Trigeminal Neurotrophic Nasal Ulcers after Resection of Large Cerebellopontine Angle Tumors: A Case Series

Introduction: Trigeminal trophic syndrome is a rare sequela of trigeminal nerve injury that is characterized by ulceration, anesthesia, and paresthesia in the distribution of the trigeminal dermatome. Ulceration is often intractable and is thought to occur due to persistent manipulation in response to paresthesias. Most commonly affected areas, in descending order, are the nasal ala, cheek, and the cornea. Prevalence is rare with reportedly approximately 200 cases having been described in literature. Diagnosis is clinical and it is imperative to exclude other causes of ulceration, such as cutaneous malignancies. Trigeminal hypoesthesia is reported to be present in cases of large compressive acoustic neuroma (stage-4 or -5 Zini-Magnan classification) in about half of cases and in similar rates after resection. In this case series, we present two patients with development of trigeminal trophic syndrome after resection of large cerebellopontine angle tumors.

Patient 1 is a 45-year-old Caucasian male who presented with a large cystic left cerebellopontine angle (CPA) mass consistent with an acoustic neuroma ([Fig. 1]). The tumor was resected via retrosigmoid approach. Patient developed an immediate HB 6/6 left facial nerve palsy and a left abducens nerve palsy. He also developed delayed left trigeminal nerve anesthesia and paresthesia. At his 10-month postoperative visit, he was noted to have developed a full-thickness left nasal ala defect at the vestibule ([Fig. 2]). Biopsies of the ala defect were obtained and were negative. The defect is currently being managed with topical therapy.

Patient 2 is a 52-year-old Caucasian male who underwent resection of a large right-sided CPA meningioma resection. He had multiple cranial nerve palsies as a sequela of his surgery (including trigeminal, facial, vestibulocochlear, and vagus). He developed a superficial right lateral nasal ala ulcer 4 months postoperatively ([Fig. 3]). This was biopsied and returned benign as ulceration with fibrosis and reactive changes. Patient did note incessant self-inflicted trauma to the area with tissues and his fingers. Patient was treated nonsurgically with education and topical therapy. The lesion resolved and was fully healed at his 3-year postoperative follow-up.

Conclusion: Trigeminal neurotrophic syndrome (TTS) is a rare sequela of trigeminal nerve injury. Although trigeminal hypoesthesia is fairly common in association with large CPA tumors, the incidence of TTS is rare. TTS may be more likely in patients with increasing density of facial numbness. Both of the patients presented in this case series has significant trigeminal anesthesia. This diagnosis should be considered in patients with facial ulceration after large posterior cranial fossa tumor resection, especially in those with dense trigeminal numbness. Time of onset and severity of ulceration can be variable. Other causes of facial ulceration, such as cutaneous malignancy, should be ruled out prior to making this diagnosis.

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Artikel online veröffentlicht:
12. Februar 2021