Rosai-Dorfman: Rare Manifestations of a Rare Disease.

Rosai-Dorfman disease (RDD) is an exceedingly rare non-Langerhans cell histiocytosis of uncertain etiology that most commonly presents in children as self-limited, painless, massive cervical lymphadenopathy. However, extranodal disease occurs in 43% of cases and has a wide range of phenotypic presentations. The pathogenesis has not been clearly understood in the literature and coupled with a wide range of clinical manifestations, early diagnosis and initiation of an appropriate treatment modality have been challenging. Herein, we describe a cohort of five cases that occurred at the same institution within a 12-month period. These cases highlight unique and atypical presentations of an already rare disease, outline the varying and tailored diagnostic and therapeutic approaches, and propose a novel environmental predisposing factor given the exceptionally high incidence at our institution over a short period of time. We emphasize the need for further investigation of predisposing factors and to discern targeted therapies that may offer benefits.

Initial Displacement of Humeral Shaft Fractures Is Associated With Failure of Nonoperative Management.

OBJECTIVES: To determine whether the initial radiographic displacement of humeral shaft fractures is associated with failure of nonoperative management. DESIGN: Retrospective cohort study. SETTING: Urban level 1 trauma center. PATIENTS/PARTICIPANTS: 106 patients with humeral shaft fractures (OTA/AO 12) initially managed nonoperatively. INTERVENTION: Functional bracing. MAIN OUTCOME MEASUREMENTS: Failure of nonoperative management, defined as conversion to surgery, malunion, and delayed union/nonunion. RESULTS: Nonoperative management failed in 33 (31%) of 106 included patients with 27 patients (25%) requiring surgery. On multivariate analysis, female sex [odds ratio (OR): 3.50, 95% confidence interval (CI): 1.09 to 11.21], American Society of Anesthesiologist classification >1 (OR: 7.16, CI: 1.95 to 26.29), initial fracture medial/lateral (ML) translation (OR: 1.09, CI: 1.01 to 1.17, per unit change), and initial fracture anterior-posterior (AP) angulation (OR: 1.09, CI: 1.02 to 1.15, per unit change) were independently associated with failure of nonoperative management. Initial fracture displacement values that maximized the sensitivity (SN) and specificity (SP) for failure included an AP angulation >11 degrees (SN 75%, SP 64%) and ML translation >12 mm (SN 55%, SP 75%). The failure rate in patients with none, 1, or both of these fracture parameters was 3.1% (1/32), 35.6% (20/56), and 66.6% (12/18), respectively. CONCLUSIONS: Nearly one-third of patients experienced failure of initial nonoperative management. Failure was found to be associated with greater initial fracture AP angulation and ML translation. Fracture displacement cut-off values were established that may be used by surgeons to counsel patients with these injuries. LEVEL OF EVIDENCE: Prognostic Level III. See Instructions for Authors for a complete description of levels of evidence.

Incorporation of Whipstitch Suture in Tibial Interference Fixation Improves Pullout in Anterior Cruciate Ligament Soft Tissue Grafts.

INTRODUCTION: Interference screw fixation of soft tissue grafts is commonly used in anterior cruciate ligament (ACL) reconstruction. The purpose of this study was to determine whether including suture material at the graft-screw interface affects ultimate fixation strength of soft tissue grafts using a tibialis anterior tendon allograft model. MATERIALS AND METHODS: Forty fresh-frozen human tibialis anterior tendon allografts were fixed to rigid polyurethane foam simulating the tibial tunnel. Twenty grafts underwent fixation with interference screws and 20 with interference bolts. Within each group, 10 grafts had suture in contact with either the screw or bolt. A load-to-failure test was then performed at a rate of 200 mm/min. RESULTS: The group of allografts with sutures in the tibial tunnel had significantly higher load to failure than the group without sutures. Using interference screw fixation, failure load of the grafts without sutures in the tunnel (535.2 ± 73.40 N) was significantly lower (P = .001) than with sutures in the tunnel (696.3 ± 110.0 N). Using interference bolt fixation, failure load of the grafts without sutures in the tunnel (613.0 ± 83.46 N) was significantly lower (P <.0001) than with sutures in the tunnel (845.8 ± 87.23 N). CONCLUSIONS: In a biomechanical model, suture within the tibial tunnel enhances fixation strength with both interference screw and bolt fixation for soft tissue tibialis anterior allografts. Additionally, there was no difference in load to failure when comparing failure of a screw with suture in the tunnel with an interference bolt without suture. Due to improved biomechanical properties, incorporation of suture in the bone-graft interface should be considered when performing soft tissue ACL allograft reconstructions. Failure at the tibial bone-graft interface is a known complication of ACL reconstruction, and incorporation of suture within the interface should be considered for improved biomechanical properties.

Squeaking Is Common and Increases Over Time Among Patients With Long-term Follow-up After Ceramic-on-ceramic THA.

BACKGROUND: Ceramic-on-ceramic (CoC) is a durable bearing with excellent wear characteristics, but squeaking remains a concern. The proportion of patients who report squeaking varies widely between studies performed at short- and mid-term follow-up. QUESTIONS/PURPOSES: (1) What proportion of patients treated with CoC THA bearings report squeaking at a minimum of 10 years of follow-up? (2) Are patient, implant, or radiographic factors associated with squeaking? (3) Are THAs that squeak more likely to be revised than those that do not? (4) Are patient-reported functional outcome scores lower between THAs that squeak and THAs that do not squeak at long-term follow-up? METHODS: Between January 1, 2003 and August 31, 2008 a total of 80 patients received THAs with third-generation alumina-on-alumina bearings at one center. Of the original 80 patients, 1% (1 of 80) had died, and 21% (17 of 80) were lost to follow-up before 10 years, leaving 62 patients for analysis at a median (range) of 14 years (11 to 16). Ceramic-on-ceramic THA represented 23% (80 of 343) of all primary THAs performed during the study period. Ceramic-on-ceramic THA was used preferentially in patients younger than 50 years of age. The mean (range) age of patients in the cohort was 44 ± 11 years (18 to 65). Sixty-eight percent (42 of 62) were men. Two separate manufacturers' implants were included. There were uncemented acetabular and femoral components included in this study. All CoC bearings were third-generation alumina-on-alumina. Squeaking was determined through a mailed questionnaire or telephone interview. The 10-question survey developed by the researchers queried patients whether audible "squeaking" could be heard from their hip replacement. Patients were asked to write in their description of the noise to distinguish squeaking from other noises not relevant to the current study. Implant information, component position, and patient demographics were obtained via chart review and postoperative radiographs reviewed by one of the investigators not involved with the index operative procedure. Using revision for any reason as an endpoint, a Kaplan-Meier analysis was performed to compare survivorship between THAs that squeaked versus those that did not. Patient-reported outcomes were surveyed using the Hip Disability and Osteoarthritis Outcome Score for Joint Replacement (HOOS JR), which comprises six items regarding patient pain and function, raw summed scores range from 0 (perfect hip health) to 24 (total hip disability). RESULTS: Squeaking was self-reported by 53% (33 of 62) of patients in this group. Squeaking was more common in patients who received the titanium-molybdenum-zirconium-iron stem than in patients with the titanium-aluminum-vanadium stem (63% [29 of 46] versus 31% [4 of 13]; odds ratio 3.8 [95% CI 1.02 to 14.4]; p = 0.046). We found no differences in the likelihood a patient would report squeaking based on component position, component size, patient age, sex, or BMI. Ten-year survivorship free from revision was not lower in patients who reported squeaking (91% [95% CI 74 to 97] versus 90% [95% CI 71 to 96]; p = 0.69). Patient-reported outcome scores (HOOS JR) were not lower in those who reported squeaking (3 ± 3 [95% CI 1.5 to 4.0] versus 3 ± 5 [95% CI 1.5 to 5.5]; p = 0.59). CONCLUSION: At long-term follow-up, we found that CoC bearing squeaking in patients who underwent THA is more common than previously reported. Survivorship was lower than expected in this cohort, and most revisions in this series were for squeaking. Although implant-dependent, surgeons should counsel patients about the potential for squeaking in CoC THA, which may occur years after index procedure. LEVEL OF EVIDENCE: Level III, therapeutic study.