Rare Case Research Articles

Crystalline keratopathy secondary to the use of ciprofloxacin after cataract surgery with confirmation by histopathological study: A case report and review of the literature

ObjectiveTo report the case of a 75-year-old patient who presented crystalline keratopathy secondary to the use of topical ciprofloxacin with histopathological verification, after cataract surgery without complications. MethodCase report with clinical and photographic follow-up, as well as slides with samples of epithelium and crystalline deposits. ResultsCorneal deposits resolved after drug suspension, topical lubricant change, and subsequent surgical debridement. The histopathological examination reported epithelial cells and basophilic particles compatible with drug precipitates. ConclusionsCrystalline keratopathy is a condition in which crystals of various kinds are deposited in the corneal epithelium and/or in the anterior stroma. It may have an infectious, pharmacological cause or, in rarer cases, corneal dystrophies. Certain factors such as a previous epithelial defect, systemic pathology with diabetes mellitus, ocular surgery and previous dry eye can favor the deposition of ciprofloxacin leading to the formation of a keratopathy.

Steroid as a Saviour and Culprit in Pemphigus Vulgaris Therapy: A Rare Case Series

Introduction: Pemphigus is a group of autoimmune dermatological life-threatening diseases of which Pemphigus Vulgaris (PV) is the most common (1-5 cases/million/year) with a mortality rate of 60-90%. PV is generally characterized by painful oral erosions, along with or without cutaneous lesions, as blisters are often misdiagnosed. The management strategy of PV includes steroids along with adjuvant therapy. Case Presentation: We report the case of three female patients with a mean age of 30.67 years who were diagnosed with pemphigus vulgaris and was on steroid therapy for a long duration. The longterm steroid therapy in all these three patients resulted in adverse drug reactions such as Cushing syndrome, oral and vaginal candidiasis. Although the steroid was found to cause ADR among these patients, the treatment with steroids and rituximab was continued. The withdrawal of steroids results in flare-up conditions, whereas the addition of rituximab serves as a steroid sparring partner in the management of PV. However, premedication was administered before the infusion of rituximab to prevent potential toxicity associated with rituximab. Further, administration of pulse therapy over oral dosing of steroids lowers the risks of ADR and long-term complications associated with steroids. Conclusion: As PV requires the management with high doses of steroids along with the immunosuppressant, patients are more prone to developing drug-related problems. Hence, the rigorous monitoring of the therapy is vital to prevent adverse effects of long-term therapy. Clinical pharmacists and pharmacologists are the most suitable pillar of the healthcare system for drug therapy management.

Eliciting a single amino acid change by vaccination generates antibody protection against group 1 and group 2 influenza A viruses

Broadly neutralizing antibodies (bnAbs) targeting the hemagglutinin (HA) stem of influenza A viruses (IAVs) tend to be effective against either group 1 or group 2 viral diversity. In rarer cases, intergroup protective bnAbs can be generated by human antibody paratopes that accommodate the conserved glycan differences between the group 1 and group 2 stems. We applied germline-engaging nanoparticle immunogens to elicit a class of cross-group bnAbs from physiological precursor frequency within a humanized mouse model. Cross-group protection depended on the presence of the human bnAb precursors within the B cell repertoire, and the vaccine-expanded antibodies enriched for an N55T substitution in the CDRH2 loop, a hallmark of the bnAb class. Structurally, this single mutation introduced a flexible fulcrum to accommodate glycosylation differences and could alone enable cross-group protection. Thus, broad IAV immunity can be expanded from the germline repertoire via minimal antigenic input and an exceptionally simple antibody development pathway.

Anterior Cruciate Ligament Reconstruction in Tibial Eminence Avulsion Fracture: A Case Series

Introduction: Ruptures of the anterior cruciate ligament (ACL) are common orthopedic injuries, especially among patients participating in sports. Anterior cruciate ligament (ACL) avulsion fracture or tibial eminence avulsion fracture is a type of avulsion fracture of the knee. This article describes several cases of ACL rupture management with eminence avulsion. Case report: This case series describes 3 cases of ACL tear with eminence avulsion. The first patient was a 31-year-old female with right knee pain since 6 months ago after a traumatic fall from a motorcycle. The second patient, a 16-year-old girl with a history of bending her legs inward during exercise. The third patient is a 49-year-old female with a history of accidents and complaints of knee pain. All three patients found ACL tear accompanied by an avulsion fracture on the tibial eminence and then undergo ACL reconstruction. Discussion: The most common mechanism of trauma, which leads to a disruption of ACL, is a trauma forcing the knee in hyperextension combined with valgus-external rotation. At times, these lesions can occur in adults; in this instance, the main cause is high-energy trauma, such as road accidents. Based on case reports and previous research, the management approach in this case is still debatable in children and adults and requires a specific approach in each case. Conclusion: The management of ACL reconstruction in patients with tibial eminence avulsion involvement is still a challenge for orthopedic surgeon. Given the rarer cases, a different approach is needed in young and adult cases. Each case requires proper management to improve the patient's functional outcome and their quality of life. Keywords: anterior cruciate ligament reconstruction, avulsion fracture, tibial eminence avulsion

Neuroophthalmic Manifestations and Management of Cryptococcal Meningitis in HIV-negative Pregnant Woman : A Rare Case

Abstract
 Introduction : Cryptococcal meningitis (CM) is Cryptococcus infection affecting mostly immunocompromise patients. However, immunocompetent and pregnant woman can also be affected. This condition can cause neuroophthalmic manifestations which may result in permanent vision loss or even death if not managed properly. However, literatures on neuroophthalmic manifestations and management of HIV-negative pregnant woman with CM are very scarce.
 Case Illustration : We present a rare case of 25 y.o. HIV-negative pregnant woman with neuroophthalmic manifestations associated with CM. The patient was consulted from neurology department with complaints of painless vision loss accompanied by diplopia, severe headache, nausea, and frequent vomitus. Ophthalmological examination showed best corrected visual acuity 20/40 in both eyes, ophthalmoplegia, and grade 4 papilledema. Lumbal puncture result was 89 cmH20. Cerebrospinal fluid (CSF) analysis showed mononuclear cells domination, increased protein, and decreased glucose. Indian ink stain showed Cryptococcus organism. Patient was given intravenous fluconazole, topical nonsteroid anti-inflammatory drug, and therapeutic lumbal punctures which result in clinical improvement.
 Discussion : CM can cause neuroophthalmic manifestations resulting from increase intracranial pressure (ICP) due to fungal load in arachnoid granulation which obstruct CSF outflow. The manifestations found in this patient were papilledema, ophthalmoplegia, and diplopia due to 6th nerve palsy. The management was multidisciplinary with obstetrics-gynecology and neurology department, focused in pathogen eradication and ICP management to prevent neuroophthalmic sequelae, with considerationof maternal-fetal life saving dilemma.
 Conclusion : Various neuroophthalmic manifestations can be involved in CM. Prompt and appropriate therapy in a rare case of HIV-negative pregnant woman with CM can decrease both maternal and fetal morbidity and mortality risk.

Research Note: A Novel Sullivan Method Projection Framework With Application to Long COVID.

Originally developed for estimating healthy life expectancy, the traditional Sullivan method continues to be a popular tool for obtaining point-in-time estimates of the population impacts of a wide range of health and social conditions. However, except in rare data-intensive cases, the method is subject to stringent stationarity assumptions, which often do not align with real-world conditions and restrict its resulting estimates and applications. In this research note, we present an expansion of the original method to apply within a population projection framework. The Sullivan method projection framework produces estimates that offer new insights about future trends in population health and social arrangements under various demographic and epidemiologic scenarios, such as the percentage of life years that population members can expect to spend with a condition of interest in a time interval under different assumptions. We demonstrate the utility of this framework using the case of long COVID, illustrating both its operation and potential to reveal insights about emergent population health challenges under various theoretically informed scenarios. The traditional Sullivan method provides a summary measure of the present, while its incorporation into a projection framework enables preparation for a variety of potential futures.

Pediatrik Bir Hastada İskemik İnmeye Neden Olan Primer Santral Sinir Sistemi Vasküliti: Nadir Bir Olgu Sunumu

Amaç: Çocukluk çağında inme edinsel beyin hasarına yol açabilen nörolojik bulgu ve semptomlarla karakterize nöropatolojik bir sendromdur. Etiyolojide sıklıkla kardiyoembolik, trombofilik, genetik hastalıklar ve nadiren serebral vaskülitler görülür. Serebral vaskülit terimi sistemik hastalıklara sekonder serebral tutulumu ve yalnızca santral sinir sitemini tutan primer vaskülitleri kapsamaktadır. Primer santral sinir sitemi vasküliti nadir görülmekle birlikte, tanısı sekonder nedenler ve sıklıkla karışan moya moya arteriopatisi, geçici serebral vazokonstrüksiyon sendromu, mikroskobik poliarterit gibi tanıların dışlanmasına dayanır.
 
 Olgu: 14 yaşında erkek hasta bayılması ve o sırada başlayan konuşma bozukluğu olması üzerine acil servise getirildi. Fizik muayenede Glasgow koma skalası skoru 15, motor kuvvetinin sağ alt ve üst ekstremitelerde 1/5, sol alt ve üst ekstremitelerde 5/5, fasiyal asimetri ve konuşmada artikülasyon bozukluğu olduğu görüldü. Sistemik muayenesinde travma bulgusu saptanmadı. Difüzyon ağırlıklı manyetik rezonans görüntülemesinde sol bazal ganglionlar, posterior frontal ve presantral giruslarda akut iskemik inme ile uyumlu difüzyon kısıtlılığı olduğu tespit edildi. Acil serviste enoksaparin (6000 ANTI-XA IU/0.6 ml subkutan 2x1) tedavisi başlanan hasta iskemik inme ön tanısı ile hastaneye yatırıldı. Hastanın bilgisayarlı beyin tomografisi, kontrastlı beyin anjiografi ve manyetik rezonans venografisinde patoloji saptanmadı. Ayrıca beyin manyetik rezonans anjiografi ve dijital subtraksiyon anjiyografide patoloji görülmedi. Olası diğer tanılar dışlandıktan sonra primer santral sinir sistemi vasküliti düşünülerek yüksek doz metilprednizolon tedavisi başlandı.
 
 Sonuç: Çocukluk çağında iskemik inme erişkinlere oranla daha az görülse de nörolojik disfonksiyon, tekrarlayan inme riski ve artmış mortalite ile ilişkilidir. Primer santral sinir sistemi vasküliti tanısı dikkatli bir ayırıcı tanı sürecine dayanan nadir bir pediatrik inme nedeni olduğu hatırlanmalıdır.

Use of Over the Scope Clips in the Management of Non-Variceal Upper GI Bleeding

Use of Over the Scope Clips in the Management of Non-Variceal Upper GI Bleeding

A Rare Case of Acute Appendicitis Associated with Endoscopic Mucosal Resection (EMR) of A Large Cecal Polyp

A Rare Case of Acute Appendicitis Associated with Endoscopic Mucosal Resection (EMR) of A Large Cecal Polyp

Osteomyelitis Tuberculosis in long bone mimicking bone malignancy: A case report

Osteomyelitis tuberculosis in developing countries is still a severe problem. Early diagnosis and treatment of osteomyelitis tuberculosis are very important because a delay in diagnosis can resemble malignancies. Osteomyelitis tuberculosis is often difficult to distinguish from a malignancy both clinically and radiologically. We present a rare case of a 10 years old boy with chief complaint was a lump around his left knee. He was treated earlier at a peripheral hospital as a malignancy process of bone, and referred to Dr. Soetomo Hospital, as a tertiary hospital, and decided to do an open biopsy in the operating room. On clinical examination was found lump and deformity with surrounding soft tissue swelling and limited ROM. On X-ray examination obtained pathological fractures and bone destruction in the distal femur with a lytic lesion on along the femur metadiaphysis without joint involvement and periosteal reaction proximally, from the examination it cannot be distinguished between osteomyelitis tuberculosis and malignancy. During the surgery, a voluminous amount of odourless pus with multiple septa surrounding the diaphysis. Pus culture and some of the tissue was taken to be examined by pathology that shows connective tissues infiltrated by inflammatory cells. Zeihl-Nielsen staining was done and obtained an image of acid-fast bacilli. Clinical and Radiological Features of osteomyelitis tuberculosis can mimic malignancy. The definitive diagnosis of tuberculosis osteomyelitis is usually made by CT-guided needle aspiration cytology and histologic examination using open biopsy. The latter has better sensitivity, especially if the acid-fast bacilli is obtained.

Neonatal Ebsteins Anomaly presenting with unique features: Rare case report

Ebstein’s anomaly (EA) of the tricuspid valve is an uncommon malformation, representing an estimate 0.3% to 0.6% of all cases of congenital heart disease. The basic defect involves a deformed and displaced tricuspid valve, resulting in symptoms that vary with age of the patient and severity of the defect. Symptoms are referable to progressive heart failure and cyanosis resulting from severe tricuspid insufficiency. Once symptoms appear the only treatment for Ebstein’s anomaly is surgical intervention. Infants with symptomatic Ebstein’s anomaly have a poor prognosis, with an expected mortality rate of 50% to 75%. Previous attempts at palliative surgical treatment in this neonatal group have produced equally dismal results. Herein, we are reporting a very sick 3 day old neonate afflicted with Ebsteins anomaly with double-outlet left ventricle (DOLV) associated with myriads of other significant congenital heart defects.

Determining the manner of death in victims in drug-induced psychosis: a case of an atypical head stab wound.

This case report presents a rare case of an atypical head stab wound suffered by a drug addict and inflicted with a screwdriver during drug-induced psychosis. It describes the diagnostic and treatment procedures in the hospital and the findings of the subsequent autopsy. It also analyzes the review of the interpretation of the CT scans made upon admission and the subsequent treatment by an independent medical review panel, which revealed signs of medical mismanagement. Therefore, it also discusses the legal consequences that the case may have involved for the attending physicians in addition to the consequences for the suspected perpetrator. The report raises many issues encountered in the case in terms of the clinical treatment and forensic determination of the manner of death in cases of injuries caused by sharp instruments and highlights the importance of comprehensive evaluation of the circumstantial evidence together with the clinical or autopsy findings, since such evidence may sometimes be overlooked in clinical practice.

Modified Senning procedure in a patient with dextrocardia with left atrial isomerism and anomalous systemic venous drainage: a rare case report.

The atrial switch procedure by Senning or Mustard technique primarily aims in correcting parallel systemic and pulmonary circulations at atrial level. This procedure may be used in late presenting D-transposition of great arteries with a deconditioned left ventricle, congenitally corrected transposition of great arteries and isolated ventricular inversion. We describe the case of a child with dextrocardia, left atrial isomerism with complex pulmonary and systemic venous drainage resulting in mixing at atrial level. She was successfully operated by modified Senning procedure performed through the left-sided atrium.

Successful management of an advanced interstitial ectopic pregnancy in a resource-limited setting: a case report

BackgroundInterstitial ectopic pregnancy is an ectopic gestation developing in the uterine part of the fallopian tube. The condition is rare and presents challenges for clinical as well as radiological diagnosis. This case report presents a rare case of interstitial ectopic pregnancy diagnosed intraoperatively.Case presentationA 36-year-old Black woman, referred from a peripheral health facility, presented at the emergency department with severe abdominal pains, vaginal spotting, nausea, and vomiting, with a 2-month history of irregular menstrual flow. Clinical and laboratory findings were suggestive of an acute abdomen likely due to a ruptured ectopic pregnancy (ultrasound was not available). An emergency exploratory laparotomy was done, which revealed a right adnexal ruptured interstitial pregnancy of a lifeless female fetus weighing 500 g (estimated mean gestational age of 22–23 weeks). The left fallopian tube looked normal. The site of rupture was repaired, followed by cleaning and closure of the abdomen. The post-operative period was uneventful, and the patient was discharged on postoperative day 7.ConclusionInterstitial pregnancies are uncommon and rarely attain advanced gestational ages, as in this case, compared with other tubal ectopic pregnancies. However, women presenting with signs of hypovolemic shock and acute abdomen, with a positive pregnancy test, warrant a high index of suspicion.

Transcatheter renal arterial embolization for intractable urinary fistula occurring after robot-assisted partial nephrectomy: a case report.

Partial nephrectomy is the standard surgical procedure for small renal tumors. Since the advent of robot-assisted partial nephrectomy (RAPN), the number of cases of renal tumors undergoing the procedure has increased exponentially. Urinary fistula is a complication of partial nephrectomy. Conservative management using ureteral stents is useful in most cases of urinary fistulas. However, some patients develop intractable urinary fistulas. Herein, we report a case in which vascular embolization was useful for treating an infected and intractable urinary fistula that developed after RAPN. A 59-year-old man was accidentally found to have a right renal tumor (approximately 3cm in diameter) during a physical examination. Pathology was clear cell carcinoma. RAPN was performed owing to the small size of the renal tumor; however, postoperatively, an intractable urinary fistula with an isolated calyx developed, which was successfully treated with transcatheter renal arterial embolization (TAE). We encountered a rare case of infected refractory urinary fistula with an isolated calyx in which TAE was successful. TAE seems useful in treating intractable urinary fistulas with an isolated calyx occurring after RAPN.

Growing Pains: A Rare Case of a Temporal Dermoid Cyst in an Adult and Review of the Literature

Dermoid cysts are common cutaneous developmental anomalies that occur due to the entrapment of ectodermal elements during embryonic closure. While the exact origin of dermoid cysts is unknown, they are theorized to originate from multipotent embryonic stem cells or translocated epithelial cells. Dermoid cysts are typically congenital but are sometimes diagnosed later in childhood and rarely in adulthood. They are reported as the most common pediatric skull tumors. Surgical excision is recommended to prevent enlargement, invasion, rupture, or infection. Temporal dermoid cysts, specifically, are extremely rare, with only a few cases having been reported in adulthood. We present a case of a 51-year old female with an enlarging left temporal mass found to be a dermoid cyst. The mass was excised, resulting in complete resolution of symptoms. This case highlights the importance of early excision, radiologic studies, and a tailored management approach based on cyst location. Our proposed treatment algorithm provides guidance for clinicians in identification and management of adult dermoid cysts.

Repositioning of herniated buccal fat pad following blunt trauma - Rare case report in pediatric patients

Herniation of the buccal fat pad in pediatric patients is a rare injury, occurring due to a breach in the buccinator muscle, often termed traumatic pseudolipoma in infants. This condition plays a crucial role in cheek contour in adults. Two case reports detailing the management of this rare injury in young children emphasize the importance of assessing mucosal perforation and considering previous trauma history for accurate diagnosis. These reports provide valuable insights for healthcare professionals, contributing to the understanding and management of uncommon medical conditions.

Spinal and cervical nodal metastases in apatient with glioblastoma.

This article presents the rare case of a54-year-old gentleman with primary glioblastoma developing multiple extracranial metastases 7months after diagnosis. Initially, the patient complained of progressive headaches, confusion, and weakness of the left arm. Magnetic resonance imaging of the brain showed aright temporoparietal tumor with substantial surrounding subcortical edema and midline shift to the left. Two consecutive craniotomies resulted in complete microsurgical resection of the lesion. Histology was consistent with aWorld Health Organization gradeIV, IDH-wildtype glioblastoma. Further treatment was standard chemoradiation including intensity-modulated radiotherapy with oral temozolomide chemotherapy. Seven months after diagnosis, the cranial lesion progressed, and the patient developed painful metastases in multiple bones and suspicious right-sided cervical lymph nodes. Immunohistochemistry and molecular signature supported the case of ametastatic glioblastoma. Further treatment was palliative radiotherapy of the spinal lesions along with symptomatic pain management. Extracranial metastasis of glioblastoma is arare complication of which only afew cases have been reported in the literature. Little is known about the precise mechanisms of tumor dissemination and the appropriate treatment.

Successful ductal stent implantation for initial palliation in two patients with aortic atresia and interrupted aortic arch.

The coexistence of aortic valve atresia and interrupted aortic arch are an extremely rare condition. In this pathology, blood flow to the ascending aorta and coronary arteries should be provided through the ductus arteriosus or collaterals originating from the descending aorta. In rare cases where bilateral ductus arteriosus is present, they can provide circulation. Here, we report two cases in which coronary arteries and ascending aorta were supplied by one ductus arteriosus and distal systemic circulation is supplied by a second ductus arteriosus in one patient and a collateral artery in the other. Initial palliation was successfully performed by bilateral pulmonary artery banding and transcatheter ductal stent implantation in both cases.

Respiratory epithelial adenomatoid hamartoma in a child with nasal polyposis. Case report

Benign and malignant formations of the nose and paranasal sinuses in the early stages of development are asymptomatic or manifest minor nonspecific signs, and therefore often remain unrecognized. Hamartoma is a tumor-like formation of a dysembriological nature, consisting of excessive tissues peculiar to the affected organ. Due to the rarity of the occurrence of hamartomas of the sinonasal region, especially in pediatric practice, their diagnosis may present certain difficulties, since they can mimic other formations, such as nasal polyps or inverted papilloma, may occur as an isolated formation in the nasal cavity, or in combination with chronic rhinosinusitis, nasal polyposis, allergic rhinosinusitis. Aim – to report a rare clinical case in pediatric practice of respiratory epithelial adenomatoid hamartoma associated with allergic rhinosinusitis and polyposis, display the relationship between the occurrence of respiratory epithelial adenomatoid hamartoma and chronic inflammation of the nasal cavity and paranasal sinuses, to carry out differential diagnosis with other exophytic aggressive lesions of the nasal cavity, combine and summarize available data on sinonasal hamartomas. The article describes a clinical case of respiratory epithelial adenomatoid hamartoma in a 17-year-old child with a previous 4-year follow-up with nasal cavity polyposis and a history of repeated polypotomy. The results of endoscopy and computed tomography of the nasal cavity and paranasal sinuses, the results of surgical intervention, as well as the conclusions of a histological examination of the tumor removed from the patient are presented. The authors analyzed the domestic and foreign literature, on the basis of which they presented a differential series of sinonasal hamartomas. Respiratory epithelial adenomatoid hamartoma is a rare pathological formation of the sinonasal region, which manifests itself as an isolated polypoid mass in the nasal cavity or as an accidental surgical finding in patients with chronic sinusitis. The association with nasal polyps supports the hypothesis that inflammation may be one of the inducing factors. Respiratory epithelial adenomatoid hamartoma, although rare, should be considered in the differential diagnosis of exophytic lesions of the nasal cavity. The tactics of treatment involves endoscopic removal of the formation within healthy tissues, which provides good long-term results, relapses are extremely rare.