Osteoarticular Infections: Younger Children With Septic Arthritis and Low Inflammatory Patterns Have a Better Prognosis in a European Cohort.

BACKGROUND: Osteoarticular infections (OAI) are associated with complications and sequelae in children, whose prediction are of great importance in improving outcomes. We aimed to design risk prediction models to identify early complications and sequelae in children with OAI. METHODS: This observational study included children (>3 months-17 years old) with acute OAI admitted to a tertiary-care pediatric hospital between 2008 and 2018. Clinical treatment, complications and sequelae were recorded. We developed a multivariable logistic predictive model for an acute complicated course (ACC) and another for sequelae. RESULTS: A total of 240 children were identified, 17.5% with ACC and 6.0% and 3.6% with sequelae at 6 and 12 months of follow-up, respectively. In the multivariable logistic predictive model for ACC, predictors were fever at admission [adjusted odds ratio (aOR): 2.98; 95% confidence interval (CI): 1.10-8.12], C-reactive protein ≥100 mg/L (aOR: 2.37; 95% CI: 1.05-5.35), osteomyelitis (aOR: 4.39; 95% CI: 2.04-9.46) and Staphylococcus aureus infection (aOR: 3.50; 95% CI: 1.39-8.77), with an area under the ROC curve of 0.831 (95% CI: 0.767-0.895). For sequelae at 6 months, predictors were age ≥4 years (aOR: 4.08; 95% CI: 1.00-16.53), C-reactive protein ≥110 mg/L (aOR: 4.59; 95% CI: 1.25-16.90), disseminated disease (aOR: 9.21; 95% CI: 1.82-46.73) and bone abscess (OR: 5.46; 95% CI: 1.23-24.21), with an area under the ROC curve of 0.887 (95% CI: 0.815-0.959). CONCLUSIONS: In our model we could identify patients at low risk for complications and sequelae, probably requiring a less aggressive approach.

Deep Vein Thrombosis after a Supracondylar Fracture of the Humerus in a Child - A Rare Complication / Trombose venosa profunda após uma fratura supracondilar do úmero em uma criança - Uma complicação rara

Abstract Deep vein thrombosis in the upper extremities is uncommon, especially in the pediatric population and in the trauma setting. The diagnosis is challenging, due to its rarity, requiring a high degree of suspicion. We describe a rare case of humeral vein thrombosis after a displaced supracondylar fracture of the humerus in a 7-year-old girl. The risk factors for thromboembolism and sequelae are also discussed. The early detection and treatment are mandatory to prevent poor outcomes, such as fatal thromboembolism.

Resumo Trombose venosa profunda nas extremidades superiores é incomum, especialmente na população pediátrica e no ambiente do trauma. O diagnóstico é desafiador, devido a sua raridade, exigindo alto grau de suspeita. Descrevemos um caso raro de trombose venosa úmera após uma fratura supracondilar deslocada do úmero em uma menina de 7 anos. Os fatores de risco para tromboembolismo e sequelas também são discutidos. A detecção e o tratamento precoces são obrigatórios para evitar desfechos ruins, como tromboembolismo fatal.

Case Report of an Osteochondral Lesion of the First Metatarsal Head in an Adolescent Patient Treated by Autograft Mosaicplasty: Surgical Technique and Decision-Making Rationale.

Introduction: First metatarsophalangeal (MTP) joint mobility is critical to a normal gait pattern; therefore, osteochondral defects (OCDs) of the first metatarsal head should be treated promptly to avoid functional limitation and progression to hallux rigidus. <15 cases of OCDs of the first MTP joint in the pediatric population, predominantly adolescents, have been published so far. The purpose of this article is to present a rare case of an adolescent first MTP joint OCD treated by the technique of autograft osteochondral mosaicplasty, which has been commonly used for OCDs of the knee and talus but scarcely described for the first MTP joint. Case Report: The case of a 13-year-old male futsal player with a post-traumatic 50 mm2 OCD of the first metatarsal head is presented. The Osteochondral Autograft Transfer System® (Arthrex Inc™, Naples, FL) was used to harvest a 10-mm diameter donor plug from the medial ipsilateral femoral condyle, then delivered to the first metatarsal head. At the 6-month follow-up, physical activity had been resumed with no pain or significant mobility limitation and an improvement of the American Orthopedic Foot and Ankle Society hallux score of 54-95 points. At the same time, a follow-up magnetic resonance showed complete incorporation of the osteochondral graft without bone edema or subchondral osteonecrosis. Conclusion: Treatment for osteochondral lesions, regardless of their location, aims to restore function by recreating the articular congruity reducing the potential for a progressive degenerative process. Multiple surgical treatment options exist for OCDs of the first metatarsal head. The treatment decision must take into consideration both patient factors and lesion factors. Based on this, a literature revision and treatment decision rationale are presented. This case demonstrated that an osteochondral transplant could be a reasonable treatment option for a traumatic, full-thickness OCD of the first metatarsal head in adolescent patients.

Bilateral hip heterotopic ossification with sciatic nerve compression on a paediatric patient-An individualized surgical approach: A case report.

BACKGROUND: Neurogenic heterotopic ossification is an acquired serious complication described in patients with central nervous system disorders and defined by bone formation in non-osseous tissue. CASE SUMMARY: We present an unusual case of a 13-yr-old boy presenting with hip pain and severe gait impairment 5 mo after the diagnosis of hemiplegia following a spontaneous intracerebral haemorrhage. Computed tomography revealed bilateral heterotopic ossification of both the paretic and the non-paretic limbs, with entrapment of the sciatic nerve. The choice of surgical or nonsurgical management of such patients depends on the timing of diagnosis, the symptoms, and the extent of maturation of the ossified lesions. Surgical resection remains the only treatment with proven, evidence-based effectiveness. The choice of surgical approach largely depends on the location of the ossified lesions. CONCLUSION: We believe the plane of dissection presented is a satisfactory option for resection of a posteromedial mass and sciatic nerve release.

Osteoarticular infections in infants under 3 months of age.

BACKGROUND: Acute osteoarticular infections (OAI) in infants under 3 months of age (≤3M) are rare and remain a diagnostic challenge. Orthopedic complications and functional sequelae have been less well described in this age group. Our aims were to evaluate trends in aetiology, management, and outcomes of OAI ≤ 3M, and to compare these younger children who have OAI with older children. METHODS: A longitudinal observational study was conducted of OAI cases admitted to tertiary care pediatric hospital from 2008 to 2018. OAI ≤ 3M was compared with children above 3 months. Clinical, microbiological, imaging, and outcome data were analyzed. RESULTS: We identified 24 (9.1%) of the 263 OAI in children under 3 months. Analyzing OAI ≤ 3M there was a twofold increase since 2014; 54% were males with a median age of 28 days (IQR: 13.5-60.0), 10 (41.7%) were premature and nine (37.5%) had healthcare-associated infections. Microbiological causes were identified in 87.5%, mostly Staphylococcus aureus (57.1%) and Group B Streptococcus (23.8%), and 25% were multidrug-resistant (5 methicillin-resistant S. aureus and 1 Enterobacter cloacae). Bacteremia (100% vs 36.8%, P = 0.037), multidrug resistant bacteria (75% vs 16, P = 0.04), and healthcare-associated infections (100% vs 26.3%, P = 0.014) were associated with sequelae. Comparing OAI ≤ 3M with older children, OAI ≤ 3M were treated with longer antibiotic courses, had more complications and sequelae (17.4% vs 3.2%, P = 0.002). CONCLUSIONS: S. aureus is still the most common cause of OAI ≤ 3M, and 25% of causative bacteria were multidrug-resistant bacteria. Complications and sequelae were more frequent in OAI ≤ 3M when compared with older children.

Distinguishing Kingella kingae from Pyogenic Acute Septic Arthritis in Young Portuguese Children.

(1) Background: We aim to identify clinical and laboratorial parameters to distinguish Kingella kingae from pyogenic septic arthritis (SA). (2) Methods: A longitudinal, observational, single-centre study of children < 5 years old with microbiological positive SA admitted to a paediatric hospital from 2013−2020 was performed. Clinical and laboratorial data at admission and at 48 h, as well as on treatment and evolution, were obtained. (3) Results: We found a total of 75 children, 44 with K. kingae and 31 with pyogenic infections (mostly MSSA, S. pneumoniae and S. pyogenes). K. kingae affected younger children with low or absent fever, low inflammatory markers and a favourable prognosis. In the univariate analyses, fever, septic look, CRP and ESR at admission and CRP at 48 h were significantly lower in K. kingae SA. In the multivariate analyses, age > 6 months ≤ 2 years, apyrexy and CRP ≤ 100 mg/L were significative, with an overall predictive positive value of 86.5%, and 88.4% for K. kingae. For this model, ROC curves were capable of differentiating (AUC 0.861, 95% CI 0.767−0.955) K. kingae SA from typical pathogens. (4) Conclusions: Age > 6 months ≤ 2 years, apyrexy and PCR ≤ 100 mg/L were the main predictive factors to distinguish K. kingae from pyogenic SA < 5 years. These data need to be validated in a larger study.

Acute haematogenous osteomyelitis in Lisbon: an unexpectedly high association with myositis and arthritis.

INTRODUCTION: Despite the current trend toward less aggressive therapeutic approaches, acute haematogenous osteomyelitis (AHO) continues to be a challenge and is associated with significant morbidity worldwide. Our aim was to assess whether compliance with the current protocol was achieved in 80% of cases, to identify complications and the associated risk factors, and to analyse trends in the aetiology and management of AHO in the paediatric population. METHODS: We conducted a longitudinal, observational, single-centre study in patients with AHO aged less than 18 years admitted to a paediatric hospital between 2008 and 2018 divided in 2 cohorts (before and after 2014). We analysed data concerning demographic and clinical characteristics and outcomes. RESULTS: The study included 71 children with AHO, 56% male, with a median age of 3 years (interquartile range, 1-11). We found a 1.8-fold increase of cases in the last 5 years. The causative agent was identified in 37% of cases: MSSA (54%), MRSA (4%), S. pyogenes (19%), K. kingae (12%), S. pneumoniae (8%), and N. meningitidis (4%). Complications were identified in 45% of patients and sequelae in 3.6%. In recent years, there was an increase in myositis (30% vs 7%; P=.02), septic arthritis (68 vs 37.2%; p=0.012) and in the proportion of patients treated for less than 4 weeks (37 vs 3.5%; p=0.012), with a similar sequelae rates. The risk factors associated with complications were age 3 or more years, C-reactive protein levels of 20mg/L or higher, time elapsed between onset and admission of 5 or more days and positive culture, although the only factor that continued to be significantly associated in the multivariate analysis was positive culture. The presence of complications was a risk factor for sequelae at 6 months. CONCLUSIONS: Our study confirms that AHO can be aggressive. The identification of risk factors for complications is essential for management.

Osteomielitis hematógena aguda en Lisboa: asociación con miositis y artritis inesperadamente alta / Acute haematogenous osteomyelitis in Lisbon: An unexpectedly high association with myositis and arthritis

Introducción: Aunque actualmente se tiende a un abordaje terapéutico menos agresivo, la osteomielitis hematógena aguda (OHA) sigue suponiendo un reto, con una morbilidad significativa a nivel mundial. El objetivo del estudio fue evaluar si se alcanzó una adherencia del 80% con el protocolo vigente, identificar las complicaciones y riesgos asociados y analizar las tendencias en la etiología y el manejo de la OHA en la población pediátrica. Métodos: Estudio observacional longitudinal unicéntrico en pacientes menores de 18 años con OHA ingresados en un hospital pediátrico entre 2008 y 2018 divididos en 2 cohortes (antes y después de 2014). Se analizaron datos demográficos, clínicos y concernientes a la evolución de la enfermedad. Resultados: El estudio incluyó a 71 niños con OHA, 56% varones, con una edad mediana de 3 años (rango intercuartílico, 1-11). Se observó una incidencia 1,8 veces mayor en los últimos 5 años. El agente causal se identificó en el 37% de los casos: SASM (54%), SARM (4%), Streptococcus pyogenes (19%), Kingella kingae (12%), Streptococcus pneumoniae (8%) y Neisseria meningitidis (4%). Se identificaron complicaciones en el 45% y secuelas en 3,6% de los pacientes. En los últimos años aumentó la incidencia de miositis (30% vs. 7%; p=0,02) y de artritis séptica (después de 2015, 68% vs. 37,2%, p=0,012), así como la proporción de pacientes con tratamiento inferior a 4 semanas (37% vs. 3,5%; p=0,012), con tasas de secuelas similares. Los factores de riesgo de complicaciones fueron la edad ≥3 años, nivel de PCR≥20mg/l, duración de los síntomas al ingreso de 5 o más días y cultivo positivo, aunque en el análisis multivariado solo se validó el cultivo positivo. La presencia de complicaciones se identificó como factor de riesgo de secuelas a los 6 meses. Conclusiones: El presente estudio confirma que la OHA puede ser agresiva. La identificación de los factores de riesgo es fundamental para su abordaje. (AU)

Introduction: Despite the current trend towards less aggressive therapeutic approaches, acute haematogenous osteomyelitis (AHO) continues to be a challenge and is associated with significant morbidity worldwide. Our aim was to determine if 80% compliance with current protocol was achieved, identify complications and associated risk factors and analyse trends in aetiology and management of AHO in children. Methods: We conducted a longitudinal, observational, single-centre study in patients with AHO aged less than 18 years admitted to a paediatric hospital, between 2008 and 2018, divided into 2 cohorts (before and after 2014). Demographic, clinical data and disease progression were analysed. Results: The study included 71 children with AHO, 56% male, with a median age of 3 years (interquartile range, 1–11). We found a 1.8-fold increase of cases in the last 5 years. The causative agent was identified in 37% of cases: MSSA (54%), MRSA (4%), Streptococcus pyogenes (19%), Kingella kingae (12%), Streptococcus pneumoniae (8%), and Neisseria meningitidis (4%). Complications were identified in 45% of patients and sequelae in 3.6%. In recent years, there was an increase in myositis (30% vs. 7%; p=0.02), septic arthritis (68% vs. 37.2%; p=0.012) and in the proportion of patients treated for less than 4 weeks (37% vs. 3.5%; p=0.012), with a similar sequelae rates. The risk factors for complications were age 3 or more years, CRP levels of 20mg/l or higher, time elapsed between onset and admission of 5 or more days and positive culture, although on multivariate analysis only positive culture was significant. The presence of complications was a risk factor for sequelae at 6 months. Conclusions: Our study confirms that AHO can be aggressive. The identification of risk factors for complications may be fundamental for management. (AU)

Pinckney fracture: do not underestimate trauma of the distal phalanx of the hallux.

Toe injuries are common in the emergency department and most of them are treated conservatively. In some circumstances, these injuries can present as a physeal fracture with concomitant soft-tissue injury affecting the nail bed and resulting in a hidden open fracture. To adequately treat these patients, a high index of suspicion is needed to diagnose and treat the open fractures and to prevent complications such as infection, osteomyelitis, malunion and premature physeal arrest.We report a case of a patient that was admitted to the hospital with a Salter-Harris type I fracture of the distal phalanx of the hallux. After confirming the diagnosis, antibiotic treatment was started and the fracture was reduced and fixed.The literature on this entity is sparse and most of the management protocols are based on its hand equivalent-the Seymour fracture, emphasising the low threshold for treating these lesions as an open fracture.

[Acute haematogenous osteomyelitis in Lisbon: An unexpectedly high association with myositis and arthritis]. / Osteomielitis hematógena aguda en Lisboa: asociación con miositis y artritis inesperadamente alta.

INTRODUCTION: Despite the current trend towards less aggressive therapeutic approaches, acute haematogenous osteomyelitis (AHO) continues to be a challenge and is associated with significant morbidity worldwide. Our aim was to determine if 80% compliance with current protocol was achieved, identify complications and associated risk factors and analyse trends in aetiology and management of AHO in children. METHODS: We conducted a longitudinal, observational, single-centre study in patients with AHO aged less than 18 years admitted to a paediatric hospital, between 2008 and 2018, divided into 2 cohorts (before and after 2014). Demographic, clinical data and disease progression were analysed. RESULTS: The study included 71 children with AHO, 56% male, with a median age of 3 years (interquartile range, 1-11). We found a 1.8-fold increase of cases in the last 5 years. The causative agent was identified in 37% of cases: MSSA (54%), MRSA (4%), Streptococcus pyogenes (19%), Kingella kingae (12%), Streptococcus pneumoniae (8%), and Neisseria meningitidis (4%). Complications were identified in 45% of patients and sequelae in 3.6%. In recent years, there was an increase in myositis (30% vs. 7%; p=0.02), septic arthritis (68% vs. 37.2%; p=0.012) and in the proportion of patients treated for less than 4 weeks (37% vs. 3.5%; p=0.012), with a similar sequelae rates. The risk factors for complications were age 3 or more years, CRP levels of 20mg/l or higher, time elapsed between onset and admission of 5 or more days and positive culture, although on multivariate analysis only positive culture was significant. The presence of complications was a risk factor for sequelae at 6 months. CONCLUSIONS: Our study confirms that AHO can be aggressive. The identification of risk factors for complications may be fundamental for management.

Kingella kingae Displaced S. aureus as the Most Common Cause of Acute Septic Arthritis in Children of All Ages.

BACKGROUND: Acute septic arthritis (SA) still remains a challenge with significant worldwide morbidity. In recent years, Kingella kingae has emerged and treatment regimens have become shorter. We aim to analyze trends in SA etiology and management and to identify risk factors for complications. METHODS: Longitudinal observational, single center study of children (<18 years old) with SA admitted to a tertiary care pediatric hospital, from 2003 to 2018, in 2 cohorts, before and after implementation of nucleic acid amplification assays (2014). Clinical, treatment and disease progression data were obtained. RESULTS: A total of 247 children were identified, with an average annual incidence of 24.9/100,000, 57.9% males with a median age of 2 (1-6) years. In the last 5 years, a 1.7-fold increase in the annual incidence, a lower median age at diagnosis and an improved microbiologic yield (49%) was noticed. K. kingae became the most frequent bacteria (51.9%) followed by MSSA (19.2%) and S. pyogenes (9.6%). Children were more often treated for fewer intravenous days (10.7 vs. 13.2 days, P = 0.01) but had more complications (20.6% vs. 11.4%, P = 0.049) with a similar sequelae rate (3.7%). Risk factors for complications were C-reactive protein ≥80 mg/L and Staphylococcus aureus infection, and for sequelae at 6 months, age ≥4 years and CRP ≥ 80 mg/L. CONCLUSIONS: The present study confirms that K. kingae was the most common causative organism of acute SA. There was a trend, although small, for decreasing antibiotic duration. Older children with high inflammatory parameters might be at higher risk of sequelae.

Sacroileítis piógena: Lecciones aprendidas de una serie de casos atípicos / Pyogenic sacroiliitis: Lessons learned from an atypical case series

Introducción: La sacroileítis piógena (SIP) es una entidad infrecuente que representa del 1 al 2% del total de las infecciones articulares en la edad pediátrica. Su diagnóstico a menudo se complica y retrasa debido a la inespecificidad de sus síntomas, signos y exploración física. Además, la identificación microbiológica puede resultar difícil debido a la alta proporción de hemocultivos negativos y los riesgos implicados en la aspiración de líquido articular en esta localización. Pacientes y métodos: Revisión retrospectiva de las historias clínicas de todos los pacientes menores de 18 años ingresados en un hospital infantil terciario con SIP en el período 2008-2016. Resultados: Se identificaron 6 casos de SIP en niños. Los hemocultivos fueron negativos, y la identificación del agente etiológico requirió aspiración de líquido sinovial en un paciente con infección por Aggregatibacter aphrophilus y pruebas específicas para la detección de agentes menos frecuentes en los pacientes restantes: Kingella kingae (n = 2), Brucella melitensis (n = 1) y Bartonella henselae (n = 1). Los pacientes recibieron regímenes de antibioterapia específica, y todos presentaron una evolución favorable y libre de secuelas durante el seguimiento. Conclusiones: A pesar del reducido tamaño muestral, nuestro estudio puso de relieve la baja efectividad del hemocultivo en el diagnóstico de la SIP pediátrica. También evidenció la necesidad de mantener un elevado índice de sospecha de los agentes atípicos y de emplear precozmente métodos diagnósticos apropiados, como las pruebas de imagen y la reacción en cadena de la polimerasa (PCR) en muestras de sangre, así como la prescripción de antibioterapia efectiva

Introduction: Pyogenic sacroiliitis (PSI) is a rare condition that amounts to 1% to 2% of all joint infections in the paediatric age group. Its diagnosis is often difficult and delayed due to its nonspecific signs, symptoms and physical findings. Also, the identification of the causative microorganism is frequently challenging due to a high proportion of negative blood cultures and the risks involved in joint aspiration in this site. Patients and methods: We performed a retrospective review of the health records of all patients aged less than 18 years admitted to a tertiary children's hospital due to PSI between 2008 and 2016. Results: We identified 6 cases of paediatric PSI. The blood cultures were negative, and the identification of the causative agent required joint fluid aspiration in one patient with infection by Aggregatibacter aphrophilus, and specific screening tests for less frequent agents in the other patients: Kingella kingae (n = 2), Brucella melitensis (n = 1) and Bartonella henselae (n = 1). The patients were treated with specific antimicrobial regimens, and all had favourable clinical outcomes and were free from sequelae during the follow-up. Conclusions: Despite the small sample size, our study evinced the low effectiveness of blood cultures for diagnosis of paediatric PSI. It also highlights the need for a high level of suspicion for atypical agents and the early use of adequate diagnostic methods, including imaging and serological testing or polymerase chain-reaction (PCR) analysis of blood samples, as well as prescription of effective antimicrobial therapy

[Pyogenic sacroiliitis: Lessons learned from an atypical case series]. / Sacroileítis piógena: Lecciones aprendidas de una serie de casos atípicos.

INTRODUCTION: Pyogenic sacroiliitis (PSI) is a rare condition that amounts to 1% to 2% of all joint infections in the paediatric age group. Its diagnosis is often difficult and delayed due to its nonspecific signs, symptoms and physical findings. Also, the identification of the causative microorganism is frequently challenging due to a high proportion of negative blood cultures and the risks involved in joint aspiration in this site. PATIENTS AND METHODS: We performed a retrospective review of the health records of all patients aged less than 18 years admitted to a tertiary children's hospital due to PSI between 2008 and 2016. RESULTS: We identified 6 cases of paediatric PSI. The blood cultures were negative, and the identification of the causative agent required joint fluid aspiration in one patient with infection by Aggregatibacter aphrophilus, and specific screening tests for less frequent agents in the other patients: Kingella kingae (n=2), Brucella melitensis (n=1) and Bartonella henselae (n=1). The patients were treated with specific antimicrobial regimens, and all had favourable clinical outcomes and were free from sequelae during the follow-up. CONCLUSIONS: Despite the small sample size, our study evinced the low effectiveness of blood cultures for diagnosis of paediatric PSI. It also highlights the need for a high level of suspicion for atypical agents and the early use of adequate diagnostic methods, including imaging and serological testing or polymerase chain-reaction (PCR) analysis of blood samples, as well as prescription of effective antimicrobial therapy.

Peri-prosthetic bone cysts after total ankle replacement. A systematic review and meta-analysis.

BACKGROUND: Periprosthetic cystic osteolysis is a well-known complication of total ankle replacement. Several theories have been proposed for its aetiology, based on individual biomechanical, radiological, histopathology and outcome studies. METHODS: Studies that met predefined inclusion/exclusion criteria were analysed to identify literature describing the presence of peri-prosthetic ankle cystic osteolysis. Quantitative data from the selected articles were combined and statistically tested in order to analyse possible relations between ankle peri-prosthetic bone cysts and specific implant characteristics. RESULTS: Twenty-one articles were elected, totalizing 2430 total ankle replacements, where 430 developed peri-prosthetic cystic osteolysis. A statistically significant association (P<.001) was found between the presence of bone cysts and non-anatomic implant configuration, hydroxyapatite-coating, mobile-bearing and non tibial-stemmed implants. No significant association existed between the type of constraining and the presence of cysts (P>.05). CONCLUSIONS: Non-anatomic, mobile-bearing, hydroxyapatite-coated and non tibial-stemmed total ankle replacements are positively associated with more periprosthetic bone cysts.

Carpal synovitis with capitate bone tuberculosis in a child.

We present a 10-year-old boy with 2-month duration non-traumatic wrist pain and inflammatory signs. Due to elevated inflammatory markers on blood tests, with an increase in radiocarpal and intercarpal joints synovial fluid and no bony lesions, the patient was submitted to wrist arthrocentesis for the suspicion of septic arthritis. The patient did not improve on conventional treatment, however. An MRI showed synovitis around the carpus and a lytic lesion of the capitate bone due to osteomyelitis. A biopsy was able to identify the causative agent as Mycobacterium tuberculosis, and the patient was treated with antibiotics. He improved significantly, with no pain and signs of normal capitate bone remodelling on the last radiograph.

Atypical presentation and management of fibrodysplasia ossificans progressiva.

We report a case of an 18-year-old woman, with bilateral acute inflammatory pain on the hip area, during the premenstrual period, and progressive increase in volume and rigidity of both hips. Bilateral exuberant soft tissue calcifications were present on the radiographic exams, and the patient also presented with bilateral short-length hallux valgus. A heterozygous mutation in the protein kinase domain of ACVR1 gene was found, allowing the diagnosis of fibrodysplasia ossificans progressive. Due to the relation between the disease flares and the premenstrual period, the patient was put into a chemically induced amenorrhea, with no new inflammatory crises since.This case illustrates the importance of an accurate diagnosis to prevent unnecessary diagnostic procedures, as well as the need to develop specific treatment strategies to address each patient's particular needs.