Differential diagnosis of rare adrenal cellular schwannomas: A case report.

BACKGROUND: Adrenal cellular schwannomas are exceptionally rare stromal tumors that are often misdiagnosed due to the lack of specific radiological, serological, or clinical features. In this report, we describe the differential diagnosis of a rare adrenal cellular schwannoma. METHODS: A 69-year-old man with a history of persistent hypertension, chronic kidney disease, hypertensive heart disease, and cardiac insufficiency was hospitalized due to bilateral lower extremity edema lasting for 3 months. Plain computed tomography at that time revealed a space-occupying lesion in the right adrenal gland. As serum levels of catecholamines, cortisol, and adrenocorticotropic hormone were within normal ranges, the edema was attributed to the chronic kidney disease and cardiac insufficiency, and the patient was referred to our hospital for surgical treatment. Contrast-enhanced computed tomography revealed heterogeneous enhancement in the adrenal mass indicating pheochromocytoma. An irregularly shaped 5 cm mass with a complete capsule in the right adrenal gland was laparoscopically resected. The postoperative histopathological diagnosis was adrenal cellular schwannoma. RESULTS: The postoperative course was unremarkable and the tumor did not recur during 5 years of follow-up. CONCLUSION: Adrenal cellular schwannoma is a very rare tumor that is extremely difficult to preoperatively diagnose. Histological and immunohistochemical analyses are required for differential diagnosis and confirmation. Cellular schwannomas can transform into malignant peripheral nerve sheath tumors, but not often. Consequently, regular postoperative follow-up is required for such patients, especially imaging.

[A hard subcutaneous swelling: not always as malignant as it seems]. / Een harde onderhuidse bult.

BACKGROUND: Nodular fasciitis is a rare, benign soft tissue tumor in young adults on the arms or trunk. CASE DESCRIPTION: A 24-year-old woman with previous pretibial melanoma in situ presented with a painless, rapidly growing subcutaneous hard swelling on the right forearm since two weeks. Ultrasound showed a subcutaneous, irregularly shaped, vascularized mass, suspicious for malignancy. Three days later, the swelling had subsided spontaneously. A new ultrasound showed a smaller, oval, sharply demarcated lesion with an inflammatory aspect. Histopathological-immunohistochemical examination of a biopsy showed a spindle cell lesion consisting of (myo)fibroblasts with some erythrocyte extravasation, consistent with nodular fasciitis. Three months later the nodule was resolved spontaneously without treatment. CONCLUSION: Nodular fasciitis should be included in the differential diagnosis in case of hard, fast-growing, subcutaneous bumps in young adults. By ultrasound, it is difficult to differentiate from a malignant soft tissue tumor. A biopsy is necessary to confirm the diagnosis with certainty.

Laser in Situ Keratomileusis Flap Free-Floating on Interface Edema in Acute Corneal Hydrops.

This case report describes a patient with a history of laser in situ keratomileusis who presented with pain, photophobia, and blurred vision secondary to a corneal flap free-floating on interface edema in the setting of acute hydrops.

Omalizumab in the treatment of Morbihan syndrome in an adolescent girl - case report and literature review.

Morbihan syndrome (MS) is characterized by solid facial edema, usually related to rosacea or acne vulgaris. The facial edema deforms the patient's features, can impair peripheral vision, and affects quality of life. Its pathophysiology remains unclear. The disease usually has a slow and chronic course. MS most commonly affects middle-aged Caucasian men with rosacea and is rare in people below 20 years of age. MS is a diagnosis of exclusion. There is no standard treatment for MS, though systemic isotretinoin and antihistamines are mainly used. We present the case of an adolescent girl with MS nonresponding to 19 months of isotretinoin treatment with add-on antihistamines. Therapy with monthly administration of omalizumab (anti-IgE) for 6 months was an effective therapeutic option, improving the quality of life. Our case is the second description of omalizumab use in Morbihan syndrome, the first in an adolescent.

Neck circumference is a highly reliable anthropometric measure in older adults requiring long-term care.

The reliability of neck circumference measurement as an assessment tool for older adults requiring long-term care remains unknown. This study aimed to evaluate the reliability of neck circumference measurement in older adults requiring long-term care, and the effect of edema on measurement error. Two physical therapists measured the neck circumference. Intraclass correlation coefficient (ICC) and Bland-Altman analyses were performed to examine the reliability of neck circumference measurement. Correlation analysis was used to evaluate the relationship between edema values (extracellular water/total body water) and neck circumference measurement difference. For inter-rater reliability of neck circumference measurement, the overall ICC (2,1) was 0.98. The upper and lower limits of the difference between examiners ranged from -0.9 to 1.2 cm. There was no association between edema values and neck circumference measurement error. Thus, measurement of the neck circumference in older adults requiring long-term care is a reliable assessment tool, with a low error rate, even in older adults with edema.

Neonatal pyocele originating from a urinary tract infection: a case report.

BACKGROUND: The origins of pyocele are primarily idiopathic, with reports suggesting the spread from intraperitoneal or hematogenous infection. However, we found no information in the literature regarding the pathogen's spread from the urinary tract. CASE PRESENTATION: We report here a case of a 23-day-old term Iranian boy (Fars ethnicity) with complaints of new-onset fever, irritability, poor feeding, right hemiscrotal erythema, and edema. Moreover, the physical examination revealed an enlarged, erythematous, tender right hemiscrotum and edematous scrotal walls. Along with leukocytosis and elevated C-reactive protein levels, the urine culture indicated an infection with Escherichia coli. However, as the color Doppler ultrasonographic examination was inconclusive, he underwent surgical exploration on which a hydrocele sac with reddish cloudy pus fluid was noted, with its culture indicating growth with the same mentioned pathogen. Therefore, an appropriate antibiotic regimen was administered, and the patient was discharged a few days later after achieving full recovery and demonstrating no urinary tract structural abnormalities. CONCLUSION: In neonatal pyocele, the spectrum of evaluating the source of the infection should also be extended to the urinary tract. Moreover, administering suitable antibiotics would produce favorable results in cases with no structural abnormalities.

TAFRO syndrome is associated with anti-SSA/Ro60 antibodies, in contrast to idiopathic castleman disease.

TAFRO syndrome is an acute systemic inflammatory disease characterized by thrombocytopenia, anasarca, fever, reticulin fibrosis/renal dysfunction, and organomegaly. There have been increasing reports that TAFRO is a disease distinct from idiopathic multicentric Castleman disease and that TAFRO patients may be positive for anti-SSA antibodies. To assess anti-SSA antibody positivity and the clinical characteristics of the two diseases, we retrospectively compared 7 TAFRO and 10 iMCD patients in our hospital. The mean age of onset of TAFRO and iMCD was 48.0 (interquartile range [IQR], 41-53) and 45.0 (IQR, 35-53) years, respectively. The TAFRO and iMCD groups had 6 (86%) and 4 (40%) male patients, respectively, and the following pretreatment laboratory values: platelet count, 3.8 (IQR, 2.2-6.4) and 35.5 (IQR, 22.2-42.8) × 104/µL, respectively; C-reactive protein, 10.2 (IQR, 6.8-21.4) and 9.5 (IQR, 6.2-13.6) mg/dL, respectively; IgG, 1431 (IQR, 1112-1815) and 4725 (IQR, 3755-5121) mg/dL, respectively. RNA immunoprecipitation (5 cases for anti-SSA) or protein array (5 cases for anti-SSA/Ro60) detected anti-SSA antibodies in six (86%) TAFRO patients but not in iMCD patients; it did not detect anti-SSB antibodies in any of the patients. None of the patients were diagnosed with Sjögren syndrome. All iMCD patients treated with tocilizumab (TCZ) responded well. Meanwhile, two of six TAFRO patients treated with TCZ showed inadequate responses; thus, both patients were switched to rituximab, following which they achieved remission. TAFRO and iMCD have different clinical features. TAFRO may be categorized as a severe phenotype of the anti-SSA antibody syndrome.

Value of Magnetic Resonance T1 Mapping in Evaluating the Early Response to Treatment for Rheumatoid Arthritis.

BACKGROUND: Rheumatoid Arthritis Magnetic Resonance Imaging Score (RAMRIS) is usually used for the semi-quantitative evaluation of joint changes in Rheumatoid Arthritis (RA). However, this method cannot evaluate early changes in bone marrow edema (BME). OBJECTIVE: To determine whether T1 mapping of wrist BME predicts early treatment response in RA. METHODS: This study prospectively enrolled 48 RA patients administered oral anti-rheumatic drugs. MRI of the most severely affected wrist was performed before and after 4 (48 patients) and 8 weeks of treatment (38 patients). Mean T1 values of BME in the lunate, triangular, and capitate bones; RAMRIS for each wrist; Erythrocyte-Sedimentation Rate (ESR); and 28-joint Disease Activity Score (DAS28)-ESR score were analyzed. Patients were divided into responders (4 weeks, 30 patients; 8 weeks, 32 patients) and non-responders (4 weeks, 18 patients; 8 weeks, 6 patients), according to EULAR response criteria. Receiver operating characteristic (ROC) curves were used to evaluate the efficacy of T1 values. RESULTS: ESR and DAS28-ESR were not correlated with T1 value and RAMRIS at each examination (P > 0.05). Changes in T1 value and DAS28-ESR relative to the baseline were moderately positively correlated with each other at 4 and 8 weeks (r = 0.555 and 0.527, respectively; P < 0.05). At 4 weeks, the change and rate of change in T1 value significantly differed between responders and non-responders (-85.63 vs. -19.92 ms; -12.89% vs. -2.81%; P < 0.05). The optimal threshold of the rate of change in T1 value at 4 weeks for predicting treatment response was -5.32% (area under the ROC curve, 0.833; sensitivity, 0.900; specificity, 0.667). CONCLUSION: T1 mapping provides a new imaging method for monitoring RA lesions; changes in wrist BME T1 values reflect early treatment response.

Inter-device agreement of sacral subepidermal oedema measurement in healthy adults during prolonged 60° head of bed elevation.

AIM: To investigate the level of agreement between the SEM 200 and Provisio® subepidermal moisture sacral delta measurements, which may indicate increased pressure injury risk, in healthy adults during 120 min of prolonged 60° head of bed elevation. This position, which requires the elevation of the patient's upper body at a 60° angle above the horizontal plane for an extended period, is used by clinicians to prevent or manage a patient's medical or surgical conditions. DESIGN: This prospective exploratory study recruited 20 healthy adults during October 2021 and collected sacral subepidermal moisture delta measurements using the SEM 200 and Provisio® devices. METHODS: Delta measurements were taken at 20-min intervals over 120 min resulting in seven data collection timepoints. Descriptive statistics and a Bland Altman plot analysis were conducted. RESULTS: A total of 280 sacral subepidermal moisture delta measurements were gathered or 140 per device. There were good levels of agreement between the two devices at baseline (T0) [mean 0.025; SD 0.137] and following 60- (T3) [mean 0.025; SD 0.111], 80- (T4) [mean -0.01; SD 0.177] and 100 min (T5) [mean 0.01; SD 0.129] of prolonged 60° head of bed elevation. Head of bed elevations can increase a patient's risk of sacral pressure injuries. In some countries, nurses have access to the SEM 200 and/or the Provisio® device, so our findings may increase nurses' confidence in the interchangeability of the device measurements, although further research is needed to confirm this. The SEM 200 and Provisio® subepidermal moisture scanners show promise in gathering similar objective pressure injury risk data which could prompt clinicians to implement prevention strategies. IMPACT: Current pressure injury risk assessment is largely subjective in nature. This quantitative study on healthy human sacral tissue found a good level of agreement in the SEM 200 and Provisio® subepidermal moisture scanners, which may increase nurses' confidence in the interchangeability of the devices in clinical practice.

[Bone marrow edema syndromes of the knee]. / Knochenmarködem-Syndrome am Kniegelenk.

Bone marrow edema represents a common finding on magnetic resonance imaging (MRI) of the knee and other joints, which can occur as a primary pathology or as a secondary phenomenon of various bone and joint pathologies. This article reviews the terminology, definition, pathology and differential diagnosis of bone marrow edema of the knee taking into consideration current concepts.

[Cockett's syndrome, a rare differential diagnosis of unilateral inferior member oedema]. / L'image du mois. Un diagnostic différentiel rare de l'œdème de membre inférieur unilatéral : le syndrome de Cockett.

We present here a typical picture of May-Thurner syndrome, rare but treatable etiology with specific treatment, of unilateral leg oedema and thromboembolic disease of the young woman.

Nous présentons ici une image typique de syndrome de Cockett, étiologie rare mais curable par un traitement spécifique, d'œdème de membre inférieur et de maladie thrombo-embolique chez la femme jeune.

National survey of patient symptoms and therapies among 707 women with a lipedema phenotype in the United States.

BACKGROUND: National survey data exploring the patient experience with lipedema are lacking. METHODS: We conducted national surveys from 2016 to 2022 of women with lipedema as well as female controls. Surveys collected information on symptomatology, pain, and therapies. We performed logistic regression comparing symptoms among those with lipedema versus controls adjusting for age and BMI. RESULTS: A total of 707 women with lipedema and 216 controls completed the surveys. Those with lipedema had a mean age of 48.6 years and mean BMI of 40.9 kg/m2. Lipedema symptom onset occurred frequently at puberty (48.0%) or pregnancy (41.2%). Compared to controls, women with lipedema were more likely to report leg swelling in heat (odds ratio [OR], 66.82; 95% CI, 33.04-135.12; p < 0.0001), easy bruising (OR, 26.23; 95% CI, 15.58-44.17; p < 0.0001), altered gait (OR, 15.54; 95% CI, 7.58-31.96; p < 0.0001), flu-like symptoms (OR, 12.99; 95% CI, 4.27-39.49; p < 0.0001), joint hypermobility (OR, 12.88; 95% CI, 6.68-24.81; p < 0.0001), cool skin (OR, 12.21; 95% CI, 5.20-28.69; p < 0.0001), varicose veins (OR, 11.29; 95% CI, 6.71-18.99; p < 0.0001), and fatigue (OR, 9.59; 95% CI, 6.10-15.09; p < 0.0001). Additionally, 70.3% had upper arm involvement, 21.2% reported foot swelling, and 16.6% reported foot pain. Most (52.2%) reported no symptom improvement with diet or exercise. Common therapies used included compression therapy (45.0%), gastric bypass (15.7%), and lower-extremity liposuction (14.0%). CONCLUSION: In a large, national, symptom survey, women with lipedema reported excess pain, swelling, and fat in the legs along with numerous symptoms beyond those classically described. Symptom responses to common therapies remain understudied.