Single dose intralesional doxycycline foam infusion promotes bone growth and resolution of an odontoid aneurysmal bone cyst.

Background: Aneurysmal bone cysts (ABCs) are locally invasive bone tumors that most commonly arise in long bone metaphyses, the vertebral column, and pelvis, often presenting in the second decade of life. ABCs can be treated with resection, radiation, arterial embolization, and intralesional curettage. More recently intralesional doxycycline foam injections, which appear to act through the inhibition of matrix metalloproteinases and angiogenesis, have been used successfully, although multiple treatments are often required with this approach. Case Description: A 13-year-old male with an incidentally discovered ABC filling much of the odontoid process but not violating the native odontoid cortex was treated with a single intralesional doxycycline foam injection delivered through a transoral approach with an excellent radiographic result. After placing a Crowe-Davis retractor, a transoral exposure of the odontoid process was performed with neuronavigation guidance. A fluoroscopy-guided Jamshidi needle biopsy was performed and via the needle doxycycline foam (2 mL 50 mg/mL doxycycline, 2 mL 25% albumin, and 1 mL Isovue 370 mixed with 5 mL of air) was infused, filling the cystic cavities of the odontoid process. The patient tolerated the operation well. Two months postoperatively, a computed tomography (CT) scan demonstrated a decrease in the size of the lesion with substantial new bone formation. Repeat CT at 6 months showed no residual cystic lucency, formation of dense new bone, and only minimal irregularity of the cortex at the prior needle biopsy site. Conclusion: This case illustrates that the use of doxycycline foam can be an excellent option when managing ABCs that cannot be resected without incurring significant morbidity.

The effect of intrathyroidal versus intraperitoneal bevacizumab on thyroid volume and vasculature flow in a rat model.

BACKGROUND: Several clinical conditions increase thyroid gland vascularity, impacting surgical blood loss. Bevacizumab has been observed to reduce thyroid function, possibly through its effect on gland angiogenesis. This study aimed to determine if bevacizumab has any effect on thyroid vascularity as measured by gland volume and superior thyroid artery (STA) flow velocity in the normal rat thyroid. METHODS: Sixteen adult female Sprague-Dawley rats were placed under general anesthesia to measure baseline thyroid gland characteristics. A Vevo 2100 high-frequency ultrasound with 40 mHz transducer was used to obtain STA flow measurements and thyroid gland dimensions. Four rats served as controls. Six rats received intrathyroidal (IT) injections and 6 received intraperitoneal (IP) injections of bevacizumab (4-5 mg/kg). After two weeks ultrasound measurements were repeated. RESULTS: Pretreatment animals displayed similar thyroid volume and vascularity. Thyroid volume decreased (62.583 vs. 42.161, P=0.004) after IP administration of bevacizumab, and blood flow measurements did not change [peak velocity 75.896 vs. 76.7, P=0.96, average velocity 45.748 vs. 43.867, P=0.88, or resistivity index (RI) 30.345 vs. 25.32, P=0.60]. IT bevacizumab did not change thyroid volume (55.229 vs. 58.16, P=0.64). The average peak (73.191 vs. 100.589 cm/s, P=0.03) and mean (45.047 vs. 62.843 m/s, P=0.03) velocities were increased, but did not differ in the RI (0.619 vs. 0.632, P=0.82). No differences were noted on VEGF or CD 31 immunohistochemical analysis. CONCLUSIONS: Single systemic administration of bevacizumab appears to decrease thyroid volume without an effect on STA flow, VEGF or CD31 staining. These preliminary findings support further study of pharmacologic intervention in thyroid conditions characterized by increased angiogenesis and vascularity, such as iodine deficiency, Graves disease, and hypothyroidism.

Management protocol for primary hyperparathyroidism in a single institution: utility of surgeon performed ultrasound.

BACKGROUND: We aimed to: (I) discover preoperative diagnostic studies, intraoperative techniques, and patient factors most predictive of cure within a single hospital system; (II) establish practice guidelines for surgical treatment of primary hyperparathyroidism to maximize outcomes based on this hospital system's performance. METHODS: A retrospective chart review was undertaken of all parathyroid-related procedures from 01/01/02 to 7/31/15 at the Veteran's Administration Hospital. RESULTS: Seventy-one patients were eligible and charts available for analysis. Preoperative studies most predictive of cure were a combination of sestamibi parathyroid scan and surgeon performed ultrasound (S-US). When studies did not agree, S-US was most often correct. Intraoperative parathyroid hormone (PTH) rapid assay was helpful in predicting cure, but added an average of 33 minutes to operating room time. Patients who had two corroborating preoperative localizing studies, one of which was S-US, that agreed with intraoperative findings, and who did not undergo intraoperative PTH confirmation enjoyed equal cure rates and shorter operating room times. Successful achievement of normal calcium was high at 95.8%. Vitamin D deficiency was prevalent in this patient population, prompting more aggressive preoperative investigation and replacement. CONCLUSIONS: A management protocol was developed based on the findings of this study: (I) obtain two preoperative localization studies, one of which is surgeon-performed ultrasound; (II) obtain preoperative vitamin D levels and supplement as indicated; and (III) in select patients who have two strongly corroborating preoperative localization studies, one of which is surgeon performed ultrasound, and intraoperative findings are consistent with the localizing studies, intraoperative PTH (IOPTH) may not be necessary.

Use of Ultrasound for Sizing Tracheoesophageal Puncture Prostheses.

Tracheoesophageal puncture (TEP) with voice prosthesis placement is the gold standard voice rehabilitation following total laryngectomy. Ultrasound may be useful to determine tracheoesophageal wall thickness, guiding prosthesis choice. Therefore, 14 patients undergoing total laryngectomy and TEP or prosthesis change with 16-mHz ultrasound measurement of the posterior tracheal wall were included. Seven patients underwent secondary TEP, 3 primary TEP, and 4 TEP changes. Six patients underwent flap reconstruction, while 8 patients were closed primarily. Average party wall thickness was 9.6 ± 1.7 mm, without a difference ( P = .08) between primary closure (10.3 ± 1.7 mm) and flap reconstruction (8.6 ± 1.4 mm). Change from the hypothesized sizing was noted in 11 patients (79%). Prosthesis size did not correlate with age (-0.19, P = .51), height (-0.12, P = .69), weight (0.26, P = .38), body mass index (0.22, P = .46), or flap status (-0.48, P = .079). These data suggest that ultrasound is beneficial in patients with distorted or less predictable anatomy (eg, flap reconstruction) but also important for those patients undergoing primary closure.

Invasive fungal sinusitis in the pediatric population: Systematic review with quantitative synthesis of the literature.

BACKGROUND: Invasive fungal sinusitis (IFS) represents an often fatal condition within the pediatric population. In an effort to characterize demographics, treatment modalities, and prognostic factors, we performed a systematic review. METHODS: We systematically reviewed EMBASE, Medline, TRIPdatabase, SCOPUS and the Cochrane database for invasive fungal nasal and sinus infections limited to individuals <18 years of age. Case series including 3 or more patients were included. Demographics, treatment and outcomes were analyzed using R Gui statistical software. RESULTS: Twelve studies met inclusion criteria (103 patients). There was male preponderance of 48.5% with median age of 11 years old. Majority of patients had underlying leukemia (44.6%). Aspergillus was the predominant organism (47%). Isolated nasal findings occurred in 14% of patients and nasal findings occurred in 49% overall. Absolute neutrophil count (ANC) of immunocompromised patients was below 600 in most patients (99%). Average and median length of neutropenia was 2 weeks. All patients were prescribed amphoterocin with 50% as single medicinal therapy. Surgery occurred in 82.8% of cases. The mortality rate was 46%. Univariate analysis identified presenting with facial pain as a negative predictor of overall mortality (OR 0.296, 95% CI: 0.104-0.843, p < 0.05). CONCLUSION: Mortality remains high in pediatric patients with IFS. An ANC of <600 occurred in the majority of immunocompromised patients at a duration of 2 weeks. Presenting with facial pain was a negative predictor of mortality. Many studies label this condition as invasive fungal sinusitis; however, approximately one seventh presented with only nasal findings and half overall had nasal involvement.

Incidence of Visualization of the Glossopharyngeal Nerve after Pediatric Tonsillectomy.

The objective was to determine the incidence of exposure of the lingual branch of the glossopharyngeal nerve during tonsillectomy with a retrospective review of surgical findings in 138 children who underwent total tonsillectomy at a tertiary medical center. Age, sex, surgical indication, tonsil size, congenital abnormalities, operative time, and surgical findings indicating the presence or absence of the glossopharyngeal nerve in the tonsillar fossa were recorded. Statistical analysis was performed with z test, t test, and Fisher's exact test. Thirty-seven nerves were observed in 28 patients, with preponderance for the left fossa (24 of 37 vs 13 of 37; P = .01). In a comparison of children with and without exposed nerves, there was no statistically significant difference in mean age (6.89 vs 7.08; P = .84), proportion of males (14 of 28 vs 54 of 110; P = 1), or proportion of 3 to 4+ tonsils (20 of 28 vs 73 of 110; P = .66). In approximately 20% of children undergoing tonsillectomy, the lateral pharyngeal musculature incompletely protected the lingual branch of the glossopharyngeal nerve from the tonsil capsule.

A new mutation of the Atoh1 gene in mice with normal life span allows analysis of inner ear and cerebellar phenotype in aging.

Atoh1 is a transcription factor that regulates neural development in multiple tissues and is conserved among species. Prior mouse models of Atoh1, though effective and important in the evolution of our understanding of the gene, have been limited by perinatal lethality. Here we describe a novel point mutation of Atoh1 (designated Atoh1(trhl) ) underlying a phenotype of trembling gait and hearing loss. Histology revealed inner ear hair cell loss and cerebellar atrophy. Auditory Brainstem Response (ABR) and Distortion Product Otoacoustic Emission (DPOAE) showed functional abnormalities in the ear. Normal lifespan and fecundity of Atoh1(trhl) mice provide a complementary model to facilitate elucidation of ATOH1 function in hearing,central nervous system and cancer biology.

Heterogeneous transmural proteoglycan distribution provides a mechanism for regulating residual stresses in the aorta.

The arterial wall contains a significant amount of charged proteoglycans, which are inhomogeneously distributed, with the greatest concentrations in the intimal and medial layers. The hypothesis of this study is that the transmural distribution of proteoglycans plays a significant role in regulating residual stresses in the arterial wall. This hypothesis was first tested theoretically, using the framework of mixture theory for charged hydrated tissues, and then verified experimentally by measuring the opening angle of rat aorta in NaCl solutions of various ionic strengths. A three-dimensional finite element model of aortic ring, using realistic values of the solid matrix shear modulus and proteoglycan fixed-charge density, yielded opening angles and changes with osmolarity comparable to values reported in the literature. Experimentally, the mean opening angle in isotonic saline (300 mosM) was 15 +/- 17 degrees and changed to 4 +/- 19 degrees and 73 +/- 18 degrees under hypertonic (2,000 mosM) and hypotonic (0 mosM) conditions, respectively (n = 16). In addition, the opening angle in isotonic (300 mosM) sucrose, an uncharged molecule, was 60 +/- 16 degrees (n = 11), suggesting that the charge effect, not cellular swelling, was the major underlying mechanism for these observations. The extent of changes in opening angle under osmotic challenges suggests that transmural heterogeneity of fixed-charge density plays a crucial role in governing the zero-stress configuration of the aorta. A significant implication of this finding is that arterial wall remodeling in response to altered wall stresses may occur via altered deposition of proteoglycans across the wall thickness, providing a novel mechanism for regulating mechanical homeostasis in vascular tissue.