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Open access

The recurrent plexiform neurofibroma of the scalp in neurofibromatosis type 1: illustrative case

Galih Indra Permana, M. Arifin Parenrengi, Wihasto Suryaningtyas, Dyah Fauziah, and Muhammad Azzam

BACKGROUND

Plexiform neurofibroma is a benign tumor of the peripheral nerves. It is an unusual variant of neurofibroma originating from all parts of the nerve. Plexiform neurofibroma is primarily pathognomonic and exhibits an unusual variant from neurofibromatosis type 1 (NF1). The possibility of malignancy and recurrence are the main reasons for long-term, close follow-up.

OBSERVATIONS

The authors report a case of a 14-year-old girl with a recurrent plexiform neurofibroma derived from the peripheral nerves, which also presented with a typical sign of NF1 disease. The aim of the tumor resection is symptomatic relief.

LESSONS

Accomplishing a good outcome can be related to good perioperative planning and a precise operative procedure. The result of anatomical pathology determines the prognosis of the patient. Clinical examination and radiological studies are needed to evaluate the recurrence of complications after surgical procedures.

Open access

Acute pulmonary edema due to occult air embolism detected on an automated anesthesia record: illustrative case

Samuel Wood and Gennadiy Fuzaylov

BACKGROUND

The authors report a case of venous air embolism (VAE) during a pediatric posterior fossa craniotomy with resulting pulmonary edema requiring postoperative ventilation. Pulmonary edema is a known but rare complication of VAE, and diagnosis and treatment are discussed.

OBSERVATIONS

The embolism was undetected during the surgical procedure, and the first clinical sign of respiratory decompensation appeared an hour after the initial insult, with imaging suggesting acute pulmonary edema. A transient but significant end-tidal carbon dioxide decrease was detected on postoperative review of the anesthesiology record.

LESSONS

This report highlights an uncommon sequela of VAE and the importance of post hoc automated record review for intraoperative event analysis.

Open access

Asymptomatic rotational vertebral artery compression in a child due to head positioning for cranial surgery: illustrative case

M. Travis Caton, Kazim Narsinh, Amanda Baker, Adib A. Abla, Jarod L. Roland, Van V. Halbach, Christine K. Fox, Heather J. Fullerton, and Steven W. Hetts

BACKGROUND

The authors recently reported a series of children with vertebral artery (VA) compression during head turning who presented with recurrent posterior circulation stroke. Whether VA compression occurs during head positioning for cranial surgery is unknown.

OBSERVATIONS

The authors report a case of a child with incidental rotational occlusion of the VA observed during surgical head positioning for treatment of an intracranial arteriovenous fistula. Intraoperative angiography showed dynamic V3 occlusion at the level of C2 with distal reconstitution via a muscular branch “jump” collateral, supplying reduced flow to the V4 segment. She had no clinical history or imaging suggesting acute or prior stroke. Sequential postoperative magnetic resonance imaging scans demonstrated signal abnormality of the left rectus capitus muscle, suggesting ischemic edema.

LESSONS

This report demonstrates that rotational VA compression during neurosurgical head positioning can occur in children but may be asymptomatic due to the presence of muscular VA–VA “jump” collaterals and contralateral VA flow. Although unilateral VA compression may be tolerated by children with codominant VAs, diligence when rotating the head away from a dominant VA is prudent during patient positioning to avoid posterior circulation ischemia or thromboembolism.

Open access

Intracranial temporal bone angiomatoid fibrous histiocytoma: illustrative case

Shivani Gillon, Jacqueline C. Junn, Emily A. Sloan, Nalin Gupta, Alyssa Reddy, and Yi Li

BACKGROUND

Angiomatoid fibrous histiocytoma (AFH) is a rare, slowly progressive neoplasm that most commonly occurs in soft tissues. AFH rarely occurs in bone such as the calvaria. The authors present a case of AFH in the petrous temporal bone, which, to their knowledge, is the first case of AFH in this location.

OBSERVATIONS

A 17-year-old girl presented with worsening positional headaches with associated tinnitus and hearing loss. Imaging demonstrated an extraaxial mass extending into the right cerebellopontine angle, with erosion of the petrous temporal bone, with features atypical for a benign process. The patient underwent retrosigmoid craniotomy for tumor resection. Pathology was consistent with a spindle cell tumor, and genetic testing further revealed an EWSR1 gene rearrangement, confirming the diagnosis of AFH. The patient was discharged with no complications. Her symptoms have resolved, and surveillance imaging has shown no evidence of recurrence.

LESSONS

The authors report the first case of AFH in the petrous temporal bone and only the second known case in the calvaria. This case illustrates the importance of the resection of masses with clinical and imaging features atypical of more benign entities such as meningiomas. It is important to keep AFH in the differential diagnosis for atypical masses in the calvaria and skull base.

Open access

Simultaneous intracranial and testicular germ cell tumors: illustrative case

Lei Han, Jie Lu, Luxiong Fang, Songtao Qi, and Ye Song

BACKGROUND

Simultaneous intracranial and testicular germ cell tumors (GCTs) are extremely rare, leading to a lack of adequate experience in their treatment. Therefore, the authors report a case of this kind of GCT.

OBSERVATIONS

A 5-year-old boy was admitted to the hospital with headache and vomiting. Computed tomography and magnetic resonance imaging suggested the possibility of a GCT in the pineal region. The value of the serum tumor marker alpha-fetoprotein (AFP) was 5,396.1 μg/L, and β-human chorionic gonadotropin levels were within the normal range. Subsequently, the tumor was removed, and the final pathological result was a mixed GCT. Therefore, chemotherapy and radiation were added. However, the authors found a testicular tumor on ultrasound at the same time, and pathology after surgery suggested a mature cystic teratoma. Following treatment, the patient recovered well, and AFP levels dropped to normal values.

LESSONS

To the authors’ knowledge, this report is the fourth case of simultaneous intracranial and testicular GCTs and the first case of a simultaneous mixed GCT in the pineal region and mature teratoma of the testis. A combination of surgery, chemotherapy, and radiation therapy for mixed GCTs in the pineal region and surgical excision for testicular reproductive cell tumors are effective in these patients, but long-term monitoring is required.

Open access

Decompression surgery for pure arterial malformations in a 15 year old with acute, progressive visual impairment: illustrative case

Katsuma Iwaki, Koichi Arimura, Ataru Nishimura, and Koji Iihara

BACKGROUND

The authors document the first case of pure arterial malformations (PAMs) of the posterior communicating artery (PCoA), which were successfully treated with microsurgical clipping of the main body of the PAMs. PAMs are defined as dilated, overlapping, and tortuous arteries with a coil-like appearance and/or a mass of arterial loops without any associated venous component. Although PAMs usually have a benign history and are often incidental findings, this case presented with acute progression of visual field impairment.

OBSERVATIONS

Because the patient’s right optic tract was affected by the loop of PAMs of the PCoA, the authors performed microsurgical clipping of the main body of the PAMs using endoscopy, which ceased the progression of symptoms without any complications.

LESSONS

There have been several reports of PAMs receiving surgical treatment for accompanying lesions. However, in this case, the lesion to the main body of PAMs was the cause of visual field impairment and was successfully treated with microsurgical clipping.

Open access

Misdiagnosis of elevation of β-hCG in cystic craniopharyngioma: illustrative case

Umaira Saleh, Liang Hooi Lim, Ihfaz Ismail, and Nasser Abd Wahab

BACKGROUND

Craniopharyngiomas and germ cell tumors (GCTs) are both rare intracranial tumors commonly present in childhood or middle age. They share similar clinical and radiological features. GCTs commonly give rise to tumor markers in the cerebrospinal fluid, hence guiding the treatment plan.

OBSERVATIONS

This article reports the case of a 5-year-old boy with a large sellar and suprasellar mass with obstructive hydrocephalus. Laboratory studies showed increased beta-human chorionic gonadotrophin (β-hCG) levels in the cystic fluid, suggestive of choriocarcinoma. He underwent 3 cycles of chemotherapy but showed a poor response. Further aspiration followed by tumor debulking was performed, and histopathological examination revealed craniopharyngioma.

LESSONS

This case report indicates that β-hCG, commonly regarded as a specific tumor marker for choriocarcinoma, is detectable in other forms of suprasellar tumors. The authors highlight clinical and radiological features of suprasellar tumors that can be misdiagnosed as intracranial GCTs. The relevance of tumor markers and indications for histopathological confirmation are discussed.

Open access

Spontaneous regression of a vein of Galen aneurysmal malformation in a pediatric patient: illustrative case

Kevin K. Kumar, Linden E. Fornoff, Robert L. Dodd, Michael P. Marks, and David S. Hong

BACKGROUND

Vein of Galen aneurysmal malformations (VGAMs) are rare congenital intracranial vascular lesions that represent 30% of all pediatric vascular anomalies. These lesions are associated with severe manifestations, including congestive heart failure, hydrocephalus, and spontaneous hemorrhage. The mainstay of management is medical stabilization followed by endovascular embolization of the lesion. Although VGAM was first reported in 1937, there are few published cases demonstrating spontaneous regression of the lesion.

OBSERVATIONS

The authors report the case of a 31-month-old female who presented with an incidentally found VGAM. After initial evaluation, including magnetic resonance imaging and angiography, the patient was lost to follow-up. Upon her return to the clinic at age 12 years, the previously identified VGAM was absent, indicative of involution of the lesion. The patient remained asymptomatic and met appropriate developmental milestones during this interval.

LESSONS

This report adds a rare case of the spontaneous resolution of VGAM to the literature. This case may suggest the presence of VGAMs that are asymptomatic, undetected, and regress within the pediatric population. Future studies may benefit from identifying imaging and angiographic findings predictive of spontaneous regression. There may be a role for conservative management in particular cases of asymptomatic and medically stable children with VGAMs.

Open access

Idiopathic dissecting cerebral aneurysm of the distal anterior cerebral artery in an infant successfully treated with aneurysmectomy: illustrative case

Suguru Nagamitsu, Natsue Kaneko, Toshikazu Nagatsuna, Hiroaki Yasuda, Manabu Urakawa, Masami Fujii, and Tetsuo Yamashita

BACKGROUND

Idiopathic dissecting cerebral aneurysms (IDCAs) are male dominant but are extremely rare in children. Many IDCAs in children are located in the posterior cerebral artery and the supraclinoid internal cervical artery. No cases of IDCA of the distal anterior cerebral artery (ACA) have been reported.

OBSERVATIONS

A previously healthy 7-month-old boy experienced afebrile seizures and presented at the authors’ hospital 1 week after the first seizure. He was not feeling well but had no neurological deficits. The authors diagnosed a ruptured aneurysm of the right distal ACA based on imaging results. He underwent emergency craniotomy to prevent re-rupture of the aneurysm. Using intraoperative indocyanine green videoangiography, the authors confirmed peripheral blood flow and then performed aneurysmectomy. Pathological examination of the aneurysm revealed a thickened intima, fragmentation of the internal elastic lamina, and a hematoma in the aneurysmal wall. The authors ultimately diagnosed IDCA because no cause was indicated, including a history of trauma. The boy recovered after surgery and was subsequently discharged with no complications.

LESSONS

The authors reported, for the first time, IDCA of the distal ACA in an infant. The trapping technique is often used for giant fusiform aneurysms in infants. Indocyanine green videoangiography is useful for evaluating peripheral blood flow during trapping in this case.

Open access

Removal of malformation in cerebral proliferative angiopathy: illustrative case

Gwang Yoon Choi, Hyuk Jai Choi, Jin Pyeong Jeon, Jin Seo Yang, Suk-Hyung Kang, and Yong-Jun Cho

BACKGROUND

Cerebral proliferative angiopathy (CPA) is a rare vascular disorder distinct from arteriovenous malformation. Because of the disorder’s rarity, there is still a controversy on the most promising treatment method for CPA. However, several meta-analysis articles suggest indirect vascularization such as encephalo-duro-arterio-synangiosis as an effective way of treating symptoms that are medically uncontrolled.

OBSERVATIONS

The authors describe a case of an 11-year-old boy with this disease, who had epilepsy that was intractable despite conservative management. The patient recovered from his symptoms after the vascular malformation was surgically removed. This is the first reported case of surgical removal in CPA.

LESSONS

Although further investigation on the best treatment for CPA is needed, the authors believe surgical intervention may also be an effective treatment modality when a patient presents with persisting symptoms.