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1.
BMJ Open Qual ; 12(Suppl 2)2023 09.
Artículo en Inglés | MEDLINE | ID: mdl-37783522

RESUMEN

BACKGROUND: Neck of femur fractures are common with associated high morbidity and mortality rates. National standards include provision of orthogeriatric care to any patient with a hip fracture. This study assessed the outcomes at 5 years following implementation of a collaborative orthogeriatric service at Southland Hospital in 2012. METHODS: Retrospective data were collected for patients aged 65 years and older admitted with a fragility hip fracture. Data were collated for 2011 (preimplementation) and 2017 (postimplementation). Demographic data and American Society of Anesthesiologists (ASA) scores were recorded to ensure comparability of the patient groups. Length of stay, postoperative complications and 30-day and 1-year mortality were assessed. RESULTS: 74 admissions with mean age at surgery of 84.2 years in 2011 and 107 admissions with mean age of 82.6 years in 2017. There was a higher proportion of ASA 2 and ASA 3 patients in 2017 compared with 2011 (p=0.036). The median length of stay in the orthopaedic ward was unchanged in the two cohorts but there was a shorter median length of stay by 6.5 days and mean length of stay by 11 days in 2017 in the rehabilitation ward (p<0.001 for both median and mean). Through logistic regression controlling for age, sex and ASA score, there was a reduction in the odds of having a complication by 12% (p<0.001). The study was too small to undertake statistical testing to calculate significant difference in overall 30-day and 1-year mortality between the groups. CONCLUSION: The orthogeriatric service has reduced the frequency of complications and length of stay on the rehabilitation ward 5 years following implementation.


Asunto(s)
Fracturas de Cadera , Ortopedia , Humanos , Anciano , Anciano de 80 o más Años , Estudios de Seguimiento , Estudios Retrospectivos , Nueva Zelanda/epidemiología , Tiempo de Internación , Fracturas de Cadera/cirugía
2.
Arch Osteoporos ; 17(1): 108, 2022 08 02.
Artículo en Inglés | MEDLINE | ID: mdl-35917039

RESUMEN

This narrative review describes efforts to improve the care and prevention of fragility fractures in New Zealand from 2012 to 2022. This includes development of clinical standards and registries to benchmark provision of care, and public awareness campaigns to promote a life-course approach to bone health. PURPOSE: This review describes the development and implementation of a systematic approach to care and prevention for New Zealanders with fragility fractures, and those at high risk of first fracture. Progression of existing initiatives and introduction of new initiatives are proposed for the period 2022 to 2030. METHODS: In 2012, Osteoporosis New Zealand developed and published a strategy with objectives relating to people who sustain hip and other fragility fractures, those at high risk of first fragility fracture or falls and all older people. The strategy also advocated formation of a national fragility fracture alliance to expedite change. RESULTS: In 2017, a previously informal national alliance was formalised under the Live Stronger for Longer programme, which includes stakeholder organisations from relevant sectors, including government, healthcare professionals, charities and the health system. Outputs of this alliance include development of Australian and New Zealand clinical guidelines, clinical standards and quality indicators and a bi-national registry that underpins efforts to improve hip fracture care. All 22 hospitals in New Zealand that operate on hip fracture patients currently submit data to the registry. An analogous approach is ongoing to improve secondary fracture prevention for people who sustain fragility fractures at other sites through nationwide access to Fracture Liaison Services. CONCLUSION: Widespread participation in national registries is enabling benchmarking against clinical standards as a means to improve the care of hip and other fragility fractures in New Zealand. An ongoing quality improvement programme is focused on eliminating unwarranted variation in delivery of secondary fracture prevention.


Asunto(s)
Fracturas de Cadera , Osteoporosis , Fracturas Osteoporóticas , Anciano , Australia , Fracturas de Cadera/prevención & control , Humanos , Nueva Zelanda/epidemiología , Osteoporosis/complicaciones , Fracturas Osteoporóticas/epidemiología , Fracturas Osteoporóticas/prevención & control , Prevención Secundaria
3.
BMC Musculoskelet Disord ; 23(1): 560, 2022 Jun 10.
Artículo en Inglés | MEDLINE | ID: mdl-35689284

RESUMEN

BACKGROUND: Elastic knee sleeves are often worn following anterior cruciate ligament reconstruction (ACLR) but their effects on movement patterns are unclear. AIM: To determine the immediate and six-week effects of wearing a knee sleeve on biomechanics of the knee during a step-down hop task. METHODS: Using a cross-over design, we estimated sagittal plane knee kinematics and kinetics and stance duration during a step-down hop for 31 participants (age 26.0 [SD 6.6] years, 15 women) after ACLR (median 16 months post-surgery) with and without wearing a knee sleeve. In a subsequent randomised clinical trial, participants in the 'Sleeve Group' (n = 9) then wore the sleeve for 6 weeks at least 1 h daily, while a 'Control Group' (n = 9) did not wear the sleeve. We used statistical parametric mapping to compare (1) knee flexion/extension angle and external flexion/extension moment trajectories between three conditions at baseline (uninjured side, unsleeved injured side and sleeved injured side); (2) within-participant changes for knee flexion angles and external flexion/extension moment trajectories from baseline to follow-up between groups. We compared discrete flexion angles and moments, and stance duration between conditions and between groups. RESULTS: Without sleeves, knee flexion was lower for the injured than the uninjured sides during mid-stance phase. When wearing the sleeve on the injured side, knee flexion increased during the loading phase of the stance phase. Discrete initial and peak knee flexion angles increased by (mean difference, 95% CIs) 2.7° (1.3, 4.1) and 3.0° (1.2, 4.9), respectively, when wearing the knee sleeve. Knee external flexion moments for the unsleeved injured sides were lower than the uninjured sides for 80% of stance phase, with no change when sleeved. The groups differenced for within-group changes in knee flexion trajectories at follow-up. Knee flexion angles increased for the Control group only. Stance duration decreased by 22% for the Sleeve group from baseline to follow-up (-89 ms; -153, -24) but not for the Controls. CONCLUSIONS: Application of knee sleeves following ACLR is associated with improved knee flexion angles during hop landing training. Longer term (daily) knee sleeve application may help improve hop stance duration, potentially indicating improved hop performance. TRIAL REGISTRATION: The trial was prospectively registered with the Australia New Zealand Clinical Trials Registry No: ACTRN12618001083280, 28/06/2018. ANZCTR.


Asunto(s)
Lesiones del Ligamento Cruzado Anterior , Reconstrucción del Ligamento Cruzado Anterior , Adulto , Lesiones del Ligamento Cruzado Anterior/cirugía , Reconstrucción del Ligamento Cruzado Anterior/efectos adversos , Fenómenos Biomecánicos , Estudios Cruzados , Femenino , Humanos , Cinética , Articulación de la Rodilla/cirugía
4.
BMC Musculoskelet Disord ; 22(1): 655, 2021 Aug 04.
Artículo en Inglés | MEDLINE | ID: mdl-34348704

RESUMEN

BACKGROUND: Rehabilitation following anterior cruciate ligament (ACL) reconstructions is based mainly on comprehensive progressive exercise programmes using a multi-dimensional approach. Elastic knee sleeves may be useful adjuncts to rehabilitation. The aim of this study was to determine the immediate and 6-week effects of wearing a knee sleeve on person-reported outcomes and function in participants who had undergone an ACL reconstruction and who had residual self-reported functional limitations. METHODS: Individuals with ACL reconstruction in the previous 6 months to 5 years were recruited. Immediate effects of a commercially-available elastic knee sleeve on single-leg horizontal hop distance were explored using a cross-over design. Following this first session, participants were randomised into a Control Group and a Sleeve Group who wore the sleeve for 6 weeks, at least 1 h daily. Outcome measures for the randomised clinical trial (RCT) were the International Knee Documentation Classification Subjective Knee Form (IKDC-SKF) score, the single-leg horizontal hop distance, and isokinetic quadriceps and hamstring peak torque. Linear mixed models were used to determine random effects. Where both limbs were measured at multiple time points, a random measurement occasion effect nested within participant was used. RESULTS: Thirty-four individuals (16 women) with ACL reconstruction completed the cross-over trial. Hop distance for the injured side during the sleeve condition increased by 3.6 % (95 % CI 0.4-6.8 %, p = 0.025). There was no evidence of differential changes between groups for the IKDC-SKF (Sleeve Group n = 15; Control Group n = 16; p = 0.327), or relative improvement in the injured side compared to the uninjured side for the physical performance measures (Sleeve Group n = 12, Control Group n = 12; three-way interaction p = 0.533 [hop distance], 0.381 [quadriceps isokinetic peak torque], and 0.592 [hamstring isokinetic peak torque]). CONCLUSIONS: Single-leg hop distance of the ACL reconstructed side improved when wearing a knee sleeve. Wearing the knee sleeve over 6 weeks did not lead to enhanced improvements in self-reported knee function, hop distance and thigh muscle strength compared to the control group. TRIAL REGISTRATION: The trial was prospectively registered with the Australia New Zealand Clinical Trials Registry No: ACTRN12618001083280 , 28 June 2018.


Asunto(s)
Lesiones del Ligamento Cruzado Anterior , Reconstrucción del Ligamento Cruzado Anterior , Lesiones del Ligamento Cruzado Anterior/diagnóstico , Lesiones del Ligamento Cruzado Anterior/cirugía , Estudios Cruzados , Femenino , Humanos , Articulación de la Rodilla/cirugía , Laboratorios , Fuerza Muscular , Músculo Cuádriceps
5.
Bone Joint J ; 102-B(12): 1735-1742, 2020 Dec.
Artículo en Inglés | MEDLINE | ID: mdl-33249893

RESUMEN

AIMS: Acetabular fractures in older adults lead to a high risk of mortality and morbidity. However, only limited data have been published documenting functional outcomes in such patients. The aims of this study were to describe outcomes in patients aged 60 years and older with operatively managed acetabular fractures, and to establish predictors of conversion to total hip arthroplasty (THA). METHODS: We conducted a retrospective, registry-based study of 80 patients aged 60 years and older with acetabular fractures treated surgically at The Alfred and Royal Melbourne Hospital. We reviewed charts and radiological investigations and performed patient interviews/examinations and functional outcome scoring. Data were provided by the Victorian Orthopaedic Trauma Outcomes Registry (VOTOR). Survival analysis was used to describe conversion to THA in the group of patients who initially underwent open reduction and internal fixation (ORIF). Multivariate regression analyses were performed to identify factors associated with conversion to THA. RESULTS: Seven patients (8.8%) had died at a median follow-up of 18 months (interquartile range (IQR) 12 to 25), of whom four were in the acute THA group. Eight patients (10%) underwent acute THA. Of the patients who underwent ORIF, 17/72 (23.6%) required conversion to THA at a median of 10.5 months (IQR 4.0 to 32.0) . After controlling for other factors, transport-related cases had an 88% lower rate of conversion to THA (hazard ratio (HR) 0.12, 95% confidence interval (CI) 0.02 to 0.91). Mean standardized Physical Component Summary Score (PCS-12) of the 12-Item Short Form Health Survey (SF-12) was comparable with the general population (age-/sex-matched) by 12 to 24 months. Over half of patients working prior to injury (14/26) returned to work by six months and two-thirds of patients (19/27) by 12 months. CONCLUSION: Patients over 60 years of age managed operatively for displaced acetabular fractures had a relatively high mortality rate and a high conversion rate to THA in the ORIF group but, overall, patients who survived had mean PCS-12 scores that improved over two years and were comparable with controls. Cite this article: Bone Joint J 2020;102-B(12):1735-1742.


Asunto(s)
Acetábulo/cirugía , Artroplastia de Reemplazo de Cadera , Fijación Interna de Fracturas/métodos , Fracturas Óseas/cirugía , Reducción Abierta , Acetábulo/lesiones , Factores de Edad , Anciano , Anciano de 80 o más Años , Artroplastia de Reemplazo de Cadera/mortalidad , Femenino , Fijación Interna de Fracturas/mortalidad , Fracturas Óseas/mortalidad , Humanos , Masculino , Persona de Mediana Edad , Reducción Abierta/métodos , Reducción Abierta/mortalidad , Sistema de Registros , Estudios Retrospectivos , Análisis de Supervivencia , Resultado del Tratamiento
6.
J Bone Joint Surg Am ; 102(Suppl 2): 8-14, 2020 Nov 04.
Artículo en Inglés | MEDLINE | ID: mdl-32453109

RESUMEN

BACKGROUND: The New Zealand Maori and Pacific ethnicities have the highest burden of slipped capital femoral epiphysis (SCFE) worldwide. Therefore, New Zealand provides a rich and unique therapeutic and research environment for this devastating hip condition. METHODS: A systematic literature review was performed using MEDLINE, PubMed, and Google Scholar from January 1, 1900, to December 31, 2019, with the following keywords: New Zealand, SCFE, slipped capital femoral epiphysis, SUFE, and slipped upper femoral epiphysis. The references were also screened. RESULTS: Eight original scientific research articles that had been published in peer-reviewed journals were identified, as well as 4 published abstracts from conference proceedings; we summarized the key findings. The New Zealand Maori and Pacific ethnicities have the highest reported burden of SCFE, with 4.2 and 5.6 times the prevalence, respectively, when compared with New Zealand European (Caucasian). Maori children are younger at presentation (p = 0.002) and more frequently present with bilateral SCFE (p = 0.05), and there is a strong link with childhood obesity. A contralateral posterior sloping angle (PSA) of ≥14° may be used as an adjunct threshold for decision-making regarding prophylactic pinning. A PSA of ≥40° was found to correlate with a higher likelihood of unsatisfactory functional outcome following SCFE pinning in situ. When conversion to total hip arthroplasty is eventually required due to SCFE, early functional outcomes are similar to those of patients with primary osteoarthritis, and revision rates and mortality rates are not significantly different. CONCLUSIONS: Landmark literature from New Zealand has been published on SCFE management, and there is a promising future of quality, impactful research. LEVEL OF EVIDENCE: Therapeutic Level IV. See Instructions for Authors for a complete description of levels of evidence.


Asunto(s)
Epífisis Desprendida de Cabeza Femoral/cirugía , Niño , Humanos , Nueva Zelanda/epidemiología , Prevalencia , Epífisis Desprendida de Cabeza Femoral/diagnóstico por imagen , Epífisis Desprendida de Cabeza Femoral/epidemiología , Epífisis Desprendida de Cabeza Femoral/etiología
7.
Front Surg ; 7: 621125, 2020.
Artículo en Inglés | MEDLINE | ID: mdl-33585549

RESUMEN

Introduction: The application of an external fixator for unstable pelvic fractures is an important component of many resuscitation protocols. Moreover, certain pelvic fractures may be treated with an external fixator without requiring further internal fixation. We report our initial clinical results with an alternate pelvic external fixator site, the lateral posterior external fixator (LPEF), and describe the surgical technique. Methods and Materials: From 2010 to 2013, we identified 27 consecutive patients (mean age 44.6 years, range 18-80 years) treated by the same surgeon (MKR) with an LPEF in a level 1 trauma center. Retrospective data collection included mechanism of injury, surgical interventions, and complications. Results: The LPEF was used in 16 patients as acute pelvic stabilization and converted at a median of 2 days (interquartile range 1-3.5) to internal fixation, whereas in 10 patients, it was used as definitive treatment and removed at a median of 48 days (interquartile range 37-64). One patient died on day 14, secondary to his severe closed head injury. The only surgical complications were two wound infections (20%, 2/10 in the group of definitive LPEFs), which resolved without sequelae after the removal of the LPEF (at 36 and 50 days) and antibiotics, one case of loss of fixation leading to the removal of the LPEF at 71 days, and one patient who had hypergranulating external fixator sites and eventually healed without any cutaneous sequelae. All fractures consolidated in a good position. Discussion: The described techniques of pelvic external fixation include the anterosuperior (iliac wing), supra-acetabular (anteroinferior), and subcristal (anterior superior iliac spine) insertion sites. The reported infection rates in definitive pelvic fracture treatment range from 20 to 40%. Due to the localization of the insertion sites, the lateral femoral cutaneous nerve is potentially at risk with the last two techniques. On the other hand, the LPEF insertion site is quite safe, as it is anatomically far from any nerves and the inguinal region, and allows easy access for laparotomy. The results in this series suggest that the lateral posterior pelvic external fixator technique is an alternative to previous techniques with a low risk of complications.

8.
JBJS Case Connect ; 8(4): e97, 2018.
Artículo en Inglés | MEDLINE | ID: mdl-30540606

RESUMEN

CASE: Congenital vertical talus (CVT) is a rare rigid flatfoot disorder with a rocker-bottom flatfoot appearance. It is characterized by hindfoot valgus and equinus, with associated midfoot dorsiflexion and forefoot abduction. We describe a patient who was born with dysmorphic features and subsequently was diagnosed with Beals contractural arachnodactyly. After the diagnosis of bilateral CVT was made, it was treated with a single-stage open reduction. There was a unilateral recurrence, which was treated with revision surgery. The patient had an excellent functional outcome. CONCLUSION: CVT often requires surgical management and may recur. To our knowledge, this is the first reported case of CVT associated with Beals contractural arachnodactyly.


Asunto(s)
Aracnodactilia , Contractura , Pie Plano/cirugía , Preescolar , Pie Plano/congénito , Pie Plano/diagnóstico por imagen , Humanos , Lactante , Procedimientos Ortopédicos
9.
SICOT J ; 4: 3, 2018.
Artículo en Inglés | MEDLINE | ID: mdl-29469803

RESUMEN

Fracture of the femoral head (OTA 31-C1.3) following anterior obturator dislocations are a challenging problem as the fractures are often communited, impacted and with loose osteochondral fragments, making surgical fixation difficult. This can result in residual articular defects if the fragments cannot be internally fixed and need be excised, predisposing to secondary osteoarthritis. Treatment options for these defects are limited, have variable results and with limited literature to guide us on outcomes due to the rarity of these injuries. Here, we describe the first use of the technique of partial femoral head resurfacing in two patients with such fractures and report on their long term outcomes.

10.
J Orthop Trauma ; 32(5): 245-250, 2018 05.
Artículo en Inglés | MEDLINE | ID: mdl-29401087

RESUMEN

OBJECTIVES: To evaluate patient-independent risk factors for aseptic femoral hypertrophic nonunion requiring exchange nailing, with particular reference to the fit of the nail at the isthmus within the canal. DESIGN: Retrospective case control study. SETTING: Level 1 trauma center. MAIN OUTCOME MEASUREMENTS: Between 2008 and 2012, 211 patients without any patient-dependent risk factors for nonunion were treated with a locked reamed intramedullary nail for a femoral shaft fracture. Twenty-three cases went on to hypertrophic nonunion requiring exchange nailing (treatment group) and 188 cases went on to union (control group). Patient-independent risk factors for exchange nailing were documented. RESULTS: Patient-independent risk factors for exchange nailing were poor fracture reduction [Odds ratio (OR): 11.5, 95% confidence interval (CI), 4.0-33.4, P < 0.001], open fracture (OR: 7.6, 95% CI, 3.0-19.6, P = 0.004), Winquist classification of 4 (OR: 4.4, 95% CI, 1.9-6.7, P = 0.016), and poor nail fit (OR: 10.3, 95% CI, 5.1-28.4, P < 0.001). Multivariate analysis revealed nail fit as an independent predictor of femoral nonunion requiring exchange nailing (OR: 11.4, 95% CI, 6.9-15.2, P < 0.001). Moreover, we found a direct relationship between increasingly poor nail fit and increased risk of exchange nailing, with the criterion occurring at a nail fit ratio <70%. CONCLUSION: When proceeding to femoral fracture reamed intramedullary nailing, we recommend a minimum nail fit of 70% at the isthmus and ideally 90% or more, to avoid surgical reintervention. LEVEL OF EVIDENCE: Prognostic Level III. See Instructions for Authors for a complete description of levels of evidence.


Asunto(s)
Clavos Ortopédicos/efectos adversos , Fracturas del Fémur/cirugía , Fémur/cirugía , Fijación Intramedular de Fracturas/efectos adversos , Fijación Intramedular de Fracturas/instrumentación , Fracturas no Consolidadas/cirugía , Adolescente , Adulto , Femenino , Fijación Intramedular de Fracturas/métodos , Curación de Fractura , Fracturas no Consolidadas/patología , Humanos , Hipertrofia , Masculino , Persona de Mediana Edad , Reoperación , Estudios Retrospectivos , Factores de Riesgo , Resultado del Tratamiento , Adulto Joven
11.
JBJS Case Connect ; 8(4): e106, 2018.
Artículo en Inglés | MEDLINE | ID: mdl-30601276

RESUMEN

CASE: We report the case of a pediatric terrible triad injury of the elbow consisting of an ulnohumeral dislocation, a radial neck fracture, and a coronoid chondral injury, which was diagnosed at 14 days following injury. We describe our surgical technique and provide details of the postoperative course, including the development of a rotational contracture following heterotopic ossification. CONCLUSION: We describe a rare and challenging injury that should be recognized early and managed surgically to maintain acceptable elbow function.


Asunto(s)
Traumatismos del Brazo/cirugía , Lesiones de Codo , Fijación Interna de Fracturas/métodos , Traumatismos del Brazo/diagnóstico por imagen , Niño , Humanos , Masculino
12.
J Pediatr Orthop ; 34(6): e27-32, 2014 Sep.
Artículo en Inglés | MEDLINE | ID: mdl-24992349

RESUMEN

BACKGROUND: Tuberous sclerosis has been associated with lower extremity lymphoedema in rare cases, only in females. Our objective was to increase present knowledge about this condition by describing this association of lower limb lymphoedema and tuberous sclerosis, however, in a male patient. METHODS: We report the case of a 5-year-old boy who presented with left lower limb congenital lymphoedema and was diagnosed with tuberous sclerosis 9 days after birth based on adequate clinical criteria. This patient's lymphoedema was severe and caused compartment syndrome requiring fasciotomies at 2 days after birth, numerous surgical interventions for chronic wounds and eventually necessitating several toe amputations. A biopsy confirmed benign vascular lymphatic malformation and genetic analysis confirmed the TSC2 mutation. A thorough literature review using the Medline database from 1948 to August 2013 was conducted to investigate the possible association between lower extremity lymphoedema and tuberous sclerosis. RESULTS: Our literature review confirms that the 5 reported cases of the association between lower extremity lymphoedema and tuberous sclerosis were all in female patients. Our case is the first, to our knowledge, to identify that the cause of lymphoedema is biopsy-proven benign vascular lymphatic malformation and to confirm that this association may occur in male patients as well. CONCLUSIONS: Although the true incidence is unknown, there exists a rare association between tuberous sclerosis and lower extremity lymphoedema, both in female and male patients, which may be severe and lead to compartment syndrome as well as other complications throughout development, including chronic wounds, eventually requiring amputations. A multidisciplinary team approach should be strongly considered. LEVEL OF EVIDENCE: Level V (case report).


Asunto(s)
Enfermedades del Prematuro , Linfedema/etiología , Esclerosis Tuberosa/complicaciones , Adulto , Niño , Preescolar , Femenino , Humanos , Recién Nacido , Recien Nacido Prematuro , Extremidad Inferior/patología , Masculino , Piel/patología , Esclerosis Tuberosa/diagnóstico
13.
Int J Surg Case Rep ; 5(2): 43-6, 2014.
Artículo en Inglés | MEDLINE | ID: mdl-24434727

RESUMEN

INTRODUCTION: Actinomycosis is known to mimic several types of neoplasms, leading to morbid surgical interventions. PRESENTATION OF CASE: We report the particular case of an extensive right para-iliac actinomycetoma presenting as a sarcoma, which to our knowledge has not yet been described in the literature, in a patient with previous ruptured appendicitis. Thanks to the collaborative work between the orthopedic and general surgeons, pathologist and microbiologist, the diagnosis of actinomycosis was made pre-operatively, saving this 15-year-old patient from a tumor resection protocol. DISCUSSION: Actinomycetomas have often been reported to present in the same way as several abdominal and gynecological neoplasms, and on rare occasions been described as mimicking other soft-tissue sarcomas, leading to unnecessary morbid tumor resection protocols. The most common cause of abdominal actinomycosis is perforated appendicitis, and may present several years later. CONCLUSION: While faced with a soft tissue mass transgressing tissue planes and possibly extending to the region of the right lower quadrant, especially with a history of previous perforated appendicitis, one should consider the possibility of an abscess caused by pathogens of intestinal origin, including the gram positive anaerobe Actinomycosis israelii. Failure to actively search for this pathogen, which is not detectable with routine staining techniques and may take up to 1-2 weeks to isolate, may lead to unnecessary morbid surgical procedures.

14.
J Pediatr Orthop ; 33(2): e10-7, 2013 Mar.
Artículo en Inglés | MEDLINE | ID: mdl-23389580

RESUMEN

OBJECTIVE: Also named Caffey disease, infantile cortical hyperostosis is a rare disease that usually affects children of a few weeks of age. The clinical picture is that of irritability, soft tissue swelling at various sites (mandible, clavicle, limbs) with local warmth, and pain on palpation. Radiographs demonstrate an important cortical thickening of the affected bony structures. There is generally spontaneous resolution of the inflammatory signs within the first few months or years of life, without future recurrence. We report the case of a recurrence in the adolescent period and conducted a thorough review of the literature to confirm this rare possibility. METHODS: We report the case of a 12-year-old female patient, presenting symptoms of pain and swelling of both forearms and legs, similar to her initial clinical picture at 1 month of age when the diagnosis of Caffey disease was made. Genetic analysis confirmed the COL1A1 mutation, the classic mutation described in the familial form of this disease. Radiologic investigation revealed new periosteal bone formation of the right and left ulnae and the left fibula, suggesting a recurrence of cortical hyperostosis. A thorough review of the literature was conducted, using the Medline database between 1948 and 2011, to confirm this hypothesis. RESULTS: The literature confirms the possibility but also the rarity of a recurrence of cortical hyperostosis in an adolescent who was diagnosed with Caffey disease in infancy. We have identified less than a dozen cases in the literature describing such a recurrence and in the majority there are no medical photographs or radiographs to support the clinical diagnosis. Our case is well documented both clinically and radiologically regarding the initial presentation and the recurrence in adolescence. CONCLUSIONS: Although very rare and poorly recognized, the diagnosis of recurrent cortical hyperostosis must be evoked in a patient who suffered from Caffey disease in infancy, and presents similar clinical characteristics in the adolescent period. LEVEL OF EVIDENCE: Level V--case report.


Asunto(s)
Colágeno Tipo I/genética , Hiperostosis Cortical Congénita/fisiopatología , Niño , Cadena alfa 1 del Colágeno Tipo I , Femenino , Humanos , Hiperostosis Cortical Congénita/diagnóstico , Hiperostosis Cortical Congénita/genética , Mutación , Recurrencia
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