American Journal of Case Reports最新文献

BACKGROUND Spontaneous hepatic hematoma and liver capsule rupture is a rare but severe complication of Hemolysis, Elevated Liver Enzyme, and Low Platelet (HELLP) syndrome, with a high mortality rate. We report a case of a pregnant woman with HELLP syndrome and liver subcapsular hematoma rupture that was diagnosed during surgery. CASE REPORT A 35-year-old woman with 34+1 weeks of pregnancy came to the emergency department due to abdominal pain for 4 days. She was diagnosed with HELLP syndrome after a blood test. She was transferred to the obstetrics department, and an emergency cesarean section was performed under general anesthesia, due to fetal distress. During the surgery, non-clotting blood was found flowing out during the suturing of the incision. We suspended the surgery and organized an emergency multidisciplinary consultation. Subcapsular liver hematoma was diagnosed after intraoperative ultrasound detection. Emergency upper abdominal laparotomy was performed, and a ruptured liver capsule and active bleeding were found. The liver capsule was sutured and blood products were infused before the patient was sent to the intensive care unit. She recovered and was discharged 12 days after surgery. No special discomfort was reported during the 30-day follow-up after surgery. CONCLUSIONS Our case emphasizes that all parturients with abdominal pain and HELLP syndrome must be screened for spontaneous hepatic hematoma, and clinicians should pay attention to whether there is rupture of the liver capsule. Multidisciplinary consultations can increase the probability of successful rescue for such patients.

BACKGROUND White sponge nevus (WSN) is rare, hereditary oral mucosa disorder. This condition usually presents as asymptomatic white coatings on mucous membranes that vary in intensity and thickness. Changes are usually observed on the oral cavity mucosa, and other mucosal sites of involvement include nasal, esophageal, and anogenital. In most cases this condition is considered benign since the plaques cause no pain, with no risk of malignant transformation. Thorough clinical and histopathological examination is indicated to exclude other similar conditions and to avoid unnecessary treatment. CASE REPORT We present a new case of white sponge nevus in a 13-year-old Polish girl with history of familial involvement. The patient had been treated with low-dosage tetracycline rinse (0.25%) for 3 months, and slight reduction in lesions was observed. Laser, ablation was performed with a CO₂ laser, the lesions recurred after half a year. It is the first published case report of WSN lesions on the oral mucosa treated with CO₂ laser. The patient remains under observation. CONCLUSIONS Because therapeutic options recommended in the literature provide only partial improvement or are unsuccessful, further research is still needed to develop treatment methods for patients with WSN. CO₂ laser ablation, although promising, cannot be regarded as an effective treatment method for white sponge nevus.

BACKGROUND In living-donor liver transplantation, biliary complications are considered an Achilles' heel. Consequently, various attempts have been made to reduce their incidence, and multiple innovations in surgical techniques have been reported. We herein report a case involving an intraoperative ultrasound cholangiogram in the recipient's abdominal cavity after reperfusion of the graft. CASE REPORT A 39-year-old male patient with decompensated alcoholic liver cirrhosis was admitted to our hospital for living-donor liver transplantation. The donor was his younger brother. Preoperative magnetic resonance cholangiopancreatography revealed no evidence of biliary anatomical variance; this could have been problematic when donating the left lobe graft. Intraoperative cholangiography showed that the left hepatic duct was sufficiently long for division, guaranteeing donor safety. Back-table observation of the bile duct revealed 3 orifices; of these, the central orifice was very small, and the corresponding bile duct was not evident on intraoperative cholangiography in donor surgery. After an injection of perfluorobutane microbubbles (Sonazoid) diluted 1000-fold into the small central orifice, the bile duct of segment 4 (B4) was clearly visualized with an intraoperative ultrasound cholangiogram. The off-label use of Sonazoid was approved by Nagasaki University Hospital. Based on this finding, we determined that all 3 openings required reconstruction and reconstructed them using a telescope reconstruction method. CONCLUSIONS We verified that intraoperative ultrasound cholangiogram is useful as a tool to confirm the anatomy of the bile duct when it is not revealed through other evaluation techniques; hence, it is a method that transplant surgeons should be familiar with.

BACKGROUND Gallstone ileus is an uncommon cause of intestinal obstruction. Rigler's classic triad for a gallstone ileus includes the following: small bowel obstruction, air in the biliary tract, and an obstructing gallstone. This triad, however, is not always observed. We present an unusual case of a gallstone present in the small bowel for several years prior to presenting with an acute obstruction. CASE REPORT A 71-year-old man presented with 3 days of lower abdominal pain, constipation, and abdominal distension, with his last reported bowel movement 3 days prior. The patient's vitals were stable, with a white blood cell count of 11.47×10⁹/L and no lactic acidosis. Abdominal exam was significant for bilateral lower-quadrant tenderness and mild distension. Findings on abdominal computed tomography revealed a large foreign body in the distal small bowel, with evidence of proximal small bowel obstruction. Review of imaging from 4 years prior incidentally revealed the foreign body more proximally in the jejunum. Laparoscopy and enterotomy were performed with removal of a 4×4 cm gallstone encased in fecal material. The patient recovered well from surgery and had no complications. CONCLUSIONS A 71-year-old man presented with lower abdominal pain and distension. Work-up revealed a small bowel obstruction secondary to a presumed foreign body, later found to be a gallstone. We present a highly unusual presentation of a gallstone ileus, with radiographic evidence of an enteric gallstone present 4 years prior, with no evidence of pneumobilia or biliary-enteric fistula in current or previous computed tomography scans.

BACKGROUND Giant coronary artery aneurysms (CAA) are extremely rare and can mimic cardiac tumors. Therefore, an unidentified mass in the heart requires a multimodality imaging approach for accurate diagnosis and guidance of further management, which for CAAs often include surgical intervention to prevent complications such as thrombosis or rupture. CASE REPORT A 37-year-old man presented with non-specific symptoms. A CT scan revealed multiple bilateral pulmonary embolisms and an indeterminate mass in the right atrium. Transthoracic echocardiography (TTE) showed a suspected cardiac tumor, and further imaging with transesophageal echocardiography (TEE), magnetic resonance imaging (MRI), and position emission tomography (PET) indicated a local inhomogeneous mass with arterial perfusion. A preoperative cardiac CT found the mass to be a giant thrombosed CAA in the proximal right coronary artery compressing the tricuspid annulus. The patient underwent successful surgical excision of the CAA along with coronary artery bypass grafting. Postoperative management included lifelong administration of acetylsalicylic acid and a 3-month course of anticoagulant therapy. Histopathology excluded systemic vasculitis, indicating a congenital etiology for the CAA. CONCLUSIONS This case illustrates the indispensable role of coronary CT angiography in accurately diagnosing and managing complex cardiac conditions. Due to the complex and diverse nature of suspected cardiac tumors, cardiac CT should always be added in the diagnostic workup to describe the coronary anatomy in relation to the tumor and to identify a differential diagnosis such as a giant coronary aneurysm.

BACKGROUND Mycobacterium marinum is a slow-growing non-tuberculous mycobacterium that is known to cause skin and soft tissue infections, even in healthy patients, and is commonly associated with fish and aquatic environments. CASE REPORT A 23-year-old man working in aquarium management presented with a chronic progression of multiple skin nodules on his right forearm and thumb. The patient was referred from the Dermatology Department to the Outpatient Clinic due to suspected skin tuberculosis, as indicated by a positive T-SPOT.TB test. A second excisional biopsy tested positive for M. marinum via PCR sequencing by the National Institute of Infectious Diseases, confirming the diagnosis. The initial treatment consisted of rifabutin at 300 mg/day and clarithromycin at 800 mg/day. However, due to liver dysfunction, the regimen was changed to moxifloxacin at 400 mg/day and rifabutin. Moxifloxacin was discontinued due to nausea. Finally, the treatment was adjusted to linezolid at 1200 mg/day and clarithromycin. The patient's skin condition improved, with the nodular lesions showing a trend toward resolution. Culturing is time-consuming, and the sensitivity can be reduced when using N-acetyl-l-cysteine-sodium hydroxide in the pre-treatment process; therefore, caution with its use is necessary. Pathological examination can initially show inflammatory changes, and granulomatous lesions with caseous necrosis are not always present. Antibiotics such as rifampicin, rifabutin, moxifloxacin, and clarithromycin are used, but there is scant evidence for treatment regimens, often resulting in prolonged monotherapy or combination therapy. CONCLUSIONS In cases presenting chronic lesions resembling multiple sporotrichoid forms, repeated biopsies are crucial due to the challenges associated with culturing.

BACKGROUND Small bowel transplantation (SBT) is a rare but life-saving surgery. However, successful full-term pregnancies in individuals with SBT are exceedingly rare due to the nutritional and immunosuppression challenges this transplant poses for pregnancy. Therefore, clear guidelines for treating pregnant SBT recipients are unavailable. Here, we report the second case of a successful pregnancy in an individual with a triple organ transplant, including SBT, highlighting the need for careful immunosuppressive management and multidisciplinary care. CASE REPORT A 20-year-old woman in the third trimester of pregnancy with a history of small bowel, liver, and pancreas transplantation at age 1 year presented with elevated liver function test results. She had been taking tacrolimus, sirolimus, and prednisone before pregnancy, with no signs of organ rejection. While sirolimus and prednisone was discontinued upon conception, laboratory test results at presentation revealed low serum tacrolimus levels. The patient had an acute kidney injury and pulmonary edema during her hospitalization and received a diagnosis of preeclampsia. She underwent a successful cesarean delivery, due to labor induction complications; however, about 1 month after hospital discharge, the patient experienced elevated liver enzymes, which was treated with high-dose steroids and adjusted tacrolimus. Sirolimus was restarted, and the patient's liver enzymes have been normalized to date. CONCLUSIONS Comprehensive multidisciplinary care, as well as monitoring and optimizing immunosuppression, are essential for pregnant SBT recipients throughout the prenatal, perinatal, and postpartum periods to mitigate risks, prevent graft rejection, and ensure positive maternal and fetal health outcomes.

BACKGROUND Obesity-hypoventilation syndrome (OHS), also known as Pickwickian syndrome, is a respiratory consequence of morbid obesity, usually treated with non-invasive positive airway pressure (PAP) therapies and weight loss. This study reports a 53-year-old woman with a body mass index of 49 kg/m² who experienced acute hypercapnic respiratory failure due to OHS. Her treatment involved mechanical ventilation, home oxygen therapy, and long-term weight loss of >30 kg. CASE REPORT A 53-year-old woman (109 kg) presented with acute hypercapnic respiratory failure due to OHS, which improved with mechanical ventilation and diuretics. After discharge from the hospital, she was treated with nocturnal non-invasive positive-pressure ventilation (NPPV) and home oxygen therapy. Over a 5-year period, following loss of >30 kg, she was re-evaluated for the discontinuation of NPPV and oxygen therapy. She was evaluated with various NPPV settings and oxygen doses, monitored by transcutaneous pressure of carbon dioxide (PtcCO₂). On NPPV, PtcCO₂ levels ≥55 mmHg were maintained within 10 min, indicating that the durations of PtcCO₂ ≥50 mmHg were too prolonged for her to be switched to continuous PAP therapy. Nonetheless, oxygen therapy was discontinued because the duration of peripheral blood oxygen saturation <90% was brief. CONCLUSIONS For patients with OHS treated with NPPV and oxygen therapy, weight loss alone may not improve hypoventilation and wean the patient from NPPV. Besides obesity, various factors influence respiratory compromise in OHS; hence, a comprehensive assessment of hypoventilation, including PtcCO₂ monitoring, is essential to determine whether NPPV withdrawal is possible after body weight loss.

BACKGROUND Orbital abscess is a severe infectious condition of the eye that can result in significant complications, including vision loss or fatality. Timely and accurate diagnosis and treatment of this condition are crucial. CASE REPORT A 45-year-old man with exophthalmos, diplopia, and decreased vision was admitted to our hospital. He had a history of chronic sinusitis. Ultrasonography showed a dark fluid area above the superior rectus muscle of the right eye, measuring approximately 5.7 ml. Orbital MRI revealed short T1 and long T2 signal shadows outside the upper muscle cone of the right orbit, with a size of about 13.2 ml. The right eyeball was compressed and moved forward, and the superior rectus muscle was also compressed. Long T2 signal shadows were observed in the right frontal sinus, maxillary sinus, and bilateral ethmoid sinuses, leading to a diagnosis of orbital subperiosteal abscess and sinusitis. We performed a fine-needle puncture and injection of antibiotics into the abscess cavity using a 5-ml syringe under the guidance of B-ultrasound. On the 7th day after surgery, the patient showed clinical improvement with decreased symptoms. His visual acuity improved from 20/40 to 20/20, and diplopia resolved. His sinusitis was treated with medication, and no recurrence of ocular symptoms was observed during the 3-year follow-up. CONCLUSIONS This report highlights the use of ultrasound-guided fine-needle puncture and injection of antibiotics into the abscess cavity for the treatment of an upper-quadrant orbital subperiosteal abscess. Timely surgical drainage and effective antibiotic therapy can help reduce the complications associated with orbital abscesses.

BACKGROUND Detection of episodes of desaturation on pulse oximetry in elderly people in community settings is now a common occurrence. Transfer of such patients to hospital by ambulance has led to a greatly increased exposure to high-dose (FiO₂ > 50%) inhaled oxygen therapy in this group. Current British Thoracic Society (BTS) guidelines recommend administration of oxygen, 15 L/min via a non-rebreather mask (NRB) to acutely hypoxemic patients with an SpO₂ below 85% on room air. In some elderly subjects, such high-dose oxygen therapy induces significant hypercapnia in the absence of an identifiable risk factor for oxygen-induced CO₂ retention. CASE REPORT This case series describes 3 very elderly (>85 years old) female patients developing acute hypercapnia shortly after initiation of high-dose inhaled oxygen therapy. In each of these cases, hypercapnia developed in the absence of an accepted risk factor for oxygen-induced CO₂ retention. In 2 cases, CO₂ narcosis resolved within hours of the establishment of controlled oxygen therapy on bi-level positive airway pressure (BPAP). The possibility of oxygen-induced CO2 retention was not considered by the treating physicians in the acute setting. CONCLUSIONS The possibility of oxygen-induced CO₂ retention should be considered in all elderly patients developing acute type II respiratory failure in the setting of high-dose oxygen therapy. Failure to recognize oxygen-induced CO₂ retention has significant implications for patient outcome and resource utilization.