Trigeminal Trophic Syndrome Research Articles

Trigeminal Trophic Syndrome With Herpetic Keratitis.

The aim of this study was to report a rare observation of trigeminal trophic syndrome presenting with concurrent herpetic keratitis. This study was a case report. A 42-year-old male with a remote history of a motor vehicle accident, traumatic brain injury, extensive facial reconstructive surgery, and traumatic optic neuropathy presented with left periorbital swelling. He was found to have extensive periorbital and total hypoesthesia in the cranial nerve V1 distribution and partial hypoesthesia in the V2 distribution. Corneal findings were highly suspicious for herpes simplex keratitis or zoster ophthalmicus. The patient was diagnosed with herpetic epithelial and stromal keratitis with periorbital ulceration secondary to Trigeminal trophic syndrome. The patient was treated broad spectrum intravenous antibiotics, intravenous acyclovir, and topical antibiotics. He was treated with topical steroids upon closure of epithelial defect. Upon discharge, the patient's vision improved from hand-motion to 20/30 at near. The patient was later fit with a custom thermoplastic facial mask to minimize further selfinjurious behavior. To our knowledge, this is the first report of trigeminal trophic syndrome presenting with concurrent herpetic keratitis. Successful treatment requires a multidisciplinary approach to provide the patient insight and create protective equipment.

A rare case of trigeminal trophic syndrome with periorbital cellulitis and full-thickness upper eyelid defect in an undiagnosed patient with human immunodeficiency virus: a case report

BackgroundTrigeminal trophic syndrome is a rare cranial and facial condition caused by damage to the central or peripheral branches of the trigeminal nerve. This syndrome consists of a triad of anesthesia, paresthesia, and crescent-shaped facial ulcer involving the ala nasi and sometimes extending to the upper lip. Although previous screening for human immunodeficiency virus in some patients with trigeminal trophic syndrome was negative, we present a unique case of trigeminal trophic syndrome who tested positive for human immunodeficiency virus with eye complications.Case presentationWe present a rare case of trigeminal trophic syndrome in a 44-year-old Black African woman who tested positive for human immunodeficiency virus. She presented with a 6-week history of progressive, persistent, and painless left sided facial and scalp ulcerations that started as small skin erosion. Diagnosis of trigeminal trophic syndrome was made on clinical grounds based on the triad of anesthesia, paresthesia, and unilateral crescent-shaped ulcer in the trigeminal dermatome and her past medical history. The ulcer healed completely after counseling and pharmacological therapy, but she later developed left periorbital cellulitis and left upper eyelid full-thickness defect.ConclusionThis is by far the first documented case of trigeminal trophic syndrome with a positive human immunodeficiency virus test. Testing for human immunodeficiency virus in patients with trigeminal trophic syndrome is necessary as this can help improve clinical management and treatment outcomes. Seeking the services of specialists remotely in resource constraint settings is beneficial for managing complications associated with trigeminal trophic syndrome.

A new meaning to 'picking your nose': the first use of N-acetylcysteine to successfully treat trigeminal trophicsyndrome.

Trigeminal trophic syndrome (TTS) is a rare neurological condition characterized by self-induced injury to the face in the distribution of the trigeminal nerve. Chronic scratching or picking cause ulceration, scarring, and tissue loss. TTS typically occurs following damage to the trigeminal nerve, often from surgical procedures, trauma, or stroke. We present a case of trigeminal trophic syndrome in an elderly lady who successfully responded to oral N-acetylcysteine. An increased awareness of this rare syndrome and potential treatment options will encourage a prompt diagnosis and possibly prevent permanent disfiguration.

P08 Shingles in two children with no history of chicken pox

Abstract We present two cases of dermatomal, vesicular eruptions in young children. Both were otherwise well with no history of immunocompromise or prior chickenpox (varicella). Child A was 3 years old and presented with painful erythema and pustules to the left side of his nose and cheek with a sharp midline demarcation. He was admitted by the paediatricians, unwell and treated with antibiotics and acyclovir for presumed impetigo or herpes simplex virus infection. However, the swab was positive for varicella zoster virus (VZV), which in this dermatomal distribution is in keeping with shingles. Child B was an 18-month-old girl who was referred with a dermatomal vesico-pustular eruption on the left forehead, scalp and nose unresponsive to antibiotics. Despite the lack of varicella history, shingles was suspected, and the child was started on aciclovir. Swabs were positive for VZV. Shingles is rare in children and usually associated with immunocompromise. It is especially rare in those with no prior history of chicken pox and of the cases in the literature, almost all have had previous vaccination to chicken pox resulting in latent infection and subsequent shingles. We surmise that our otherwise well patients, with no prior vaccination, had previous asymptomatic chicken pox. There was a delay in the diagnosis of patient A who went on to develop trigeminal trophic syndrome with recurrent paranasal ulceration over 18 months. We highlight these cases to make clinicians aware of the possibility of shingles even in very young, immunocompetent children with no prior history of chickenpox.

A RARE CAUSE OF TRIGEMINAL THROPHIC SYNDROME: ISCHEMIC CRANIAL NEUROPATHY

Trigeminal trophic syndrome (TTS) is a rare disease characterized by trigeminal anesthesia, facial paresthesia, and ulcerated lesions on the face as a result of damage to the trigeminal nerve due to different causes. Ulceration is most commonly seen as a single lesion on the nasal wing. Multiple ulcerated lesions are rarely encountered in the regions at trigeminal nerve dermatome. Here, a case of TTS presenting with multiple facial ulcerations after ischemic cranial neuropathy is reported.

PS12 A new meaning to ‘picking your nose’: the use of N-acetylcysteine to treat trigeminal trophic syndrome

Abstract Trigeminal trophic syndrome (TTS) is a rare condition in which patients induce unilateral facial ulceration by persistent physical manipulation. Peripheral or central damage to the trigeminal nerve causes anaesthesia, paraesthesia, formication or pruritus. Compulsive skin picking leads to ulceration of the ala nasi, cheek, scalp or mouth in the distribution of the trigeminal nerve. Patients with TTS are often subjected to extensive investigations and can be misdiagnosed as having a psychocutaneous disorder, such as dermatitis artefacta, skin-picking disorder or delusional parasitosis. We describe a 90-year-old woman who developed TTS following rhizotomy and radiofrequency treatment for trigeminal neuralgia. Underlying infectious, malignant and vasculitic causes for her ala nasi and cheek ulceration were ruled out through six incisional biopsies and two punch biopsies over 8 years. Previous treatment of TTS has included patient education and physical barriers (e.g. face masks and occlusive dressings). Treatment to date has been challenging, with trials of medications to address paraesthesia, including carbamazepine, gabapentin and amitriptyline. This patient re-presented to dermatology in 2022 with significantly worsened ulceration to her cheek and severe loss of nasal cartilage. In September 2022, the patient was started on N-acetylcysteine (NAC) 600 mg twice daily, and within 3 months, there was a considerable improvement. This drug is a mucolytic that has historically been used as an antidote for paracetamol overdose. It has antioxidant properties by replenishing glutathione and maintaining redox balance within cells. It also modulates neurotransmitters and can increase glutamate and alter dopamine levels. In addition, it has anti-inflammatory action by inhibiting interleukins (ILs), including IL-6 (Adil M, Amin SS. N-acetylcysteine in dermatology. Indian J Dermatol Venereol Leprol 2018; 84:652). In recent years, NAC has been reported to be successful in small case series and case reports among patients with trichotillomania, skin-picking disorders, onychotillomania, toxic epidermal necrolysis, alopecia, contact dermatitis, atopic dermatitis and wound healing (Grant JE, Chamberlain SR, Redden SA, Leppink EW. N-Acetylcysteine in the treatment of excoriation disorder. JAMA Psychiatry 2016; 73:490). It is an inexpensive drug that appears to be well tolerated and effective. To our knowledge, this is the first case report of the use of NAC in TTS. In summary, this was a case of TTS in a 90-year-old woman who successfully responded to NAC. The drug has antioxidant, anti-inflammatory and neurotransmitter modulatory effects. Reliable randomized controlled studies are needed to confirm the efficacy and optimum dose of NAC. Increased awareness of this rare syndrome and potential treatment options will ensure prompt diagnosis and prevention of permanent disfiguration.

Trigeminal trophic syndrome in the wake of a meningioma resection: A rare postoperative complication

Trigeminal trophic syndrome in the wake of a meningioma resection: A rare postoperative complication

Trigeminal trophic syndrome, a rare and often overlooked cause of facial ulceration: a case report and literature review

Trigeminal trophic syndrome (TTS) is an uncommon and relatively unknown cause of facial ulceration that occurs after damage to the trigeminal nerve. It characteristically involves non-healing facial ulceration(s) with accompanying anesthesia, paresthesia, and dysesthesia along the distribution of a trigeminal dermatome. The ulcerations are believed to be self-induced in response to paresthesia. The disease is most common in middle-aged women, manifesting as a unilateral crescent-shaped ulceration on the ala nasi, with sparing of the nasal tip. The diagnosis is clinical and mostly based on exclusion of other possible causes of facial ulcerations, with emphasis on neoplasms, infection-associated vasculitis, and factitial disorders. There are no specific histological or laboratory signs. There is no standard treatment protocol; however, a number of different successful strategies have been reported, including pharmaceutical and surgical interventions, transcutaneous nerve stimulation, and simple occlusion dressings. Due to the self-inflicted nature of this disorder, the cornerstone of management is patient education with behavioral modification. Here, we report a case of TTS following herpes zoster ophthalmicus and review the current literature on this subject.

Trigeminal Trophic Syndrome.

Trigeminal trophic syndrome (TTS) is a rare cause of facial ulceration resulting from self-manipulation of skin following peripheral or central injury to the trigeminal system. The average period between the trigeminal nerve injury and the onset of ulcer varies from several weeks to many years.[1] The syndrome is characterized by a triad of anesthesia, paresthesia, and a secondary persistent or recurrent facial ulceration. The common dermatological causes are Hansen’s disease and herpes zoster.[2,3] Timely recognition of TTS will promote its correct diagnosis and better management. The condition has been reported to be associated with Alzheimer’s disease and meningioma.[4] We describe TTS in a 70-year-old male who was presented with a persistent linear ulcer on the forehead and severe scarring involving the forehead, upper eyelid, and left nose [Figure 1], following an episode of herpes zoster affecting the ophthalmic division of the trigeminal nerve 1 year ago.Figure 1: Linear ulcer on the forehead and scarring involving the forehead upper eyelid and left side of the noseDeclaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.

Chronic Skin Picking: Trigeminal Trophic Syndrome in the Setting of Meningioma.

Chronic Skin Picking: Trigeminal Trophic Syndrome in the Setting of Meningioma.

Post-stroke corneal and facial ulcerations: trigeminal trophic syndrome.

Post-stroke corneal and facial ulcerations: trigeminal trophic syndrome.

Nasal Ulceration in Trigeminal Trophic Syndrome.

Facial Plastic Surgery & Aesthetic MedicineVol. 24, No. 6 ObservationNasal Ulceration in Trigeminal Trophic SyndromeTerrence K. Pleasant, Andrew Benvenuto, and Jeffrey S. MoyerTerrence K. PleasantDepartment of Otolaryngology- Head and Neck Surgery, University of Michigan Medical School, Ann Arbor, Michigan, USA.Search for more papers by this author, Andrew Benvenuto*Address correspondence to: Andrew Benvenuto, BS, University of Michigan Medical School, 1500 E Medical Center Drive, TC 1904, Ann Arbor, MI 48109-0624, USA, E-mail Address: abenvenu@med.umich.eduhttps://orcid.org/0000-0003-4663-2938University of Michigan Medical School, Ann Arbor, Michigan, USA.Search for more papers by this author, and Jeffrey S. Moyerhttps://orcid.org/0000-0002-0435-9200Department of Otolaryngology- Head and Neck Surgery, University of Michigan Medical School, Ann Arbor, Michigan, USA.Search for more papers by this authorPublished Online:8 Nov 2022https://doi.org/10.1089/fpsam.2021.0417AboutSectionsView articleView Full TextPDF/EPUB Permissions & CitationsPermissionsDownload CitationsTrack CitationsAdd to favorites Back To Publication ShareShare onFacebookTwitterLinked InRedditEmail View articleFiguresReferencesRelatedDetails Volume 24Issue 6Dec 2022 InformationCopyright 2022, American Academy of Facial Plastic and Reconstructive Surgery, Inc.To cite this article:Terrence K. Pleasant, Andrew Benvenuto, and Jeffrey S. Moyer.Nasal Ulceration in Trigeminal Trophic Syndrome.Facial Plastic Surgery & Aesthetic Medicine.Dec 2022.506-508.http://doi.org/10.1089/fpsam.2021.0417Published in Volume: 24 Issue 6: November 8, 2022Online Ahead of Print:May 12, 2022PDF download

A rare case of a folded nasolabial flap for a full-thickness alar defect due to trigeminal trophic syndrome after herpes zoster infection

A 51-year-old man was referred for a skin defect with paresthesia and decreased pain in his right ala. He had a history of herpes zoster infection 3 weeks previously. Excisional biopsy and culture showed no evidence of skin malignancy or infection. He was diagnosed with trigeminal trophic syndrome (TTS) after herpes zoster infection. A two-stage folded nasolabial flap was used to reconstruct the defect. The patient was satisfied with the cosmetic outcomes of surgery, and there was no evidence of recurrence during the follow-up. TTS, a rare skin condition that presents with paresthesia, anesthesia, and facial ulceration, occurs after trigeminal nerve injury, which can be caused by herpes zoster infection in rare cases. TTS is an often-persistent condition with a high recurrence rate, causing cosmetic problems. Some surgical methods have been attempted, but our case presents the first use of a folded nasolabial flap to treat TTS. The flap was symmetric, without any nostril collapse. It is also noteworthy that the outcome was excellent with only a skin flap that did not use a bony framework. Herein, we report a rare case of TTS that occurred after herpes zoster infection and was successfully treated with a folded nasolabial flap.

Scalp dysesthesia: a neuropathic phenomenon.

Scalp dysesthesia is an abnormal sensation of the scalp in the absence of cutaneous disease. It is characterized by a burning and/or itching sensation and can be related to a variety of neurogenic or psychogenic causes. This condition is extremely bothersome and is also common- especially among the geriatric population, in women, in patients with diabetes mellitus, and patients with psychiatric history. However, despite its prevalence in many populations, there are limited data about its causes and characteristics. Given its limited cutaneous manifestations, it is also easily misdiagnosed and an underrecognized cause of scalp pruritus in the dermatological community. Therefore, education on scalp dysesthesia is paramount to helping physicians identify and provide appropriate treatment for these patients. This review focuses predominantly on the neurogenic causes (with a brief review of psychogenic itch) of scalp dysesthesia and the therapeutics that have been found to be effective for this condition. Neurogenic causes of scalp dysesthesia occur with damage to the central or peripheral pathways of itch sensation, resulting in modification and heightened sensitivity of nerves that result in abnormal sensations in the absence of or out of proportion to external stimuli. A comprehensive review of etiologies is provided here, ranging from lesions to the central nervous system caused by cervical spine disease, trigeminal trophic syndrome, tumor, stroke, and multiple sclerosis, to small-fiber neuropathies caused by diabetes, brow lifts, keloid, and burn scarring. Recently, there have also been reports of scalp dysesthesias associated with post-infectious COVID-19. Treatment options tailored toward disease severity and different causes of disease will also be discussed. By elucidating the different mechanisms and therapeutic treatments of scalp dysesthesia, we hope to provide clinicians with the tools to identify and treat this condition as well as encourage further research into its etiologies and therapeutics.

Trigeminal Trophic Syndrome: A Comprehensive Review of a Surgical Approach.

Trigeminal trophic syndrome (TTS) is an exceedingly rare disease that causes facial ulceration, most commonly at the nasal ala. The overall incidence of TTS is not known, with less than 150 cases published in the literature.We searched the United States National Library of Medicine National Institutes of Health (PubMed) using the terms "Trigeminal Trophic Syndrome" and "TTS" as keywords. Publications in all languages were included if an English abstract was available. We reviewed 111 cases of TTS described in 75 publications from 1979 to 2021.Fourteen cases involved surgical reconstruction. Of these, the lesions completely healed in 7 cases (50%), recurred in 5 (36%), and were unspecified in 2 (14%). Reconstruction was completed in a 2 to 3-stage approach in 6 cases; of these, healing without recurrence was observed in 5 cases (83%). When donor tissue from the affected side was used as a basis for reconstruction, healing without recurrence was observed in 2 cases (50%). This is in comparison to the use of contralateral, sensate tissue in which there was healing without recurrence in all 3 cases (100%).The surgical management of TTS remains a topic of controversy. The rates of success remain comparable despite the use of various flap types. However, the use of contralateral, sensate flaps and a staged surgical approach appears to be effective based upon the best available evidence in the literature. Further prospective orretrospective controlled studies are necessary to make more reliable recommendations, though may be challenging given the rarity of TTS.

Postherpetic Trigeminal Trophic Syndrome: A Case Report

Trigeminal trophic syndrome (TTS) is a rare condition characterized by anesthesia, paresthesia, and facial ulceration involving the trigeminal dermatome secondary to self-manipulation of the skin after a peripheral or central injury to the trigeminal nerve or its branches. Differential diagnosis of TTS includes conditions presenting with chronic facial ulceration, such as various infectious diseases, malignancy, vasculitis, pyoderma gangrenosum and dermatitis artefacta. We report a case of postherpetic TTS and highlight the importance of early diagnosis and prompt treatment of this condition, which may commonly be misdiagnosed.

A Case of Anterior Nasal Atresia Caused by Trigeminal Trophic Syndrome after Herpes Zoster

A Case of Anterior Nasal Atresia Caused by Trigeminal Trophic Syndrome after Herpes Zoster

One-stage reconstruction of full-thickness alar defects with a folded nasolabial island flap.

BackgroundThe reconstruction of large full-thickness alar defects requires complex surgical procedures that are usually performed in two stages, with concomitant disadvantages in terms of patient trauma, surgical risk, and cost. This study presents a functional folded nasolabial island flap (FNIF) that can be used to repair large-sized full-thickness alar defects in a straightforward manner.MethodsThis retrospective study included seven patients who received a FNIF for a full-thickness alar defect between January 2007 and December 2020. The FNIF is different from the conventional nasolabial flap in that it is folded and twisted to achieve nostril reconstruction with a satisfactory three-dimensional mucosal lining in a single stage. The cosmetic and functional results of FNIF were evaluated by both patients and physicians.ResultsThe age ranged from 51 to 82 years (mean, 65.6 years). The causes of the defects were squamous cell carcinoma, basal cell carcinoma, and trigeminal trophic syndrome. The nostril lining did not collapse, there was no hypertrophic scarring, and air movement through the nostrils on the flap side was normal. Overall, FNIF produced excellent aesthetic and functional outcomes, with minimal patient discomfort. There were no postoperative complications.ConclusionCompared with existing reconstruction methods for large full-thickness alar defects, FNIF can easily achieve aesthetic and functional success in a single-stage procedure. It provides satisfactory results for both the patient and the surgeon.

Treatment of Trigeminal Trophic Syndrome using Scalp Based Flap Reconstruction: Case Report

Trigeminal trophic syndrome, or trigeminal neurotrophic ulceration, is an uncommon cause of persistent or recurrent facial ulceration, paresthesia, and anesthesia following self-manipulation of the skin, most commonly affecting the maxillary branch of the trigeminal nerve in the ala nasi region. We present a case of a 63-year-old with chronic ulceration of the right frontoparietal scalp involving the ophthalmic branch of the trigeminal nerve following herpes zoster infection. The patient was successfully treated with a fasciocutaneous transposition flap and splitthickness skin graft; these procedures allowed us to avoid the diseased ophthalmic nerve distribution. This patient’s postoperative course was uneventful, and the flap and skin graft healed without further ulceration of the involved area at 1 year follow-up.

Trigeminal trophic syndrome in a child – An unusual case report from Syria

The trigeminal trophic syndrome (TTS) is a rare form of cutaneous ulceration characterized by anesthesia, paresthesia and a secondary persistent facial ulceration. It is uncommon in children. However, we report an unusual case of TTS in a 5- year-old child with Moebius syndrome ,extending over the right nostril and then the other nostril was infected. The diagnosis of TTS was based on clinical history, tissue biopsy and serologic evaluation. The ulcer was treated with multiple antibiotics with no improvement. We describe this case because of very few pediatric cases of TTS published in the past , and to increase the awareness of this syndrome among dermatologists.