Journal of Medical Case Reports最新文献

Background: The spontaneous rupture of an artery aneurysm during the perinatal period is considered a serious complication associated with the physiological alteration by pregnancy and delivery. The rupture of an ovarian artery aneurysm is rare and leads to rapid retroperitoneal hemorrhage. Here, we report one case complicated by postpartum hypertensive disorder of pregnancy associated with massive bleeding into retroperitoneal cavity by the spontaneous rupture of ovarian artery aneurysm after caesarean section, and reviewed previous literature.

Case presentation: A 41-year-old Japanese woman was referred to our hospital four days after undergoing cesarean delivery because of hemorrhagic shock with abdominal and right flank pain. Contrast-enhanced computed tomography revealed a large retroperitoneal hematoma. Angiography showed a ruptured right ovarian artery aneurysm, anastomosing with a dilated uterine artery. She was successfully treated with transcatheter arterial embolization. She was diagnosed with postpartum hypertensive disorder of pregnancy 3 days after the embolization.

Conclusion: The rupture of ovarian aneurysm can occur during the perinatal period, although it has not been widely understood among obstetricians. Multiparity and hypertensive disorder of pregnancy might be risk factors for the rupture of an ovarian aneurysm. Early diagnosis and therapy, such as transcatheter arterial embolization in preparation for an emergency laparotomy, are crucially important for the management of this fatal disease.

Background: Although vaginal stenosis following pelvic or vaginal radiotherapy for cancer treatment is a common complication, postpartum vaginal stenosis is a very rare obstetric complication. We report two cases of postpartum vaginal stenosis.

Case presentation: The first was case was a 30-year-old para-I Ethiopian woman who presented with a history of foul-smelling vaginal discharge on her 11th postpartum day. Pelvic magnetic resonance imaging (MRI) demonstrated a 1.4 cm distal vaginal stenosis. The second case was a 39-years-old para-II Ethiopian woman who presented with history of amenorrhea and dysparunia of 20 months duration. Abdominopelvic ultrasound examination revealed hematometrocolpos secondary to postinflammatory vaginal stenosis, and 3300 ml of hematocolpometra was drained. In both cases, adhesion excision with successful vaginal reconstruction was performed.

Conclusion: Postpartum vaginal stenosis is a rare obstetric complication and vaginal adhesions excision with regular vaginal dilation is the recommended management for it.

Background: Cogan syndrome is a rare autoimmune systemic vasculitis presenting with interstitial keratitis and audiovestibular symptoms. The atypical form, characterized by more extensive ocular lesions with audiovestibular symptoms appearing with a longer delay and more frequent systemic features, is usually underdiagnosed, delaying treatment.

Case presentation: We report the case of a 30-year-old Mediterranean female who presented recurrent left red and painful eye. The evolution in flare-ups during 3 years, associated with a nonspecific biological inflammatory response, motivated ocular biopsies demonstrating diffuse inflammatory changes from the cornea to the sclera. Since Morocco is an endemic region for tuberculosis, the laboratory tests were expanded to identify the latent forms based on the tuberculin skin test and QuantiFERON TB Gold in tube assay, both positive for Mycobacterium tuberculosis infection. Thus, antibacillary chemotherapy was started for 6 months, with reappearance of the symptoms at the end of treatment. Three years later, the patient presented isolated and fluctuating audiovestibular Ménière-like symptoms, with progressive sensorineural hearing loss. The nonstabilization under usual medical treatment along with her diffuse inflammatory ocular lesions led to the diagnosis of atypical Cogan syndrome. The patient received long-term corticosteroid with notable improvement.

Conclusion: Cogan syndrome is a rare autoimmune disease that should be considered when faced with ocular and audiovestibular manifestations, even in its atypical form, to provide early and adequate treatment, which is the main prognosis factor in the control of irreversible lesions. Thus, interdisciplinary collaboration is fundamental along with screening for other infectious and systemic disorders that should include tuberculosis.

Background: Primary hyperparathyroidism is an endocrine disease and a common cause of nonmalignant hypercalcemia, often discovered incidentally in asymptomatic patients. The case reported herein illustrates that significant hormonal imbalances can present with unexpectedly mild clinical manifestations.

Case presentation: We describe a 47-year-old Caucasian male with two episodes of kidney stones and otherwise no symptoms of severe hypercalcemia. In our case, fasting emerged as a potential risk factor for the development of renal stones. The diagnosis was primary hyperparathyroidism. The intervention consisted of preoperative fluids and cinacalcet while awaiting surgery. Despite initial stability, the patient experienced a subsequent increase in ionized calcium and parathyroid hormone levels, becoming symptomatic with fatigue, tremors, and heart palpitations. This progression led to a diagnosis of a hypercalcemic crisis, and the plan was accelerated. An acute parathyroid scintigraphy and an ultrasound was performed and revealed a giant intrathoracic parathyroid adenoma. After surgical removal of the adenoma, he developed hungry bones and biochemical remission. The patient has received three extracorporeal shock wave lithotripsy treatments for a stone in the upper left kidney, but as of a computed tomography scan on 9 February 2024, the stone remains unchanged. Owing to the stone's resistance to extracorporeal shock wave lithotripsy and the absence of symptoms, no further treatment is planned. Endoscopic laser treatment (retrograde intrarenal surgery) is an option, but for now, the situation will be monitored.

Conclusion: This case report underscores that extremely elevated levels of calcium and parathyroid hormone along with a rare giant parathyroid adenoma may not necessarily present with symptoms indicative of a calcium crisis. Additionally, management of such rare giant parathyroid adenomas requires careful monitoring and a tailored approach to address potential postsurgical complications such as hungry bone syndrome.

Background: Hyperthyroidism, caused by metastatic differentiated thyroid cancer, is a rare condition that can be difficult to diagnose. Thyrotoxicosis and metastatic disease regarding functional metastasis increase morbidity and mortality in patients with functional metastasis and need to be treated. This study aims to present a case of hyper-functional metastasis of thyroid cancer to analyze its pathological features, diagnostic procedures, and treatment options and to gather and examine recent cases of hyper-functional metastasis of thyroid cancer.

Case presentation: A 26-year-old Iranian woman presenting with hyperthyroidism and a solid cystic nodule measuring 14.5 × 15.7 × 19.6 mm in the left thyroid lobe underwent thyroid surgery revealing papillary thyroid carcinoma. Despite lymph node metastasis, she refused further surgery and opted for lymph node radiofrequency ablation and radioactive iodine therapy. Subsequent follow-ups showed no recurrence of lymphadenopathy, normalized thyroid function, and decreasing triglyceride levels, with the patient under surveillance.

Conclusion: Radioactive iodine is a first-line treatment option for patients with hyperactive thyroid cancer presenting with metastatic disease. Another therapeutic option is surgery, which is performed to maximize the reduction of thyroid tissue, lymph nodes, and distant metastasis.

Background: Malrotation of the bowel due to imperfect embryologic development is a rare condition with a wide spectrum of resulting anatomical variations. Similar conditions are achieved in the adult by derotating the bowel via the Cattell-Braasch maneuver. However, possible preparational bias might compromise the resulting topography.

Case presentation: We present a case of atypical malrotation of the bowel in a cadaver study using a 96-year-old Caucasian male specimen with incidental finding of the pathology post mortem with no known surgical intervention in the abdomen during his lifetime. We compare the topography and abdominal layers with the anatomy of a 98-year-old Caucasian female specimen where the Cattell-Braasch maneuver was used to revert the embryologic development.

Conclusions: Reverting the embryologic development in the adult via Cattell-Braasch maneuver enables to mirror inherent malrotation and reestablishes the position of the bowel prior to its rotation. The Cattell-Braasch maneuver is further validated in this study by showing that it is able to demonstrate essential layers for surgical interventions without damaging their integrity. Atypical malrotation unmasks those fascial border-like layers, which are often hidden due to adhesions and fusing of tissue during the usual embryologic development. Developmental defects present a chance to explore essential surgical layers that are otherwise masked by artifacts due to fusion of layers of connective tissue.

Background: Catatonia is a potentially life-threatening condition that is characterized by psychiatric and motor disturbances, such as negativism, hypomotility, bradykinesia, and unusual movements. The diagnosis is based on clinical examination and occurs in both pediatric and adult patients and is associated with an increased mortality. Catatonia is associated with psychiatric illnesses such as schizophrenia, major depression, encephalitis, and bipolar disorder. The physiopathology of catatonia is complex and not fully understood. There is an ongoing debate in the medical community whether catatonia is an independent syndrome, or secondary to other mental illnesses. This case presentation is unique, as there are few reports describing cases of isolated catatonic syndrome in the absence of any other psychiatric or medical condition with a delayed onset caused by recreational drug abuse.

Case presentation: We present the case of a 17-year-old Caucasian athletic girl with no previous contact with child and adolescent psychiatry, nor any previous drug abuse. After recreational intake of drugs, there was a delay of approximately 7 days, before the patient searched care with symptoms that were at a later stage recognized as catatonia. Treatment with a high dose of lorazepam in combination with memantine and lithium resulted in a regression of the symptoms. After 6 weeks the patient could be discharged from the hospital almost fully recovered.

Conclusions: An acute onset of psychomotor symptoms without any previous history of mental illnesses must be addressed early as a potential catatonic syndrome. Delayed onset of catatonic symptoms after intake of drugs should not be overlooked, and we here suggest that mephedrone might be capable of inducing delayed catatonia. It is feasible to use memantine as an adjuvant to the treatment of catatonia in adolescents.

Background: Facial nerve baroparesis is a peripheral facial nerve injury resulting from barotrauma during activities such as diving, aviation, and mountain climbing. This condition occurs when increased pressure in the middle ear affects the facial nerve, leading to facial palsy. Despite being documented in otolaryngology literature, facial nerve baroparesis remains underreported and often misunderstood. Enhanced awareness and education about this condition are essential for effectively managing affected patients, helping to mitigate unnecessary panic and procedures during episodes.

Case presentation: We present two cases of facial nerve baroparesis that occurred during flights. The first case involves a 25-year-old Arab male with no significant medical history who experienced a transient, one-time episode of right-sided facial nerve baroparesis during a flight from Kuwait to Syria; he reported nasal congestion and a sore throat prior to the flight, with the episode lasting approximately 30 min before resolving spontaneously. The second case is a 30-year-old Arab female with a history of chronic allergic rhinitis, who has experienced recurrent episodes of left-sided facial nerve baroparesis for the past 2 years, occurring during nearly every flight; each episode lasted around 5 min, caused severe facial pain, and also resolved spontaneously. Both patients exhibited varying degrees of Eustachian tube dysfunction. Treatment for the recurrent case included maneuvers to alleviate ear pressure and the use of nasal decongestants, which improved symptoms and decreased the frequency of episodes.

Conclusion: Facial nerve baroparesis is frequently underestimated due to its temporary nature, yet it remains a significant cause of facial nerve palsy, especially after changes in pressure. Although the underlying mechanisms are not completely understood, dysfunction of the Eustachian tube is believed to be a contributing factor. Identifying the characteristic features of baroparesis-such as rapid onset, brief duration, and bilateral involvement of facial muscles-is essential for effective management. This highlights the necessity of educating healthcare professionals and flight personnel about this condition to prevent unnecessary treatments. For recurrent cases, management strategies should focus on alleviating Eustachian tube dysfunction.

Background: Cerebrospinal fluid rhinorrhea is a rare condition characterized by the abnormal leakage of cerebrospinal fluid from the intracranial space into the sinonasal cavity. It includes various etiologies, including traumatic, iatrogenic, and spontaneous causes, each with distinct epidemiological and clinical characteristics.

Case presentation: A 40-year-old Asian female presented with a three-month history of watery discharge from her left nostril and dull headaches localized to the left side of her head. Despite the initial diagnosis of allergic rhinitis, symptoms persisted, leading to further evaluation and eventual diagnosis of cerebrospinal fluid rhinorrhea. Detailed history, physical examination, and diagnostic tests including fluid analysis and imaging confirmed the diagnosis. The patient underwent a successful surgical repair after failed conservative management, highlighting the importance of timely intervention.

Conclusion: Cerebrospinal fluid rhinorrhea poses significant risks if left untreated, including meningitis. Prompt recognition, accurate diagnosis, and individualized treatment strategies are crucial in mitigating complications and improving patient outcomes. A multidisciplinary approach, incorporating both conservative and surgical interventions tailored to the underlying cause, is essential for the successful management of cerebrospinal fluid rhinorrhea.

Background: Allogeneic hematopoietic stem cell transplantation is a definitive cure for eligible patients with thalassemia major, and calcineurin inhibitors are essential for preventing graft-versus-host disease. Although invaluable, there are few reports of life-threatening hypersensitivity reactions associated with calcineurin inhibitors. These reactions are generally rare but seem to be more prevalent among patients with thalassemia.

Case presentation: Herein, we retrospectively report four cases of patients with thalassemia major who developed hypersensitivity reactions to parenteral cyclosporine. The cases include one 19-year-old Caucasian female and three Caucasian males, aged 17, 10, and 20 years, respectively. The patients exhibited symptoms of varying severity, necessitating different management strategies. The reactions occurred either immediately or within a few minutes after the onset of cyclosporine infusion and were often worsened by rechallenge. In all cases, cyclosporine was eventually replaced with tacrolimus or sirolimus. A comprehensive literature review was conducted to investigate the basis of severe immunoglobulin E-mediated hypersensitivity reactions to calcineurin inhibitors in patients with thalassemia major undergoing hematopoietic stem cell transplantation.

Conclusions: Several immunogenic factors may potentially increase the susceptibility of these patients to hypersensitivity reactions to Cremophor-containing medications. While severe reactions to calcineurin inhibitors remain rare, clinicians should be aware of the potential for serious adverse events in patients with thalassemia.