Herpes zoster-associated trigeminal trophic syndrome: a case report and review

Abstract

The case of a 75-year-old Caucasian woman with a trigeminal trophic syndrome (TTS) is presented and discussed. TTS of the ala nasi subsequent to a herpes zoster infection has not been described previously. We provide a review of the literature with insights into the pathogenesis and current therapeutic strategies. Keywords Trigeminal trophic syndrome · Ala nasi lesion · Herpes zoster infection · ReviewCase report A 75-year-old Caucasian woman was referred to our department with a right ala nasi lesion (Fig. 1). She had a history of breast carcinoma, which had been treated with surgery and radio-chemotherapy. Three years ago, herpes zoster affected the second division of the right trigeminal nerve. Subsequently, the patient developed postherpetic neuralgia, which was treated with carbamazepine. The initial anterior rhinoscopy showed a small mucosal fissure of the right nasal ala. Treatment consisted of local application of polymyxin-B ointment. Three months later, the patient returned with a distinct lesion, which expanded within weeks and was associated with recurrent bleeding. The following physical examination revealed a crescent lesion of about 2 cm in length with a concomitant loss of a significant portion of the right ala nasi and a complete anesthesia of the maxillary dermatome. The patient complained about numbness, paresthesia and the psychosocial impact of the lesion. We ruled out a malignant lesion with biopsies, and computed tomography was performed (Fig. 2). Two biopsies from the remaining ala and the lateral nasal margin confirmed the lack of malignancy. Histopathologically, the hyperkeratotic epidermis did not have any irregularities or signs of malignant transformation. Considering the patient's history of herpes zoster in the same right maxillary dermatome and the persistence of anesthesia, we assumed a trophic malfunction, e.g., trigeminal trophic syndrome (TTS). To our knowledge, TTS of the ala nasi has not been previously reported subsequent to a herpes zoster infection. After a thorough assessment and review of the literature, we decided to Eur Arch Otorhinolaryngol (2003) 260 : 86-90

Trigeminal trophic syndrome of all three nerve branches: an underrecognized complication after brain surgery

“…Other causes, such as infarctions of the brainstem, intracranial meningiomas, acoustic neuromas, astrocytomas, ophthalmic herpes zoster, syringobulbia, vertebrobasilar insufficiency, postencephalitic parkinsonism, and head trauma, are less common. 2,7,12,14,19,26 Idiopathic TTS has also been described. 19 Although psychiatric factors are important in factitious dermatitis, they are not considered important in TTS.…”

“…Trigeminal trophic syndrome is more common in women and the elderly; the average age of such patients is 57 years. 14,30 Self-induced trauma (chronic manipulation of sensationless skin) is believed to produce the tissue destruction; 7,28 therefore, the use of the term "trophic" is misleading. Patients often have paresthesias in the affected area, which prompt touching, rubbing, or picking to relieve the perceived irritation.…”

Trigeminal trophic syndrome

“…The differential diagnosis of the skin ulceration is given by Licschel, 5 and includes malignancies, vasculitites, infections, and insect bites. However, this patient best fits the description of TTS.…”

“…However, this patient best fits the description of TTS. There have been a number of case reports and reviews of the literature on TTS, 1–7 and Loveman 6 is generally credited for characterizing the disorder.…”

“…Pathology is conjectured as being self‐induced trauma secondary to paraesthesias causing patients to rub the affected area. Abnormal sympathetic tone has also been postulated, 2 in addition to abnormalities affecting nerve growth factors 5 …”