18F-FDG PET imaging for treatment response assessment and management guidance in patients with skull base osteomyelitis.

OBJECTIVE: The objective of this study is to evaluate the role of hybrid 18F-FDG PET for treatment response assessment and management guidance in patients with skull base osteomyelitis. MATERIALS AND METHODS: Retrospectively, 33 patients, with at least a baseline and follow-up PET (computed tomography/MRI) scan, were included. Parameters like standardized uptake value (SUV) max, erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP) (initial, follow-up, percentage change) were analysed and outcomes based on 18F-FDG PET reports were classified into resolution (a), partial response (b), progression (c) and stable disease (d). The clinical course and response on anatomical imaging were also compared with 18F-FDG PET. RESULTS: There was mild correlation between initial SUV with ESR (0.338) and CRP (0.384). Moderate correlation was seen between follow-up SUV and CRP (0.619), percentage change in SUV max (PC SUV) with percentage change in ESR (0.456) and CRP (0.668). Mean PC SUV was 70% (a), 35% (b), -40% (c) and -18% (d), respectively. 48% (16/33) (resolution, progression, stable disease) patients had clear management change with 18F-FDG PET with either escalation or stopping of antibiotics/antifungals. Management decision in partial response group (52%, 17/33) was taken clinically. On retrospective PC SUV analysis, treatment continuation group (8 patients) showed 20% decrease, whereas the group that was only monitored further (9 patients) had 48% reduction in SUV. CONCLUSION: 18F-FDG PET showed a moderate association with clinical markers used in follow-up of patients with skull base osteomyelitis and is a reliable investigation for assessment of disease status. This can be used as a guide along with clinical evaluation for de-escalation of treatment.

68 Ga-Citrate PET/CT Imaging in the Diagnosis of Skull Base Osteomyelitis and Its Usefulness in the Assessment of Treatment Response.

PURPOSE: 68 Ga-citrate PET/CT imaging in the diagnosis of skull base osteomyelitis (SBO) and its usefulness in the assessment of treatment response. METHODS: Eighteen patients underwent 68 Ga-citrate PET/CT scans for suspected SBO for diagnosis/assessment of treatment response. RESULTS: Of 18 patients, 16 patients had a positive study for SBO, and 2 patients had a negative study. Scan findings were correlated with clinical, biochemical, microbiological, and radiological parameters. CONCLUSION: 68 Ga-citrate PET/CT is a promising tool in the diagnosis and management of skull base osteomyelitis.

Acute musculoskeletal infection in children: assessment and management.

Musculoskeletal infection in children is challenging to treat, and includes septic arthritis, deep tissue infection, osteomyelitis, discitis and pyomyositis. Delays to diagnosis and management, and under-treatment can be life-threatening and result in chronic disability. The British Orthopaedic Association Standards for Trauma include critical steps in the timely diagnosis and management of acute musculoskeletal infection in children, the principles of acute clinical care and the service delivery requirements to appropriately manage this cohort of patients. Orthopaedic and paediatric services are likely to encounter cases of acute musculoskeletal infection in children and thus an awareness and thorough understanding of the British Orthopaedic Association Standards for Trauma guidelines is essential. This article reviews these guidelines and associated published evidence for the management of children with acute musculoskeletal infection.

Analysis of the epidemiological status, microbiology, treatment methods and financial burden of hematogenous osteomyelitis based on 259 patients in Northwest China.

Background: The incidence of hematogenous osteomyelitis is on the rise, and the prognosis is poor. There has been no large-scale epidemiological analysis of hematogenous osteomyelitis in the world, and the treatment method is still controversial. Methods: A retrospective case study method was used to collect and analyze clinical data obtained from patients with hematogenous osteomyelitis in a tertiary hospital in Northwest China from January 1, 2011, to December 31, 2020. The aim of this study was to investigate the epidemiological status, microbiological characteristics, treatment and financial burden of hematogenous osteomyelitis in Northwest China to explore the therapeutic effects of different treatment methods, elucidate the epidemiological characteristics of hematogenous osteomyelitis and to provide a basis for the choice of treatment. Results: We included 259 patients with hematogenous osteomyelitis, including 96 patients with acute hematogenous osteomyelitis and 163 patients with chronic hematogenous osteomyelitis. The cause of the disease was not obvious in most patients, the sex ratio of males to females was 1.98, and the three most common infected sites were the tibia, femur and phalanx. Regarding preoperative serum inflammatory markers, the rate of positivity for ESR was the highest at 67.58%. Among pathogenic microorganisms, Staphylococcus aureus was the most common. Regarding the financial burden, the median total cost per patient was 25,754 RMB, and medications accounted for the largest proportion of the main costs. Conclusions: The most common pathogen associated with HO infection was MSSA. Oxacillin has good PK and PD and is recommended as the first-line drug. Some blood-borne bone infections may lead to complications, such as pulmonary infection through bacteremia, which requires early detection to avoid a missed diagnosis. Regarding surgical intervention, debridement plus absorbable calcium sulfate bone cement and calcium sulfate calcium phosphate bone cement exclusion have achieved good therapeutic effects, but they are worthy of further in-depth research. Regarding the financial burden, the median total cost per patient was 25,754 RMB. The financial burden of blood-borne osteomyelitis was lower than that of traumatic osteomyelitis. Among the main costs, drugs accounted for the largest proportion.

Chronic Nonbacterial Osteomyelitis: Insights into Pathogenesis, Assessment, and Treatment.

Chronic nonbacterial osteomyelitis, or its most severe form, chronic recurrent multifocal osteomyelitis, is an autoinflammatory bone disease that causes skeletal inflammation characterized by bone pain and swelling that primarily affects children. It is a diagnosis of exclusion and its clinical presentation may mimic underlying infectious processes and malignancy. Clinical suspicion for this diagnosis and timely referral to pediatric rheumatology is crucial to achieve earlier diagnosis, appropriate treatment, and improved quality of life of affected patients and families. This article focuses on recent insights into the pathogenesis of chronic nonbacterial osteomyelitis and outlines recent advances and ongoing research.

Incidence of Osteomyelitis in Sacral Decubitus Ulcers and Recommendations for Management.

Stage-4 pressure ulcers are defined as ulcerations that violate the fascia and expose underlying bone, muscle, and tendon. Exposed bone is always colonized by bacteria, but this does not necessarily lead to osteomyelitis. The rates of osteomyelitis in exposed bone in stage-4 pressure ulcers range from 14% to 86%.There has been no evidence that the presence of osteomyelitis leads to higher complication rates following flap coverage. There has been no evidence that bone biopsy and preoperative treatment of osteomyelitis have any benefit before flap coverage.

Health Care Costs of Post-traumatic Osteomyelitis in China: Current Situation and Influencing Factors.

BACKGROUND: Currently, very limited information is available regarding the economic burdens of patients with extremity post-traumatic osteomyelitis (OM). This study aimed to investigate direct health care costs and utilization for inpatients with extremity post-traumatic OM and analyze its constituent ratios and influencing factors in Southern China. METHODS: We searched in the electronic medical record system for inpatients who had received surgical interventions at our department between 2013 and 2016 for extremity post-traumatic OM. Data of direct health care costs incurred during their hospitalizations were collected in six main categories (service, diagnosis, treatment, materials, pharmaceuticals, and miscellaneous expenses). In addition, data of total medical costs for contemporaneous inpatients with non-post-traumatic OM were also collected as controls. RESULTS: A total of 278 post-traumatic OM and 10,420 controls were included. The median cost for the post-traumatic OM inpatients was $10,504 US dollars, 4.8-fold higher than that for those with non-post-traumatic OM ($2189, P < 0.001). The direct cost in the category of materials accounted for the largest proportion (61%), followed by that in pharmaceuticals (12%) and treatment (11%). The median number of hospital admissions for post-traumatic OM patients was 1 time, with a median length-of-stay of 22 d. The most influencing factors for the health care costs of the post-traumatic OM inpatients were use of an external fixator ($16,016 for those who used versus $4956 for those who did not, P < 0.001), external fixator type ($19,563 for ring fixator versus $14,966 for rail fixator, P < 0.001), infection site ($13,755 for tibia, $14,216 for femur and $5673 for calcaneus, P < 0.001), and infection-associated injury type ($12,890 for infection after open fracture versus $8087 for infection after closed fracture, P = 0.001). CONCLUSIONS: An unexpectedly large proportion of the direct health care costs for inpatients with extremity post-traumatic OM went to cover an external fixator, with expenses for pharmaceuticals and treatment accounting for only a little more than the tenth of the total health care costs. Use of external fixator, external fixator type, infection site, and infection-associated injury type directly influenced the health care costs.

Organisational and financial consequences of the early discharge of patients treated for acute bacterial skin and skin structure infection and osteomyelitis in infectious disease departments in Greece, Italy and Spain: a scenario analysis.

OBJECTIVE: The aim of the analysis is to assess the organisational and economic consequences of adopting an early discharge strategy for the treatment of acute bacterial skin and skin structure infection (ABSSSI) and osteomyelitis within infectious disease departments. SETTING: Infectious disease departments in Greece, Italy and Spain. PARTICIPANTS: No patients were involved in the analysis performed. INTERVENTIONS: An analytic framework was developed to consider two alternative scenarios: standard hospitalisation care or an early discharge strategy for patients hospitalised due to ABSSSI and osteomyelitis, from the perspective of the National Health Services of Greece, Italy and Spain. The variables considered were: the number of annual hospitalisations eligible for early discharge, the antibiotic treatments considered (ie, oral antibiotics and intravenous long-acting antibiotics), diagnosis-related group (DRG) reimbursements, number of days of hospitalisation, incidence and costs of hospital-acquired infections, additional follow-up visits and intravenous administrations. Data were based on published literature and expert opinions. PRIMARY AND SECONDARY OUTCOME MEASURES: Number of days of hospitalisation avoided and direct medical costs avoided. RESULTS: The total number of days of hospitalisation avoided on a yearly basis would be between 2216 and 5595 in Greece (-8/-21 hospital beds), between 15 848 and 38 444 in Italy (-57/-135 hospital beds) and between 7529 and 23 520 in Spain (-27/-85 hospital beds). From an economic perspective, the impact of the early discharge scenario is a reduction between €45 036 and €149 552 in Greece, a reduction between €182 132 and €437 990 in Italy and a reduction between €292 284 and €884 035 in Spain. CONCLUSIONS: The early discharge strategy presented would have a positive organisational impact on National Health Services, leading to potential savings in beds, and to a reduction of hospital-acquired infections and costs.

Nationwide epidemiologic study for pediatric osteomyelitis and septic arthritis in South Korea: A cross-sectional study of national health insurance review and assessment service.

Epidemiologic studies of pediatric bone and joint infection have been done mostly by developed Western countries, and such results could be expected to be biased. Therefore, an epidemiologic study to identify the distinct features of an Eastern country would be beneficial to improve their health outcomes and to reduce health care cost. A study was planned to investigate the epidemiology of pediatric osteomyelitis and septic arthritis in South Korea and to find out epidemiologic factors related with the occurrence of surgery.We conducted a cross-sectional study among hospitalized pediatric patients (<18 years old) with osteomyelitis and septic arthritis using nationwide cohort based on the Health Insurance Review and Assessment Service (HIRA) from 2008 to 2016. Percentage of hospitalization was additionally calculated according to several epidemiologic factors including age, month, site of infection, and region of residence. Logistic regression analysis was performed to find out the association between epidemiologic factors and occurrence of surgeryAnnual hospitalization rates (per 100,000) in our country were 7.8 to 9.1 for osteomyelitis, and 11.9 to 20.8 for septic arthritis. Frequent sites of osteomyelitis and septic arthritis were pelvis and lower extremity. The hospitalization rates of osteomyelitis and septic arthritis showed increasing trend with age. After multivariate logistic regression analysis, female gender, children, and adolescents rather than infants, rural area rather than urban area were associated with significantly increased odd ratios for surgery.The hospitalization rate of septic arthritis (11.9 to 20.8 per 100,000) in South Korea was higher than the Western countries (1.1 to 11 per 100,000), and the hospitalization rate of osteomyelitis (peak at 12 years) and septic arthritis (peak at 17 years) showed increasing trend with age, which was different from those of the developed Western countries showing higher incidence in children aged under 5 years. After multivariate logistic regression analysis, our study identified female gender, higher age group including children, and adolescents, and rural residence as epidemiologic risk factors associated with surgery.

The CRIOAc healthcare network in France: A nationwide Health Ministry program to improve the management of bone and joint infection.

BACKGROUND: Bone and joint infections (BJIs) have a major clinical and economic impact in industrialized countries. Its management requires a multidisciplinary approach, and a great experience for the most complicated cases to limit treatment failure, motor disability and amputation risk. To our best knowledge there is not currently national specific organization dedicated to manage BJI. Is it possible to build at a national level, a network involving orthopaedic surgeons, infectiologists and microbiologists performing locally multidisciplinary meetings to facilitate the recruitment and the management of patients with complex bone and joint infection in regional centers? HYPOTHESIS: A national healthcare network with regional labeled centers creates a dynamic that improves the recruitment, the management, the education, and the clinical research in the field of complex BJI. PATIENTS AND METHODS: We describe the history of this unique national healthcare network and how it works, specify the missions confided to the CRIOAcs, evaluate the activity of the network over the first decade, and finally discuss perspectives. RESULTS: The labelling of 24 centers in the CRIOAc network allowed for a meshing of the territory, with the possibility of management of complex BJI in each region of France. A dedicated secure national online information system was designed and used to facilitate decision-making during multidisciplinary consultation meetings. Since October 2012 to June 2017, 4553 multidisciplinary consultation meetings have been performed in the structures belonging to the network, with 34,607 cases discussed in 19,961 individual. Prosthetic joint infections represented 38% (7585/19,961) of all BJIs. Among all the cases discussed, the rate of complexity was of 61% (21,110/34,607) (related to antibiotic resistance, infection recurrence, patient co morbidities). A national scientific meeting was created and a national postgraduate diploma in the field of BJI was launched in 2014. The promotion of education, clinical research and interactivity between each academic discipline and between each labeled centers across the country has synergized the strengths and have greatly facilitated the management of patients with BJI. DISCUSSION: The setting up of the CRIOAc network in France took time, and has a cost for the French Ministry of Health. However, this network has greatly facilitated the management of BJI in France, and allowed to concentrate the management of complex BJI in centers that have significantly gained skills. There is, to our knowledge, no other exemple of such nationwide network in the field of BJI. LEVEL OF EVIDENCE: IV, case series without control group.

Use of a Clinical Care Algorithm to Improve Care for Children With Hematogenous Osteomyelitis.

BACKGROUND AND OBJECTIVES: Acute hematogenous osteomyelitis (AHO) causes significant morbidity in children. Quality improvement (QI) methods have been used to successfully improve care and decrease costs through standardization for numerous conditions, including pediatric AHO. We embarked on a QI initiative to standardize our approach to the inpatient management of AHO, with a global aim of reducing inpatient costs. METHODS: We used existing literature and local consensus to develop a care algorithm for the inpatient management of AHO. We used the Model for Improvement as the framework for the project, which included process mapping, failure mode analysis, and key driver identification. We engaged with institutional providers to achieve at least 80% consensus regarding specific key drivers and tested various interventions to support uptake of the care algorithm. RESULTS: Fifty-seven patients were included. There were 31 patients in the preintervention cohort and 26 in the postintervention cohort, of whom 19 were managed per the algorithm. Mean inpatient charges decreased from $45 718 in the preintervention cohort to $32 895 in the postintervention cohort; length of stay did not change. Adherence to recommended empirical antimicrobial agents trended upward. CONCLUSIONS: A simple and low-cost QI project was used to safely decrease the cost of inpatient care for pediatric AHO at a tertiary care children's hospital. A robust local consensus process proved to be a key component in the uptake of standardization.

Disease burden and social impact of pediatric chronic nonbacterial osteomyelitis from the patient and family perspective.

BACKGROUND: Chronic nonbacterial osteomyelitis (CNO) is an autoinflammatory bone disorder that if left untreated can result in bone destruction and severe continuing pain due to persistent inflammation. The impact this chronic disease has on the daily lives of affected children and their families is not well known. The purpose of this study is to understand the disease burden and socioeconomic and psychological impact of CNO from the patients' and families' perspectives and identify areas of improvement for patient care and reduced disease burden based on patients' and families' responses. METHODS: Participants were invited through a social media platform group and at clinic visits at Stanford Children's Health. An online survey was administered to patients with a diagnosis of CNO made at < 22 years of age and/or the parent/guardian of a patient with CNO diagnosis made at < 22 years of age. RESULTS: There was a total of 284 survey participants. The median age at CNO diagnosis was 10 years (range 2-22+). Median time from first CNO symptom to diagnosis was 2 years. Antibiotics were used in 35% of patients prior to CNO diagnosis; of these, 24% received antibiotics for greater than 6 months. Between 25 and 61% reported a negative effect of CNO on relationships, school/work performance, or finances; and 19-50% reported effects on psychosocial well-being. The majority agreed patients' performance with daily tasks and hobbies was challenged by pain, fatigue and physical limitation related to CNO. CONCLUSIONS: Patients with CNO experienced on average a 2-year delay in diagnosis and receiving effective treatments. At least 25% reported problems with relationships, school, work, finances and well-being due to CNO. Recognition of these challenges emphasizes the need to increase awareness of this disease and address the socioeconomic stressors and mental health issues in order to provide optimal care of children with CNO.

Acute and Chronic Suppurative Osteomyelitis of the Jaws: A 10-Year Review and Assessment of Treatment Outcome.

PURPOSE: To review cases of suppurative osteomyelitis of the jaws treated at the Massachusetts General Hospital (Boston, MA) over a 10-year period, evaluate specifics of disease presentation, and answer the clinical question: are there identifiable variables associated with treatment outcome? MATERIALS AND METHODS: A retrospective cohort study was completed using patients treated for suppurative osteomyelitis of the jaws at the Massachusetts General Hospital from April 2006 to October 2016. Inclusion criteria were a diagnosis of suppurative osteomyelitis of the jaw, age older than 18 years, and complete medical records. Patients with nonsuppurative disease, radiation history, or antiresorptive exposure were excluded. Candidate variables included demographic information, medical and dental history, presenting signs and symptoms, and radiologic and laboratory findings. The outcome variable was successful treatment, defined as resolution of symptoms and radiographic evidence of healing after initial treatment. Appropriate statistical analyses were performed with significance set a P value less than .05. RESULTS: Forty-two patients met the inclusion criteria. Mean age was 53 years (range, 20 to 80 yr) and 26 were women (62%). Common comorbidities included cardiovascular disease (52%), tobacco use (45%), and psychiatric disorders (45%). Pain (90%), swelling (86%), and neurosensory change (50%) were the most common findings. Common microbacterial isolates included Streptococcus milleri (74%) and coagulase-negative Staphylococcus species (43%), which showed marked antibiotic resistance. Surgical debridement was the most common intervention (93%). Successful treatment was found in 86%. Of 6 persistent cases, 4 resolved with a second debridement and continued antibiotics and 2 required resection. Increased white blood cell (WBC) count at presentation (P = .005) and associated psychiatric diagnoses (P = .037) were statistically associated with unsuccessful initial treatment. CONCLUSION: The results of this study indicate that antibiotic resistance is commonly encountered in this patient population, although it was not associated with unsuccessful outcome. Patients presenting with increased WBC count and concurrent psychiatric comorbidities required protracted treatment.

The Hyperbaric Oxygen Therapy Registry: Driving quality and demonstrating compliance.

OBJECTIVE: To provide an update on the status of provider participation in the US Wound Registry (USWR) and its specialty registry the Hyperbaric Oxygen Therapy Registry (HBOTR), which provide much-needed national benchmarking and quality measurement services for hyperbaric medicine. METHODS: Providers can meet many requirements of the Merit-Based Incentive Payment System (MIPS) and simultaneously participate in the HBOTR by transmitting Continuity of Care Documents (CCDs) directly from their certified electronic health record (EHR) or by reporting hyperbaric quality measures, the specifications for which are available free of charge for download from the registry website as electronic clinical quality measures for installation into any certified EHR. Computerized systems parse the structured data transmitted to the USWR. Patients undergoing hyperbaric oxygen (HBO2) therapy are allocated to the HBOTR and stored in that specialty registry database. The data can be queried for benchmarking, quality reporting, public policy, or specialized data projects. RESULTS: Since January 2012, 917,758 clinic visits have captured the data of 199,158 patients in the USWR, 3,697 of whom underwent HBO2 therapy. Among 27,404 patients with 62,843 diabetic foot ulcers (DFUs) captured, 9,908 DFUs (15.7%) were treated with HBO2 therapy. Between January 2016 and September 2018, the benchmark rate for the 1,000 DFUs treated with HBO2 was 7.3%, with an average of 28 treatments per patient. There are 2,100 providers who report data to the USWR by transmitting CCDs from their EHR and 688 who submit quality measure data, 300 (43.6%) of whom transmit HBO2 quality data.

Comparison of Methicillin-resistant Versus Susceptible Staphylococcus aureus Pediatric Osteomyelitis.

BACKGROUND: The incidence of methicillin-resistant Staphylococcus aureus (MRSA) pediatric osteomyelitis has risen and been associated with a more severe clinical course than methicillin-susceptible Staphylococcus aureus (MSSA) infections. National databases have been underutilized to describe these trends. We compared demographics, clinical course, and outcomes for patients with MRSA versus MSSA osteomyelitis. METHODS: We queried the 2009 and 2012 Healthcare Cost and Utilization Project Kids Inpatient Database for discharge records with diagnosis codes for osteomyelitis and S. aureus. We explored demographics predicting MRSA and evaluated MRSA versus MSSA as predictors of clinical outcomes including surgery, sepsis, thrombophlebitis, length of stay, and total charges. RESULTS: A total of 4214 discharge records were included. Of those, 2602 (61.7%) had MSSA and 1612 (38.3%) had MRSA infections. Patients at Southern and Midwestern hospitals were more likely to have MRSA than those at Northeastern hospitals. Medicaid patients' odds of MRSA were higher than those with private insurance, and black patients were more likely to have MRSA compared with white patients. MRSA patients were more likely to undergo multiple surgeries compared with MSSA patients and were more likely to have complications including severe sepsis, thrombophlebitis, and pulmonary embolism. Patients with MRSA had longer lengths of stay than those with MSSA and higher total charges after controlling for length of stay. CONCLUSION: Review of a national database demonstrates MRSA is more prevalent in the South and Midwest regions and among black patients. MRSA patients have more surgeries, complications, and longer lengths of stay. LEVEL OF EVIDENCE: Level III.

Epidemiology and Management of Acute, Uncomplicated Septic Arthritis and Osteomyelitis: Spanish Multicenter Study.

BACKGROUND: Acute osteoarticular infection (OAI) is a potentially severe disease. The aim of this study was to evaluate the etiology, clinical characteristics and therapeutic approach of OAI in children in Spain. METHODS: Medical records from children <14 years with OAI from 25 hospitals between 2008 and 2012 were reviewed. Confirmed osteomyelitis (OM) and septic arthritis (SA) required a positive bacterial isolate; otherwise, they were considered probable. Probable SA with <40,000 cells/mm in joint fluid was not included. RESULTS: A total of 641 children were evaluated. Two hundred and ninety-nine cases (46%) were OM, 232 (36%) SA, 77 (12%) osteoarthritis and 33 (5%) spondylodiscitis. Children with OM were older (63 vs. 43 months for SA; P < 0.001). Magnetic resonance imaging and bone scintigraphy had the highest yield for OM diagnosis (94%). Arthrocentesis was performed in 96% of SA. A microorganism was isolated in 246 patients (38%: 33% OM vs. 55% SA; P < 0.001): Staphylococcus aureus was the most common (63%), followed by Kingella kingae (15%) and Streptococcus pyogenes (9%). Ninety-five percent of children initially received IV antibiotics, mostly cefotaxime + cloxacillin (60%) or cloxacillin (40%). Total treatment duration was 38 (±31) days for OM and 28 (±16) days for SA (P < 0.0001). Twenty percent of children with OM (46% because of complications) and 53% with SA (95% initial arthrotomy) underwent surgery. Patients with SA were compared according to initial arthrotomy (n = 123) versus arthrocentesis (n = 109), and no clinical differences were observed, except for higher rate of hip SA in the former (50% vs. 9%; P < 0.001). Children with arthrocentesis had less sequelae [6.6% vs. 1%; P = 0.03, odds ratio = 0.58 (95% confidence interval: 0.45-0.76)], but not in the multivariate analysis. CONCLUSIONS: This is the largest pediatric cohort of OAI in Spain. S. aureus was the most common isolate, although K. kingae was recovered in a high proportion of cases. Conservative management was applied in half of the patients. There was a low rate of sequelae, even with nonsurgical approaches.

Multidisciplinary assessment of congenital insensitivity to pain syndrome.

BACKGROUND: Congenital insensitivity to pain and anhidrosis (CIPA) is a rare clinical condition characterized by the absence of normal subjective and objective responses to noxious stimuli in patients with intact central and peripheral nervous systems. CASE PRESENTATIONS: Two patients with CIPA are reported. The first patient was a 13-year-old girl who presented to our hospital with multiple joint destructions secondary to osteomyelitis. The second patient was a 10-year-old boy who presented with multiple hand lesions and right leg osteomyelitis. Our patients were treated with multiple debridements and intravenous antibiotics according to our hospital protocol. CONCLUSION: Early recognition of the disease is important. The treatment for this condition is focused more on the prevention of bone injuries and joint infection, as opposed to a cure. There are no standard techniques or guidelines available to treat this rare disease. Overall, effective CIPA treatment is built around family education and patient training.