EXPERIENCES IN THE SUCCESSFUL MANAGEMENT OF FIVE CASES OF STERNOMASTOID MUSCULAR TORTICOLLIS

Congenital muscular Torticollis is a rare musculoskeletal disorder characterized by unilateral shortening of the Sternocleidomastoid muscle. Producing deformity of the neck with ?exion of the neck towards the same side and the chin facing the opposite shoulder. We treated six cases, and of which ?ve are due to congenital Sternomastoid shortening (unilateral) and another case with fusion of cervical vertebrae causing Torticollis and Klippel Feil Syndrome with cleft of the soft palate which was also repaired.

A rare case of spasmus nutans in an infant

@#We report a case of an otherwise healthy 23-month-old boy who presented with nystagmus, head shaking, and abnormal head posture suggestive of spasmus nutans. Neuro-ophthalmologic exam revealed bilateral, low-amplitude, high-frequency, horizontal, disconjugate nystagmus that was more prominent in one eye along with head shaking and a head tilt or face turn. The rest of the exam and the systemic physical examination were normal. Magnetic resonance imaging of the brain did not disclose optic pathway glioma, which has been reported to cause spasmus nutans-like disease. Electroretinogram (ERG) was also recommended to rule out occult retinopathies. However, it was not done due to unavailability of the appropriate corneal electrode for his age. Instead, close follow-up was advised to monitor spontaneous improvement or resolution, or until the child comes of age that he can undergo ERG. This case highlights the management approach and rationale of patients with presumed spasmus nutans. Recognition of the triad of spasmus nutans allows for quick diagnosis and more focused and efficient investigation.

Application of 99Tc m-MIBI SPECT/CT imaging in patients with primary cervical dystonia / 中华核医学与分子影像杂志

Objective:To evaluate the value of 99Tc m-methoxyisobutylisonitrile(MIBI) SPECT/CT imaging for the identification of dystonic muscles in patients with primary cervical dystonia (PCD). Methods:A total of 10 patients with PCD (3 males, 7 females, age (47.3±9.9) years) and 10 healthy subjects (4 males, 6 females, age (43.5±9.4) years; control group) between August 2019 and October 2021 in China-Japan Friendship Hospital were enrolled prospectively. All subjects underwent 99Tc m-MIBI SPECT/CT scan. The SUV max of 8 bilateral representative muscles, including rectus capitis posterior major, obliquus capitis inferior, splenius capitis, semispinalis, sternocleidomastoid, trapezius, musculus scalenus muscle and levator scapulae were evaluated in control group. In PCD group, muscles with abnormal uptake were determined. ROI was drawn and SUV max was measured. Independent-sample t test was used to analyze the differences of SUV max between normal and abnormal muscles. The detecting rates of neck MRI and SPECT/CT for abnormal muscles were analyzed by χ2 test. Results:Normal muscles of healthy subjects showed mild symmetrical radioactivity distribution, with the SUV max of 1.10±0.19. A total of 60 muscles with abnormal uptake in 10 patients were found, including 7 rectus capitis posterior major, 10 obliquus capitis inferior, 8 splenius capitis, 8 semispinalis, 10 sternocleidomastoid, 5 trapezius, 3 musculus scalenus muscle and 9 levator scapulae. The SUV max of muscles with abnormal uptake was 1.81±0.43, which was higher than that of normal muscles ( t=17.05, P<0.001). Only 30 pieces abnormal hypertrophy muscle were found by neck MRI, and the detecting rate was much lower than that of SPECT/CT (18.75%(30/160) vs 37.50%(60/160); χ2=28.03, P<0.001). Conclusion:99Tc m-MIBI SPECT/CT may be a useful method for identifying dystonic muscles and a guide to precision therapy in patients with PCD.

Características, diagnóstico y tratamiento de la tortícolis muscular congénita

Introducción: La tortícolis muscular congénita es una entidad clínica que se hace evidente al nacimiento o poco después, presenta un amplio espectro de secuelas; algunas de estas, una vez establecidas, pueden requerir complejas y costosas correcciones quirúrgicas. we Objetivo: Caracterizar la tortícolis muscular congénita según elementos clínicos, diagnósticos y terapéuticos. Métodos: Se realizó una búsqueda de literatura relevante sobre el tema en el primer cuatrimestre de 2021. Se utilizaron como buscadores de información científica: Pubmed/Medline, SciELO, Scopus y ScienceDirect, así como fuentes oficiales como, China CDC, CDC y FDA. La estrategia de búsqueda incluyó los siguientes términos como palabras clave: tortícolis muscular congénita, complicaciones dentofaciales y psicológicas, plagiocefalia posicional. Se evaluaron artículos de revisión, de investigación y páginas web que, en general, tenían menos de 10 años de publicados, en idioma español e inglés, y que hicieran referencia específicamente al tema de estudio a través del título. Fueron excluidos los artículos que no cumplieron con estas condiciones. Esto permitió el estudio de 90 referencias bibliográficas, de las cuales 30 se citaron en el presente artículo. Conclusiones: La tortícolis muscular congénita es una enfermedad de observancia frecuente, su diagnóstico clínico y por exámenes complementarios debe hacerse en los primeros meses de vida. La detección y tratamiento rehabilitador precoz constituyen las armas fundamentales para evitar sus secuelas dentofaciales y psicológicas.

Introduction: Congenital muscular torticollis is a clinical entity that becomes evident at birth or shortly thereafter. It presents a wide spectrum of sequelae. Some of these, once established, may require complex and costly surgical corrections. Objective: To characterize congenital muscular torticollis according to clinical, diagnostic and therapeutic elements. Methods: A search for relevant literature on the subject was carried out in the first third of 2021. As information search engines Pubmed/Medline, SciELO, Scopus and ScienceDirect were used; as well as official sources, such as China CDC, CDC and FDA. The search strategy included the following keywords: tortícolis muscular congénita [congenital muscular torticollis], complicaciones dentofaciales y psicológicas [dentofacial and psychological complications], plagiocefalia posicional [positional plagiocephaly]. Review articles, research articles and web pages, in Spanish and in English, were assessed by considering that they had generally been published within less than ten years and that they referred, within their title, specifically to the topic of study. The articles that did not meet these conditions were excluded. This allowed the study of ninety bibliographic references, thirty of which were cited in the present article. Conclusions: Congenital muscular torticollis is a frequently observed disease, which should be diagnosed, clinically and by complementary tests, in the first months of life. Early identification and rehabilitation treatment are the fundamental weapons to avoid its dentofacial and psychological sequelae.

Fibromatosis de Colli: Caso clínico / Fibromatosis Colli: A case report

Resumen Introducción: La Fibromatosis Colli es un tumor fibroso congénito benigno que se desarrolla en el músculo esternocleidomastoideo. Se manifiesta clínicamente por hinchazón del cuello y restricción de movimiento del cuello (tortícolis). Objetivo: Describir los aspectos diagnósticos y terapéuticos fundamentales de la Fibromatosis de Colli o Tortícolis Muscular Congénita. Caso Clínico: Se presenta un caso de neonato de sexo femenino de 21 días de nacida mediante parto en presentación podálica. Al examen físico se encuentra masa en cuello de 1,5*2 cm indolora e inclinación de la cabeza hacia la derecha, diagnosticado con ultrasonografía y tomografía de cuello contrastado, sin otros hallazgos. Conclusiones: La Fibromatosis de Colli es una patología que pasa desapercibida en el examen posparto del recién nacido. Es importante que el personal médico la identifique, evitando así complicaciones futuras, como asimetría craneofacial, escoliosis cervical y torácica o alteraciones oculares importantes.

Abstract Introduction: Fibromatosis Colli is a benign congenital fibrous tumor, which develops in the sternocleidomastoid muscle. Its clinical manifestations encompass neck swelling and torticollis (restriction of neck movements). Objective: The aim of this report is to describe the fundamental diagnostic and therapeutic aspects of Fibromatosis Colli or Congenital Muscular Torticollis. Clinical Case: A case of a 21-day-old female neonate is presented, with the presence of a right cervical mass (1.5*2 cm) and the head tilted to the right without other associated symptoms found in the physical examination. A relevant antecedent is breech presentation during the birth. The diagnosis is obtained through ultrasonography and contrasted neck tomography. Conclusions: Fibromatosis Colli is a pathology that could be unnoticed in the postpartum examination of the newborn. It is important that medical personnel identify it in order to avoid future complications, such as craniofacial asymmetry, cervical and thoracic scoliosis, or important ocular alterations.

Caracterización clínica, radiológica y epidemiológica de niños con secuelas de tortícolis muscular congénita / Clinical, radiological, and epidemiological characterization of children with sequelae of congenital muscular torticollis / Caracterização clínica, radiológica e epidemiológica de crianças com sequelas de torcicolo muscular congênito

RESUMEN Introducción: Las secuelas de la tortícolis muscular congénita en niños tiene un amplio espectro, algunas de las cuales, una vez establecidas pueden requerir complejas y costosas correcciones quirúrgicas. Objetivo: Caracterizar las secuelas de tortícolis muscular congénita según elementos clínicos, radiológicos y epidemiológicos en niños atendidos en Santiago de Cuba en el periodo 2017-2020. Método: Estudio prospectivo-analítico de caso-control en 112 niños con dicha enfermedad, separados en casos (24 niños) y controles (88 niños). El procesamiento de datos implicó análisis de frecuencias, cálculo de Ji-cuadrado, identificación de factores asociados a variable dependiente, cálculo del valor de p y Odds ratio. Resultados: Se obtuvo predominio de población masculina en casos (70,8 %) y controles (68,2 %), con asociación estadística entre edad ≥ 6 meses al momento del diagnóstico y presencia de complicaciones (OR: 2,4-20,4; p=0,00). Existió asociación estadística entre macrosomía al nacer y presencia de complicaciones en 25,0 % de casos y 11,4 % de controles (OR: 1,9-12,5; p=0,02). Hubo asociación estadística entre inicio tardío del tratamiento rehabilitador y complicaciones (OR: 2,86-21,3; p=0,00). Conclusiones: Se observó predominio de complicaciones orgánicas o comorbilidades en varones, siendo más probable el incremento significativo de secuelas en estos y cuando se inicia el tratamiento médico después de los 6 meses de edad. Las complicaciones más frecuentes fueron: asimetría facial, plagiocefalia y asociación de dos o más secuelas.

ABSTRACT Introduction: Sequelae of congenital muscular torticollis in children have a wide spectrum, some of which, set already, may require complex and costly surgical corrections. Objective: To characterize the sequelae of congenital muscular torticollis according to clinical, radiological, and epidemiological elements in children attended in Santiago de Cuba from 2017 to 2020. Method: Prospective-analytical case-control study in 112 children with this disease, separated in cases (24 children) and controls (88 children). Data processing involved frequency analysis, calculation of chi-square, identification of factors associated with dependent variable, calculation of the p-value and OR. Results: The male population predominated in cases (70.8%) and controls (68.2%), with a statistical association between age ≥ 6 months at diagnosis and the presence of complications (OR: 2.4-20.4; p=0.00). There was a statistical association between macrosomia at birth and the presence of complications in 25.0 % of cases and 11.4 % of controls (OR: 1.9-12.5; p=0.02). There was a statistical association between late initiation of rehabilitation treatment and complications (OR: 2.86-21.3; p=0.00). Conclusions: A predominance of organic complications or comorbidities was observed in males, with a significant increase of sequelae in this group and also when medical treatment is started after 6 months of age. The most frequent complications were facial asymmetry, plagiocephaly and association of two or more sequelae.

RESUMO Introdução: As sequelas do torcicolo muscular congênito em crianças têm amplo espectro, algumas das quais, uma vez estabelecidas, podem exigir correções cirúrgicas complexas e onerosas. Objetivo: Caracterizar as sequelas do torcicolo muscular congênito segundo elementos clínicos, radiológicos e epidemiológicos em crianças atendidas em Santiago de Cuba no período 2017-2020. Método: Estudo prospectivo-analítico caso-controle em 112 crianças com essa doença, separadas em casos (24 crianças) e controles (88 crianças). O processamento dos dados envolveu análise de frequência, cálculo do Qui-quadrado, identificação dos fatores associados à variável dependente, cálculo do valor de p e Odss ratio. Resultados: Obteve-se predominância da população masculina nos casos (70,8%) e controles (68,2%), com associação estatística entre idade ≥ 6 meses no momento do diagnóstico e presença de complicações (OR: 2,4-20,4; p=0,00). Houve associação estatística entre macrossomia ao nascimento e presença de complicações em 25,0% dos casos e 11,4% dos controles (OR: 1,9-12,5; p=0,02). Houve associação estatística entre início tardio do tratamento de reabilitação e complicações (OR: 2,86-21,3; p=0,00). Conclusões: Observou-se predominância de complicações orgânicas ou comorbidades no sexo masculino, com aumento significativo de sequelas sendo mais provável nestes e quando o tratamento médico é iniciado após os 6 meses de idade. As complicações mais frequentes foram: assimetria facial, plagiocefalia e associação de duas ou mais sequelas.

Subluxación rotatoria atloaxoidea o síndrome de Grisel. A propósito de un caso / Rotatory subluxation of the atlantoaxial joint or Grisel's syndrome. Case report

La tortícolis es una contractura involuntaria unilateral del esternocleidomastoideo y de la musculatura posterior del cuello que causa la inclinación de la cabeza hacia un lado. Se han descrito múltiples causas de torticolis siendo una de ellas la subluxación atloaxoidea atraumática asociada a un proceso de carácter inflamatorio en la región de cabeza y cuello, llamada síndrome de Grisel. Aunque la mayoría de los pacientes suelen recuperarse sin secuelas tras tratamiento médico existe la posibilidad de complicaciones graves con déficits funcionales, de ahí la importancia de la realización de un diagnóstico y tratamiento precoz.

Torticollis is an unilateral involuntary contracture of the sternocleidomastoid and posterior neck musculature that causes the head tilts to the one side. Multiple causes of torticolis have been described, one of them being the non-traumatic rotatory subluxation of the atlantoaxial joint associated with an inflammatory process in the head and neck region, called Grisel's syndrome. Although most patients usually recover without sequels after medical treatment, there is the possibility of serious complications with functional deficits, that is why the importance of an early diagnosis and treatment of this potology.

WU Lian-zhong's experience in treating spasmodic torticollis by Kaiqiao Shunjin method / 中国针灸

Professor WU Lian-zhong's experience in treating spasmodic torticollis by Kaiqiao Shunjin method (resuscitation and regulating muscle) is summarized in this paper. The pathogenesis of spasmodic torticollis is the occluded brain orifices and delirium, qi disorder of meridian tendons, specifically divided into five categories: damp-heat, liver-yang hyperactivity, liver-kidney yin deficiency, deficiency of the governor vessel, excess of the governor vessel. The treatment should be based on the symptoms and the root causes, the symptoms should be the main treatment, and the root cause should be treated based on syndrome differentiation. The main treatment is Kaiqiao Shunjin method (resuscitation and regulating muscle), and to take the chief (five heart acupoints-Shuigou [GV 26], Laogong [PC 8], Yongquan [KI 1]), deputy (Yintang [GV 24+], Shangxing [GV 23] through Baihui [GV 20], Ximen [PC 4]), assistant (Fenglong [ST 40], Lianquan [CV 23], combined with tongue needle prick), envoy (Hanyan [GB 4]) as the basic main acupoints, at the same time cooperate with local acupoints to remove knots and accumulation. Finally, syndrome differentiation is adopted to dredge meridians and disperse knots, regulate the governor vessel, and nourish yin and dispel wind.

Hanger reflex and cervical dystonia-negative sensory trick in normal person / 中华神经科杂志

Hanger reflex is a common phenomenon in the population that involuntary head and neck rotation or skew occurs when applying pressure to the forehead, tempus or occipitalia with an ordinary wire hanger tightly around the head. The hanger reflex is consistent with the characteristics of sensory tricks in dystonia, and the phenomenon induced by it in normal people should be regarded as negative sensory tricks in dystonia. The mechanism of dystonia behind the hanger reflex and its therapeutic potential deserve more clinicians′ attention.

Traumatic atlantoaxial rotatory dislocation in the pediatric population / Luxação rotacional traumática de c1-c2 na população pediátrica / Luxación rotatoria traumática de c1-c2 en población pediátrica

ABSTRACT Objective: The treatment of C1-C2 rotatory dislocation remains controversial and surgery is rare. Surgical treatment is indicated when the injury satisfies the instability criteria or when it cannot be reduced. The objective of this study is to analyze the principles and the adaptations necessary for treating these injuries in the pediatric population. Methods: A retrospective case series study. Three cases of patients diagnosed with traumatic C1-C2 rotatory dislocation and treated surgically in our hospital were studied. Through critical analysis of the available literature, a practical guide was proposed to establish the principles and competencies for the treatment of these injuries. Results: The operated cases were female patients between 8 and 16 years of age, with a diagnosis of traumatic atlantoaxial dislocation. Two patients required preoperative skeletal traction with halo. All patients underwent posterior instrumented arthrodesis, two with a transarticular screw technique and one with mass and C2 isthmic (Göel-Harms) screws. Conclusion:. It is essential to determine if the injury is stable and reducible. We recommend treating this type of injury keeping the criteria and competencies related to the stability, alignment, biology and function of the spine in mind. Level of evidence IV; Case series.

RESUMO Objetivo: O tratamento da luxação rotacional de C1-C2 permanece controverso, e a cirurgia é rara. O tratamento cirúrgico é indicado quando a lesão satisfaz os critérios de instabilidade ou quando não pode ser reduzida. O objetivo deste estudo é analisar os princípios e a adequação necessários para tratar essas lesões na população pediátrica. Métodos: Estudo retrospectivo de série de casos. Foram estudados três casos em pacientes tratados cirurgicamente em nosso hospital com diagnóstico de luxação rotacional traumática de C1-C2. Por meio de análise crítica da literatura disponível, foi proposto um guia prático para estabelecer os princípios e a adequação do tratamento dessas lesões. Resultados: Os casos submetidos à cirurgia foram pacientes do sexo feminino, entre 8 e 16 anos de idade, com diagnóstico de luxação atlantoaxial traumática. Duas pacientes precisaram de tração esquelética pré-operatória com halo. Todas as pacientes foram submetidas à artrodese instrumentada por via posterior, duas com técnica de parafuso transarticular e uma com parafusos de massa e pedículo e lâmina em C2 (técnica de Göel-Harms). Conclusões: É essencial determinar se a lesão é estável e se pode ser reduzida. Recomenda-se tratar esse tipo de lesão tendo em mente os critérios e a adequação relacionados com estabilidade, alinhamento, biologia e função da coluna vertebral. Nível de evidência IV; Série de casos.

RESUMEN Objetivo: El tratamiento de la luxación rotatoria de C1-C2 permanece controversial y la cirugía es rara. Se indica tratamiento quirúrgico cuándo la lesión cumple criterios de inestabilidad o cuándo es considerada irreductible. El objetivo de este estudio es revisar los principios y competencias necesarios para tratar esas lesiones en la población pediátrica. Métodos: Estudio retrospectivo de serie de casos. Se estudian tres casos en pacientes tratados quirúrgicamente en nuestro hospital con diagnóstico de luxación rotatoria de C1-C2 traumática. A través del análisis crítico de la literatura disponible se elabora un esquema práctico para establecer los principios y competencias para el abordaje de estas lesiones. Resultados: Los casos intervenidos fueron pacientes de sexo femenino entre 8 y 16 años, con diagnóstico de luxación atlantoaxoidea traumática. Dos pacientes requirieron tracción esquelética preoperatoria con halo. A todas las pacientes se les practicó artrodesis instrumentada por vía posterior, dos con técnica de tornillos transarticulares y una con tornillos de masa e ístmicos de C2 (Göel-Harms). Conclusiones: Resulta imprescindible determinar si la lesión es estable y reductible. Siempre abordar este tipo de lesiones teniendo presentes los criterios y competencias relacionados con la estabilidad, alineación, biología y función de la columna vertebral. Nivel de Evidencia IV; Serie de casos.

Effect of botulinum toxin type a in congenital muscular torticollis / 中华医学美学美容杂志

Objective:To discuss the clinical results of treatment for congenital muscular torticollis with different injection points of botulinum toxin type A and traditional conservative method, and to expound the safety and effectiveness of this way.Methods:From January 2018 to December 2018, 60 cases aged from one month to six months with congenital muscular torticollis who visited the outpatient clinic of plastic surgery in Children＇s Hospital of Quanzhou, a teaching hospital of FuJian Medical University, and there were 38 males and 22 females. The treatment groups were divided into two groups according to random number method that were treated with botulinum toxin type A: the single point group was given one point injection, and the three-point group was injected with three points, while the control group was treated with traditional conservative treatment (mainly manual stretching exercises). The number of cases in each group was 20.Results:The differences of muscle thickness and muscle length were repeatedly measured at different time points in the same group and variance analysis conducted. The results showed that there were statistical significances among the two indicators at different time points in each group ( P<0.05). Least significant difference (LSD) was further adopted for pairwise comparison between indicators at different time points in each group and the differences were statistically significant ( P<0.05). In the comparison of treatment effect of different groups after twelve months follow-up, the cure rate was 85% (17 cases) in the single point group, 95 % (19 cases) in the three-point group, and 80 % (16 cases) in the control group, there was no significant difference among three groups ( P>0.05). Conclusions:Local injection of botulinum toxin type A is a safe and effective treatment option for congenital muscular torticollis, which can achieve the same clinical effect as traditional conservative treatment.

Congenital muscular torticollis severity classifications in the diagnosis and prognosis of infants′ congenital muscular torticollis / 中华物理医学与康复杂志

Objective:To explore the value of congenital muscular torticollis severity classification scores (CMT-SCSTs) in the diagnosis and prognosis of infants′ congenital muscular torticollis (CMT).Methods:A total of 89 CMT children were examined using ultrasound to determine the size of the affected side and the contralateral sternocleidomastoid mass or the abnormal area of the muscle fringe echo from the head and neck. They were also rated using CMT-SCST scoring. They were then given 15 minutes of massage once a day and 5 minutes postural correction twice a day for 3 to 6 months. They were reviewed using ultrasound and the CMT-SCST 3 and 6 months later.Results:The number of treatments was negatively correlated with the initial CMT-SCST score. For those with a baseline CMT-SCST score ≥7 it was effective after 3 and 6 months. Those with a baseline CMT-SCST score ≥9 were almost cured after 3 and 6 months. Gender, age, signs of hip dysplasia, and comorbid global developmental delay (GDD) were not consistently related to the severity and the outcome of CMT. The CMT-SCST was more sensitive in prognosis than the CMT-SCS published in 2013.Conclusions:CMT-SCST scoring provides a theoretical foundation for the diagnosis and treatment and evaluation of infants with CMT. It deserves wider popularity and application in clinical practice.

Clinical analysis and prognosis of reoperation of congenital muscular torticollis in 12 children / 中华实用儿科临床杂志

Objective:To explore the clinical characteristics and short-term prognosis of reoperation of congenital muscular torticollis (CMT).Methods:A total of 12 cases of recurrent CMT treated in the First Affiliated Hospital of Zhengzhou University from May 2015 to May 2018 were recruited, involving 9 boys and 3 girls with the mean age of 13.2 (8-16) years.The causes of disease recurrence were analyzed, and the postoperative appearance of the children was observed by follow-up.In addition, the prognosis of CMT was evaluated by patient satisfaction survey and Lee score criteria.Results:Among the 12 patients, 10 cases were treated in other hospitals and 2 cases in this hospital.The age of the first surgery ranged from 6 months to 2.2 years.Surgical methods included minimally invasive small needle knife (2 cases), single head release of small incision (5 cases), simple amputation (7 cases) and broken end silk thread ligation (4 cases). During the second surgery, contracture and adhesion of sternocleidomastoid muscle at varying degrees were confirmed, including contracture and adhesion of platysma muscle (3 cases), difficulty in separation of jugular sheath (8 cases), and repair of rupture of internal jugular vein (2 cases). Bipolar release of sternocleidomastoid muscle was performed in all surgeries, and tension-free alignment of the neck to the midline was the complete basis for release.The postoperative appearance of all children was satisfied.They were followed up for an average of 2.3 (1.2-3.0) years.Nine and 3 cases graded more than 90 and 80 patient satisfaction scores, respectively.Lee scores reflected the range of motion of head and neck, and the distance and degree of head deviation from the midline, which yielded excellent, good and fair in 12 cases, 8 cases and 3 cases, respectively.Conclusions:Incomplete release is believed as the basic cause of recurrence in children with CMT.Meanwhile, muscle scar adhesion, early operative age, minimally invasive incision and lack of postoperative rehabilitation may also be the direct factors for CMT recurrence.For recurrent patients, satisfactory therapeutic effect can be achieved through bipolar release, especially complete release of the lower pole, combined with postoperative neck traction, fixation and rehabilitation exercise.

Summary on / 中国针灸

The thinking and experience of professor

Prognostic Factors in Recurrent Congenital Muscular Torticollis

@#Introduction: Congenital muscular torticollis (CMT), primarily resulting from unilateral shortening and fibrosis of the sternocleidomastoid muscle. One of the common surgical complications is recurrent deformity. However, the associations between unipolar or bipolar release, age of the patient, and the recurrence of the disease are unclear. Therefore, the purpose of this study was to evaluate the factors associated with recurrence after surgery. Materials and Methods: A retrospective review was performed in 47 patients who were diagnosed with CMT and had been treated surgically with unipolar or bipolar release between January 2007 and December 2015. Demographic data (sex, sides, surgical technique, age at time of surgery, period of follow-up, complications and recurrence) were recorded. Results: Forty-seven patients with an average age of 8.7 years old at time of surgery. Twenty-six patients had rightsided muscular torticollis, while 21 had left-sided. The average follow-up time was 2 years (range, 2–4 years). The average age of unipolar release was 8.8 years old (range, 2- 18 years old), while the average age of bipolar release was 8.7 years old (range, 2–13 years old). Recurrence occurred in 11 patients (9 in unipolar and 2 in bipolar release). Sex, side of deformity, type of surgery and age at time of surgery showed no statistically significant as a factor for recurrence rate, however recurrence of unipolar more than bipolar surgery was nearly two times revealing clinical significance. Conclusions: Sex, side of deformity, type of surgery and age at time of surgery were not associated with the recurrence deformity.

Tortícolis como forma de presentación de quiste de la bolsa de Blake. Diagnóstico ecográfico en atención primaria / Torticolis as a form of presentation of Blake's pouch cyst. Ultrasound diagnosis in primary care

La tortícolis es un signo clínico definido por la inclinación lateral del cuello y rotación de la cabeza, que puede ser fija o flexible y acompañarse o no de dolor cervical. Aparece en trastornos de diferente complejidad. Ante un caso de tortícolis, es preciso realizar una historia clínica cuidadosa y un examen físico completo, y, en caso de ser persistente, solicitar pruebas de imagen.Se hace referencia a una causa de tortícolis no descrita en la literatura. Se trata de una tumoración quística compresiva en la fosa craneal posterior, quiste de la bolsa de Blake, en una lactante pequeña diagnosticada mediante ecografía en la consulta de Pediatría de Atención Primaria. Tras el diagnóstico, se remitió al centro hospitalario de referencia, donde se intervino de urgencia por Neurocirugía Pediátrica, mediante fenestración de la tumoración por ventriculostomía endoscópica y derivación ventrículo-peritoneal. Actualmente, se encuentra asintomática y sin secuelas.

Torticollis is a clinical sign defined by the lateral inclination of the neck and rotation of the head, which can be fixed or flexible and accompanied or not by cervical pain. It appears in disorders of different complexity. In a case of torticollis it is necessary to carry out a careful medical history and a complete physical examination and, if persistent, request imaging tests.Reference is made to a cause of torticollis not described in the literature. This is a compressive cystic tumor in the posterior cranial fossa, Blake's pouch cyst, in a small infant diagnosed by ultrasound in the Primary Care Pediatrics office. After diagnosis, she was referred to the referral hospital, where emergency intervention was performed by pediatric neurosurgery, by fenestration of the tumor by endoscopic ventriculostomy and ventriculo-peritoneal shunt. She is currently asymptomatic and without sequelae.

The effect of cognitive task on postural stability in cervical dystonia / O efeito da tarefa cognitiva sobre a estabilidade postural na distonia cervical

ABSTRACT Background: Cervical dystonia (CD) is the most common form of focal dystonia. It is not known exactly whether abnormal head postures in cervical dystonia cause balance problems. Dual-tasking is a common every-day life situation. Objective: We aimed to evaluate postural stability (PS) in patients with CD and the effect of cognitive task on PS. As a secondary aim, we evaluated the effect of onabotulinum toxin A (BoNT) injection on PS. Methods: A total of 24 patients with CD who were on BoNT treatment for at least one year and 23 healthy controls were included. Posturographic analyses were carried out in all the subjects on static posturography platform under four different conditions: eyes open, eyes closed, tandem stance and cognitive task. In patients, posturographic analysis was carried out just before the BoNT injections and was repeated four weeks later. Results: Before treatment, the anterior-posterior sway was significantly higher in CD patients with the eyes open condition compared to the controls (p=0.03). Cognitive task significantly affected several sway velocities. Tandem stance significantly affected many sway parameters, whereas the eyes closed condition did not. After treatment, only two parameters in tandem stance and one in cognitive task improved within the patient group, in a pairwise comparison. Conclusions: Postural control is impaired in CD patients probably due to the impaired proprioceptive and sensorimotor integration. In reference to dual task theories possibly due to divided attention and task prioritization, cognitive dual-task and harder postural task disturbes the PS in these patients.

RESUMO Introdução: A distonia cervical (DC) é a forma mais comum de distonia focal. Não se sabe exatamente se posturas anormais da cabeça na DC causam problemas de equilíbrio. A execução de duas tarefas simultaneamente é situação comum da vida cotidiana. Objetivo: Avaliar a estabilidade postural (EP) em pacientes com DC e o efeito da tarefa cognitiva na EP. Como objetivo secundário, avaliamos o efeito da toxina onabotulínica A (BoNT) na EP. Métodos: Foram incluídos 24 pacientes com DC em tratamento com BoNT por pelo menos um ano e 23 controles saudáveis. As análises posturográficas foram realizadas em todos os sujeitos na plataforma de posturografia estática sob quatro condições diferentes: olhos abertos, olhos fechados, postura tandem e tarefa cognitiva. Nos pacientes, a análise posturográfica foi realizada imediatamente antes das injeções de BoNT e após quatro semanas. Resultados: Antes do tratamento, a oscilação ântero-posterior era significativamente maior nos pacientes com DC com os olhos abertos quando comparados aos controles (p=0,03). A tarefa cognitiva interferiu significativamente nas velocidades de oscilação. A postura tandem afetou significativamente muitos parâmetros de oscilação, enquanto a condição de olhos fechados não. Após o tratamento, apenas dois parâmetros na posição tandem e um na tarefa cognitiva melhoraram no grupo de pacientes. Conclusões: O controle postural é prejudicado em pacientes com DC, provavelmente devido à comprometida integração proprioceptiva e sensório-motora. Em referência às teorias de dupla-tarefa, possivelmente devido à atenção dividida e à priorização de tarefas, a dupla-tarefa cognitiva e a tarefa postural mais difíceis perturbam o EP nesses pacientes.

Nistagmo secundario a albinismo con compromiso ocular en paciente femenina: un desafío diagnóstico / Nystagmus secondary to albinism with ocular involvement in a female: a diagnostic challenge

INTRODUCCIÓN: El nistagmo infantil es infrecuente y representa un desafío diagnóstico para el pediatra. El albinismo es una de sus principales causas, siendo difícil de sospechar en ausencia de compromiso cutáneo evidente, especialmente en pacientes femeninas, debido a que tipo de herencia del albinismo ocular. OBJETIVO: Describir un caso de nistagmo secundario a albinismo con compromiso ocular aislado en paciente femenina, para discutir el enfoque diagnóstico pediátrico. CASO CLÍNICO: Paciente fe menino de 3 semanas de vida, sin antecedentes mórbidos, derivada a neuropediatra y oftalmólogo por movimientos oculares paroxísticos desde las 2 semanas, con estudio con electroencefalograma e imágenes cerebrales normales. A los 3 meses se confirmó translucencia iridiana, nistagmo y astigmatismo hipermetrópico. La valuación dermatológica descartó compromiso cutáneo. Evolucionó con inclinación cefálica hacia abajo y retraso del desarrollo de la coordinación, fue manejada con lentes de corrección y kinesioterapia. A los 3 años, destacaba mejoría de la agudeza visual, disminución del nistagmo y neurodesarrollo normal. La evaluación oftalmológica de ambos padres fue normal y no había antecedentes de nistagmo o albinismo en la familia. Por decisión de los padres no se realizó estudio genético. CONCLUSIÓN: El diagnóstico de nistagmo secundario a compromiso ocular del albinismo, aún en ausencia de afección cutánea, es clínico; el estudio genético permite confirmar la etiología, sin ser un examen imprescindible, a menos que se considere la planificación familiar. La pesquisa oportuna e intervención multidisciplinaria determinan un mejor pronóstico.

INTRODUCTION: Infantile nystagmus is an infrequent condition that represents a diagnostic challenge for the pediatri cian. Albinism is one of its main causes, being difficult to suspect in the absence of evident cutaneous involvement, especially in female patients, due to the inheritance type of ocular albinism. OBJECTIVE: To describe a case of nystagmus secondary to albinism with isolated ocular involvement in a female patient, in order to provide tools for pediatric approach and diagnosis. CLINICAL CASE: Three- weeks-old female patient, without morbid history, referred to a pediatric neurosurgeon and ophthal mologist due to paroxysmal eye movements since 2 weeks of age. The electroencephalogram and brain images were normal. In follow-up monitoring at 3 months, iris translucency, nystagmus, and hypermetropic astigmatism were confirmed. Dermatologic evaluation ruled out cutaneous invol vement. The patient developed cephalic downward inclination and coordination development de lay was confirmed, the patient was handled with corrective lenses and kinesiotherapy. In follow-up monitoring at 3 years, there was an improvement in visual acuity, decreased nystagmus and normal neurodevelopment. The ophthalmological evaluation of both parents was normal and there was no history of nystagmus or albinism in the family. Upon her parents' decision, no genetic study was ca rried out. CONCLUSION: The diagnosis of nystagmus secondary to ocular albinism, even in the absence of cutaneous involvement, is clinical. The genetic study allows confirming the etiology, without being an essential examination, unless family planning is considered. Timely research and multidisciplinary intervention determine a better prognosis.

Effect of Integrative Medicine on Infant with Congenital Muscular Torticollis / 中国康复理论与实践

Objective:To study the efficacy of integrative medicine on congenital muscular torticollis. Methods:From October, 2017 to September, 2019, 80 children with congenital muscular torticollis were divided into group 1 (n = 40) and group 2 (n = 40) according to different treatment schemes. Group 1 received comprehensive physiotherapy, including passive stretching, head control training, posture correction and family rehabilitation. Group 2 received Tuina in addition. Before and six months after treatment, the root mean square (RMS) of surface electromyography of bilateral sternocleidomastoid muscles in supine neutral position, neck rotation and stretch, and the range of motion of passive neck rotation and lateral flexion, and the angle of head deviation from the midline to the affected side were compared. Results:Before treatment, the RMS of sternocleidomastoid muscle in each position was lower in the affected side than in the healthy side (P < 0.01), and the range of motion of neck when lateral flexion to the healthy side and rotation to the affected side was less than that of the other side (P < 0.01) in both groups; however, no significant difference was found between two groups (P > 0.05). After treatment, the RMS of EMG of sternocleidomastoid muscle in each position, and the range of motion of neck when lateral flexion to the healthy side and rotation to the affected side improved (|t| > 3.290, P < 0.01) in both groups, and were better in group 2 than in group 1 (t > 2.401, P < 0.05); the angle of head deviation from the midline to the affected side significantly decreaed (t > 15.075, P < 0.001) in both groups, and was significantly less in group 2 than in group 1 (t = -4.971, P < 0.001). Conclusion:Integrative medicine is effective on infant with congenital muscular torticollis, which is superior to comprehensive physiotherapy only.

Algunos aspectos clínicos e imagenológicos de la tortícolis muscular congénita en menores de 2 años / Some clinical and imaging features of congenital muscular torticollis in children aged under two years

Introducción: La curación de las secuelas dentofaciales de la tortícolis congénita, una vez establecidas, solo puede lograrse mediante cirugías. La identificación temprana de los pacientes aquejados de esta dolencia y un tratamiento precoz es imprescindible, pues de ello dependerá la evolución a largo plazo de los mismos. Objetivo: Identificar los factores que pueden tener utilidad desde la perspectiva de la prevención primordial de la tortícolis congénita. Métodos: Se realizó una investigación observacional, longitudinal y prospectiva en pacientes con diagnóstico de tortícolis muscular congénita atendidos en el Policlínico de Especialidades del Hospital Infantil Sur de Santiago de Cuba, en el periodo de septiembre de 2016 a septiembre de 2018. La muestra fue de 20 pacientes con dicho diagnóstico. Las variables fueron agrupadas en clínicas e imagenológicas. Resultados: Predominaron los pacientes del sexo masculino, 14 casos (70 por ciento). El diagnóstico de la mayoría de los casos fue precoz, 3 (15 por ciento) en la etapa neonatal y 14 (70 por ciento) entre los 29 días y 6 meses. Las complicaciones más temidas tales como la asimetría facial, la deformidad plagiocefálica, la distopia orbitaria y auricular aparecieron sobre todo asociadas a un diagnóstico y tratamiento tardío. La tomografía axial computarizada solo superó a las radiografías para descubrir complicaciones neurológicas. Conclusiones: La atención a temprana edad es la mejor opción terapéutica para evitar complicaciones en niños con tortícolis muscular congénita(AU)

Introduction: Once established, dentofacial sequels of congenital torticollis can only be healed by surgery. Early identification of sufferers and timely treatment are indispensable to achieve a favorable long-term evolution. Objective: Identify potentially useful factors from the perspective of fundamental prevention of congenital torticollis. Methods: An observational longitudinal prospective study was conducted of patients diagnosed with congenital muscular torticollis attending the Secondary Care Polyclinic at the South Children's Hospital in Santiago de Cuba from September 2016 to September 2018. The sample was 20 patients diagnosed with the disease. The variables analyzed were grouped into clinical and imaging. Results: There was a predominance of male patients with 14 cases (70 percent). Diagnosis of most cases was performed early: 3 (15 percent) at the neonatal stage and 14 (70 percent) between 29 days and 6 months. The most feared complications, such as facial asymmetry, plagiocephalic deformity, and orbital and auricular dystopia, were mainly found to be associated to late diagnosis and treatment. Computed axial tomography only surpassed radiography in spotting neurological complications. Conclusions: Early care is the best therapeutic option to prevent complications in children with congenital muscular torticollis(AU)