidentally detected liver abscess-like lesions.
This case describes a 61-year-old woman with autoimmune autonomic ganglionopathy (AAG) on long-term intravenous immunoglobulin (IVIG) who developed progressive weakness and sensory loss, ultimately diagnosed as chronic inflammatory demyelinating polyneuropathy (CIDP). Initially evaluated for Guillain-Barré syndrome, cerebrospinal fluid analysis revealed albuminocytologic dissociation without central lesions on MRI. Despite IVIG therapy, symptoms worsened, and electrodiagnostic testing demonstrated demyelination with conduction block and secondary axonal loss, confirming CIDP. Subsequent plasma exchange therapy led to gradual improvement in strength, reflexes, and ambulation while autonomic function remained stable. The coexistence of CIDP and AAG is exceedingly rare and emphasizes the potential for shared autoimmune mechanisms within the peripheral nervous system. This case highlights the importance of recognizing overlapping autoimmune neuropathies, particularly when new or progressive symptoms arise despite appropriate therapy and adapting immunomodulatory treatment accordingly to optimize long-term outcomes.
Various drainage methods have been used for unresectable malignant hilar biliary obstruction (MHBO). However, controlling cholangitis and obstructive jaundice is still challenging. We performed a phase I/II study of endoscopic ultrasound-guided hepaticogastrostomy (EUS-HGS) as primary drainage for MHBO. This prospective Phase I/II study included 20 patients requiring biliary drainage for MHBO recruited from June 2021 to December 2023. The primary endpoint was safety. The secondary endpoints were clinical efficacy, reintervention methods, and frequency. The types of bile duct stenosis were Bismuth type I/II/IIIa/IIIb/IV; 4/1/5/3/7 cases. Fifteen cases (75%) had cholangitis before the EUS-HGS was performed. Technical success rate; 100% (20/20), clinical success rate; 75% (15/20). The four cases in which clinical success was not achieved were Bismuth type IV with cholangitis and obstructive jaundice, and all cases required additional drainage for the right bile duct.Early adverse events were stent migration: one case (severe) and peritonitis: one case (mild). The safety of EUS-HGS for the primary drainage of unresectable MHBO is acceptable. Regarding clinical efficacy, based on the results observed in patients with Bismuth type IV obstruction, we acknowledge that further investigation is warranted. Trial Registration: UMIN-CTR: UMIN000047702.