Journal of Medical Case Reports最新文献

Background: The coexistence of pheochromocytoma and hyperaldosteronism is a rare and clinically significant finding with diagnostic challenges that need to be considered in the workup of patients with hypertension.

Case presentation: This case report describes a 47-year-old Iranian man who initially presented with cold symptoms, chills, and headaches. Despite being diagnosed with panic disorder, his symptoms worsened, leading to a systolic blood pressure crisis. The results indicated elevated levels of 24-hour urine vanillylmandelic acid, metanephrine, and normetanephrine, suggesting increased catecholamine levels. An increase in serum aldosterone was also observed. Further evaluation revealed a 4 cm left adrenal mass and subsequent tests confirmed the diagnosis of pheochromocytoma and hyperaldosteronism. The patient underwent left adrenal gland resection, resulting in complete resolution of symptoms and normalization of test results.

Conclusion: This case highlights the importance of considering rare coexisting endocrine disorders in patients presenting with hypertension. Appropriate diagnosis and management of concomitant pheochromocytoma and hyperaldosteronism are crucial for favorable outcomes and may offer insights into potential overlaps in disease pathways.

Background: Interventional therapy of trigeminal neuropathic pain has been well documented; however, intraoperative monitoring and management of pain hypersensitivity remains barely reported, which may pose a great challenge for pain physicians as well as anesthesiologists.

Case presentation: A 77-year-old Han Chinese male, who suffered from severe craniofacial postherpetic neuralgia, underwent pulsed radiofrequency of trigeminal ganglion in the authors' department twice. The authors successfully placed a radiofrequency needle through the foramen ovale during the first procedure with local anesthesia and intravenous sedation (dexmedetomidine). The patient reported about 50% pain reduction postoperatively, and the second procedure was performed 1 week later. However, the intraoperative administration of sedative agents was suspended owing to hemodynamic instability during the second session. As a result, the patient displayed hypersensitivity to the percutaneous operation under local anesthesia and the authors failed to place the needle inside the Meckel's cave for uncontrollable breakthrough pain. The patient still needed to take oral medication for pain control, oxycodone (10-20 mg, every 12 hours) and pregabalin (75 mg, two times a day) in the last follow-up at 1.5 years after discharge.

Conclusion: The authors report a failure case of percutaneous puncturing operation with trigeminal neuropathic pain, potentially caused by intraoperative sensitization. It is essential to monitor and prevent hypersensitivity to both innoxious and noxious stimuli in patients with neuropathic pain syndrome, especially at surgical sites close to the area of nerve injury.

Background: Undifferentiated pleomorphic sarcoma, previously called malignant fibrous histiocytoma, is a type of malignant mesenchymal tumor (sarcoma) of soft tissue and sometimes bone. It is uncommon in the oral cavity and very sporadic in the maxillary sinus. Microscopic diagnosis of this malignancy in the maxillary sinus can be very challenging, because there is a range of features that may overlap with other benign and malignant tumors.

Case presentation: In this paper, we report a case of undifferentiated pleomorphic sarcoma in the maxillary sinus of a 61-year-old Iranian man who was referred to the maxillofacial surgery ward due to pain and swelling of the upper jaw and visual problems in the right eye. In the initial incisional biopsy, peripheral giant cell granuloma was diagnosed in the hospital service. Yet, on request of the surgeon, during reviewing the slides in the oral pathology service of the School of Dentistry, and using an immunohistochemical method, undifferentiated pleomorphic sarcoma was diagnosed. In this paper, a case of undifferentiated pleomorphic sarcoma in the maxillary sinus is reported, with an emphasis on the management of its problems and diagnostic errors.

Conclusion: This study reviews the challenges and histopathological diagnostic errors of this uncommon tumor in this rare location. This lesion may be similar to other malignant tumors, and its correct diagnosis requires a detailed and complete examination.

Background: Poikiloderma with neutropenia is a rare genetic disorder primarily characterized by the presence of poikiloderma and congenital chronic neutropenia. Mutations in the C16orf57 gene, which encodes the USB1 protein, are implicated as the underlying cause of poikiloderma with neutropenia.

Case presentation: Our patient, an 11-year-old Syrian male child who presented with poikiloderma, palmoplantar keratoderma, pachyonychia, recurrent infections, and neutropenia, is considered to be the first documented case in Syria. Clinical examinations, laboratory tests, radiographic imaging, and genetic analyses have been conducted, with the latter being essential and definitive for diagnosis.

Conclusion: This study aimed to evaluate whether poikiloderma with neutropenia should be considered for differential diagnosis because of its diagnostic complexity, emphasizing the importance of follow-up for the early identification of potential complications.

Introduction: Amyand's hernia, an uncommon condition characterized by the presence of the appendix within an inguinal hernial sac (< 1% incidence), poses diagnostic and therapeutic challenges. Often it is an intraoperative finding, with almost no clinical symptoms.

Case presentation: This is a case of an Indian male in his early 80 years, diagnosed with bilateral direct inguinal hernias, one of which contained a noninflamed appendix. Given the thinned out abdominal wall, dense adhesions, and no demarcation between layers, the decision to proceed with a modified Bassini's with Lichtenstein mesh repair without appendectomy, guided by intraoperative findings and the Losanoff-Basson Classification, reflecting the complex interplay between individual patient factors and intraoperative considerations. The patient did well during his postoperative stay and was in good health on a 45 day follow-up, with no complaints suggestive of recurrence or obstruction.

Conclusion: This case underscores the importance of tailored management strategies and highlights, especially in cases where recurrence and postoperative wall integrity are in question, the ongoing need for research to refine treatment guidelines for Amyand's hernia, especially in cases of appendicitis not diagnosed preoperatively.

Background: The diagnosis and management of female genital conditions (Rodriguez et al. in Clin Anat 34(1):103-107, 2020. https://doi.org/10.1002/ca.23654 ) are often challenging. The atypical presentations, combined with patient hesitancy to be subjected to an examination by a male urologist, are factors that limit a timely diagnosis. Para-urethral cysts (Pastor and Chmel in Int Urogynecol J 29(5):621-629, 2018. https://doi.org/10.1007/s00192-017-3527-9 ) are often incidentally detected by gynecologists during pelvic examination for other reasons. Patients rarely present with complaints of lower urinary tract symptoms and dyspareunia affecting sexual life. Diagnosis in most instances can be made by physical examination but often a detailed evaluation with ultrasonography, voiding cystourethrogram, computed tomography, or magnetic resonance imaging is needed. The definitive management of symptomatic para-urethral cysts is through surgical excision.

Objective: This report aims to reflect clinically upon a rare pathology of the female genital system.

Case presentation: We present the case of a 36-year-old, sexually active, Indian (Asian) woman with a 6-month history of progressively worsening lower urinary tract symptoms, consisting of dysuria, post-micturition dribble, increased urination frequency, and significant dyspareunia. Physical examination in the lithotomy position revealed a cystic lesion located in the midline slightly to the left of the anterior vaginal wall. Magnetic resonance imaging also revealed a T2/T1 hyperintense lesion located below the level of the pubic symphysis. The patient was posted for exploration under anesthesia and the cyst was excised completely. The histopathology findings were consistent with para-urethral gland cyst with ulceration and squamous metaplasia.

Conclusion: Any lower urinary tract symptoms in a woman needs thorough clinical examination. Association of para-urethral cyst with lower urinary tract symptoms and dyspareunia is rare, and if present, always warrants surgical excision.

Background: Superficial acral fibromyxoma is a noncancerous, benign tumor of soft tissue with an unidentified origin. Occurrences of abnormalities on the palm are less frequently documented.

Case report presentation: A 47-year-old East Asian woman presented with a palm tumor on her left knuckle that had been present for 4 months. Four months prior to treatment, a reddish nodule appeared at the site due to continuous friction, but the patient did not pay attention to it. Upon physical examination, a pale-reddish nodule measuring approximately 0.8 × 0.8 cm was observed on the flexion side of the fourth metacarpophalangeal joint of the left hand. The nodule was nearly round, exhibited distinct margins, featured slight surface keratosis, and displayed a collar-like alteration at its periphery. The stroma showed significant fibrous proliferation and a myxoid matrix. The cellular composition consisted primarily of spindle-shaped fibroblasts, which were locally arranged in bundles within the stroma. Sparse slender blood vessels were distributed along with observed red blood cell extravasation. Immunohistochemistry of the tumor showed CD34 (+), CD99 (-), desmin (-), epithelial membrane antigen (-). Extensive resection of the mass was performed.

Conclusion: A rounded mass on the palm of the hand may be superficial acral fibromyxoma. The optimal approach for treating superficial acral fibromyxoma involves complete surgical removal of the mass followed by postoperative monitoring.

Background: Horseshoe kidney is the most common renal fusion anomaly, occurring in approximately 1 in 500 individuals worldwide. It is characterized by abnormalities in kidney position, rotation, and vascular supply. While often asymptomatic, horseshoe kidneys can lead to urological complications, primarily due to ureteric obstruction and impaired urinary drainage.Although ureteropelvic junction obstruction (UPJO) is uncommon in horseshoe kidneys, its occurrence presents unique diagnostic and management challenges. This case explores the etiology, clinical presentation, and surgical management of UPJO in a patient with a horseshoe kidney, highlighting the critical role of urologists in addressing these anatomical and functional complexities.

Case presentation: We report the case of a 3-month-old African male infant who presented with a two-month history of excessive crying and symmetrical abdominal distension. Radiological evaluation revealed a horseshoe kidney with decreased parenchymal thickness in the left kidney and marked hydronephrosis of the pelvis and calyces, with abrupt cessation at the left ureteropelvic junction. Surgical correction of the obstruction was performed successfully. The patient was followed for six months, during which he remained symptom-free with normal renal function tests.

Conclusion: Ureteropelvic junction obstruction in a horseshoe kidney presents unique anatomical challenges. Symptomatic cases require thorough imaging for diagnosis and surgical planning. Pyeloplasty is the typical surgical intervention, meticulously planned to address these complexities. This ensures effective relief of obstructions and proper urinary drainage.

Background: While the potentially hazardous effects of intraocular perfluorocarbon gases during air travel have been recognized, the equivalent risk of intraocular air tamponade is less known and has, to the best of our knowledge, not been reported yet.

Case presentation: A 52-year-old white female experienced a complete loss of vision and pain in her left eye during air travel following pars plana vitrectomy with air tamponade. Clinical and multimodal imaging findings only a few hours after emergency landing indicated a transient central retinal artery occlusion due to a significant increase in intraocular pressure during the flight.

Conclusion: Air travel, even with a relatively small volume of residual air tamponade, can lead to potentially serious complications.

Background: Antibiomania is the manifestation of manic symptoms secondary to taking an antibiotic, which is a rare side effect. In these cases, the antibiotics most often incriminated are macrolides and quinolones, but to our knowledge, there are no published cases of antibiomania secondary to cotrimoxazole. Furthermore, we also provide an update of pharmacovigilance data concerning antibiomania through a search of the World Health Organization (WHO) database.

Case presentation: We present the case of a 66-year-old Caucasian man with history of a brief psychotic episode 16 years prior. He received levofloxacin for a urinary infection with Klebsiella pneumoniae, with a switch to cotrimoxazole after 5 days of treatment. Shortly after the introduction of cotrimoxazole, the patient presented with a significant flow of speech, which was incoherent. After 2 days of treatment, levofloxacin was restarted, but given the lack of improvement in symptoms, the patient was hospitalized. Levofloxacin was quickly replaced by ceftriaxone for 3 days without improvement, thus a psychiatric examination was requested. This examination identified manic symptoms, and so psychotropic treatment combining 5 mg of haloperidol and 75 mg of loxapine per day was initiated. Subsequently, there was a gradual improvement in symptoms within a week. Moreover, the results of the various investigations that were carried out came back normal, which this led to suspicion of an iatrogenic cause (antibiotherapy).

Discussion: We discuss the latest pharmacovigilance data and several etiopathogenic hypotheses suggested in literature, with growing interest concerning the microbiota-gut-brain axis. It appears important to pay particular attention when taking antibiotics in the context of bipolar disorder because this association seems to precipitate the appearance of secondary mania. Management consists of stopping the responsible antibiotic, with the possibility of adding psychotropic treatment.

Conclusion: It is important to be aware of antibiomania to adopt appropriate and early treatment. We think that this case report could be useful as a clinical reminder and could possibly spark lines of research.