Pediatric-onset trigeminal autonomic cephalalgias: A systematic review and meta-analysis

Cephalalgia ◽  
2021 ◽  
pp. 033310242110275
Author(s):  
Ankita Ghosh ◽  
Emma Silva ◽  
Mark J Burish

Background and objective There are five headache disorders composing the trigeminal autonomic cephalalgias (cluster headache, paroxysmal hemicrania, short-lasting unilateral neuralgiform headache attacks with conjunctival injection and tearing (SUNCT), short-lasting unilateral neuralgiform headache attacks with cranial autonomic symptoms (SUNA), and hemicrania continua). Little is known about these disorders in the pediatric population. The objectives of this study are to report the full age ranges of pediatric trigeminal autonomic cephalalgias and to determine if pediatric-onset trigeminal autonomic cephalalgias display similar signs and symptoms as adult onset. Methods Search criteria in Medline Ovid, Embase, PsycINFO, and Cochrane Library were created by a librarian. The remainder of the steps were independently performed by two neurologists using PRISMA guidelines. Inclusion criteria for titles and abstracts were articles discussing cases of trigeminal autonomic cephalalgias with age of onset 18 or younger, as well as any epidemiological report on trigeminal autonomic cephalalgias (as age of onset data was often found in the results section but not in the title or abstract). Data extracted included age of onset, sex, and International Classification of Headache Disorders criteria for trigeminal autonomic cephalalgias (including pain location, duration, frequency, autonomic features, restlessness) and some migraine criteria (photophobia, phonophobia, and nausea). Studies that did not meet full criteria for trigeminal autonomic cephalalgias were examined separately as “atypical trigeminal autonomic cephalalgias”; secondary headaches were excluded from this category. Results In all, 1788 studies were searched, 86 met inclusion criteria, and most ( 56 ) examined cluster headache. In cluster headache, onset occurred at every pediatric age (range 1–18 years) with a full range of associated features. Autonomic and restlessness features were less common in pediatric patients, while migrainous features (nausea, photophobia, and phonophobia) were found at similar rates. The sex ratio of pediatric-onset cluster headache (1.8, 79 male and 43 female) may be lower than that of adult-onset cluster headache. Data for other trigeminal autonomic cephalalgias, while more limited, displayed most of the full range of official criteria. The data for atypical trigeminal autonomic cephalalgias were also limited, but the most common deviations from the official criteria were abnormal frequencies and locations of attacks. Conclusions Trigeminal autonomic cephalalgias can start early in life and have similar features to adult-onset trigeminal autonomic cephalalgias. Specifically, pediatric-onset cluster headache patients display the full range of each criterion for cluster headache (except maximum frequency of six instead of eight attacks per day). However, cranial autonomic features and restlessness occur at a lower rate in pediatrics. Additional information is needed for the other trigeminal autonomic cephalalgias. As for expanding the ICHD-3 criteria for pediatric-onset trigeminal autonomic cephalalgias, we have only preliminary data from atypical cases, which suggests that the frequency and location of attacks sometimes extend beyond the official criteria. Trial Registration: This study was registered as a systematic review in PROSPERO (registration number CRD42020165256).

Cephalalgia ◽  
2013 ◽  
Vol 34 (3) ◽  
pp. 231-235 ◽  
Author(s):  
Andreas Totzeck ◽  
Hans-Christoph Diener ◽  
Charly Gaul

Introduction The trigeminal autonomic cephalalgias (TACs) subsume four primary headache disorders. Hemicrania continua is increasingly regarded as an additional TAC. In rare cases patients may present with two different TACs or a TAC and hemicrania continua. Cases We report four patients with two different TACs or one TAC and hemicrania continua. Two patients presented with cluster headache and paroxysmal hemicrania, one patient with cluster headache and hemicrania continua, and one patient suffered from cluster headache and SUNCT. Discussion While the International Classification of Headache Disorders (ICHD-II) proposes specific diagnostic criteria, the variability of clinical presentation may make clear diagnosis difficult. All patients fulfilled the ICHD-II criteria. The manifestation of two different TACs or hemicrania continua in one patient is uncommon but possible and should be taken into account especially when chronic headache patients present with changing headache symptoms.


Author(s):  
Thijs H. Dirkx ◽  
Peter J. Koehler

The trigeminal autonomic cephalalgias (TACs), including cluster headache, paroxysmal hemicrania, SUNCT (short-lasting unilateral neuralgiform headache attacks with conjunctival injection and tearing), SUNA (with cranial autonomic symptoms), and hemicrania continua, belong to the primary headaches. They are characterized by severe unilateral headache attacks in association with ipsilateral cranial autonomic features. Cluster headache is the most frequent of the TACs. The other TACs are rare, but epidemiological data are scarce and variable. The various types of TAC are distinguished not only by differences in attack frequency and duration, but also by differences with respect to treatment response. The typical headache syndromes, fulfilling the International Classification of Headache Disorders-3 criteria, have also been described in association with other disorders and imaging is required to exclude intracranial pathology in all newly diagnosed patients.


Cephalalgia ◽  
2003 ◽  
Vol 23 (9) ◽  
pp. 929-930 ◽  
Author(s):  
C Lisotto ◽  
F Mainardi ◽  
F Maggioni ◽  
G Zanchin

The trigeminal autonomic cephalgias (TACs) are characterized by short-lasting unilateral headaches with autonomic features (1). They include four headache disorders, cluster headache (CH), paroxysmal hemicrania (PH), SUNCT syndrome and hemicrania continua (HC). The coexistence of different ipsilateral TACs in the same patient has been previously reported in six published cases (2-6). In five of these patients an association of CH and PH was noted (2-5). The two varieties of attacks occurred separately in three patients, while their simultaneous occurrence was observed in two cases. In another patient the successive occurrence of trigeminal neuralgia, SUNCT syndrome, PH and CH in one active headache period was noted (6). All the reported cases concerned male patients. We describe what we believe to be the first case of coexistence of two different contralateral TACs.


Cephalalgia ◽  
2008 ◽  
Vol 28 (6) ◽  
pp. 626-630 ◽  
Author(s):  
P Irimia ◽  
E Cittadini ◽  
K Paemeleire ◽  
AS Cohen ◽  
PJ Goadsby

Our objective was to compare the presence of self-reported unilateral photophobia or phonophobia, or both, during headache attacks comparing patients with trigeminal autonomic cephalalgias (TACs)—including cluster headache, shortlasting unilateral neuralgiform headache attacks with conjunctival injection and tearing (SUNCT) and paroxysmal hemicrania—or hemicrania continua, and other headache types. We conducted a prospective study in patients attending a referral out-patient clinic over 5 months and those admitted for an intramuscular indomethacin test. Two hundred and six patients were included. In episodic migraine patients, two of 54 (4%) reported unilateral photophobia or phonophobia, or both. In chronic migraine patients, six of 48 (13%) complained of unilateral photophobia or phonophobia, or both, whereas none of the 24 patients with medication-overuse headache reported these unilateral symptoms, although these patients all had clinical symptoms suggesting the diagnosis of migraine. Only three of 22 patients (14%) suffering from new daily persistent headache (NDPH) experienced unilateral photophobia or phonophobia. In chronic cluster headache 10 of 21 patients (48%) had unilateral photophobia or phonophobia, or both, and this symptom appeared in four of five patients (80%) with episodic cluster headache. Unilateral photophobia or phonophobia, or both, were reported by six of 11 patients (55%) with hemicrania continua, five of nine (56%) with SUNCT, and four of six (67%) with chronic paroxysmal hemicrania. Unilateral phonophobia or photophobia, or both, are more frequent in TACs and hemicrania continua than in migraine and NDPH. The presence of these unilateral symptoms may be clinically useful in the differential diagnosis of primary headaches.


Cephalalgia ◽  
2017 ◽  
Vol 38 (4) ◽  
pp. 786-793 ◽  
Author(s):  
Maria-Eliza R Aguila ◽  
Trudy Rebbeck ◽  
Kristofferson G Mendoza ◽  
Mary-Grace L De La Peña ◽  
Andrew M Leaver

Background Clear definitions of study populations in clinical trials may facilitate application of evidence to clinical populations. This review aimed to explore definitions of study populations in clinical trials on migraine, tension-type headache, cluster headache, and cervicogenic headache. Methods We performed a systematic review of clinical trials investigating treatment efficacy for migraine, tension-type headache, cluster headache, and cervicogenic headache. We extracted data on diagnosis, inclusion criteria and baseline headache characteristics. Results Of the 229 studies reviewed, 205 studies (89.5%) defined their populations in adherence to the International Classification of Headache Disorders (ICHD) criteria. Some studies ( n = 127, 55.5%) specified diagnosing through interview, clinical examination and diary entry. The most commonly reported inclusion criteria were pain intensity for migraine and tension-type headache studies ( n = 123, 66.1% and n = 21, 67.7%, respectively), episode frequency ( n = 5, 71.4%) for cluster headache studies, and neck-related pain for cervicogenic headache studies ( n = 3, 60%). Few studies reported details on the extent to which diagnostic criteria were present at baseline. Conclusions ICHD is routinely used in defining populations in headache studies. Details of baseline headache characteristics were not as consistently reported.


Cephalalgia ◽  
2021 ◽  
pp. 033310242110304
Author(s):  
Kuan-Po Peng ◽  
Marlene Schellong ◽  
Arne May

Objective The presence of aura is rare in cluster headache, and even rarer in other trigeminal autonomic cephalalgias. We hypothesized that the presence of aura in patients with trigeminal autonomic cephalalgias is frequently an epiphenomenon and mediated by comorbid migraine with aura. Methods The study retrospectively reviewed 480 patients with trigeminal autonomic cephalalgia in a tertiary medical center for 10 years. Phenotypes and temporal correlation of aura with headache were analyzed. Trigeminal autonomic cephalalgia patients with aura were further followed up in a structured telephone interview. Results Seventeen patients with aura (3.5%) were identified from 480 patients with trigeminal autonomic cephalalgia, including nine with cluster headache, one with paroxysmal hemicrania, three with hemicrania continua, and four with probable trigeminal autonomic cephalalgia. Compared to trigeminal autonomic cephalalgia patients without aura, trigeminal autonomic cephalalgia patients with aura were more likely to have a concomitant diagnosis of migraine with aura (odds ratio [OR] = 109.0, 95% CI 30.9–383.0, p < 0.001); whereas the risk of migraine without aura remains similar between both groups (OR = 1.10, 95% CI = 0.14–8.59, p = 0.931). Aura was more frequently accompanied with migraine-like attacks, but not trigeminal autonomic cephalalgia attacks. Interpretation In most patients with trigeminal autonomic cephalalgia, the presence of aura is mediated by the comorbidity of migraine with aura. Aura directly related to trigeminal autonomic cephalalgia attack may exist but remains rare. Our results suggest that aura may not be involved in the pathophysiology of trigeminal autonomic cephalalgia.


2013 ◽  
Vol 40 (11) ◽  
pp. 1797-1805 ◽  
Author(s):  
Deepak R. Jadon ◽  
Athimalaipet V. Ramanan ◽  
Raj Sengupta

Ankylosing spondylitis (AS) has 2 main modes of onset: juvenile-onset AS (JoAS) and adult-onset AS (AoAS). It is not known whether JoAS is a subtype of AS, or AS modulated by early age of onset and longer disease duration. We performed a systematic review of the literature, identifying 12 articles and 1 abstract directly comparing JoAS and AoAS cohorts, with observational study design. Patients with JoAS appear to have more peripheral joint involvement both clinically and radiographically (especially knees and ankles) and more root joint involvement (hips and shoulders); they are more likely to proceed to hip arthroplasty and often initially present with peripheral rather than axial symptoms. Patients with AoAS appear to have more axial symptoms and radiographic disease, particularly in the lumbar spine, and worse axial metrology. In terms of other characteristics, more evidence is needed to confidently state whether JoAS and AoAS are different.


2010 ◽  
Vol 68 (4) ◽  
pp. 627-631 ◽  
Author(s):  
Vanise Amaral ◽  
Gabriel R. de Freitas ◽  
Bruno C.B Rodrigues ◽  
Daniel de H Christoph ◽  
Carlos A. de Pinho ◽  
...  

Patent foramen ovale (PFO), a relatively common abnormality in adults, has been associated with migraine. Few studies also linked PFO with cluster headache (CH). To verify whether right-to-left shunt (RLS) is related to headaches other than migraine and CH, we used transcranial Doppler following microbubbles injection to detect shunts in 24 CH, 7 paroxysmal hemicrania (PH), one SUNCT, two hemicrania continua (HC) patients; and 34 matched controls. RLS was significantly more frequent in CH than in controls (54% vs. 25%, p=0.03), particularly above the age of 50. In the HC+PH+SUNCT group, RLS was found in 6 patients and in 2 controls (p=0.08). Smoking as well as the Epworth Sleepiness Scale correlated significantly with CH, smoking being more frequent in patients with RLS. PFO may be non-specifically related to trigeminal autonomic cephalalgias and HC. The headache phenotype in PFO patients probably depends on individual susceptibility to circulating trigger factors.


RMD Open ◽  
2020 ◽  
Vol 6 (2) ◽  
pp. e001196
Author(s):  
Matthew Turk ◽  
Jacqueline Hayworth ◽  
Tatiana Nevskaya ◽  
Janet Pope

ObjectivesThis meta-analysis investigated the frequency of ocular involvement in childhood and adult spondyloarthritis (SpA).MethodsA systematic review of the literature was conducted. Medline, Web of Science and Cochrane databases were searched upto October 2018 identifying publications related to SpA, including ankylosing spondylitis (AS) with ocular conditions (OC) (uveitis, iritis, retinitis, chorioretinitis and other ocular involvement). The rates of OC were extracted and random effects models estimated their frequency. Heterogeneity was evaluated using I2. Inclusion criteria were studies in SpA of either children or adults who included a frequency of OC.Results3164 studies were identified, and 41 analysed which included frequencies of uveitis/iritis. Other OC were too infrequent to analyse. A pooled random effects model showed that the prevalence of uveitis was 24% in adult AS (23 studies, 11 943 patients), 10% in adult psoriatic arthritis (PsA) (9 studies, 1817) and 17% in undifferentiated adult SpA (9 studies, 6568 patients). In juveniles with AS, the prevalence of uveitis was 27% (8 studies, 927 patients), in juvenile PsA it was 16% (5 studies, N=498) and in juvenile undifferentiated SpA, uveitis occurred in 7% (2 studies, 1531 patients). In all evaluated SpA subgroups, there were no statistical differences in the frequency of uveitis between juveniles and adults.ConclusionsUveitis in adult versus child-onset SpA is similar in AS but more common in adult-onset undifferentiated SpA, and less frequent in adult-onset PsA compared to child-onset PsA, but the differences were not significant.


2018 ◽  
Vol 32 (07) ◽  
pp. 673-685
Author(s):  
Ajaykumar Shanmugaraj ◽  
Darren de SA ◽  
Matthew M. Skelly ◽  
Andrew Duong ◽  
Nicole Simunovic ◽  
...  

AbstractThe purpose of this systematic review is to ascertain the risk profile of allografts in primary anterior cruciate ligament reconstruction (ACLR) of skeletally immature patients. Three databases (PubMed, EMBASE, and MEDLINE) were searched for articles addressing primary ACLR in skeletally immature patients (i.e., open femoral and tibial physes). Inclusion criteria encompassed the use of allograft tissue with available postoperative outcomes data. The methodological index for non-randomized studies (MINORS) was used to assess all studies. Descriptive statistics such as means, 95% confidence intervals and standard deviations are presented where applicable. A total of 3,852 studies were screened, with 9 studies of a total of 406 skeletally immature patients (mean age 14.9 ± 1.2 years) satisfying inclusion criteria. The majority (98%) of included patients underwent complete transphyseal ACLR. Where specified, allograft options included Achilles tendon (AT) (66.5%), tibialis anterior tendon (7.6%), bone–patellar tendon (2.5%), and fascia lata (1.0%). The use of a bone block for the AT was reported in one patient (0.2%). Postoperatively, and where specified, patients achieved full range of motion (12.1%), had good Lysholm scores of 94 to 100 (8.1%), and a return to preinjury level athletic participation of 82.9% (8.4%). Complications (13.3%) included graft failures (7.9%), nonrevision reoperation (4.7%), and a combined leg length discrepancy and angular (valgus and extension) deformity (0.2%). There were no reported incidences of disease transmission. Although failure rates of primary allograft ACL reconstruction are acceptable compared with other studies of mainly autograft use in this young, high-risk population, there was a very low rate of clinically significant physeal damage. However, the relatively low quality of the included studies limits the ability to recommend routine use of allograft for ACLR in the skeletally immature patient. More robust studies with long-term follow-up data are necessary to better ascertain the influence of allograft choice on postoperative outcomes for these young patients. This is a Level IV study, systematic review of Levels III and IV studies.


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