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Self-Trephination in Cranial Excoriation Disorder. BACKGROUND AND IMPORTANCE: Trephination is a procedure in which a small hole is made in the skull. Rare cases of self-trephination by individuals seeking medical benefit have been reported. Excoriation disorder is a compulsive skin-picking condition in which an individual self-inflicts cutaneous lesions. Left untreated, severe excoriation disorder can pose significant health risks. CLINICAL PRESENTATION: Here, we describe 5 patients who presented with self-trephination due to a severe form of compulsive cranial excoriation at 2 neighboring academic medical centers over a 4-year period. We review the clinical presentation of self-trephination in cranial excoriation disorder and associated risk factors, surgical and nonsurgical interventions, complications of the disease, treatments, and mortality. Defining clinical characteristics include repetitive self-induced destruction of the scalp and skull with entry into the intracranial compartment, frequent psychiatric comorbidities, infection or injury of the brain with consequent neurological morbidity or mortality, and frequent treatment failures because of poor adherence. CONCLUSION: Self-trephination in cranial excoriation disorder is a severe neuropsychological disorder and neurosurgical emergency that exposes the brain and is often life-threatening. Appropriate therapy requires antibiotics, surgical debridement and repair of the wound, and concomitant effective psychiatric management of the underlying compulsion, including the use of antidepressants and behavioral therapy.

A 47-year-old female patient with a history of anxiety, depression, post-traumatic stress disorder, and remote intravenous drug use presented with headaches, vision changes, and photophobia. She reported finding “chunks of meat” on her pillow and having her “scalp peel off” while combing her hair. She admitted to skin picking, sometimes using tweezers. Examination revealed a 10 × 7 cm scalp wound with a skull defect and exposed brain (Figure 1A and 1B). She was admitted to the hospital, and broad-spectrum antibiotics were administered. She underwent scalp excision with bilateral craniectomy and duraplasty, followed by latissimus free flap reconstruction. Cultures grew Staphylococcus epidermidis and Corynebacterium. She was evaluated by psychiatry and prescribed risperidone, trazodone, hydroxyzine, and fluoxetine. Psychiatry deemed her to have medical decision-making capacity. She declined inpatient rehabilitation and was discharged home.

simus free flap reconstruction. Cultures grew Staphylococcus epidermidis and Corynebacterium. She was evaluated by psychiatry and prescribed risperidone, trazodone, hydroxyzine, and fluoxetine. Psychiatry deemed her to have medical decision-making capacity. She declined inpatient rehabilitation and was discharged home. Clinical and radiographic images for patients 1 and 2. A, Photograph of self-inflicted frontal calvarial excoriation measuring 10 cm × 7 cm. B, Sagittal noncontrast T1-weighted MRI at initial presentation demonstrating a soft tissue wound with exposed calvarium and a small vertex skull defect. C, After scalp reconstruction, Patient 1 re-presented with flap decimation and scalp wound breakdown with exposed brain. D, Sagittal noncontrast T1-weighted MRI reveals exposed brain and frontal lobe cerebritis. E, Two months later, patient 1 returned with sepsis and a re-excoriated scalp wound with exposed brain, purulent drainage and maggots in situ. F, Sagittal noncontrast T1-weighted MRI demonstrating pneumocephalus, scattered blood products and cerebral edema consistent with extensive cerebritis. G, Photograph of patient 2 with self-inflicted vertex frontal scalp wound on initial presentation. H, Coronal T1-weighted MRI with gadolinium demonstrating a soft tissue wound with a calvarial defect and magnetic susceptibility artifact in the superior right frontal lobe. I, One year after being lost to follow-up, patient 2 re-presented with expansion of the frontal cranial wound. J, Coronal T1-weighted MRI with gadolinium revealing a calvarial defect with development of an underlying contrast-enhancing parenchymal lesion. K, Intraoperative photograph of the surgical repair showing scalp debridement and reconstruction with a rotational flap and allograft. L, Photograph of patient 2 at 2 months postoperatively showing a well healed incision.

ealing a calvarial defect with development of an underlying contrast-enhancing parenchymal lesion. K, Intraoperative photograph of the surgical repair showing scalp debridement and reconstruction with a rotational flap and allograft. L, Photograph of patient 2 at 2 months postoperatively showing a well healed incision. Five weeks postoperatively, the patient was referred to the emergency department by a visiting nurse who observed her picking at the surgical site. On examination, she was somnolent but arousable. She complained of several days of worsening headaches and dizziness. Assessment showed new wound breakdown with exposed brain and cerebrospinal fluid leakage (Figure 1C). Imaging demonstrated multifocal intraparenchymal hemorrhage with gas in the frontal lobe, consistent with cerebritis (Figure 1D). She was given antibiotics, soft restraints, and a helmet to prevent further excoriation. She underwent wound revision with fascia lata autograft and synthetic bilayer wound matrix. Postoperatively, guardianship was pursued but dismissed in court, and she was again discharged home. One month after discharge, she presented with recurrent falls, altered mental status, and an enlarged scalp wound. Imaging revealed frontal lobe encephalomalacia without new mass or hemorrhage. She was admitted and placed on remote video monitoring because of persistent attempts to pick at her wound. She was transferred to psychiatry for management of the excoriation disorder and discharged home 3 weeks later.

enlarged scalp wound. Imaging revealed frontal lobe encephalomalacia without new mass or hemorrhage. She was admitted and placed on remote video monitoring because of persistent attempts to pick at her wound. She was transferred to psychiatry for management of the excoriation disorder and discharged home 3 weeks later. Two months later, she presented to the emergency department after she was found down in a large amount of dried blood. She was hypotensive, hypoxic, and hypothermic. On examination, she was obtunded and found to have a 10 × 6 cm vertex scalp wound with exposed brain, maggots in situ, and malodorous purulent drainage (Figure 1E). A brain MRI showed vasogenic edema, pneumocephalus, and blood products bilaterally, consistent with cerebritis (Figure 1F). Because of the extent of infection, her poor neurological function, and history of multiple failed attempts to manage the wound surgically, further surgical management was felt to be futile. She was admitted for medical management of sepsis and diffuse cerebritis. Three days later, the patient lost all brainstem reflexes and expired. A 61-year-old male patient with a history of bipolar disorder and remote head trauma presented with headaches, tinnitus, vision changes, and a bifrontal scalp wound exceeding 5 cm (Figure 1G). He reported headache relief after manipulating the wound with tools (either a clothes pin or drill bit) and eliciting clear drainage from the wound. Physical examination was otherwise unremarkable. A brain MRI demonstrated an underlying calvarial defect with intracranial extension (Figure 1H).

ceeding 5 cm (Figure 1G). He reported headache relief after manipulating the wound with tools (either a clothes pin or drill bit) and eliciting clear drainage from the wound. Physical examination was otherwise unremarkable. A brain MRI demonstrated an underlying calvarial defect with intracranial extension (Figure 1H). Recommendations were made for lumbar puncture and consultations with psychiatry and plastic surgery. The patient reported immediate relief of head “pressure” after lumbar puncture. He began behavioral therapy but did not follow-up with plastic surgery. One year later, he returned with speech difficulty, behavioral changes, and an enlarged calvarial defect (Figure 1I). A brain MRI revealed a new contrast-enhancing lesion underneath the cranial defect, concerning for cerebral abscess (Figure 1J). He was admitted for antibiotic therapy. Several days later, he underwent scalp debridement, allograft placement, and advancement of a rotational flap (Figure 1K and 1L). He was discharged and noted at follow-up to be enrolled in behavioral therapy with a well-healed wound.

, concerning for cerebral abscess (Figure 1J). He was admitted for antibiotic therapy. Several days later, he underwent scalp debridement, allograft placement, and advancement of a rotational flap (Figure 1K and 1L). He was discharged and noted at follow-up to be enrolled in behavioral therapy with a well-healed wound. A 26-year-old female patient with a history of dermatillomania, chronic skull osteomyelitis, and active substance use disorder (cannabinoids, cocaine, and opiates) presented with headaches, vomiting, fevers, photophobia, and hyperacusis. The patient had previously undergone a scalp flap closure procedure one year prior, but did not follow-up. On examination, she had a large scalp and cranial defect with scattered brain hemorrhages and an associated encephalocele (Figure 2A). She was neurologically intact. A brain MRI showed cerebritis, a subdural fluid collection concerning for empyema, and superior sagittal sinus thrombosis (Figure 2B).

did not follow-up. On examination, she had a large scalp and cranial defect with scattered brain hemorrhages and an associated encephalocele (Figure 2A). She was neurologically intact. A brain MRI showed cerebritis, a subdural fluid collection concerning for empyema, and superior sagittal sinus thrombosis (Figure 2B). Clinical and radiographic images for patients 3, 4, and 5. A, Photograph of patient 3 presenting with an encephalocele herniating through a self-inflicted cranial excoriation wound with an associated calvarial defect. B, Coronal T1-weighted MRI with contrast demonstrating a bilateral scalp wound with bilateral skull defects and a left frontal herniating encephalocele. C, Photograph of cranial wound with associated encephalocele after 6 weeks of antibiotic therapy but before surgical repair. D, Photograph of cranial wound at follow-up visit for suture removal after reconstructive surgery showing rotational flap and skin graft. The patient's wound appeared to be healing well. E, Photograph of patient 4 at presentation with a small parietal scalp wound with yellow crusting. F, Sagittal T1-weighted MRI with contrast demonstrating a peripherally contrast-enhancing left anterior parietal lesion concerning for cerebral abscess. G, Emergent computed tomography of the head with contrast after the patient had a seizure and developed hemiparesis before surgical intervention. H, Intraoperative photograph of patient 4 showing pus upon opening the abscess during surgical evacuation. I, Photograph of patient 5 at presentation demonstrating an open gangrenous skull defect underlying an unhealed scalp ulcer. J, Coronal T1-weighted MRI with contrast demonstrating focal pachymeningeal enhancement and thickening underlying the scalp wound. K, Axial T2-weighted MRI 1 week later demonstrating extensive T2 hyperintensity in the adjacent left temporal lobe, concerning for cerebritis. L, Photograph of patient 5 at 2 months postoperatively showing a well-healed incision.

demonstrating focal pachymeningeal enhancement and thickening underlying the scalp wound. K, Axial T2-weighted MRI 1 week later demonstrating extensive T2 hyperintensity in the adjacent left temporal lobe, concerning for cerebritis. L, Photograph of patient 5 at 2 months postoperatively showing a well-healed incision. She was treated with 6 weeks of inpatient antibiotics for osteomyelitis and intracranial infection. Throughout the admission, the patient picked at the encephalocele. The subdural empyema resolved, and her scalp began to heal (Figure 2C). After resolution of the infection, she was taken for wound debridement, duraplasty, and a rotational scalp flap. A cranioplasty was placed after healing of the rotational flap. Intraoperative cultures were positive for Gram-positive rods. She remained in the hospital for another 2 weeks for antibiotic therapy. Psychiatry medically managed the excoriation disorder throughout the hospitalization. At a subsequent clinic visit, her wound was well-healed (Figure 2D).

ealing of the rotational flap. Intraoperative cultures were positive for Gram-positive rods. She remained in the hospital for another 2 weeks for antibiotic therapy. Psychiatry medically managed the excoriation disorder throughout the hospitalization. At a subsequent clinic visit, her wound was well-healed (Figure 2D). A 45-year-old female patient with a history of active polysubstance abuse (cocaine, alcohol, and cannabis), depression, anxiety, hypertension, and chronic hepatitis C presented with fevers, chills, headaches, and a generalized seizure. She was neurologically intact but had a 1-cm scalp lesion (Figure 2E) caused by picking at her skin because of the recent stress of her son's death. A brain MRI revealed a peripherally enhancing lesion with diffusion restriction and edema in the postcentral gyrus underlying the scalp ulceration and calvarial defect, concerning for cerebral abscess (Figure 2F).

cm scalp lesion (Figure 2E) caused by picking at her skin because of the recent stress of her son's death. A brain MRI revealed a peripherally enhancing lesion with diffusion restriction and edema in the postcentral gyrus underlying the scalp ulceration and calvarial defect, concerning for cerebral abscess (Figure 2F). Consultations with neurosurgery, plastic surgery, and psychiatry were obtained and surgery was planned. Before surgery, she suffered another seizure and developed right hemiplegia. A head computed tomography demonstrated abscess rupture (Figure 2G). She was taken emergently for a craniotomy and abscess evacuation (Figure 2H). The scalp was debrided, and the skull defect was closed with titanium mesh, followed by primary scalp closure. Cultures grew Staphylococcus aureus, Actinomyces odontolyticus, Peptostreptococcus micros, Staphylococcus hominis, Streptococcus constellatus, and Group A Streptococcus. She was treated with intravenous antibiotics, started on fluoxetine, and discharged to a rehabilitation center. Four months postoperatively, her motor function was improved. However, she resumed picking at the surgical site. She continued with psychiatric care and showed behavioral improvement.

d Group A Streptococcus. She was treated with intravenous antibiotics, started on fluoxetine, and discharged to a rehabilitation center. Four months postoperatively, her motor function was improved. However, she resumed picking at the surgical site. She continued with psychiatric care and showed behavioral improvement. A 51-year-old female patient with a history of multiple skin ulcers, depression, anxiety, hearing loss, and chronic left facial paralysis presented with an open scalp and skull defect. Evaluation by dermatology determined that the scalp lesion was due to repetitive scalp trauma. The patient reported months of headaches with photosensitivity. On examination, she was neurologically intact and had a 4-cm temporal scalp wound with underlying osseous erosion (Figure 2I). A brain MRI revealed contrast enhancement and edema of the temporal bone with associated pachymeningeal enhancement and hyperintensities in the adjacent temporal and frontoparietal regions (Figure 2J). Consultation with neurosurgery, otolaryngology, plastic surgery, and psychiatry were obtained. Psychiatric evaluation ruled out acute psychiatric symptoms or factitious disorder, and she was continued on bupropion.

A 51-year-old female patient with a history of multiple skin ulcers, depression, anxiety, hearing loss, and chronic left facial paralysis presented with an open scalp and skull defect. Evaluation by dermatology determined that the scalp lesion was due to repetitive scalp trauma. The patient reported months of headaches with photosensitivity. On examination, she was neurologically intact and had a 4-cm temporal scalp wound with underlying osseous erosion (Figure 2I). A brain MRI revealed contrast enhancement and edema of the temporal bone with associated pachymeningeal enhancement and hyperintensities in the adjacent temporal and frontoparietal regions (Figure 2J). Consultation with neurosurgery, otolaryngology, plastic surgery, and psychiatry were obtained. Psychiatric evaluation ruled out acute psychiatric symptoms or factitious disorder, and she was continued on bupropion. Before surgery, a repeat MRI showed increased signal abnormalities in the adjacent temporal lobe, concerning for cerebritis (Figure 2K). She underwent drainage of the cerebral abscess, scalp debridement, and a rotational scalp flap. Intraoperative cultures grew Streptococcus mitis, Streptococcus intermedius, Staphylococcus epidermidis, and Candida glabata. Postoperatively, she was placed on intravenous antibiotics. At 2 months postoperatively, the wound was well healed (Figure 2L).

enlarged scalp wound. Imaging revealed frontal lobe encephalomalacia without new mass or hemorrhage. She was admitted and placed on remote video monitoring because of persistent attempts to pick at her wound. She was transferred to psychiatry for management of the excoriation disorder and discharged home 3 weeks later. Two months later, she presented to the emergency department after she was found down in a large amount of dried blood. She was hypotensive, hypoxic, and hypothermic. On examination, she was obtunded and found to have a 10 × 6 cm vertex scalp wound with exposed brain, maggots in situ, and malodorous purulent drainage (Figure 1E). A brain MRI showed vasogenic edema, pneumocephalus, and blood products bilaterally, consistent with cerebritis (Figure 1F). Because of the extent of infection, her poor neurological function, and history of multiple failed attempts to manage the wound surgically, further surgical management was felt to be futile. She was admitted for medical management of sepsis and diffuse cerebritis. Three days later, the patient lost all brainstem reflexes and expired.

A 61-year-old male patient with a history of bipolar disorder and remote head trauma presented with headaches, tinnitus, vision changes, and a bifrontal scalp wound exceeding 5 cm (Figure 1G). He reported headache relief after manipulating the wound with tools (either a clothes pin or drill bit) and eliciting clear drainage from the wound. Physical examination was otherwise unremarkable. A brain MRI demonstrated an underlying calvarial defect with intracranial extension (Figure 1H). Recommendations were made for lumbar puncture and consultations with psychiatry and plastic surgery. The patient reported immediate relief of head “pressure” after lumbar puncture. He began behavioral therapy but did not follow-up with plastic surgery. One year later, he returned with speech difficulty, behavioral changes, and an enlarged calvarial defect (Figure 1I). A brain MRI revealed a new contrast-enhancing lesion underneath the cranial defect, concerning for cerebral abscess (Figure 1J). He was admitted for antibiotic therapy. Several days later, he underwent scalp debridement, allograft placement, and advancement of a rotational flap (Figure 1K and 1L). He was discharged and noted at follow-up to be enrolled in behavioral therapy with a well-healed wound.

A 26-year-old female patient with a history of dermatillomania, chronic skull osteomyelitis, and active substance use disorder (cannabinoids, cocaine, and opiates) presented with headaches, vomiting, fevers, photophobia, and hyperacusis. The patient had previously undergone a scalp flap closure procedure one year prior, but did not follow-up. On examination, she had a large scalp and cranial defect with scattered brain hemorrhages and an associated encephalocele (Figure 2A). She was neurologically intact. A brain MRI showed cerebritis, a subdural fluid collection concerning for empyema, and superior sagittal sinus thrombosis (Figure 2B).

A 45-year-old female patient with a history of active polysubstance abuse (cocaine, alcohol, and cannabis), depression, anxiety, hypertension, and chronic hepatitis C presented with fevers, chills, headaches, and a generalized seizure. She was neurologically intact but had a 1-cm scalp lesion (Figure 2E) caused by picking at her skin because of the recent stress of her son's death. A brain MRI revealed a peripherally enhancing lesion with diffusion restriction and edema in the postcentral gyrus underlying the scalp ulceration and calvarial defect, concerning for cerebral abscess (Figure 2F). Consultations with neurosurgery, plastic surgery, and psychiatry were obtained and surgery was planned. Before surgery, she suffered another seizure and developed right hemiplegia. A head computed tomography demonstrated abscess rupture (Figure 2G). She was taken emergently for a craniotomy and abscess evacuation (Figure 2H). The scalp was debrided, and the skull defect was closed with titanium mesh, followed by primary scalp closure. Cultures grew Staphylococcus aureus, Actinomyces odontolyticus, Peptostreptococcus micros, Staphylococcus hominis, Streptococcus constellatus, and Group A Streptococcus. She was treated with intravenous antibiotics, started on fluoxetine, and discharged to a rehabilitation center. Four months postoperatively, her motor function was improved. However, she resumed picking at the surgical site. She continued with psychiatric care and showed behavioral improvement.

We report a series of 5 patients who presented to 2 academic medical centers over a 4-year period with neurosurgical complications from self-trephination occurring in the context of severe excoriation disorder involving the cranium (Table). We have termed this entity self-trephination in cranial excoriation disorder (STICED). Common features among the 5 cases include a history of preexisting psychiatric disorder, chronic repetitive cranial excoriation, the use of tools to gain entry into the intracranial compartment (eg, drill bits or tweezers), life-threatening exposure of the neural elements to infection or injury, initial treatment refusal or subsequent relapse-related sabotage of surgical repair of the cranial defect, and poor medical adherence. Interestingly, these patients tolerated exposure of the brain for an extended period before developing symptomatic infection, presumably due to immune containment of infection during gradual barrier compromise. The literature on the severity, treatment, and outcomes of STICED is sparse. However, a recent case report described 2 cases of STICED in which both patients had a history of substance abuse, underwent surgical intervention with scalp reconstruction, and relapsed because of self-manipulation of the surgical site.8 Patient Demographics and Outcomes I & D, incision and drainage; STICED, self-trephination in cranial excoriation disorder. Summary of patient demographics and clinical outcomes of patients presenting with STICED at 2 neighboring academic medical centers over the span of 4 y.

We report a series of 5 patients who presented to 2 academic medical centers over a 4-year period with neurosurgical complications from self-trephination occurring in the context of severe excoriation disorder involving the cranium (Table). We have termed this entity self-trephination in cranial excoriation disorder (STICED). Common features among the 5 cases include a history of preexisting psychiatric disorder, chronic repetitive cranial excoriation, the use of tools to gain entry into the intracranial compartment (eg, drill bits or tweezers), life-threatening exposure of the neural elements to infection or injury, initial treatment refusal or subsequent relapse-related sabotage of surgical repair of the cranial defect, and poor medical adherence. Interestingly, these patients tolerated exposure of the brain for an extended period before developing symptomatic infection, presumably due to immune containment of infection during gradual barrier compromise. The literature on the severity, treatment, and outcomes of STICED is sparse. However, a recent case report described 2 cases of STICED in which both patients had a history of substance abuse, underwent surgical intervention with scalp reconstruction, and relapsed because of self-manipulation of the surgical site.8 Patient Demographics and Outcomes I & D, incision and drainage; STICED, self-trephination in cranial excoriation disorder. Summary of patient demographics and clinical outcomes of patients presenting with STICED at 2 neighboring academic medical centers over the span of 4 y. In our experience, surgical wound closure in the absence of psychiatric and behavioral treatment was associated with poor outcomes. Common among our cases was the need for psychiatric medications and behavioral therapy to treat the underlying compulsion and the use of medical restraints, helmets, and/or video monitoring. Treatment of excoriation disorder traditionally involves a combination of cognitive behavioral therapy and neuropharmacological management (most frequently lamotrigine, selective serotonin reuptake inhibitors, or other antidepressants), although clinical trials have demonstrated inconsistent efficacy.3,9-11 Recently, glutamatergic modification with N-acetyl cysteine has shown promise in adults12 and children.13 Furthermore, stereotactic neuromodulation using ablation, stimulation, or radiation approaches have demonstrated clinical improvement among patients with refractory obsessive-compulsive disorder and/or self-injurious behaviors.14-18 Ongoing research has identified the anterior cingulate gyrus and ventral capsule as promising targets for deep brain stimulation for the management of obsessive-compulsive disorder, although these approaches are generally reserved for refractory cases.19-22 At present, ethical challenges and limited access prevent these treatments from being considered as standard of care. Nevertheless, advancements in functional neurosurgery provide an encouraging outlook for these complex patients.

Patients with STICED have the potential for extremely poor prognosis, and the success of surgical interventions depends on concomitant antibiotic therapy, surgical debridement and repair of the cranial defect, and effective psychiatric care. Surgical wound repair in the absence of comprehensive psychiatric and behavioral management is associated with readmission, morbidity, and mortality.