Radial nerve palsy caused by a rapidly growing intramuscular hematoma in an infant with biliary atresia: a case report.

BACKGROUND: Biliary atresia (BA) is a rare cause of persistent jaundice in infants that can result in vitamin K malabsorption and vitamin K deficiency bleeding (VKDB). We present an infant with BA who developed a rapidly growing intramuscular hematoma in her upper arm after a vaccination which caused a radial nerve palsy. CASE PRESENTATION: An 82-day-old girl was referred to our hospital because of a rapidly growing left upper arm mass. She had received three doses of oral vitamin K before age 1 month. At age 66 days, she received a pneumococcal vaccination in her left upper arm. On presentation, she showed no left wrist or finger extension. Blood examination revealed direct hyperbilirubinemia, liver dysfunction, and coagulation abnormalities, indicating obstructive jaundice. Magnetic resonance imaging showed a hematoma in the left triceps brachii. Abdominal ultrasonography revealed an atrophic gallbladder and the triangular cord sign anterior to the portal vein bifurcation. BA was confirmed on cholangiography. VKDB resulting from BA in conjunction with vaccination in the left upper arm were considered the cause of the hematoma. The hematoma was considered the cause of her radial nerve palsy. Although she underwent Kasai hepatic portoenterostomy at age 82 days, the obstructive jaundice did not sufficiently improve. She then underwent living-related liver transplantation at age 8 months. The wrist drop was still present at age 1 year despite hematoma resolution. CONCLUSIONS: Delayed detection of BA and inadequate prevention of VKDB can result in permanent peripheral neuropathy.

Bacteremia Caused by Kalamiella piersonii Found in an Infant During the Course of Gastrointestinal Food Allergy.

Kalamiella piersonii is rare pathogen, and its pathogenicity to humans has been unknown. We describe an infant with bacteremia caused by Kalamiella piersonii. The patient was a 2-month-old girl presented with diarrhea, poor oral intake, and vomiting. The patient was tentatively diagnosed with acute enterocolitis. After admission, the patient developed a fever and blood culture yielded Gram-negative cocci, first determined to be Pantoea septica by matrix-assisted laser desorption/ionization time-of-flight mass spectrometry. However, genetic analysis of 16S rRNA allowed its identification as Kalamiella piersonii (GenBank accession number is OQ547240). Other housekeeping genes such as gyrB, rpoB, and atpD also identified the isolated strain as Kalamiella piersonii. The patient was successfully treated with cefotaxime without sequelae. Later, the patient was diagnosed as non-IgE-mediated gastrointestinal food allergy. Our experience indicated that Kalamiella piersonii is a potential human pathogen that can cause invasive infections even in infants and children. Identification of Kalamiella piersonii is difficult with routine conventional tests, and detailed studies including genetic analyses are necessary to clarify the pathogenicity of Kalamiella piersonii in humans.

Supramalleolar Lateral Opening-Wedge Osteotomy in Conjunction With Progressive Collapsing Foot Deformity Reconstruction for PCFD With Ankle Instability.

BACKGROUND: Reconstruction of progressive collapsing foot deformity (PCFD) with ankle instability (PCFD class E) remains a substantial challenge to orthopaedic surgeons. We report the outcomes of PCFD class E treated by means of a relatively standard PCFD foot reconstruction approach with the addition of a supramalleolar lateral opening-wedge osteotomy. METHODS: We conducted a retrospective study of 13 patients (15 ankles) who underwent this procedure for PCFD class E between 2010 and 2021. Mean follow-up time was 3.8 (range, 2-6.3) years. Clinical assessment was based on the Japanese Society for Surgery of the Foot (JSSF) ankle/hindfoot scale as well as a Self-Administered Foot Evaluation Questionnaire (SAFE-Q). Radiographic assessments, including identification of the mechanical ankle joint axis point and a modified valgus Takakura-Tanaka osteoarthritis grade, were recorded. RESULTS: The mean JSSF score improved significantly from 45.2 preoperatively to 83.9 postoperatively (P < .001). SAFE-Q scores at the final follow-up were 70.1 for the pain and pain-related subscale, 75.4 for the physical functioning and daily living subscale, 83.0 for the social functioning subscale, 74.4 for the shoe-related subscale, and 78.1 for the general health and well-being subscale. Radiographic measurements showed improvement in the tibiotalar tilt angle (average improvement: 5.4 degrees); orientation of the talus joint line relative to the ground (average improvement: 14.0 degrees); anteroposterior talus-first metatarsal angle (average improvement: 11.2 degrees), talonavicular coverage angle (average improvement: 21.6 degrees), and lateral talus-first metatarsal angle (average improvement: 17.0 degrees). Postoperatively, the mechanical ankle joint axis point moved medially for all patients and into the medial half of the ankle joint for 7 patients. The modified osteoarthritis stage improved postoperatively in most cases. CONCLUSION: In this select and relatively small group of patients who had a supramalleolar lateral opening-wedge osteotomy combined with PCFD reconstruction, we measured general improvement in JSSF scores and radiographic variables. Long-term durability of the procedure remains unknown. This procedure may be an option for preserving the ankle joint in treatment of PCFD class E with osteoarthritis of the ankle. LEVEL OF EVIDENCE: Level IV, therapeutic.

Neonatal intrahepatic cholestasis caused by citrin deficiency with no hepatic steatosis: a case report.

BACKGROUND: Neonatal intrahepatic cholestasis caused by citrin deficiency (NICCD) is a common form of neonatal jaundice. Histopathological examination of the liver in patients with NICCD typically shows fatty liver, steatohepatitis, and liver fibrosis. Jaundice and fatty liver often improve by 1 year of age. We herein describe a girl who was diagnosed with NICCD based on an SLC25A13 mutation, although no fatty deposits were found on pathologic examination of the liver. CASE PRESENTATION: The patient in this case was a 3-month-old girl. At 2 months of age, she presented with jaundice, discolored stools, and poor weight gain and was found to have hyperbilirubinemia. Cholangiography revealed that she did not have biliary atresia. A laparoscopic liver biopsy was performed, and liver histopathology showed no fatty deposits. Genetic analysis revealed a compound heterozygous mutation in SLC25A13, and she was diagnosed with NICCD. She was given medium-chain triglyceride milk and gained weight. She resumed consumption of normal milk and breast milk, and her stool color improved. She was discharged at 4 months of age with adequate weight gain and a lower total bilirubin concentration. She was in good condition after discharge and showed normal development at the time of outpatient follow-up. CONCLUSIONS: We experienced a case of NICCD in a patient without fatty liver. This case illustrates that the absence of hepatic steatosis in neonatal cholestasis does not rule out NICCD.

Fixation of the osteochondral talar fragment yields good results regardless of lesion size or chronicity.

PURPOSE: Osteochondral talar lesions, regardless of their size and/or chronicity, are, at our hospital, now treated by fixation of the fragment if the talar dome cartilage is judged to be healthy. The retrospective study described herein was conducted to assess clinical outcomes of this treatment strategy. METHODS: The study group comprised 44 patients (18 men and 26 women) with 45 such talar lesions. In all cases, the osteochondral fragment was reduced and fixed with bone harvested from the osteotomy site and shaped into peg(s) (one to four pegs per lesion). Median follow-up was 2.1 years (1-9 years). The lesion area was measured on computed tomography arthrographs, and the Japanese Society for Surgery of the Foot (JSSF) ankle/hindfoot scale was used to evaluate postoperative outcomes. Clinical failure was defined as a JSSF score < 80 points. RESULTS: The mean JSSF score improved significantly from 63.5 points preoperatively to 93.0 postoperatively (p < 0.001). Treatment failure occurred in only one ankle (0.02%). The mean lesion area was 51.2 mm2 (range 5-147 mm2). Correlation between lesion areas and the postoperative JSSF scores was weak (r = - 0.133). Correlation between the time of the trauma to the time of fixation surgery and the postoperative JSSF scores was also weak (r = 0.042). Radiographic outcomes were good for 28 ankles, fair for 10, and poor for 7. CONCLUSION: Fixation of the lesion fragment, regardless of size and/or chronicity, appears to be appropriate in cases of an osteochondral talar lesion. LEVEL OF EVIDENCE: IV.

Anatomical Pathology of Subtle Lisfranc Injury.

The extent and patterns of Lisfranc joint complex disruption in subtle Lisfranc injuries have not been well clarified. We reviewed the direct intraoperative findings for 87 patients, examined computed tomography images that had been obtained preoperatively for 73 of the patients, and classified the injuries according to the Kaar  et al. criteria as the transverse type (instability between the first cuneiform [C1] and the second metatarsal [M2] and between the second cuneiform [C2] and M2) or longitudinal type (instability between C1 and M2 and between C1 and C2). Our patients' injuries were classified as follows: longitudinal type (38%), transverse type (30%), transverse type and first tarsometatarsal (TMT) joint injury (20%), longitudinal type plus transverse type (7%), longitudinal type and first TMT joint injury (3%), and longitudinal type, transverse type, and first TMT joint injury (2%). In 11 patients, the longitudinal injury extended into the naviculo-first cuneiform joint. In 41 (56%) of the 73 patients for whom CT images were obtained, 1 or more fractures (not counting small avulsion fragments between C1 and M2) were found. Orthopedic surgeons should be aware of the various injury patterns possible in cases of subtle Lisfranc injury.

T1ρ mapping of articular cartilage grafts after autologous osteochondral transplantation for osteochondral lesions of the talus: A longitudinal evaluation.

BACKGROUND: Clinical results of autologous osteochondral transplantation (AOT) for treatment of osteochondral lesions of the talus have been mixed. T1ρ imaging can be used to noninvasively detect early cartilage degeneration. PURPOSE OR HYPOTHESIS: To quantitatively assess, by means of T1ρ imaging, changes over time in the biochemical health of grafted cartilage after AOT for osteochondral lesions of the talus. STUDY TYPE: Retrosepctive case series. POPULATION: The study group comprised nine patients who underwent AOT for an osteochondral lesion of the talus and in whom T1ρ mapping was performed 1 and 2 years postoperatively. FIELD STRENGTH/SEQUENCE: 3 Tesla. T1ρ-weighted turbo field echo. ASSESSMENT: The mean T1ρ value of full-thickness cartilage at the repair site and that of full-thickness cartilage elsewhere in the same image (far-field cartilage) were determined. Clinical assessment was based on the American Orthopaedic Foot & Ankle Society (AOFAS) scale. Correlation between the T1ρ ratios (grafted-to-far-field cartilage T1ρ values) and clinical outcomes was examined. STATISTICAL TESTS: Mixed effects model. Pearson correlation analysis. RESULTS: At 1 year, a significant difference existed between the mean T1ρ value of the grafted cartilage (57.0 ± 7.7 ms) and that of the far-field cartilage (41.8 ± 4.6 ms) (P < 0.001). At 2 years, the mean T1ρ value of the grafted cartilage (49.1 ± 6.4 ms) was significantly lower than that at 1 year (P = 0.011). Moderate negative correlation was found between the 1-year T1ρ ratio and 1-year AOFAS score (r = -0.60) and between the 2-year T1ρ ratio and 2-year AOFAS score (r = -0.50). DATA CONCLUSION: Our observation of substantial restoration of the proteoglycan content of the grafted cartilage approximately 2 years after AOT for osteochondral lesions of the talus indicates that the content changes gradually and that the cartilage reparation process is slower than previously believed. LEVEL OF EVIDENCE: 3 Technical Efficacy: Stage 3 J. MAGN. RESON. IMAGING 2018;48:398-403.

SigCH, an extracytoplasmic function sigma factor of Porphyromonas gingivalis regulates the expression of cdhR and hmuYR.

Extracytoplasmic function (ECF) sigma factors play an important role in the bacterial response to various environmental stresses. Porphyromonas gingivalis, a prominent etiological agent in human periodontitis, possesses six putative ECF sigma factors. So far, information is limited on the ECF sigma factor, PGN_0319. The aim of this study was to investigate the role of PGN_0319 (SigCH) of P. gingivalis, focusing on the regulation of hmuY and hmuR, which encode outer-membrane proteins involved in hemin utilization, and cdhR, a transcriptional regulator of hmuYR. First, we evaluated the gene expression profile of the sigCH mutant by DNA microarray. Among the genes with altered expression levels, those involved in hemin utilization were downregulated in the sigCH mutant. To verify the microarray data, quantitative reverse transcription PCR analysis was performed. The RNA samples used were obtained from bacterial cells grown to early-log phase, in which sigCH expression in the wild type was significantly higher than that in mid-log and late-log phases. The expression levels of hmuY, hmuR, and cdhR were significantly decreased in the sigCH mutant compared to wild type. Transcription of these genes was restored in a sigCH complemented strain. Compared to the wild type, the sigCH mutant showed reduced growth in log phase under hemin-limiting conditions. Electrophoretic mobility shift assays showed that recombinant SigCH protein bound to the promoter region of hmuY and cdhR. These results suggest that SigCH plays an important role in the early growth of P. gingivalis, and directly regulates cdhR and hmuYR, thereby playing a potential role in the mechanisms of hemin utilization by P. gingivalis.

In vitro Effect of Mouthrinse Containing Essential Oils on Proliferation and Migration of Gingival Epithelial Cells.

We aimed to investigate in vitro the effects of mouthrinses containing essential oils (EOs) on proliferation and migration of gingival epithelial cells. Human gingival epithelial cells were treated with predetermined dilutions of commercially available EO mouthrinses with or without ethanol and a mouthrinse containing cetyl pyridinium chloride (CPC) for 60 s. Cell proliferation was evaluated using WST-1 assay. Cell migration was assessed using a wound closure model. Within 10 s of exposure to EO mouthrinse without ethanol, the epithelial cells became aberrant and shrank. No statistically significant difference in cell migration or proliferation was observed among cells pretreated by the EO mouthrinse with ethanol, CPC mouthrinse and control (phosphate buffered saline). In contrast, the EO mouthrinse without ethanol significantly reduced cell proliferation (p < 0.001) to approximately 20% relative to control. As for the EO mouthrinse without ethanol, it was not possible to assess its effect on cell migration using this model, because treated cells could be easily detached from the culture plate upon scratch, possibly because of the surfactant ingredient in the formulation. Within the limitations of the study, the EO mouthrinse with ethanol exerted no inhibitory effect on proliferation and migration of the gingival epithelial cells. Copyright © 2016 John Wiley & Sons, Ltd.

Effect of a mouthrinse containing rice peptide CL(14-25) on early dental plaque regrowth: a randomized crossover pilot study.

BACKGROUND: We aimed to evaluate clinically the effect of mouthrinse containing a rice peptide on early dental plaque regrowth. METHODS: The study was designed as a double-masked, two-group crossover randomized pilot trial, involving 10 periodontally healthy volunteers. After receiving a professional tooth cleaning at baseline, over the next 3 days each participant refrained from all oral hygiene measures and had two daily rinses with 20 ml of the test mouthrinse containing 0.4 % rice peptide CL(14-25) or placebo rinse. At the end of each experimental period, plaque score was assessed using the modified Volpe's method, and the participants filled out a questionnaire. Each participant underwent a 7-day washout period followed by a second allocation. The plaque score was the primary outcome of the study and participant perception was the secondary outcome. RESULTS: No adverse effects were observed in the participants during the study. Clinically, the mean plaque score of the examined teeth was significantly lower in the test group (2.44 ± 0.74, CI: 1.91-2.96) than the placebo group (2.65 ± 0.63, CI: 2.20-3.10) (P < 0.05). When analyzed according to the type of teeth, a significantly lower score of the premolars/molars was observed in the test group (2.39 ± 0.68, CI: 2.08-2.71) than that in the placebo group (2.66 ± 0.58, CI: 2.39-2.93) (P < 0.05). CONCLUSIONS: The mouthrinse containing 0.4 % rice peptide CL(14-25) was effective in reducing the early regrowth of dental plaque. However, clinical relevance of this efficacy needs to be validated in a future large-scale study. TRIAL REGISTRATION: UMIN Clinical Trials Registry (UMIN-CTR) R000014000. Date of formal registration: November 1, 2013.

Weight-bearing-line analysis in supramalleolar osteotomy for varus-type osteoarthritis of the ankle.

BACKGROUND: We determined the preoperative and postoperative passing points of the mechanical axis of the lower limb at the level of the tibial plafond using a new method involving a full-length standing posteroanterior radiograph that includes the calcaneus (a hip-to-calcaneus radiograph) and correlated them to the clinical results after supramalleolar osteotomy for ankle osteoarthritis. METHODS: We reviewed the hip-to-calcaneus radiographs of fifty lower limbs of forty-one patients treated for lower limb malalignment at our institution. The mechanical axis point of the ankle was the point at which the mechanical axis divides the coronal length of the plafond, expressed as a percentage. Four independent observers performed all measurements twice. Supramalleolar tibial osteotomy was performed in twenty-seven ankles (twenty-four patients) to treat moderate varus-type osteoarthritis of the ankle. The mean follow-up period was 2.8 years (range, two to 5.3 years). Clinical assessment was based on the American Orthopaedic Foot & Ankle Society (AOFAS) scale. RESULTS: Interobserver and intraobserver reliability in identifying the mechanical ankle joint axis point were very high. The mean postoperative mechanical axis point was 50% (range, 13% to 70%) in ankles for which the preoperative point was ≤0%, whereas the mean postoperative point was 81% (range, 48% to 113%) in ankles for which the preoperative point was >0%. The mean change in AOFAS score was significantly less for patients with a preoperative point of ≤0% than for those with a preoperative point of >0% (p=0.004). Improvement was significantly greater in ankles with a postoperative mechanical ankle joint axis point of ≥80% than in ankles with a postoperative mechanical ankle joint axis point of <60% (p=0.030). CONCLUSIONS: Traditional tibial correction resulted in great variation in the locations of the postoperative mechanical ankle joint axis point. In ankles with the preoperative point more medial than the tibial plafond, the point was insufficiently moved to the lateral side, and the clinical outcomes were less satisfactory.

Changes in antimicrobial susceptibility profile and prevalence of quinolone low-sensitive strains in subgingival plaque from acute periodontal lesions after systemic administration of sitafloxacin.

This study aimed to assess changes in antimicrobial susceptibilities of subgingival bacteria in acute periodontal lesions following systemic administration of a new-generation fluoroquinolone, sitafloxacin and to monitor the occurrence and fate of quinolone low-sensitive strains. Patients with acute phase of chronic periodontitis were subjected to microbiological assessment of their subgingival plaque samples at baseline (A1). Sitafloxacin was then administered systemically (100 mg/day for 5 days). The microbiological examinations were repeated one week after administration (A2). Susceptibilities of clinical isolates from acute sites to various antimicrobials were determined using broth and agar dilution methods. At A2, subgingival bacteria with low sensitivity to levofloxacin were identified in four patients, and they were subjected to a follow-up microbiological examination at on the average 12 months after sitafloxacin administration (A3). The patients received initial and supportive periodontal therapy during the period A2 to A3. From the examined subgingival sites, 8 and 19 clinical isolates were obtained at A2 and A3, respectively. Some Streptococcus strains isolated at A2 were found to be resistant to levofloxacin (MIC 16-64 µg/ml), azithromycin (MIC 2->128 µg/ml) or clarithromycin (MIC 1->32 µg/ml). At A3, isolated streptococci were highly susceptible to levofloxacin (MIC 0.5-2 µg/ml), while those resistant to azithromycin or clarithromycin were still isolated. It is suggested that the presence of the quinolone low-sensitive strains in initially acute lesions after sitafloxacin administration was transient, and they do not persist in the subgingival milieu during the periodontal therapy.

Effects of systemic administration of sitafloxacin on subgingival microflora and antimicrobial susceptibility profile in acute periodontal lesions.

The aim of this study was to assess the effect(s) of systemic administration of sitafloxacin on subgingival microbial profiles of acute periodontal lesions. Antimicrobial susceptibility of clinical isolates was also investigated. Patients with acute phases of chronic periodontitis were subjected to clinical examination and microbiological assessment of their subgingival plaque samples by culture technique. Sitafloxacin was then administered (100 mg/day for 5 days) systemically. The clinical and microbiological examinations were repeated 6-8 days after administration. Susceptibilities of clinical isolates to various antimicrobials were determined using the broth and agar dilution methods. From the sampled sites in 30 participants, a total of 355 clinical isolates (34 different bacterial species) were isolated and identified. Parvimonas micra, Prevotella intermedia and Streptococcus mitis were the most prevalent cultivable bacteria in acute sites. Systemic administration of sitafloxacin yielded a significant improvement in clinical and microbiological parameters. Among the antimicrobials tested, sitafloxacin was the most potent against the clinical isolates with an MIC90 of 0.12 µg/ml at baseline. After administration, most clinical isolates were still highly susceptible to sitafloxacin although some increase in MICs was observed. The results suggest that systemic administration of sitafloxacin is effective against subgingival bacteria isolated from acute periodontal lesions.

Prevalence of Aggregatibacter actinomycetemcomitans, Porphyromonas gingivalis and Tannerella forsythia in Japanese patients with generalized chronic and aggressive periodontitis.

This study aimed to investigate the prevalence and levels of major periodontal pathogens, Aggregatibacter actinomycetemcomitans, Porphyromonas gingivalis and Tannerella forsythia in subgingival plaque samples of a group of Japanese patients with aggressive periodontitis (AgP) and chronic periodontitis (CP). A total of 40 patients with clinical diagnosis of AgP or CP and 10 periodontally healthy volunteers were subjected to clinical and microbiological analysis. Subgingival plaque samples were analyzed for A. actinomycetemcomitans, P. gingivalis and T. forsythia with a real-time polymerase chain reaction (PCR) technique. The prevalence of P. gingivalis and T. forsythia was relatively high in patients with periodontitis: over 60% of AgP or CP patients harbored these pathogens whereas they were not detected in the subgingival plaque samples from periodontally healthy individuals. P. gingivalis and T. forsythia were relatively frequently detected together in AgP and CP patients. No significant differences in the prevalence or level of the 3 pathogens were found between periodontitis groups. The proportion of T. forsythia was approximately 4-fold higher in CP group than in AgP group (P = 0.02). In periodontitis patients, a significant positive correlation was found between periodontal parameters (probing depth and clinical attachment level) and the numbers of total bacteria, P. gingivalis and T. forsythia. No distinct pattern of the subgingival profile of these pathogens was discerned between the two disease entities, except for the difference in the proportion of T. forsythia. The red complex bacteria, P. gingivalis and T. forsythia were highly prevalent in this population of Japanese AgP and CP patients, collaborating their roles in periodontitis.