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Phantosmia and Migraine with and Without Headache

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Phantosmia and Migraine with and Without Headache ISSN 0017-8748 Headache doi: 10.1111/head.12890 VC 2016 American Headache Society Published by Wiley Periodicals, Inc. Expert Opinions Phantosmia and Migraine With and Without Headache Yasmin I. Jion, MD; Brian M. Grosberg, MD; Randolph W. Evans, MD Phantosmia is a rare migraine aura. We present two cases of phantosmias occurring before migraine headaches and also without headaches. To our knowledge, these are the third and fourth cases of phantosmias ever reported due to migraine aura without headache. Key words: phantosmia, olfactory hallucination, migraine, headache (Headache 2016;00:00-00) CASE HISTORIES take acetaminophen with relief in 1–2 hours. Stress Case 1.—This is a 53-year-old female with a 9- was a trigger. month history of smelling a dirty dog smell as often as She saw a cardiologist about 1 month after 2–3 times a day or up to 4 days without the smell with onset and had a CT of the brain without contrast an average duration of 5 minutes (range 30 seconds to on 1/19/15 that was normal. She saw two ENT phy- 1 hour) followed by a headache about every 2 weeks sicians who found normal exams. but most of the time without an associated headache. Past medical history of hyperlipidemia on prav- She described a bifrontal throbbing headache with an astatin. Family history: sister has migraine. Neuro- intensity of 6/10 associated with nausea, light and noise logical examination was normal. sensitivity but no vomiting. She always takes ibuprofen MRI of the brain with and without contrast was with relief in 1 hour. During the episodes, there is no normal. EEG was normal. A complete blood count, alteration of consciousness. She has no triggers. She chemistry profile, thyroid functions, erythrocyte sedi- denied depression, anxiety, or increased stress. mentation rate, antinuclear antibody, rheumatoid Prior to 9 months ago, she had occasional head- arthritis factor, and Sjogren’s antibodies were negative. aches since her 20s described as bitemporal aching She declined a trial of migraine preventive with an intensity of 7/10 associated with light and medication. noise sensitivity but no nausea or aura. She would Case 2.—This is a 69-year-old female with a his- tory of headaches since age 35 that have been occurring about 6 days per month the prior 12 weeks since starting onabotulinum toxin A 2 years From the National Neuroscience Institute, Singapore (Y.I. Jion); Hartford Healthcare Headache Center, Wethersfield, previously and before about 4–6 days per week CT, USA (B.M. Grosberg); Department of Neurology, Bay- since age 45–50 years. She described a top of the lor College of Medicine, Houston, TX, USA (R.W. Evans). head and/or then left or right sided throbbing with Address all correspondence to Y.I. Jion, National Neurosci- an intensity of 7–10/10 associated with nausea, light ence Institute, 11 Jalan Tan Tock Seng, 308433 Singapore, and noise sensitivity but no vomiting or visual, email [email protected] Accepted for publication June 21, 2016. Conflict of Interest: None. 1 2 Month 2016 sensory, or language aura. She took naratriptan chronic migraine with onabotulinum toxin A with relief in about 3–4 hours. Change of weather injections. was a trigger. Alterations in the sense of smell can be broadly For the past 20 years, once every 1–2 weeks, classified into quantitative dysfunction (hyperosmia, she would smell something burning like a campfire hyposmia, and anosmia) and qualitative dysfunction 90% of the time and like burning rubber 10% of (parosmia and phantosmia).1,2 A distortion of the the time lasting 15 minutes to 2 hours. Three times, perceived odor is termed parosmia or troposmia, first about 18 years ago and last about 1 month while the perception of an odor when there is no ago, she had a menthol smell lasting about 1 hour odorant stimulus present in the environment is to 2 days. The smell would precede a typical head- termed phantosmia, cacosmia, or olfactory halluci- ache 1 hour to a few hours later. She also reported nation.3,4 Phantosmia typically lasts longer than a occasional episodes of the abnormal smell occurring few seconds, while olfactory hallucination usually without a headache. Since starting onabotulinum lasts only a few seconds.4,5 toxin A injections, the frequency of olfactory hallu- The occurrence of abnormal perception of odor cinations decreased to every few months. When she has long been described in ancient times as early as went 10 months between injections, the hallucina- 131 A.D. by Aretaeus, a Cappodocian: “a heavy smell tions increased after 5 months to about twice a sometimes preceded the accession of a paroxysm.”6 month. In migraine, the majority of patients have normal Propranolol, venlafaxine, divalproex, and verap- olfactory function.7 During a migraine episode, olfac- amil were not effective for prevention. MRI of the tory acuity may be impaired with a minority exhibit- brain in 2015 showed non-specific white matter ing microsmia or hyposmia during acute attacks and abnormalities. decreased olfactory sensitivity.7,8 However, a height- Past medical history of hyperlidemia and fibro- ened sense of smell/olfactory hypersensitivity has also myalgia. Recent nuclear cardiac stress test negative. been described during both a migraine attack and Neurological examination was normal. between attacks in up to 46 and 35%, respectively.9–11 Questions. What is the diagnosis? How com- These patients tend to have greater frequency of mon is this disorder in adults and children? What migraines, odor-induced migraines and visual hyper- might be the mechanism? How do you distinguish sensitivity. About 50% of migraineurs report that phantosmias due to migraine from other disorders? odors can also trigger their migraine attacks and olfac- What is the treatment? tory hallucinations sometimes develop during What Is the Diagnosis?.—Case 1 presents with a migraine.9,12–15 There is suggestion of the role of the phantosmia of a dirty dog lasting 30 seconds to 1 piriform cortex and antero-superior temporal gyrus in hour with an average of 5 minutes sometimes fol- olfactory hypersensitivity in migraine, as well as dys- lowed by a migraine headache and often occurring function in central olfactory processing.8,16,17 without a headache. MRI of the brain and EEG The description of phantosmia in both cases were normal. meets the criteria of aura, 5–60 minutes, followed Case 2 presents with a 34 year history of by headache and is consistent with similar case migraine without aura and a 20 year history of a series and not consistent with epilepsy as discussed burning smell lasting 15–120 minutes and rarely a below.18 Case 2 did have some episodes of phantos- menthol smell before a migraine headache. She mia lasting more than 1 hour which is not uncom- also reported occasional episodes of the burning mon for various migraine auras and phantosmia smell occurring without a headache. MRI of the (discussed below).19 Interestingly, the International brain showed only non-specific white matter abnor- Classification of Headache Disorders (ICHD) does malities. The frequency of the episodes of phantos- not yet recognize the presence of olfactory aura as mias significantly decreased after treatment of a migrainous aura.20 Headache 3 How Common Is This Disorder in Adults and interestingly found to predict the development of Children?.—Phantosmia has been found to occur in clinical psychosis at a 10-year reevaluation.33 Olfac- 6% of community dwelling adults and up to 12% in tory hallucination has also been described in a tertiary otolaryngology clinic.21,22 The majority depression (19–33%) and in up to 10% of non- had concomitant anosmia/hyposmia, and most demented Parkinson’s disease patients, though phantosmias presented with no history of upper prevalence is less (5%) in those without accompa- respiratory tract infection, head trauma or aging nying visual and auditory hallucination.35,36 In tem- (idiopathic).1,4,22 A typical history begins in a wom- poral lobe epilepsy, estimated prevalence ranges an between the ages of 15–30 years old, who no- from 0.6 to 30%.35,37–40 A higher prevalence has tices odor perception that is not appreciated by been reported in those with head injury and those others. Odors last 5–20 minutes each time and with decreased smell and taste acuity in up to resolve spontaneously. Recurrent episodes may 40–60% of patients.35 occur more frequently and may last longer subse- What Might Be the Mechanism?.—The mecha- quently. They may be perceived arising from one nism behind phantosmia has not been clearly eluci- or both nostrils, and resolve with sleep, Valsalva dated. Theories of abnormal peripheral as well as maneuver, or occlusion or instrumentation of the central mechanisms have been postulated.2,4,5 nostril.4 Proponents of the peripheral theory highlight The first case of olfactory hallucination in phantosmia being worse in the nostril with the migraine was described in 1982.12 The late Oliver poorer olfactory ability, the fact that phantosmia can Sacks also reported seeing several migraine patients be eliminated by occluding air flow and anesthetiz- with hallucinations of smell associated with forced ing the olfactory mucosa in the affected nostril. His- reminiscence and feelings of deja vu.23 Since then, topathological studies also demonstrated decreased multiple case reports have highlighted this phenome- number of neurons, greater ratio of immature to non with migraines.13,20,24–27 Estimated prevalence mature neurons and disordered growth of olfactory ranged from 0.1% in consecutive migraine
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