Intraoral Pain in Patients with Primary Intraosseous Cavernous Hemangiomas (PICHs)

Trigeminal Neuralgia (TN) is a condition associated with severe facial pain that could easily be associated with common dental issues. However, severe facial pain can also be the result of Primary Intraosseous Cavernous Hemangiomas (PICHs), a rare condition in which benign tumors made up of blood vessels grow in bones that make up the skull or spine. The pain can often be confused with dental issues; therefore, the overlap between dental pain and TN-related pain poses a significant clinical challenge, emphasizing the importance of comprehensive diagnostic and examination procedures that include physical examination, imaging studies, medical history, and electrophysiological testing. This study presents the case of a 74-year-old patient who was misdiagnosed with dental problems instead of TN. The misdiagnosis puts the patient through unnecessary painful dental procedures, which stresses the importance of medical practitioners using comprehensive diagnostic procedures.

Introduction

Lesions of the skull, particularly in the medullary space, such as primary intraosseous cavernous hemangiomas (PICHs), present a diverse range of clinical challenges in medicine. They are rare lesions, representing approximately 0.2% of all bone tumors and predominantly occurring in the parietal and frontal bones [1], [2]. PICHs are more prevalent among women in their second to fourth decades [2]–[4] and often grow slowly, leading to symptoms such as headaches or palpable masses. However, they can remain asymptomatic until they expand sufficiently to cause symptoms [3]. One notable consequence of PICHs is the development of neuropathic conditions, such as trigeminal neuralgia (TN). TN is a severe neuropathic disorder characterized by sudden, sharp facial pain, often resulting from nerve compression or irritation. The proximity of cavernous hemangiomas to cranial nerves, particularly in the parietal and sphenoid regions, can lead to compression and symptoms such as facial pain that might be misinterpreted as dental pain [5].

Studies have shown that PICHs can compress adjacent neurovascular structures, leading to symptoms such as numbness, weakness, and motor dysfunction [6]. Additionally, patients with PICHs may experience seizures, headaches, or cognitive impairment due to the potential impact on surrounding brain tissue.

The trigeminal nerve (TN) is one of the major cranial nerves (CN), and it has a special pattern of pain distribution. CN V originates from the brainstem, particularly the pons, and possesses sensory and motor components [7]. Three main branches innervate the facial sensation of the face, mouth, and jaw. Transmission of sensations like pain, temperature, and touch perceived from the face to the cranium is controlled by the Ophthalmic branch (V1), Maxillary branch (V2), and Mandibular branch (V3) [8]. Patients often experience chronic, sudden, and severe pain described as electrical shock-like impulses on the face, impeding daily functionality like speaking, eating, or brushing teeth [9]. Based on clinical observations, the most common association in patients with TN is seen with nerve compression, leading to a damaged myelin sheath.

The connection between trigeminal neuralgia and oral facial pain stems from the intertwined anatomy and its function in sensory information transmission. The pain episodes are frequently triggered by special stimuli, like eating, talking, or even gentle touch to the face. Based on extensive research, oral facial pain comes from abnormal electrical impulses or damage of the myelin sheet caused by demyelination along the afferent pathway [9]. With compression of the trigeminal nerve located at the root of entry, hyperactivity of the pain fibers is observed [10]. There is novel research on the altered connectivity and structuring of the central pain-related circuits that suggest a link between oral facial pain and TN due to the shared pathways. Since the trigeminal nerve controls the sensation of the face, chronic conditions like Multiple Sclerosis, nerve injuries, or Persistent Idiopathic Facial Pain (PIFP) can arbitrarily be confused with TN. Here, the same sensory processing and location are involved. This area, the trigeminal nucleus complex in the brainstem, serves as the relay station for the sensations carried back to the brain once encountered on the face [11].

Although TN epitomizes as a neuropathic cause of orofacial pain, there are other causes, like dental disorders, temporomandibular joint dysfunctions, and musculoskeletal issues, that can embody the pain-transmitting pathway as well. In clinical practice, TN is diagnosed as neurovascular compression, where secondary causes are complicated by inflammation, tumors, or trauma [7]. A wide range of treatments are deemed as potential therapeutic options. These include anticonvulsant medications, nerve blocks, or, in severe cases, surgical interventions to help alleviate nerve compression. Furthermore, peripheral nerve ablation, focused radiation, and decompression of the nerves provide a high rate of relief with varying adverse outcomes [7].

The overlap between TN and dental pain is a significant clinical challenge. Neuropathic dental pain triggered by TN can complicate diagnosis and treatment, leading to diagnostic confusion with odontogenic pain. Furthermore, the similarities in anatomical distribution between TN-related pain and dental pain can make it challenging for clinicians to distinguish between these two conditions, potentially leading to misdiagnosis or delayed diagnosis [12].

The relationship between PICHs, TN, and dental pain is multifaceted and warrants further investigation. Clinically, patients with PICHs and TN may present a range of symptoms, including facial pain, numbness, and tingling [13]. The primary imaging modality for TN diagnosis is Magnetic Resonance Imaging (MRI), surpassing computer tomography (CT) as the resolution of MRI is superior to that of CT scans [14]. A more contemporary imaging method called magnetic resonance angiography, or MRA makes it possible to see the relevant region’s vascular structure without the need for contrast agents [14]. MRAs can help identify the presence and extent of these lesions but may not always detect the underlying neural compression that contributes to TN symptoms.

Studies have shown that PICHs can compress adjacent neurovascular structures, leading to symptoms such as numbness, weakness, and motor dysfunction [6]. Additionally, patients with PICHs may experience seizures, headaches, or cognitive impairment due to the potential impact on surrounding brain tissue.

Understanding the relationship between PICHs, TN, and dental pain is critical to ensure accurate diagnosis and effective treatment. This requires a multidisciplinary approach involving clinicians from various specialties, including neurology, dentistry, and radiology. A comprehensive diagnostic workup should include a thorough medical history, physical examination, imaging studies, and, potentially, electrophysiological testing. Treatment options may include pain management strategies, such as medication or injections, surgical decompression of the nerve, or referral to a specialist for further evaluation and management.

Case Report

A 74-year-old female patient presented to the orofacial pain clinic due to persistent dental and facial pain while chewing and talking. The intraoral pain on the left side of the oral cavity began one year before she arrived at the clinic. Following a history of multiple dental treatments, the initial pain was localized to tooth number 17 (lower third molar), prompting its extraction due to severe pain and suspected dental caries. Subsequent treatment with root canal therapy for tooth number 18 (lower left second molar) temporarily alleviated her symptoms; however, the pain recurred after extensive RCT treatment. As a result, tooth number 19 (lower left first molar) was also extracted due to persistent pain.

Given the persistence of pain despite dental interventions, the treating dentist referred the patient to our clinic for further evaluation. Upon examination, the patient described sharp pains lasting only a few seconds to minute, localized to the left side of her face and lower posterior teeth area (molars) and, with occasional pain referred to the upper left molar region as well. The comprehensive examination revealed pain originating from the masseter muscle, primarily affecting tooth number 15 (upper left first molar) with the retromolar area. However, the patient’s pain was distinct from its initial presentation, characterized by a more electric shock-like quality. The trigeminal nerve examination did not replicate the original electrical shocking pain on the lower posterior quadrant during the first examination.

Based on clinical findings, medical/dental history, and patient’s history of the pain, duration of the pain, and characteristic of the pain history, the patient was diagnosed with Trigeminal nerve disorder, unspecified (possible TN), and myofascial pain (masticatory).

The patient underwent MRI/MRA imaging at the referral neurologist’s office to aid in diagnosis. The results of the MRI showed a “circumscribed 9 mm lesion in the medullary space of the left parietal bone–can represent hemangioma.”

After reviewing the MRI results with the patient, treatment options and next steps were discussed. The patient was subsequently referred back to the neurologist for further evaluation. A subsequent MRA scan led to the diagnosis of Trigeminal neuralgia. As part of treatment, the patient was referred to a neurosurgeon, and based on the patient’s condition, they decided that the patient would begin taking Carbamazepine 100 mg twice daily. However, due to increased pain, the neurologist decided to add the following:

1. Carbamazepine 100 mg 12-hour Tablet extended release

• 2 pills in the morning

• 2 pills at night

2. Duloxetine 60 mg DR Capsule

• 1 pill in the afternoon

3. Pregabalin 75 mg Capsule extended release

• 1 pill in the morning

• 1 pill at night.

Our clinic followed up with the patient on multiple occasions to ensure her pain levels were manageable and that she remained under the care of her neurologist, and in case of persistent pain, she would go back to the neurosurgeon for surgical options.

Conclusion

In conclusion, the relationship between primary intraosseous cavernous hemangiomas (PICHs), trigeminal neuralgia (TN), and dental pain is complex and multifaceted. PICHs are rare bone tumors that can compress adjacent neurovascular structures, leading to symptoms such as numbness, weakness, and motor dysfunction. In some cases, these lesions can cause neuropathic conditions like TN, characterized by severe facial pain, often triggered by special stimuli such as eating or talking.

The overlap between TN-related pain and dental pain is a significant clinical challenge, as both conditions share similar anatomical distributions and sensory processing pathways. Clinicians must be aware of this overlap to avoid misdiagnosis or delayed diagnosis, which can lead to inadequate treatment and poor patient outcomes.

A comprehensive diagnostic workup involving medical history, physical examination, imaging studies, and electrophysiological testing is essential for accurate diagnosis and effective treatment. Treatment options may include pain management strategies, such as medication or injections, surgical decompression of the nerve, or referral to a specialist for further evaluation and management.

In this case report, a 74-year-old female patient was diagnosed with Trigeminal neuralgia following imaging studies that revealed a PICH compressing adjacent neurovascular structures. The patient’s pain levels were managed with Carbamazepine, highlighting the importance of prompt diagnosis and effective treatment to alleviate suffering and improve quality of life.

Future research should focus on understanding the relationship between PICHs, TN, and dental pain in more detail, including the mechanisms underlying this complex interplay. A multidisciplinary approach involving clinicians from various specialities is essential for ensuring accurate diagnosis and effective treatment, and further investigation into the clinical presentation and management of these conditions will be crucial for improving patient outcomes.