A case of bilateral adrenal infarction with preserved adrenal function diagnosed by dual-energy computed tomography.

We report a case wherein adrenal function remained preserved despite bilateral adrenal infarction, as evidenced by dual-energy computed tomography (DECT) iodine density images. The patient was a 37-year-old man with a history of antiphospholipid syndrome concomitant with systemic lupus erythematosus. The patient underwent contrast-enhanced DECT, which revealed bilateral adrenal infarction. Laboratory tests revealed preserved adrenal function. On the iodine density images, the infarcted and noninfarcted areas in the adrenal glands were visually different. The volume of the non-infarcted area was 8.9 mL, which was 41% of the total adrenal volume. DECT may be a useful complementary tool for assessing the preservation of adrenal function.

Assessing Lateralization Index of Adrenal Venous Sampling for Surgical Indication in Primary Aldosteronism.

BACKGROUND: Clinical practice guidelines recommend the Lateralization Index (LI) as the standard for determining surgical eligibility in primary aldosteronism (PA). Our goal was to identify the optimal LI cut-offs in adrenal venous sampling (AVS) for diagnosing PA that is amenable to surgical cure. METHODS: We conducted a retrospective international cohort study across 16 institutions in 11 countries, including 1,550 patients with PA who underwent AVS, with and/or without ACTH stimulation. The establishment of optimal cut-offs was informed by a survey of 82 PA patients in Japan, aimed at determining the LI cut-off aligned with patient expectations for a surgical cure rate. RESULTS: The survey revealed that a median cure rate expectation of 80% would motivate PA patients towards undergoing adrenalectomy. The optimal LI cut-offs achieving an adjusted positive predictive value (PPV) of 80% were identified as 3.8 for unstimulated AVS and 3.4 for ACTH-stimulated AVS. Furthermore, a contralateral ratio of less than 0.4 and the detection of an adrenal nodule on CT imaging were identified as independent predictors of surgically curable PA. Incorporating these factors with the optimal LI cut-offs, the adjusted PPV increased to 96.6% for unstimulated AVS and 89.6% for ACTH-stimulated AVS. No clear differences in predictive ability between unstimulated and ACTH-stimulated LI were found. CONCLUSIONS AND RELEVANCE: The present study clarified the optimal LI cut-offs for without and with ACTH stimulation. The presence of contralateral suppression and adrenal nodule on CT imaging seems to provide additional available information besides LI for surgical indication.

Association between stopping renin-angiotensin system inhibitors immediately before hemodialysis initiation and subsequent cardiovascular events.

It is controversial whether renin-angiotensin system inhibitors (RASIs) should be stopped in patients with advanced chronic kidney disease (CKD). Recently, it was reported that stopping RASIs in advanced CKD was associated with increased mortality and cardiovascular (CV) events; however, it remains unclear whether stopping RASIs before dialysis initiation affects clinical outcomes after dialysis, which this study aimed to evaluate. In this multicenter prospective cohort study in Japan, we included 717 patients (mean age, 67 years; 68% male) who had a nephrology care duration ≥90 days, initiated hemodialysis, and used RASIs 3 months before hemodialysis initiation. The multivariable adjusted Cox models were used to compare mortality and CV event risk between 650 (91%) patients who continued RASIs until hemodialysis initiation and 67 (9.3%) patients who stopped RASIs. During a median follow-up period of 3.5 years, 170 (24%) patients died and 228 (32%) experienced CV events. Compared with continuing RASIs, stopping RASIs was unassociated with mortality (adjusted hazard ratio [aHR]: 0.82; 95% confidence interval [CI]: 0.50-1.34) but was associated with higher CV events (aHR: 1.59; 95% CI: 1.06-2.38). Subgroup analyses showed that the risk of stopping RASIs for CV events was particularly high in patients aged <75 years, with a significant interaction between stopping RASIs and age. This study revealed that patients who stopped RASIs immediately before dialysis initiation were associated with subsequent higher CV events. Active screening for CV disease may be especially beneficial for these patients.

Factors affecting stress shielding and osteolysis after reverse shoulder arthroplasty: A multicenter study in a Japanese population.

BACKGROUND: Stress shielding and osteolysis around the humeral stem after reverse shoulder arthroplasty causes loosening and periprosthetic fractures and reduces bone stock during revision surgery. In Japanese patients, who have relatively small bodies, different characteristics may exist regarding the occurrence of these changes compared with the characteristics of Westerners, who have relatively larger frames. The purpose of this multicenter study was to investigate the incidence and clarify the predictors of stress shielding and osteolysis in Japanese individuals who underwent reverse shoulder arthroplasty. METHODS: The occurrence of stress shielding and osteolysis was investigated in 135 shoulders that had undergone reverse shoulder arthroplasty at least 2 years prior in five Japanese hospitals. During post-surgical follow-up, which was conducted every 3 months, the locations of the stress shielding occurrences, such as cortical thinning and osteopenia (which primarily occurred in zones 1, 2, and 7, where 1 is the greater tuberosity and 7 is the calcar part), spot weld, and condensation lines, were recorded. Cases without any abnormal findings on radiographs obtained up to ≥2 years after surgery were regarded as having no abnormalities. Finally, the predictors of cortical thinning and proximal humeral osteolysis were assessed using univariate and multivariate regression analyses. RESULTS: Cortical thinning and osteopenia occurred in 68 shoulders, a condensation line occurred in 37 shoulders, and spot weld occurred in 23 shoulders. In particular, greater tuberosity and calcar osteolysis occurred in 40 and 47 shoulders, respectively. Long stem, cementless stem, and a larger proximal filling ratio were independent predictors of cortical thinning and osteopenia, whereas a cementless stem, larger metaphysis diameter, and a larger proximal filling ratio were associated with proximal humeral osteolysis. CONCLUSIONS: The predictors of stress shielding and osteolysis included the use of long stems, cementless stems, larger proximal filling ratios, and larger metaphysis diameters. LEVEL OF EVIDENCE: retrospective comparative study (Level III).

Clinical characteristics of anti-GBM disease with thrombotic microangiopathy: a case report and literature review.

The coexistence of anti-glomerular basement membrane (anti-GBM) disease with thrombotic microangiopathy (TMA) is rarely encountered, and the clinical characteristics of this phenomenon are not well known.A 76-year-old Japanese woman with a history of idiopathic pulmonary disease was diagnosed with anti-GBM disease due to rapidly progressive glomerulonephritis and a positive anti-GBM antibody test result. We treated the patient with hemodialysis, glucocorticoids, and plasmapheresis. During treatment, the patient suddenly became comatose. TMA was then diagnosed because of thrombocytopenia and microangiopathic hemolytic anemia. The activity of a disintegrin-like and metalloproteinase with thrombospondin type 1 motif 13 (ADAMTS-13) was retained at 48%. Although we continued the treatment, the patient died of respiratory failure. An autopsy revealed the cause of respiratory failure to be an acute exacerbation of interstitial pneumonia. The clinical findings of the renal specimen indicated anti-GBM disease; however, there were no lesions suggestive of TMA. A genetic test did not reveal an apparent genetic mutation of the atypical hemolytic uremic syndrome.We conducted a literature review of past case reports of anti-GBM disease with TMA. The following clinical characteristics were obtained. First, 75% of the cases were reported in Asia. Second, TMA tended to appear during the treatment course for anti-GBM disease and usually resolved within 12 weeks. Third, ADAMTS-13 activity was retained above 10% in 90% of the cases. Fourth, central nervous system manifestations occurred in more than half of the patients. Fifth, the renal outcome was very poor. Further studies are required to understand the pathophysiology of this phenomenon.

Long-term conservative treatment outcomes for midshaft clavicle fractures: a 10-to-30-year follow-up.

BACKGROUND: Few reports exist on the long-term outcomes of midshaft clavicle fracture conservative treatments. Therefore, this study investigated the long-term outcome of this treatment in patients with midshaft clavicle fractures. METHODS: Patients were treated conservatively for midshaft clavicle fractures with a figure-of-eight bandage between 10 and 30 years ago. Subsequently, a telephone survey was used to follow-up these patients, and 38 were successfully evaluated. The mean term after trauma was 17.0 years. Afterward, the American Shoulder and Elbow Surgeons Shoulder (ASES) score and Shoulder pain and disability index (SPADI) on the affected and unaffected sides were calculated based on the filled questionnaires. We defined patients whose ASES and SPADI on the affected side were worse than the unaffected side as the symptomatic group. Furthermore, plain radiographs measured proportional changes in clavicular length and displacement. RESULTS: The ASES scores of the affected side were significantly lower than those of the unaffected side, and the SPADI of the affected side was significantly higher than that of the unaffected side. Furthermore, the symptomatic group's proportional changes in clavicular length and displacement were significantly larger than the asymptomatic group. CONCLUSION: The affected shoulder side was more symptomatic than the unaffected side 10 to 30 years after the trauma when midshaft clavicle fractures were treated conservatively. Moreover, several patients became symptomatic for fractures with a larger proportional change in clavicular length or displacement.

Sudden-onset hypertension leading to the diagnosis of unilateral hydronephrosis due to ureteropelvic junction obstruction.

We present a case of a 41-year-old female who developed hypertension over a three-month period and was subsequently diagnosed with ureteropelvic junction obstruction (UPJO). The patient came to our department with elevated blood pressure. Blood examinations revealed normal renal function, hypokalemia and increased renin-angiotensin system (RAS) activity, as indicated by elevated level of plasma renin activity and plasma aldosterone level. A computed tomography imaging further revealed dilation of the left renal pelvis, atrophy of the left kidney, and indications of obstruction at the junction between the renal pelvis and ureter. Surgical intervention in the form of a left pyeloplasty successfully resolved the unilateral hydronephrosis, corrected the elevated RAS activity, normalized the blood pressure, and ameliorated the hypokalemia. This case emphasizes that elevated blood pressure might be the sole clinical indication of hydronephrosis. It's crucial to consider hydronephrosis due to UPJO as a potential cause, especially when diagnosing hypertension associated with RAS hyperactivity in young adults. It also highlights the effectiveness of surgical intervention in treating hypertension in such scenarios.

Efficacy of the shoelace technique for extremity fasciotomy wounds due to compartment syndrome.

BACKGROUND: The shoelace technique for compartment syndrome allows application of sustained tightening tension to an entire wound and intermittent tightening of the shoelace without requiring its replacement or anesthesia. We retrospectively evaluated the usefulness of the shoelace technique in the management of extremity fasciotomy wounds before and after its introduction in our institution. METHODS: We targeted 25 patients who were diagnosed as having compartment syndrome and underwent extremity fasciotomy at our hospital from April 2012 to December 2021. The N group, comprising 12 patients treated without the shoelace technique, and the S group, comprising 13 patients treated with the shoelace technique, were compared retrospectively for each outcome. RESULTS: There were no significant differences between the two groups in patient background. Compared with the N group patients, all of the S group patients avoided skin grafting (S group: n = 0, 0%; N group: n = 6, 50.0%; p < 0.01). However, there was no significant difference in the number of days to final wound closure (S group: 39.5 [IQR 24.3-58.0] days; N group: 24.0 [IQR 18.5-31.0] days, p = 0.06). CONCLUSIONS: We considered the shoelace technique to be a useful wound closure method for fasciotomy wounds caused by compartment syndrome because it can significantly reduce the need for skin grafting and tends to shorten the wound closure period.

Neuroblastoma suppressor of tumorigenicity 1 is associated with the severity of interstitial fibrosis and kidney function decline in IgA nephropathy.

INTRODUCTION: Recently, circulating neuroblastoma suppressor of tumorigenicity 1 (NBL1) was shown to be strongly associated with kidney disease progression and histological lesions in patients with diabetic kidney disease. This study aimed to examine whether serum NBL1 level was also associated with kidney function and renal histological findings in patients with IgA nephropathy. METHODS: We evaluated the levels of NBL1 in 109 patients with newly diagnosed biopsy-proven primary IgAN, between 2009 and 2018, at the Nihon University School of Medicine Itabashi Hospital, Tokyo, Japan, using serum obtained immediately before the renal biopsy, and examined the relationship between serum NBL1, renal function and renal histological findings assessed using the Oxford Classification (MEST score). Furthermore, we analyzed the association of serum NBL1 with kidney function decline over time in patients with IgA nephropathy who had follow-up data on the estimated glomerular filtration rate (n = 76). RESULTS: Serum NBL1 levels in patients with newly diagnosed IgA nephropathy were elevated, as compared to those in healthy individuals (n = 93). Logistic regression analysis demonstrated that the serum NBL1 level was independently and significantly associated with tubular atrophy/interstitial fibrosis. Immunohistochemical staining revealed that NBL1 was highly expressed in the tubulointerstitium. Furthermore, Spearman's rank correlation identified a significant correlation between serum NBL1 level and estimated glomerular filtration rate slope. CONCLUSIONS: The serum NBL1 level was significantly associated with the severity of renal interstitial fibrosis and kidney disease progression in patients with newly diagnosed IgA nephropathy. Thus, circulating NBL1 may serve as a good biomarker for evaluating renal interstitial fibrosis and the risk of kidney disease progression.

Impact of a change to a novel chemiluminescent immunoassay for measuring plasma aldosterone on the diagnosis of primary aldosteronism.

In Japan, the standard method for measuring plasma aldosterone concentration (PAC) for primary aldosteronism (PA) diagnosis was changed from radioimmunoassay (RIA) to a novel chemiluminescent enzyme immunoassay (CLEIA). The purpose of this study is to simulate the possible impact of the change on PA diagnosis. This retrospective study assessed 2,289 PA patients. PACs measured by conventional RIA were transformed to estimated PACs (CLEIA) as follows: RIA (pg/mL) = 1.174 × CLEIA (pg/mL) + 42.3. We applied the estimated PAC (CLEIA) to the conventional cut-off of aldosterone-to-renin activity ratio ≥200 for screening and captopril challenge test (CCT) and PAC ≥60 pg/mL for saline infusion test (SIT). Application of the estimated PAC to screening and confirmatory tests decreased the number of PA diagnoses by 36% (743/2,065) on CCT and 52% (578/1,104) on SIT (discrepant cases). Among the discrepant cases, 87% (548/628) of CCT and 87% (452/522) of SIT were bilateral on adrenal venous sampling (AVS). Surgically treatable aldosterone-producing adenomas (APAs) were observed in 6% (36/579) and 5% (23/472) of discrepant cases on CCT and SIT, respectively; most were characterized by hypokalemia and/or adrenal nodule on CT imaging. Application of the PAC measured by the novel CLEIA to conventional cut-offs decreases the number of PA diagnoses. Although most discrepant cases were bilateral on AVS, there are some APA cases that were characterized by hypokalemia and/or adrenal tumor on CT. Further studies which evaluate PACs measured by both RIA and CLEIA for each patient are needed to identify new cut-offs for PAC measured by CLEIA.

[A case of pancreatic adenosquamous carcinoma successfully treated by chemoradiotherapy].

Pancreatic adenosquamous carcinoma is a rare primary pancreas malignant tumor with very poor prognosis, for which there is no standard treatment. The case was of a 71-year-old woman who was admitted to the hospital with jaundice. A pancreatic head tumor was found, and pancreatic adenosquamous carcinoma was diagnosed in EUS-FNA. Despite confirmed distant metastasis, a multidisciplinary treatment centered on chemoradiotherapy gave her a 28-month prognosis.

Predictive correction of serum sodium concentration with formulas derived from the Edelman equation in patients with severe hyponatremia.

Severe hyponatremia can cause life-threatening cerebral edema. Treatment comprises rapid elevation of serum sodium concentration; however, overcorrection can result in osmotic demyelination. This study investigated potential factors, including predictive correction based on the Edelman equation, associated with appropriate correction in 221 patients with a serum sodium concentration ≤ 120 mEq/L who were admitted to a hospital in Nagoya, Japan. Appropriate correction was defined as an elevation in serum sodium concentration in the range of 4-10 mEq/L in the first 24 h and within 18 mEq/L in the first 48 h after the start of the correction. Appropriate corrections were made in 132 (59.7%) of the 221 patients. Multivariate analysis revealed that predictive correction with an infusate and fluid loss formula derived from the Edelman equation was associated with appropriate correction of serum sodium concentration (adjusted odds ratio, 7.84; 95% confidence interval, 2.97-20.64). Relative without its use, the predictive equation results in a lower proportion of undercorrection (14.3% vs. 48.0%, respectively) and overcorrection (1.0% vs. 12.2%, respectively). These results suggest that predictive correction of serum sodium concentrations using the formula derived from the Edelman equation can play an essential role in the appropriate management of patients with severe hyponatremia.

Remnant-Preserved Single-Bundle Anterior Cruciate Ligament Augmentation in Multi-ligamentous Knee Reconstruction.

This study was conducted to present remnant-preserving anterior cruciate ligament (ACL) augmentation as a useful option for partial ACL injury in multiligament knee injury (MLKI) cases, which may also contribute to conserving graft resources. The present study involved patients diagnosed with MLKI at our institute from Spring 2006 to February 2021. A total of 71 MLKI cases were provided surgery due to knee instability and disability. For every patient, an arthroscopic diagnostic was performed to ensure that ACL tear and a remnant were present. When the ACL remnant was classified into group 2, 3, or 4 of Nakamae's classification, remnant-preserved single bundle ACL augmentation was performed. Graft selection and the combination of injured ligaments were evaluated. The side-to-side difference under an anterior tibial load of 134 N with an arthrometer and the leg symmetry index at 60 degrees/s were measured. The present procedure was performed for five cases (male/female: 4/1, mean age: 33.6 years). The mean follow-up period was 26.4 months. The combination of torn ligaments was as follows: 3 cases of ACL + medial collateral ligament, one case of ACL + posterior cruciate ligament, and one case of ACL + posterolateral corner. An ACL augmentation graft was performed using an ipsilateral gracilis tendon in 2 cases, a contralateral full semitendinosus tendon in 2 cases, and the ipsilateral distal 1/2 of the semitendinosus tendon in 1 case. The mean side-to-side difference was 1.07 ± 0.4 mm. The mean leg symmetry index was 82.6 ± 12.2% in knee extension and 96.3 ± 9.9% in knee flexion. Although the present study was a small case series, the remnant-preserved single-bundle ACL augmentation for MLKI surgery provided good clinical outcome and conserved the graft resource. Even in the MLKI case, this technique is one of the useful surgical options. The level of evidence of this study is level IV (case series).

Biomechanical effect of metal augment and bone graft on cup stability for acetabular reconstruction of total hip arthroplasty in hip dysplasia: a finite element analysis.

BACKGROUND: Different methods of acetabular reconstruction with total hip arthroplasty (THA) for Crowe II and III of adult developmental dysplasia of the hip (DDH) acetabular bone defect have been implemented clinically. However, the biomechanical effect of different augmented materials for acetabular reconstruction in THA on shell stability has never been discussed. METHODS: In the present study, autologous bone graft (BG)and metal (Ti6Al4V) augment (MA) were simulated with several acetabular bone defect models of DDH in THA. The contact pressure and micromotion between the shell and host bone were measured for evaluating the shell stability using a finite element method. RESULTS: The peak contact stress between shell and host bone was higher in the MA situation (12.45 vs 8.71 MPa). And the load transfer path was different, for BG models, the high local contact stresses were found at the junction of bone graft and host bone while for MA models the concentrated contact stresses were at the surface of MA. The peak relative micromotion between shell and host bone was higher in the MA situation (12.61 vs 11.13 µm). However, the peak micromotion decreased in the contact interface of MA and cup compared to the BG models. CONCLUSIONS: The higher micromotion was found in MA models, however, enough for bone ingrowth, and direct stronger fixation was achieved in the MA-cup interface. Thus, we recommended the MA can be used as an option, even for Crowe III, however, the decision should be made from clinical follow-up results.

Herpes zoster meningitis in a rheumatoid arthritis patient treated with tofacitinib.

A man in his 70s with rheumatoid arthritis presented with seizures and coma and was transferred to our emergency department. Two months prior to admission, he started to take tofacitinib 10 mg/day. On admission, we noted a rash with a blister on the forehead, and herpes zoster was diagnosed. Cerebrospinal fluid examination suggested meningitis. An MRI of the brain showed no abnormality. Based on these findings, he was suspected with herpes zoster meningitis. We discontinued tofacitinib and treated the patient with intravenous acyclovir for 2 weeks. He regained complete consciousness, but right forehead skin lesion, severe vision loss in the right eye and right facial nerve paralysis remained as sequelae. Six weeks after admission, we restarted tofacitinib with oral valaciclovir as antiviral prophylaxis. Two years after admission, we administered Shingrix, an adjuvant recombinant vaccine for herpes zoster, and discontinued oral valaciclovir.

Effect of total hip arthroplasty on improving locomotive syndrome in hip disease patients: A prospective cohort study focused on total clinical decision limits stage 3.

BACKGROUND: In 2020, the Japanese Orthopaedic Association established a new stage 3 in clinical decision limits (CDL) to evaluate the stage of locomotive syndrome (LS). This study focused on total CDL stage 3 with the aim of investigating indicators related to improvements in total CDL by evaluating the improvement of LS in patients who underwent total hip arthroplasty (THA). METHODS: Of the 125 patients who underwent THA at our hospital, the subjects of the analysis were 105 patients determined to be total CDL stage 3 in an evaluation performed before THA. LS was evaluated using the stand-up test, two-step test, and 25-Question Geriatric Locomotive Function Scale (GLFS-25). Indicators related to improvements in total CDL were also investigated. All evaluation items were measured before THA and three months after THA. RESULTS: Before THA, all subjects (n = 105) were classified as total CDL stage 3. Three months after THA, improvements in total CDL were seen in 49 subjects (46.7%). The results of stepwise multiple logistic regression analysis showed that the before THA stand-up test and GLFS-25 were significantly related to improvements in total CDL. CONCLUSIONS: Three months after THA, improvements in LS were seen in approximately half of the subjects. The stand-up test and GLFS-25 can be used as indicators of improvement in total CDL. DESIGN: Prospective cohort study design.