Migraine is a chronic neurological disorder characterized by headaches and extracephalic symptoms. We report a 73-year-old male patient with a history of migraines as well as several other chronic conditions including abdominal pain accompanied by nausea and vomiting, pain and ecchymosis of the limbs, dysmetropsia, syncope, and melena due to telangiectasia of the sigmoid colon. After a thorough evaluation of the migraine condition, we hypothesized that the patient’s melena due to telangiectasia of the sigmoid colon might in fact be a migraine-related phenomenon. In this report, we discuss a possible mechanism for melena due to telangiectasia in migraine patients, as well as “tips” for identifying subtle and/or unreported clinical features of migraine conditions

Ishii, Hiroshi

Kakisaka, Yosuke

English

Tokushima University Institutional Repository

298CASE REPORTCase study of a migraine patient with multiple extracephalic symptoms including melena due to telangiectasia of the sigmoid colon : a possible new migraine-associated symptomYosuke Kakisaka1, and Hiroshi Ishii21Department of Epileptology, Tohoku University Graduate School of Medicine, Sendai, Miyagi, Japan, 2Department of Neurology and Psychi-atry, Aino Hospital, Osaka, JapanAbstract : Migraine is a chronic neurological disorder characterized by headaches and extracephalic symp-toms. We report a 73-year-old male patient with a history of migraines as well as several other chronic condi-tions including abdominal pain accompanied by nausea and vomiting, pain and ecchymosis of the limbs, dys-metropsia, syncope, and melena due to telangiectasia of the sigmoid colon. After a thorough evaluation of the migraine condition, we hypothesized that the patient’s melena due to telangiectasia of the sigmoid colon might in fact be a migraine-related phenomenon. In this report, we discuss a possible mechanism for melena due to telangiectasia in migraine patients, as well as “tips” for identifying subtle and/or unreported clinical features of migraine conditions. J. Med. Invest. 70 : 298-300, February, 2023Keywords : migraine, extracephalic symptoms, telangiectasiaINTRODUCTION Migraine is a chronic neurological disorder characterized by headaches (1) as well as extracephalic symptoms, some of which, including cyclic vomiting syndrome (2) and abdominal migraines (3), are known as migraine-equivalents. In this report, we present a case of migraine co-present with various symptoms including abdominal pain accompanied by nausea and vomiting, pain and ecchymosis of the limbs, dysmetropsia, syncope, and melena due to telangiectasia of the sigmoid colon. After a thor-ough evaluation, we reached a hypothesis that could explain all of these symptoms through migraine-associated mechanisms. In order to minimize the risk of underdiagnosis, we emphasize the importance, in such cases, of evaluating many different as-pects of migraine, all of which may not be obvious even to skilled physicians. CASE REPORTA 73-year-old male patient presented to our outpatient clinic with episodic headaches and various recurrent symptoms in-cluding abdominal pain with nausea and vomiting, pain in the limbs accompanied by ecchymosis, dysmetropsia, syncope, and melena due to telangiectasia of the sigmoid colon. The clinical course of the patient is shown in Figure 1. The patient’s family history showed a consistent presence of migraines in the ma-ternal family line. Beginning at age six, the patient began to experience severe periumbilical pain lasting approximately six hours, accompanied by nausea and vomiting. The patient also experienced episodic pain and ecchymosis of limbs without any obvious traumatic triggers. These symptoms always remitted spontaneously. The patient received supportive care because a medical investigation conducted between the episodes found no apparent abnormalities. Beginning at age 12, the patient began experiencing odd new episodes that lasted approximately one hour each. Regarding these episodes, the patient recounted : “I abruptly felt as if I became a 10-foot-tall giant.” The patient also suffered several episodes of loss of consciousness triggered by mental stress, which were diagnosed as neutrally-mediated syn-cope. The patient was evaluated using electroencephalography several times, all of which showed no apparent abnormalities. Beginning at age 17, the patient started experiencing episodes of melena, which were treated with supportive care only. The patient then underwent a resection of the sigmoid colon due to a massive melena at age 23. Based on histopathological evaluation of this resected section, the patient was diagnosed with telangi-ectasia as a root cause of melena. At age 23, the patient began experiencing severe, throbbing unilateral headaches typically lasting around six hours without aura, although the headache was transiently accompanied by antecedent visual auras lasting approximately one hour from the age of 39 to 60. These head-aches were also accompanied by photophobia. These episodes initially occurred weekly, although their frequencies gradually decreased to monthly after age 60 without any preventive medications. Findings of the patient’s physical and neurological examinations between attacks were unremarkable. No abnor-mality was indicated on brain MRI. According to the Interna-tional Classification of Headache Disorders 3 (ICHD-3), these types of headaches were compatible either with 1.1 : Migraine without Aura or 1.2 : Migraine with Aura. We hypothesized that all the above-mentioned symptoms could be explained by migraine-associated mechanisms, as detailed below.DISCUSSIONThis report describes a variety of migraine-associated extra-cephalic symptoms, most of which could easily be overlooked in many clinical settings. We believe that this report will facilitate The Journal of Medical Investigation    Vol. 70  2023　　　　　　　　　　　　　　　　　　　　　　　　　　　　　　　　Received for publication November 11, 2022 ; accepted February 3, 2023.Address correspondence and reprint requests to Yosuke Kakisaka, MD, Department of Epileptology, Tohoku University Graduate School of Medicine, 2-1 Seiryo-machi, Aoba-ku, Sendai, Miyagi 980-8575, Japan and Fax : +81-22-717-7346. E-mail : yosuke.kakisaka@epilepsy.med.tohoku.ac.jp299The Journal of Medical Investigation   Vol. 70  February  2023the identification of a variety of migraine-associated features. This may contribute to better management of patients with mi-graine in the future.We believe that this patient’s symptoms can all be considered migraine-associated. His abdominal pain can be considered a form of abdominal migraine (3). An abdominal migraine attack can appear without an accompanying migraine headache (4, 5). Similarly, abdominal pain due to the mechanism of primary stabbing headache (a form of primary headache, which is re-ported to somewhat relate with migraine) can appear without stabbing cephalic pain (6). His body pain and accompanying subcutaneous bleeding could be symptoms of migrainous corpal-gia (7, 8) and migraine-associated ecchymosis (4, 9), respectively. Dysmetropsia or “Alice in Wonder Syndrome” has also been reported to be associated with migraines (10). We believe that recognition of these peculiar forms of migraine is important to prevent diagnostic delay. We believe that the patient’s syncope can have some mecha-nism in common with migraine, as reported by Daas et al., who concluded that vasovagal syncope, a form of neurally mediated syncope, is associated with migraine (11). They reviewed three generations of a family with comorbid vasovagal syncope and migraine. It included 21 subjects. Eleven of the 14 subjects with a diagnosis of migraine (78%) also had vasovagal syncope, and 11 of the 12 subjects with vasovagal syncope (92%) also had migraine (11).  Moreover, our patient’s intestinal hemorrhage due to tel-angiectasia of the sigmoid colon could also be explained by a migraine-related mechanism, similar to that of another case previously reported by Christensen et al. (12). In their meta-anal-ysis, they found that the prevalence of migraines in patients with rosacea, a form of telangiectasia, was higher than in those without rosacea, although the authors could not find a detailed mechanism underlying the two conditions (12). More similar case-studies are needed in order to confirm our hypothesis, as, to the best of our knowledge, no other cases of intestinal hem-orrhaging due to telangiectasia in migraine patients have been reported. This may be a novel aspect to consider in the setting of migraine patient care. However, we do not insist that all mi-graine patients with abdominal pain should receive colonoscopy. We understand that history focused on migraine will be import-ant, especially when the migraine patient’s abdominal pain is 1) repetitive, 2) severe, 3) localized to the midline (or periumblical region), 4) remitting within 2–72 h without specific intervention, and 5) each episode shows similar profile (stereotypic) (1).The mechanism of occurrence of abdominal migraine may be speculated from that of migraine. Migraine is a condition with neuronal sensitization represented by allodynia (pain due to a stimulus that does not usually provoke pain) and hyperalgesia (increased pain from a stimulus that usually provokes pain) (13, 14). With an aim to clarify the mechanism of migraine-associ-ated abdominal pain under this context, it may be reasonable to say that non-painful stimulus for non-hypersensitive brain (i.e., physiological intestinal movements) can cause prominent abdominal pain in the sensitized condition.In future cases of this nature, we recommend a more “active” approach to history-taking (15). In other words, even if a patient does not report symptoms that may be related to migraine, the physician should still attempt to ascertain the presence or ab-sence of candidate symptoms. This can occur even in situations where symptoms appear to be “distant” from the headache (e.g., abdominal pain in migraine patients). If physicians begin to adopt a more wholistic view of migraine treatment, in which any symptom may potentially be a feature of the patient’s migraine, and attempt to develop unitary hypotheses for patients’ condi-tions, we believe they may find subtle and / or unreported mi-graine-associated extracephalic symptoms, which may in turn contribute to better patient management. CONFLICT OF INTERESTNone. REFERENCE1. Headache Classification Subcommittee of the International Headache Society. The International Classification of Head-ache Disorders, 3rd Edition. Cephalalgia 33 : 629-808, 20132. Kakisaka Y, Wakusawa K, Sato I, Haginoya K, Uematsu M, Hirose M, Munakata M, Sato T, Tsuchiya S : Successful treatment with sumatriptan in a case with cyclic vomiting syndrome combined with 18q- syndrome. J Child Neurol Figure 1.300 Y. Kakisaka and H. Ishii  Melena due to telangiectasia in migraine24 : 1561-1563, 20093. Kakisaka Y, Uematsu M, Wang ZI, Haginoya K : Abdomi-nal Migraine Reviewed from Both Central and Peripheral Aspects. World J Exp Med 2 : 75-77, 20124. Kakisaka Y, Wakusawa K, Haginoya K, Uematsu M, Tsuchiya S : Abdominal migraine associated with ecchy-mosis of the legs and buttocks : does the symptom imply an unknown mechanism of migraine? Tohoku J Exp Med 221 : 49-51, 20105. Kakisaka Y, Jin K, Kato K, Iwasaki M, Nakasato N : Tem-poral intermittent rhythmic delta activity and abdominal migraine. Neurol Sci 35 : 627-8, 20146. Kakisaka Y, Kano S, Hino-Fukuyo N, Uematsu M, Kure S : Pediatric-onset extracephalic stabbing pain. J Child Neurol 29 : NP47-8, 20147. Guiloff RJ, Fruns M : Limb pain in migraine and cluster headache. J Neurol Neurosurg Psychiatry 51 : 1022-1031, 1988 8. Kakisaka Y, Ohara T, Katayama S, Suzuki T, Hino-Fukuyo N, Uematsu M, Kure S : Lower back pain as a symptom of migrainous corpalgia. J Child Neurol 28 : 676-677, 20139. DeBroff BM, Spierings ELH : Migraine associated with periorbital ecchymosis. Headache 30 : 260-263,199010. Mastria G, Mancini V, Viganò A, Di Piero V : Alice in Wonderland Syndrome : A Clinical and Pathophysiological Review. Biomed Res Int 2016 : 8243145, 201611. Daas A, Mimouni-Bloch A, Rosenthal S, Shuper A : Famil-ial vasovagal syncope associated with migraine. Pediatr Neurol 40 : 27-30, 200912. Christensen CE, Andersen FS, Wienholtz N, Egeberg A, Thyssen JP, Ashina M : The relationship between migraine and rosacea : Systematic review and meta-analysis. Cepha-lalgia 38 : 1387-1398, 201813. Jensen TS, Finnerup NB : Allodynia and hyperalgesia in neuropathic pain : clinical manifestations and mechanisms. Lancet Neurol 13 : 924-35, 201414. Clemow DB, Johnson KW, Hochstetler HM, Ossipov MH, Hake AM, Blumenfeld AM : Lasmiditan mechanism of ac-tion - review of a selective 5-HT1F agonist. J Headache Pain 21 : 71, 202015. Kakisaka Y, Ohara T, Katayama S, Suzuki T, Hino-Fukuyo N, Uematsu M, Kure S : Another case of lower back pain associated with migraine : The importance of specific ques-tions. J Child Neurol 28 : 680, 2013

Melena due to telangiectasia in migraine

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Melena due to telangiectasia in migraine

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