Search

Purpose: A consistent relationship between CMT and DDH has been established. However, whether the presence of CMT has effect on clinical outcomes of DDH remains unanswered. This study aimed to evaluate clinical features and management of DDH in patients with CMT., Methods: We reviewed the medical records of 54 patients (91 hips) simultaneously diagnosed with DDH and CMT and included 54 patients (97 hips) with only DDH by random number method as the control group. Clinical and examination outcomes were recorded and compared, respectively., Results: The mean follow-up was 18.1 ± 14.5 months (range, 2 to 72 mo). In the DDH and CMT group, the age of initial treatment was lower (100.8 ± 92.3 vs. 229.0 ± 157.4 d, P < 0.001), and the rate of male significantly increased (42.6% vs. 11.1%, P < 0.001). Before treatment, development degree of hip was better than that of the DDH group. Therefore, patients with CMT had approximately 14 times greater odds of conservative treatment success than children in the DDH group (OR, 13.84; 95% CI, 3.16 to 60.60)., Conclusion: We concluded that CMT was not the risk factor for the failure of treatment. On the contrary, the identification of CMT may be beneficial to early discovery and treatment of DDH when we remain vigilant to perform physical exam and screen imaging of DDH for patients with CMT., (© 2021. The Author(s) under exclusive licence to SICOT aisbl.)

Torticollis is encountered often in the paediatric setting and should be considered a presenting symptom, rather than a diagnosis. Aetiologies of torticollis are numerous, and the nomenclature describing underlying diagnosis can be confusing. Furthermore, children with torticollis typically present in the first instance to primary or secondary care rather than to the subspecialist. These factors can contribute to erroneous treatment of this patient-group which could be time critical in some instances. In this review, we discuss the common causes for torticollis and propose a simple clinical assessment tool and early management scheme that will assist in the differential-diagnosis and treatment pathway of this challenging condition.Conclusion: Torticollis can be the initial presentation of various conditions. The diagnosis and management tools provided in this article can aid in guiding paediatricians as to the correct initial management, imaging, and specialist referral. What is Known: • Torticollis in childhood is a very common presenting symptom with numerous aetiologies. • Management is complex, requires multiple clinical and imaging examinations, and is usually performed by non-specialized professionals. What is New: • A new, simple clinical-assessment tool under the acronym PINCH designed to aid paediatric general practitioners in diagnosing correctly the aetiology of torticollis. • A practical management scheme to aid in the treatment pathway of children with torticollis., (© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Atlantoaxial rotatory subluxation/fixation (AARS/F) is a rare cause of torticollis in children. The aim of our study is to investigate all possible clinical and radiological prognostic factors in children with AARS/F. We retrospectively reviewed all cervical spine CT scans of children with AARS/F treated in our Hospital over the last 15 years. AARS/F was classified according to Fielding and Hawkins classification and C1-C2 rotation-degree was calculated. Moreover, two orthopedic surgeons reviewed all clinical reports of these children. All patients were conservatively treated (cervical traction/neck collar). An early recovery was considered in patients with complete clinical/radiological healing at 3 months follow-up, while a late recovery was considered in patients with disease persistence or relapse at 3 months follow-up or earlier. Fifty-five patients with diagnosis of AARS/F were included in the study (mean age = 8.5 years old - 25F, 30M). In 9/55 subjects (16.4%), a late recovery was observed. The presence of a concomitant infection or inflammation in the head and neck region (Grisel's syndrome) was significantly associated with a late recovery (p < .001). Also, the type of AARS/F (p = .019), according to the Fielding and Hawkins classification, and C1-C2 rotation-degree (p = .027) were significantly correlated with the recovery time.Conclusion: In patients with AARS/F, the presence of a concomitant infection/inflammation in the head and neck region is the most important prognostic factor and it is associated with a late recovery. The Fielding and Hawkins classification and C1-C2 rotation-degree well correlate with patients' recovery time. What is Known: • Atlantoaxial rotatory subluxation/fixation (AARS/F) is a rare cause of torticollis in children and CT is the most useful imaging tool for diagnosis and classification of AARS/F. • Conservative treatments are effective in the majority of pediatric patients with AARS/F. What is New: • The presence of a concomitant infection/inflammation in the head and neck region associated with AARS/F (Grisel's syndrome) is the most important prognostic factor and it is associated with a late recovery. • C1-C2 rotation-degrees, as well as Fielding and Hawkins classification system, well correlate with patients' recovery time.

Ocular disease is one of the causes of abnormal head positioning. Conventionally, the behavioral characteristics of ocular torticollis patients are different from those of non-ocular torticollis patients, though research addressing the significance of this difference is yet limited. This prospective, cross-sectional study aims to develop a questionnaire based on the clinical features in children with abnormal head posture being ocularly assessed. Children aged ≥ 6 months who visited our rehabilitation medicine clinic with a chief complaint of abnormal head posture were included. Patients with apparent orthopedic and neurological diseases were excluded. A 10-item questionnaire was developed to analyze the behavioral characteristics of patients. The patients were divided into ocular and non-ocular torticollis groups according to ophthalmologic examination results. Thirty-four and 13 patients were assigned to the non-ocular torticollis and ocular torticollis groups, respectively. Five questions were finally selected and the questionnaire was scored as the sum of the scores for the each questions (yes = 1 point, no = 0 point). The median (interquartile range) score of the ocular torticollis group (3.0 (3.0-4.0)) was significantly higher than that of the non-ocular torticollis group (2.0 (1.0-3.0); p = 0.000).Conclusion: Our parent-reported torticollis assessment questionnaire may be useful for screening ocular torticollis. What is known: • Ocular disease is one of the various causes of abnormal head positioning. • The behavioral characteristics of patients with ocular torticollis are different from those of patients with non-ocular torticollis; research on this matter is limited. What is new: • We delveloped a questionnaire to differentiate ocular and postural torticollis and the score of the questionnaire was different between patients with or without ocular disease. • The questionnaire based on behavioral characteristics may help screening and determining the need of ophthalmic evaluation in patients with torticollis.

Sandifer syndrome is a rare complication of gastro-oesophageal reflux disease (GERD) when a patient presents with extraoesophageal symptoms, typically neurological. The aim of this study was to review the existing literature and describe a typical presentation and most appropriate investigations and management for the Sandifer syndrome. A comprehensive literature search was performed via PubMed, Cochrane Library and NHS Evidence databases. Twenty-seven cases and observational studies were identified. The literature demonstrates that presenting symptoms of Sandifer's may include any combination of abnormal movements and/or positioning of head, neck, trunk and upper limbs, seizure-like episodes, ocular symptoms, irritability, developmental and growth delay and iron-deficiency anaemia. A 24-h oesophageal pH monitoring was positive in all the cases of Sandifer's where it was performed, while upper GI endoscopy ± biopsy and barium swallow were diagnostic only in a subset of cases. Successful treatment of the underlying gastro-oesophageal pathology led to a complete or near-complete resolution of the neurological symptoms in all of the cases.Conclusion: It is evident from the literature that many patients with Sandifer syndrome were originally misdiagnosed with various neuropsychiatric diagnoses that led to unnecessary testing and ineffective medications with significant side effects. Earlier diagnosis of Sandifer's would have allowed to avoid them.What is Known:• Sandifer syndrome is a rare complication of gastro-oesophageal reflux disease (GERD) when a patient presents with extraoesophageal symptoms, typically neurological.• It may be difficult to recognise due to its non-specific presentation and lack of gastrointestinal symptoms.What is New:• Based on the review of 44 clinical cases of suspected Sandifer syndrome, the clinical picture was clarified: the presenting symptoms of Sandifer's may include any combination of abnormal movements and/or positioning of head, neck, trunk and upper limbs, seizure-like episodes, ocular symptoms, irritability, developmental and growth delay and iron-deficiency anaemia.• Successful treatment of the underlying gastro-oesophageal pathology led to a complete or near-complete resolution of the neurological symptoms in all of the reviewed cases.

Grisel's syndrome is known as a very rare complication of ENT surgery. It is described as non-traumatic atlantoaxial rotatory subluxation, often seen after tonsillectomy or adenoidectomy in children. Therapy is staged according to the Fielding classification. We report the case of a 9-year-old female patient with Grisel's syndrome after otoplasty. The diagnosis was confirmed by CT scan. Manual reposition was performed under general anaesthesia, followed by temporary immobilization with a Minerva orthesis.

Grisel syndrome or atlantoaxial non-traumatic subluxation is a rare entity, poorly described in pediatric literature, although it is a pathology that usually appears in young children. The typical presentation is a torticollis with a previous surgical antecedent (mainly a surgery of the ear, nose, and throat area like adenoidectomy) or after an upper tract respiratory infection. A prompt diagnosis is essential for a successful evolution, and the treatment in most cases is conservative. We report a case of an 8-year-old girl with a typical evolution of this unusual complication. In our case, the most important element for diagnosis was the 3D CT scanner, and the treatment was conservative with a successful outcome. Pediatricians should be aware of this rare but potentially serious entity to establish the correct treatment and avoid complications.

Torticollis in children is a frequently encountered problem. Whereas non-traumatic torticollis usually allows a non-urgent work-up, traumatic torticollis-even in the absence of neurologic abnormalities-needs prompt analysis for structural damage of the spinal column and myelum. Even a trauma as mild as a head-over-heels may result in atlanto-axial subluxation as demonstrated by this patient as described in this case report. In all children with post-traumatic torticollis, further analysis is needed. Atlanto-axial subluxation without neurological or radiological abnormalities can be treated conservatively with a soft collar and analgesics.

We describe the case of a 6-year-old girl with post-traumatic torticollis after falling on her head. The suspected fractures of the dens axis and/or atlas were ruled out after performing CT and MRI examinations as well as dynamic fluoroscopy. Radiological findings showed no further instability but there was a congenital non-fusion of the posterior arch and an age-appropriate non-fused anterior arch of the atlas. In addition to discoligamental injuries and fractures, congenital anomalies and normal variants of the immature anatomy of the cervical spine should also be considered in the diagnosis of the pediatric cervical spine after trauma.

Atlanto-axial fixed subluxation (Grisel's syndrome) is an uncommon complication of upper neck inflammatory processes and head and neck surgery. We present the case of a 6 year old patient who developed a Grisel's syndrome after an upper respiratory infection. X-rays and CT scans demonstrated a fixed C1 and C2 subluxation which was treated with reposition and application of a Halofixateur. We give a review of the pathogenesis, diagnosis and treatment of this rare syndrome.

Torticollis in children can be due to atlanto-axial rotatory subluxation. If diagnosis, based on plain lateral radiography and on computed tomography, and specific treatment are delayed, reduction becomes difficult to accomplish. Atlanto-axial fusion might be indicated to cure the instability. Thus, a diagnostic delay may prevent restoration of cervical spine function. In acquired torticollis in children, radiographic evaluation is indicated if the symptoms are not relieved within 1 week, but immediate radiography is indicated in traumatic torticollis.

Two patients aged 2 and 13 years old are presented, both of whom had torticollis caused by atlanto-axial rotatory subluxation. The correct diagnosis was delayed for 3 months as a result of inappropriate radiologic evaluation. In each patient increased atlanto-dental distance, anterolisthesis of the atlas, and malrotation of the atlas relative to the axis were found on a simple lateral projection. Treatment, consisting in reduction under anaesthesia and plaster fixation for 8 weeks, was successful in the smaller child, whereas redislocation occurred in the other patient. It is imperative to exclude by early radiological examination skeletal abnormality in the presence of persisting acquired torticollis in children.

Acute, painful torticollis in a child is not uncommon. When no local changes can explain the disease, atlanto-axial rotary fixation should be considered. The position of the head is typical but the roentgenological diagnosis is difficult, and as early treatment is mandatory, it should be initiated even if the roentgenogram is "normal". We present a review of nine patients.

Thirty-eight patients with muscular torticollis were treated by a partial resection of the sternocleidomastoid muscle between 1970 and 1981. Clinical evaluations were performed on 35 of these patients who were followed up for 10 years (average of 11.3 years). Of 31 patients under 5 years of age at operation, good results according to Canale's criteria were obtained in 28 (90%). In four patients aged 6 years or more at operation, three showed good results. Postoperatively, massive cotton bandaging was applied with the neck in the neutral position for 3 weeks. A plaster cast, brace, or physiotherapy involving active motion training was not necessary.

Without any known specific cause a 3-year-old boy developed a torticollis. Although the symptoms were treated immediately there was no improvement. Four months later X-rays showed that the dens-atlas distance was enlarged. In spite of this, he was treated by tenotomy and intensive physiotherapy, but the torticollis remained. Two years later a tomogram and a CT showed a complete rotatory dislocation between atlas and axis. At this stage it was impossible to treat the original cause. Physiotherapy was continued to improve the position of the head.

Pediatric cardiologists are often consulted to evaluate continuous murmurs in children. This case report describes a child with a congenital vertebral-jugular fistula who presented with torticollis and a continuous murmur. The differential diagnosis of continuous murmurs in this setting is discussed.

A girl aged 4 years and 5 months was admitted because of vomiting, poor appetite, epigastrial pain, weight loss and an abnormal posturing of the head and neck since the age of 3 1/2 years. This abnormal posturing consisted of maximal lateral flexion of the head and neck to the left. A number of investigations including EEG and CT-Scan of the head, had been performed repeatedly, and despite normal results various neuropsychiatric diagnoses were made. In our hospital massive gastrooesophageal reflux and oesophagitis were demonstrated and the diagnosis of Sandifer's syndrome was established. A fundoplication was performed and the child is free of symptoms since.

A statistical study was done to determine the incidence of congenital torticollis. The aetiological relationship between torticollis and obstetrical paralysis was analysed in an attempt to clarify the possible cause of torticollis. There were forty-three cases of torticollis in the 2,160 babies delivered at the Otsu Red Cross Hospital from 1978 to 1981. The overall incidence of torticollis was 1.9%; 1.2% in cephalic presentations, 6.5% in double breech presentations, 4.2% in footling presentations and 34.0% in single (frank) breech presentations. In addition, forty-three cases of obstetrical paralysis treated in Kyoto University Hospital from 1961 to 1981 were reviewed. Torticollis was found in 26% of the cephalic presentations (6 out of 23) and in 80% of the breech presentations (16 out of 20) respectively. These findings suggest that a foetal posture with both the knees and the cervical spine extended seems to be closely related to the development of torticollis, which indicates that a direct cause may be stretching of the sternocleidomastoid muscle during delivery.