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      Headache in a patient with Klinefelter’s syndrome and hyperostosis frontalis interna

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          Hyperostosis frontalis interna (HFI) has been reported in older women, but reports in men are rare. We present a novel case of migraine headache in a gentleman with Klinefelter’s syndrome and HFI, along with a discussion of possible pathophysiologic mechanisms underlying both the headache and the HFI.

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          Most cited references 13

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          The International Classification of Headache Disorders: 2nd edition.

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            Migraine is associated with magnetic resonance imaging white matter abnormalities: a meta-analysis.

            There is controversy as to whether migraine is associated with white matter abnormalities (WMAs) on magnetic resonance images. These abnormalities may be important as a risk factor for future stroke. Further, it is controversial whether any increased risk of WMAs is attributable to comorbidities such as vascular disease. A meta-analysis of published case-control studies was undertaken to address the relationship between migraine and magnetic resonance imaging WMAs. Seven studies were identified. Data from studies reporting the incidence of magnetic resonance imaging WMAs in those with migraine and appropriate control populations were used to calculate odds ratios for WMAs in migraine for each study. A stratified meta-analysis was performed using studies that did and did not exclude subjects with disease comorbidities. The summary odds ratio shows that those with migraine are at increased risk for WMAs (odds ratio, 3.9 [95% confidence interval, 2.26-6.72]). The risk does not differ between studies that included subjects with comorbidities and those that did not. This meta-analysis demonstrates that subjects with migraine are at higher risk of having WMAs on magnetic resonance images than those without migraine. This increased risk is present even in younger individuals who do not have co-occurring cerebrovascular disease risk factors. Prospective studies are needed to determine whether the increased risk of stroke in migraine is mediated or foreshadowed by the presence of WMAs.
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              Hyperostosis frontalis interna: an anthropological perspective.

              Hyperostosis frontalis interna (HFI) is manifested by the accretion of bone on the inner table of the frontal bone. Despite the vast literature on HFI, ambiguity exists as to its etiology, osteogenesis, demography, and history. This stimulated the present broad-scale study of HFI which included the evaluation of 1,706 early 20th century skulls (1,007 males and 699 females) from the Hamann-Todd and Terry human osteological collections, as well as 2,019 pre-19th century East-Mediterranean, Amerindian, and Central European skulls. In addition, 72 cadavers were dissected for gross inspection and histology. Special attention was paid to the relationship of the brain and meninges to endocranial lesions. HFI is an independent condition, not a symptom of a more generalized syndrome as suggested in the past. It can appear in a variety of forms but each is the result of the same process and probably of the same etiology. Investigators' previous failure to recognize the mild stages of HFI (types A and B) as an early form of the general HFI process led to erroneous statistics and interpretations of observations. HFI should also be considered a phenomenon separate from HCI, hyperostosis cranialis diffusa (HCD), and other endostoses, even when it appears in association with them. To avoid ambiguity and facilitate the description of cranial hyperostoses, uniform nomenclature (HFI, HCD) has been recommended. HFI is rarely seen in historic populations, regardless of geographical origin. It is most commonly found among females and is believed to be associated with prolonged estrogen stimulation. While its magnitude of manifestation and frequency are much higher in females, HFI is not a purely female phenomenon. Males with hormonal disturbances such as atrophic testis were found to manifest HFI type D. HFI is associated with age insofar as it is much less frequent in females under 40 years of age. Although advanced cases of HFI (types C and D) have been observed in individuals as young as 40 years of age, it is more frequently found after age 60. The frequency of HFI type D will not increase from age 60. Type-predicted analysis by cohort reveals significant ethnic differences. Changes in African American (AA) females appear earlier in life and progress more rapidly than in European American (EA) females. Analysis of radiographs shows a discrepancy between the anatomic prevalence of HFI and its radiological recognition, which is very poor for mild cases. This apparently resulted in the misconceptions that HFI is entirely an old-age phenomenon, and that it is exclusively female. Histological analysis shows that the inner table along with the closely attached dural layer play a major role in the osteogenesis of HFI. Contrary to previous models, no evidence for diploe or ectocranial plate involvement was found. Cadaver study suggests that the predilection for the frontal area may be related to an altered blood supply and/or vascular stretching.

                Author and article information

                +1-734-936-0263 , +1-734-936-8967 , sindhur@med.umich.edu
                J Headache Pain
                J Headache Pain
                The Journal of Headache and Pain
                Springer-Verlag (Milan )
                10 December 2007
                December 2007
                : 8
                : 6
                : 342-344
                [1 ]Department of Neurology, University of Michigan at Ann Arbor, 300 North Ingalls St., #3D06, Ann Arbor, MI 48109 USA
                [2 ]Department of Neurology, University of California, Los Angeles, CA USA
                © Springer-Verlag Italia 2007
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