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Relief from cluster headaches following extraction of an ipsilateral infected tooth.

Abstract

A 60-year-old man with a 7-year history of cluster headaches was seen by an oral surgeon for evaluation of pain in the left upper second molar ipsilateral to the side affected by the headaches. During extraction of the tooth, infection, decay, and inflammation were discovered. Since the extraction in November 2008, the patient has experienced one episode of cluster headaches as of April 2013.

Introduction

Cluster headache is a primary trigeminal-autonomic cephalalgia that exhibits circadian and circannual periodicity. (1,2) The pain is unilateral and typically most severe in the retro-orbital and temporal areas of the affected side. (2) Cluster headache is frequently confused with migraine headache and sinus disease, which can lead to a significant diagnostic delay in many patients. (3,4) However, unlike migraine and sinus disease, cluster headache is characterized by unique timing and by abnormalities of both the sympathetic and parasympathetic divisions of the autonomic nervous system. (5)

The etiology of cluster headache is unknown. Its episodic consistency implicates a possible dysfunctional central pacemaker, (2) with primary defects of the central nervous system located in the regulating centers of the anterior hypothalamus. (6) An inflammatory mechanism has also been proposed, as a lesion in the ipsilateral cavernous sinus could explain many of the symptoms of cluster headache. (7)

There are few reports of tooth extraction resolving cluster headaches? In such cases, irritation of cranial nerves V and VII and the sympathetic postganglionic fibers of the superior cervical ganglion might have caused the symptoms associated with cluster headache. We report the case of a patient whose cluster headaches ceased after removal of the left upper second molar ipsilateral to the side affected by the headaches.

Case report

A 60-year-old man presented to an otolaryngologist in 1998 for evaluation and treatment of sinus headaches. His headaches typically lasted for 10 to 30 minutes, and they were accompanied by facial sweating and rhinorrhea. He described the pain as debilitating and of moderate severity. In 2000, he underwent endoscopic maxillary sinus surgery on the left side, and his headaches were alleviated for approximately 1 year.

From 2001 to 2006, the patient was treated by a general practitioner for what was diagnosed as sinus headaches. Magnetic resonance imaging (MRI) of his brain indicated nonspecific white-matter changes and inflammatory changes in the sinuses and fluid in the left mastoid air cells. The pain did not subside, and in May 2006 he was referred to a neurologist, who diagnosed him with cluster headache. He was treated with a prednisone regimen, and he was also given eletriptan for management of the acute attacks. The prednisone worked initially, but it lost its effect over a period of several months.

The patient described the pain as excruciating and of extreme severity. It manifested on the left side, primarily in the retro-orbital, zygomatic, and dental areas. Autonomic comorbidities included ptosis and blurred vision in the left eye, lacrimation, and facial sweating. Episodes occurred with seasonal regularity, beginning in November and ending in February; they were typically more common when the weather was dry, and they were most severe in the winter months. A typical episode would last 7 to 10 days, with 1 to 3 attacks per day, each lasting 45 to 180 minutes.

In January 2007, the patient stopped smoking. He had been a smoker since the age of 19 years. He experienced no change in cluster headache frequency after cessation.

In March 2007, the patient was seen by an oral surgeon for evaluation of bilateral upper third molar (# 1 and # 16) pain. The #1 molar had some sensitivity and decay, and the #16 molar had generalized periodontal recession. Osteitis and infection around # 16 were discovered upon extraction, and the root protruded into the maxillary sinus (figure 1).

In November 2008, the patient returned to the oral surgeon for evaluation of left upper second molar pain (#15) (figure 2). Inspection revealed signs of decay and periodontitis, and the tooth was subsequently extracted. Following the extraction, the patient did not experience another cluster headache episode until February 2011. This was the only episode he has experienced as of April 2013.

Discussion

Cluster headache remains difficult to diagnose and treat. In the absence of a precisely defined etiology, consideration of cases such as the present case may be beneficial in elucidating possible pathogenic mechanisms. As reports of tooth extraction leading to relief from cluster headaches are extremely rare, it was important that we reviewed the patient's entire history to confirm that he did indeed have cluster headaches and not another disease such as sinusitis, which can present with similar symptoms. Seasonal regularity and autonomic dysfunction during attacks are classic indicators of cluster headache. The duration and consistency of the attacks are also characteristic. (2,5)

It is interesting that prednisone initially provided some relief for our patient. However, this is not uncommon, and it can reinforce a false diagnosis of sinusitis. (1)

Factors that can rule out a diagnosis of migraine include unilateral pain and alack of photophobia or phonophobia during attacks, as well as the aforementioned seasonal regularity and autonomic dysfunction. Differences in episode duration and severity distinguished the cluster headaches in our patient from the sinus headaches that he had experienced previously.

Cluster headaches have multiple triggers. (9) Considering the notable decrease in cluster headache frequency in the present case--from yearly for 7 years to only once over 5 years--it appears that the infected tooth could have been an important trigger. There are several possible explanations for this patient's relief from cluster headaches following the tooth extraction. First, the relief could have been purely incidental. As the patient was 60 years old at the time of extraction and is now 65, his cluster headaches may simply be subsiding, as is common in patients between 65 and 70 years of age. (6) Alternately, the superior alveolar nerve could have been damaged during the tooth extraction; however, if this were the case, one would suspect a sensory deficit, and there was no discernible sensory deficit in this patient.

Lastly, extracting the tooth might have eliminated one trigger of the cluster headaches. Scorticati et al reported the case of a 34-year-old woman whose cluster headaches were relieved after a metallic foreign body was removed from her ipsilateral maxillary sinus. (10) In a case similar to ours, Romoli and Cudia described a 36-year-old man whose cluster headaches were relieved upon extraction of an impacted wisdom tooth# As in our case, that patient had experienced cluster headaches for years with no dental symptoms.

In the present case, dental signs that might have been seen on x-ray were not available because the patient had a highly sensitive gag reflex, which prevented routine x-rays from being taken at regular dentist visits. Also, it is interesting that the earlier extraction of the left upper third molar did not relieve his cluster headache. Even so, it is possible that the infection of the second molar might have provided the chronic inflammation necessary to sustain the cluster headache symptoms.

Pain of dental origin is poorly localized and highly variable, and it can simulate any painful syndrome. (11) Consequently, patients who undergo tooth extraction for facial pain or headache experience variable responses with little predictability. While a cause-and-effect relationship is difficult to prove, it is possible that irritation of the maxillary sinus in our patient might have led to irritation of the superior alveolar branch of the maxillary division of the trigeminal nerve. Takeshima et al reported on 2 patients who exhibited cluster headache symptoms secondary to paranasal sinusitis. (12) Acute or chronic sinusitis can irritate the afferent fibers of the trigeminal nerve, which can lead to pain. The trigeminal ganglion contains bipolar substance P neurons, (12-14) which innervate the maxillary sinus via the pterygopalatine nerve. Irritation of trigeminal substance P neurons in the maxillary sinus could result in symptoms of cluster headache associated with vasodilation. (15,16)

The response to steroid therapy observed in our case supports the idea that inflammation was present. Gawel et al demonstrated increased uptake in the cavernous sinus during gallium single-photon emission computed tomography. (17) It may be possible that the tooth extraction in our patient relieved the inflammation that had been caused by the infection of the ipsilateral second molar.

The present case illustrates the importance of continuing investigations into the mechanisms and triggers of cluster headaches. Although progress has been made in outlining the criteria for diagnosis, the possible etiologies and pathogenic mechanisms are not well defined, and the case we present here, while seemingly unusual, may be more common than previously thought.

In evaluating cluster headache, examining a patient's dental and sinus history and anatomy might offer insight into one possible, and surgically treatable, trigger. It is imperative that we emphasize the importance of dental inspection rather than dental extraction. In cases such as ours, where the molar was infected and decayed, extraction was warranted. However, experimental extraction is not recommended.

References

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(13.) Lundblad L, Lundberg JM, Brodin E, Anggard A. Origin and distribution of capsaicin-sensitive substance P-immunoreactive nerves in the nasal mucosa. Acta Otolaryngol 1983;96(5-6):485-93.

(14.) Uddman R, Maim L, Sundler E Substance P-containing nerve fibers in the nasal mucosa. Arch Otorhinolaryngol 1983;238(1):9-16.

(15.) Sicuteri F, Raino L, Geppetti P. Substance P and endogenous opioids: How and where they could play a role in cluster headache. Cephalalgia 1983;3(Suppl 1):143-5.

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Matthew R. Hoffman, PhD; Timothy M. McCulloch, MD

From the Division of Otolaryngology-Head and Neck Surgery, Department of Surgery, University of Wisconsin School of Medicine and Public Health, Madison.

Corresponding author: Timothy McCulloch, MD, Box 7375 Clinical Science Center-H4, 600 Highland Ave., Madison, WI 53792. Email: mccull@surgery.wisc.edu
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Title Annotation:ORIGINAL ARTICLE
Author:Hoffman, Matthew R.; McCulloch, Timothy M.
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Date:Jun 1, 2013
Words:1919
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