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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