Bilateral Paralysis Research Articles

Giant Hydatic Cyst in a 2-Year-Old Infant

Background: Infantile cerebral hydatidosis is a serious infection and represents a major problem of public health that threatens the economy of countries in which it is endemic. It imposes a large-scale prophylaxis based on the interruption of the parasite cycle. Case description: We present a case of a 2-year-old girl with no medical history living in a rural region (Sidi Ifni), where contact with dogs, sheep, and cattle is not uncommon. One month before her admission, increased intracranial pressure (ICP) symptoms appeared made of headaches, vomiting, and visual disturbances. 15 days later, motor deficits and impaired consciousness were installed. The clinical examination found a sleepy infant with a Glascow Coma Scale at 14, left hemiparesis at 2/5 (Medical Research Council score), and bilateral 6th nerve palsy, A brain CT scan was performed, showing a round hypodense intraparenchymal lesion at the right frontal lobe. The intervention was performed to remove the gliant hydatic cyst according to the Dowling-Orlando technique. Postoperatively the patient had no motor deficit. The histopathological exam confirmed the diagnosis of an echinococcus granulosis cyst. Conclusion: Presentation of such a giant cyst in a 2-year-old infant is very rare. In such cases, genotype analysis of the parasite should be performed. The surveillance of dogs and periodic testing of their stools, with treatment if they are found to be infected, and preventing their access to raw offal at slaughterhouses and farms, will be effective in controlling the menace of hydatidosis.

Sudden Onset Auditory Neuropathy Spectrum Disorder: A Rare Case Report of Brown Vialetto Van Laere Syndrome.

Brown Vialetto Van Laere syndrome is a rare disorder characterized by progressive pontobulbar palsy with bilateral sensorineural hearing loss and lower cranial nerve palsies. Fifty-eight cases have been reported in the last hundred years. As the most common presenting complaint of this disorder is hearing loss, audiological evaluation plays a vital role in pointing towards and narrowing its diagnosis. We present a case report of a 12-year male child affected by this disorder.

448. PREDICTORS OF RECURRENT LARYNGEAL NERVE PALSY RECOVERY FOLLOWING ESOPHAGECTOMY

Abstract Background Recurrent laryngeal nerve (RLN) palsy is a serious complication of esophagectomy that significantly impacts patients’ quality of life. This condition can be permanent or temporary, and identifying the factors associated with recovery can guide treatment options and manage patient expectations. We aimed to investigate the predictors associated with recovery from RLN palsy after esophagectomy. Methods We selected patients who underwent esophagectomy between 2003 and 2022, had a minimum survival and follow-up of 6 months, and experienced right, left, or bilateral RLN palsy from a prospectively managed database. Recovery was defined as restoration of vocal fold mobility within 6 months, irrespective of dysphonia symptoms while permanent injury was defined as persistent vocal cord palsy after six months. Univariate and multivariable logistic regression analyses were used to identify associations between variables and the likelihood of recovery. Results The overall RLN palsy rate was 14.5%. Ninety-five esophagectomy patients with RLN palsy were included. The mean age at operation was 64.4 years. Fifty-two patients (54.7%) recovered from vocal cord palsy. Sixty-four patients (67.4%) underwent minimally-invasive or video-assisted thoracoscopic esophagectomy. On multivariable analysis, patients who underwent neoadjuvant therapy (OR = 0.250, P = 0.012), vocal fold augmentation (OR = 0.283, P = 0.009), or reconstruction via the retrosternal route (OR = 0.326, P = 0.036) or right chest (OR = 0.088, P = 0.046) with reference to the orthotopic route, were less likely to recover from RLN palsy. Conclusion Neoadjuvant therapy, vocal fold augmentation and the route of esophageal reconstruction were significant predictors of recovery from RLN palsy after esophagectomy.

797 Short-Term Review of the Presentations and Outcomes of Vocal Fold Palsy Cases Identified at a Large DGH

Abstract Aim Due to the convoluted course of the recurrent laryngeal nerves, vocal cord palsy can be a manifestation of pathologies that cross multiple surgical and medical specialties. The primary role of the vocal folds is that of airway protection. Therefore, it is important that all relevant clinicians understand this clinical scenario in order to prevent aspiration pneumonia in their patients. Method We reviewed eight months of referrals and diagnoses of patients with vocal cord paresis or palsy. This was in the setting of a tertiary Head & Neck service within a large DGH. We assessed the aetiology, management, and outcome of these cases. Results Eleven new cases were identified over the course of eight months. One was due to intrathoracic vascular pathology; one breast cancer; two from head & neck cancer; one bilateral palsy due to upper-GI cancer surgery; four from thyroid-related pathology; one lung cancer; and one idiopathic. To date, ten of eleven cases appear permanent in nature. One patient underwent in-clinic injection laryngoplasty. Two patients (18%) died within six months of diagnosis, both predictable deaths based on baseline pathology. Conclusions Our experience demonstrated the relevance of ENT (Head & Neck), Vascular, Breast, Upper-GI and Thoracic surgeons as well as medical colleagues in Endocrinology, Respiratory Medicine, and Rheumatology. We present diagnostic and management strategies to help surgical trainees across the specialties identify, manage, and refer patients to prevent complication, improve quality of life and provide access to relatively novel procedures such as awake (local anaesthetic) injection laryngoplasty.

Spasm of the near reflex misdiagnosed as bilateral VI nerve palsy

Spasm of the near reflex misdiagnosed as bilateral VI nerve palsy

Subtotal Submucosal Arytenoidectomy with Lateralization Sutures (SMALS) Covered by a Mucosal Flap.

Glottic airway stenosis (GAWS) may result from bilateral paralysis (BVFP) or posterior glottic stenosis (PGS). Since the glottis is the principal airway sphincter, surgeons shift on the balance between airway, aspirations, and voice. We aim to describe our surgical technique and outcome of the SMALS procedure for GAWS correction. A retrospective cohort of patients who underwent SMALS for PGS between 2018 and 2021. SMALS involves: endoscopic submucosal subtotal arytenoidectmy (preserving medial mucosal flap) and lateralization sutures. The sutures lateralize the mucosal flap to cover the arytenoidectomy bed without lateralization of the membranous vocal fold; expanding the posterior glottis, while preserving a relatively good voice. Covering the arytenoidectomy bed enhances healing. Medical and surgical data, airway, voice, and swallowing symptoms were collected. Relative glottic opening area (RGOA) and relative glottic insufficiency area (RGIA) were calculated. Eleven PGS patients who underwent 15 SMALS were included (4 bilateral), all patients had post-intubation PGS, 1 patient also had prior radiation to the larynx. All patients were tracheostomy-dependent. There were no major complications. No granulation or retracting scar was observed at follow-up. None had a persistent voice or swallowing disability. Successful outcome (decannulation) was achieved in 8 (73%); RGOA increased in all (Δ = 0.37; p = 0.003), while RGIA remained relatively stable (Δ = 0.02; p = 0.055). SMALS is a safe and effective, novel modification of the classic arytenoidectomy, for GAWS correction that can be easily applied and may expand the airway without significant glottic insufficiency symptoms. 4 Laryngoscope, 134:353-360, 2024.

Unraveling the enigma of Adamkiewicz: exploring the prevalence, anatomical variability, and clinical impact in spinal embolization procedures for bone metastases.

The artery of Adamkiewicz (AKA) is vital for spinal cord blood supply. Its role in embolization procedures for bone metastases can cause serious complications. We explored its prevalence, anatomical variation, and effect on spinal embolization using N-butyl cyanoacrylate (NBCA) in patients with bone metastases. To understand the impact and variability of AKA in spinal embolizations in cases of bone metastases to reduce complications and improve patient outcomes. We examined data from 454 patients who underwent spinal embolization with NBCA between 2009 and 2018. The presence, anastomoses, and tumor features of AKA were assessed via pre-procedure imaging and angiography. Complications were classified per the CIRSE Classification System. AKA was found in 22.8% of patients, predominantly left-sided and originating from T8 to L1. Direct and indirect anastomoses were present in 66.6% and 33.4% of patients, respectively. Extra-compartmental invasion was linked with direct anastomosis (P = 0.004). High-grade complications were rare but included one instance of bilateral lower limb paralysis. Partial embolization was necessary in 22.8% of cases due to AKA. The study underscores the need for rigorous preoperative evaluation of AKA origin and anastomoses in patients undergoing spinal embolization for bone metastases. Given the significant presence of AKA and related anastomoses, especially with renal extra-compartmental tumors, caution is advised to reduce complications and optimize patient outcomes. Further research is required for best practice guidelines involving bone metastases and AKA.

NEIM-15 AN EXCEPTION TO THE METASTATIC RULE: A NON-ENHANCING MIDBRAIN LESION DUE TO METASTATIC FIBROMYXOID SARCOMA WITH EXTENSIVEINFLAMMATORY INFILTRATE

Abstract Metastases from extra-CNS sites typically enhance on MR after the administration of gadolinium-based contrast agents due to disruption of the blood-brain barrier. Fibromyxoid sarcoma involving the brain is extremely rare, with only a few reported cases manifesting as well-circumscribed enhancing masses. Here, we report an extremely rare non-enhancing brain metastasis secondary to an already rare sarcoma. A 44-year-old woman with gluteal fibromyxoid sarcoma metastatic to cervical paraspinal muscles and adrenal gland treated with ipilimumab/nivolumab presented with diplopia and was found to have an 8 mm T2-hyperintense midbrain lesion without enhancement or mass effect. An inflammatory etiology associated with checkpoint inhibitor therapy was suspected, and she was treated with IV methylprednisolone 1 g daily for 3 days followed by an oral prednisone taper, IV immunoglobulin, and plasma exchange without improvement. The midbrain lesion steadily grew over 15 months of follow up, eventually involving the bilateral medial thalami and demonstrating diffusion restriction in the midbrain, but never showing gadolinium enhancement. The imaging was reminiscent of peripheral brainstem non-enhancing band-like lesions reported in leptomeningeal lung cancer, however CSF cytology was repeatedly normal, as was extensive infectious and inflammatory testing. Her neurologic disability continued to progress, with development of bilateral oculomotor nerve palsies, dysarthria, and depressed level of alertness, devolving into non-convulsive status epilepticus requiring intubation. She died six months after onset of neurologic symptoms. At autopsy, histologic and immunohistochemical evaluation revealed fibromyxoid sarcoma (MUC-4 positive, TAOK1-FUS fusion) centered on the periventricular aqueduct with accompanying disseminated single-cell infiltration throughout the midbrain. The background midbrain and periventricular parenchyma were notable for voluminous microglial activation and histiocytic infiltrate. In sum, this case represents a radiographically unique sarcoma brain metastasis which on pathologic exam showed robust inflammatory changes far out of proportion to the tumor present.

Glomus jugulare tumor presenting as papilledema – A rare case report

A 54-year-old male presented with headache and blurring of vision in both eyes for 2 months. Ocular examination showed bilateral lateral rectus palsy with papilledema, suggestive of raised intra-cranial pressure. A computed tomography scan of the brain revealed an expansile infiltrating soft tissue lesion causing lysis of the right petrous and jugular fossa, suggestive of a Jugular fossa tumor. The patient’s blood pressure was high at all times despite regular medications because of the glomus tumor’s supposed release of catecholamines. Elective surgery was planned; however, the patient developed a fatal tumor rupture within 4 months of diagnosis, for which emergency neurosurgical intervention and intensive care support were given, but he succumbed to death. Tumors obstructing the jugular outflow can cause increased intra-cranial pressure and vision loss, requiring prompt neurosurgical intervention.

Parálisis facial periférica bilateral como presentación inicial del síndrome de Guillain-Barré: informe de un caso

El síndrome de Guillain-Barré (SGB) es un grupo heterogéneo de alteraciones neurológicas relacionadas que afectan los nervios periféricos del cuerpo. Generalmente se produce después de una infección aguda que favorece a la aparición de una respuesta autoinmune contra las moléculas de los gangliósidos de la mielina de los nervios periféricos. La parálisis facial periférica bilateral es una manifestación neurológica rara (0,3%-2% de los casos) y puede deberse a múltiples etiologías, entre las que se destaca el SGB. Se presenta el caso de un paciente de 39 años que consultó al servicio de urgencias del hospital Universitario del Caribe por parálisis facial bilateral asociada a deterioro motor progresivo y disautonomía. Se realizaron estudios que documentan SGB y se inicia manejo médico con inmunoglobulina intravenosa. El paciente presenta mejoría clínica notoria de su cuadro clínico y es dado de alta del hospital. La parálisis facial periférica bilateral es una entidad neurológica rara que puede asociarse con el síndrome de Guillain-Barré. Es necesario hacer el diagnóstico diferencial respecto de otras entidades clínicas que cursan con parálisis facial periférica bilateral. Se debe iniciar precozmente con inmunoglobulina intravenosa para aumentar la probabilidad de recuperación. El pronóstico es bueno en la mayoría de los casos.

Bilateral sixth nerve palsy with subdural hematoma: a unique presentation of B12 deficiency.

Vitamin B12 is inextricably associated with the development and maintenance of neuronal functions. It is classically associated with subacute combined degeneration and peripheral neuropathy; however, cranial neuropathy is uncommon. We observed the rarest neurological manifestation of B12 deficiency. A 12 months infant had history of lethargy, irritability, anorexia, paleness, vomiting, and neurodevelopmental delay for 2 months. He also developed inattention and altered sleep pattern. His mother noticed bilateral inward rotation of both eyes. On examination, the infant had bilateral lateral rectus palsy. The infant was found to have anemia (7.7g/dL) and severe B12 deficiency (74pg/mL). On MRI, there was cerebral atrophy, subdural hematoma (SDH) and wide cisternal spaces and sulci. On supplementation with cobalamin, he improved clinically though mild restriction of lateral gaze on the left side persists. Follow up MRI showed significant improvement in cerebral atrophy with resolution of SDH. To date, such clinical presentation of B12 deficiency has never been reported. The authors suggest B12 supplementation for at risk population esp at antenatal stage and lactating mothers in national programs. The treatment of this condition should be initiated early to prevent long term sequelae.

INDICATIONS AND COMPLICATIONS OF TRACHEOSTOMY AT A TERTIARY CARE INSTITUTE: A PROSPECTIVE AND OBSERVATIONAL STUDY

Objective: The aim of the present study was to present an analysis of indications and complications of tracheostomy in tracheostomized patients. Methods: This was a single center, hospital-based prospective, non-randomized, and non-comparative study involving 60 tracheostomized patients carried out from February 2021 to March 2022 at Geetanjali Medical College and Hospital, Udaipur, India. Results: Out of 60 tracheostomies performed most common indications were Laryngo-tracheal trauma (26.7%), organophosphorus poisoning (25%), malignancy of Larynx (11.67%), post-intubation stenosis (6.7%), bilateral abductor paralysis (6.7%), head injury (6.7%), meningitis (5%), acute laryngo-traheo-bronchitis (3.33%), hypoxic-ischemic-encephalopathy (3.33%), cerebrovascular accidents (1.67%), faucial diptheria (1.67%), and GB syndrome (1.67%). Out of 60 tracheostomies performed, 18 complications were encountered. The most common complication was granulations of tracheostoma which occurred in 5 cases (8.3%), three cases of tube occlusion (5%), three cases had difficult decannulation (5%), three cases developed infection of stoma (5%), two cases developed surgical emphysema (3.3%), one case of tracheostomy scar hypertrophy (1.6%), and one case developed tracheal stenosis (1.6%). Conclusion: Tracheostomy is the procedure of choice especially in cases. Where it is needed to provide prolonged positive pressure ventilation and tracheobronchial toilet. Thorough anatomical knowledge of trachea and adjacent structures, skill of surgeon, sterile technique, meticulous surgery, use of high-volume low pressure cuffed Portex tracheostomy tubes, and good bedside tracheostomy care results in minimal complications. Complications of tracheostomy are largely preventable.

頸髄損傷に合併した経鼻胃管症候群の1例(A case of nasogastric tube syndrome with cervical spinal injury)

経鼻胃管症候群とは，経鼻胃管留置後に咽頭痛，両側声帯の外転麻痺，喉頭浮腫を発症する症候群である。両側声帯麻痺を認めれば気道緊急を要し，しばしば致命的となる。症例は46歳の男性。墜落外傷により当院搬送となった。精査の結果，外傷性くも膜下出血，第6・7頸椎・第6–10胸椎骨折，第6・7頸髄・第6–8胸髄損傷と診断し，入院加療を開始した。第2病日麻痺性イレウスを発症し，経鼻胃管を留置し，減圧加療を開始した。第11病日吸気性喘鳴を聴取し，酸素8L/分投与下でSpO2（経皮的酸素分圧）88%まで低下を認めた。喉頭ファイバースコピー検査で両側声帯麻痺を認め，気道緊急として経口気管挿管を施行し，人工呼吸管理を開始した。第14病日気管切開術を施行した。第39病日声帯麻痺の治癒を確認した。第45病日リハビリテーション目的に転院した。 Nasogastric tube syndrome presents as throat pain, bilateral abductor paralysis of the vocal cords and pharyngeal edema after nasogastric tube placement. This syndrome often requires emergency airway management due to bilateral abductor paralysis and thus can be fatal. A 46–year–old man was transported to the emergency room due to trauma from a fall. We diagnosed traumatic subarachnoid hemorrhage, cervical and thoracic spine fractures, and cervical and thoracic spinal cord injury, and admitted him. On hospital day 2, a nasogastric tube was inserted due to paralytic ileus. On day 11, wheezing was heard on exhalation, and his SpO2 decreased to 88% on oxygen at 8L/min. Laryngeal fiberscopy revealed bilateral vocal cord paralysis, so he was intubated orally. A tracheotomy was performed on day 14. The vocal cord paralysis had healed by day 39, and on day 45, he was transferred to another hospital for rehabilitation. 経鼻胃管症候群とは，経鼻胃管留置後に咽頭痛，両側声帯の外転麻痺，喉頭浮腫を発症する症候群である。両側声帯麻痺を認めれば緊急気道確保を要し，しばしば致命的となる。今回，頸髄損傷を背景として経鼻胃管留置中に両側声帯麻痺を発症した1例を経験したので報告する。 当論文は当院倫理委員会の承諾を受け，個人情報保護法に基づいて匿名化を行っている。また，患者およびその家族から論文の出版に関する同意を得ている。 患 者：49歳の男性。身長180cm，体重91.6kg，BMI 28.3 既往歴：統合失調症 内服薬：アリピプラゾール，フルニトラゼパム，ゾルピデム酒石塩酸，クエチアピンフマル酸塩，アムロジピンベシル酸塩 現病歴：自宅3階からの飛び降りによる転落外傷で受傷した。目撃者が救急要請し，当院救急搬送となった。 来院時現症：気道は開通，呼吸音 清，呼吸様式 正常，呼吸数16/分，SpO2 97%（酸素10L/分 投与下），血圧85/45mmHg，脈拍72/分，Glasgow coma scale E3V5M6，瞳孔径2mm/2mm，対光反射は両側迅速，体温36.2度（腋窩温）であった。 身体所見：後頭部に約20cmの裂創を認めるが活動性出血は認めなかった。右膝・両踵に擦過傷を認めた。徒手筋力テストは両上下肢いずれもMMT 3以上であるが，意思疎通がとれず，詳細な評価は困難であった。持続勃起や肛門括約筋の弛緩は認めなかった。 血液検査：WBC 17,300/µL（Neut 89.4%），Hb 16.4g/dL，Plt 34.9×104/mm3，PT–INR 1.17，Fibrinogen 180mg/dL，APTT 22.2秒，D–dimer 41.21µg/mL 動脈血液ガス分析（酸素マスク10L/分投与下）：pH 7.359，PaCO2 38.0mmHg，PaO2 148.1mmHg，HCO3− 20.9mmol/L，B.E. −4.0mmol/L，lactate 29.2mg/dL 全身CT画像検査：外傷性くも膜下出血，第6・7頸椎棘突起骨折，左第4・5肋骨骨折，第6–8・10胸椎椎体骨折を認めた。 来院時ショックの状態であり，神経原性ショックの可能性を念頭に置いて診療したが，輸液負荷のみで血圧は安定し，入院加療を開始した。血液検査でのWBCおよびD–dimerの上昇は外傷に起因するものと判断した。第2病日，腸蠕動音減弱および腹部レントゲン検査で小腸の拡張を認め，麻痺性イレウスと診断し，減圧目的に20Frの経鼻胃管を留置した。第3病日下肢の筋力低下の訴えがあり，頭部・頸椎・胸椎MRI撮像したところ，T2強調像で第6椎体前面および第6–7頸髄内，第6–8胸髄内の高信号を認め，第6–7頸髄損傷，第6–8胸髄損傷と診断した（Fig. 1）。頸椎カラー装着継続で保存加療の方針とした。リハビリテーション目的の転院調整を開始しようとしたが，第11病日突然の吸気性喘鳴を認め，ボスミン筋肉注射を行うも改善を認めなかった。喉頭ファイバースコピー検査で両側声帯麻痺と診断した（Fig. 2A）。次第にSpO2は低下し，酸素8L/分 投与下にSpO2 88%前後まで酸素化低下を認めた。その時点での動脈血液ガス分析では，pH 7.501，PaCO2 48.8mmHg，PaO2 67.9mmHg，HCO3− 37.3mmol/L，B.E. 12.5mmol/L，lactate 11.8mg/dLと低酸素血症，高二酸化炭素血症を呈していた。経口気管挿管による気道確保を行い，人工呼吸管理を開始した。その後は原因検索を行ったが器質的疾患はいずれも否定的で，経鼻胃管症候群と診断した。麻痺性イレウスは遷延しており，経鼻胃管の抜去は困難であったため，第13病日経鼻胃管を18Frに入れ替えた。長期の気管挿管となることが予想され，第14病日気管切開術を施行した。腸蠕動音正常，腹部レントゲン検査で小腸の拡張の改善を認め，麻痺性イレウスの治癒と判断し，第16病日栄養投与目的に経鼻胃管を10Frに入れ替えた。第39病日喉頭ファイバースコピー検査で声帯麻痺の治癒を確認した（Fig. 2B）。精神状態悪化による昏迷状態となっており，気管切開チューブはスピーチカニューレへの入れ替えにとどめた。第45病日頸椎カラーは装着を継続しリハビリテーション目的に転院した（injury severity score: 17，probability of survival: 0.97972であった）。 Magnetic resonance imaging of the cervical and thoracic vertebrae. MRI（T2）revealed spinal cord injury at the sixth and seventh cervical vertebrae and from the sixth to eighth thoracic vertebrae (red arrow), and 5–mm retropharyngeal hematoma (yellow arrow). Laryngeal fiberscopy. A: Laryngeal fiberscopy showed abnormal abducton of the vocal cords. (during the phase of expiration (left) / inspiration (right)) B: Laryngeal fiberscopy showed recovery of the vocal cords from abductor disorder. (during the phase of expiration (left) / inspiration (right)) Cervical computed tomography (CT). Computed tomography revealed a 6–mm retropharyngeal hematoma (red arrow). 経鼻胃管による咽頭への影響は1939年Wangensteen 1により初めて報告され，その後しばしば症例報告が散見される程度であった。1981年Sofferman 2は，経鼻胃管留置中に咽頭痛，片側あるいは両側の声帯の外転機能不全を来す症候群を経鼻胃管症候群と定義した。診断は，喉頭ファイバースコピー検査で声帯麻痺と診断し，かつ原因となりうる疾患を除外したうえで，臨床的に診断することが多い。発症のタイミングは経鼻胃管を留置して12時間後から抜去2週間後まで多様である 3。 病態は完全には解明されておらず，機序として，①咳嗽や嚥下の際に固定された経鼻胃管と咽頭が垂直方向へ摩擦が起きること，②仰臥位で輪状後部が頸椎側から経鼻胃管により圧迫される，③輪状咽頭筋の収縮が経鼻胃管を脆弱で薄い輪状軟骨後板へ持続的に引き上げる，といったことが推測されている 2。その結果，経鼻胃管による輪状後部への持続的圧迫から結果的に潰瘍形成や感染，虚血が生じ，後輪状披裂筋の機能不全が生じると考えられている 2, 4。糖尿病や低免疫状態，低栄養，加齢に伴う粘膜弾性の低下，パーキンソン症候群，脳血管障害に伴う生理的な嚥下運動の減少による除圧頻度の減少，輪状軟骨レベルでの骨棘突出がリスク因子と言われている 2, 5, 6。経鼻胃管の太さがリスク因子とする報告はない。桜井ら 7による10Frの経鼻胃管を留置していた4例の報告があり，決して細いチューブがリスクを軽減するわけではないと考えられる。またチューブの径が太い方が輪状後部への影響を与える可能性は高く，リスクとなりうると考えるが，今後検討すべき事項である。Friedmanらは，経鼻胃管が声帯部を正中で走行するのは約6%と少ないものの，動物モデルで側方を走行するより正中を走行する方が炎症を生じやすい可能性があると報告している 8。 本症例において，初療時のCT画像や第3病日のMRI画像では声帯自体や輪状軟骨後部を含め声門周囲の直接的な外傷性変化は指摘できなかった。咽頭後間隙の血腫は第1病日のCTで6mm，第3病日のMRIで5mmであった（Fig. 1, 3）。リスク因子は入院後に発症した麻痺性イレウスの影響による低栄養のみであった。経鼻胃管はCT画像で確認する限り，声門部では側方を通過していた。喉頭ファイバースコピー検査の際も側方を通過しているのを確認できた。入院後は頸髄損傷のため，仰臥位での安静時間がほとんどであった。しかし，頸髄損傷で経鼻胃管の留置が必要であった患者はこれまで多く経験しているが，当院で経鼻胃管症候群と診断したのは初めてであり，頸髄損傷などで輪状軟骨レベルでの損傷がある外傷症例での経鼻胃管症候群の報告は，我々が渉猟する限り見られなかった。以上から，本症例では低栄養状態や長期臥床の影響だけでなく，リスク因子として知られている輪状軟骨レベルの骨棘突出と同様の機序で，頸部外傷による咽頭後間隙の血腫・炎症性変化が経鼻胃管による輪状後部への刺激を増強し，炎症や虚血を起こしたのではないかと考えられた。頸髄損傷に偶発的に合併した可能性は否定できないが，椎体前面の外傷性変化が一因となり，経鼻胃管症候群を発症しうることが示唆された。椎体前面に血腫を形成する頸髄損傷の症例には重症度の差がある。軽症例では，通常経口摂取が可能で経鼻胃管が留置される可能性は高くない。一方，重症例では，腹式呼吸や喀痰喀出困難を原因とした呼吸不全に対する気管挿管のタイミングで経鼻胃管が留置されることが多い。その後十分な声帯の評価が行われずに気道確保や気管切開が行われていることが多く，経過中に経鼻胃管症候群を発症していても認知されていない可能性がある。本症例は腹式呼吸を呈するような重症例ではなかったものの，イレウスを発症したことで経鼻胃管が留置され，頸椎前面の外傷性炎症と相まって，経鼻胃管症候群の発症に至った。それに加え，咳嗽減弱や喀痰喀出困難をそれまで認めていなかったという経過から，本症に気づくことができたのではないかと考えた。 治療は経鼻胃管の抜去が必須とされている 9。その他，抗菌薬やPPI，副腎皮質ステロイドなどが投与された報告はあるが，確立した治療法はない。声帯外転障害が改善せず気管カニューレが抜去できないこともある 3。本症例では，治療上経鼻胃管を抜去することは困難であり，まずは経鼻胃管を減圧の治療の障害にならない範囲で細いものに入れ替えたのちに，経鼻胃管を留置している限り短期間での治癒は見込めないと判断し，気管切開術を行った。発症から29日後に声帯麻痺の治癒を確認できた。検索する限り今まで経鼻胃管を抜去せずに経鼻胃管症候群が治癒したという報告は見られなかった。治癒までの期間は長くなるかもしれないが，本症例のように治療上必要であれば経鼻胃管を抜去せずとも治癒する可能性があることが示唆された。 本症例は外傷症例の亜急性期に発生した吸気性喘鳴を契機に，各疾患を除外してから経鼻胃管症候群と診断できた。経鼻胃管症候群は比較的近年に定義された症候群であり，とくに2010年以降の症例報告が増加している 7, 9。これに関しては，経鼻胃管の留置数が著明に増加しているわけではないと考えられることから，発症する頻度が増加したわけではなく，この疾患の概念が認識され始めたことに影響しているものと推測された。対応が遅れると致命的となりうるだけでなく，本疾患を鑑別に入れておかないと経鼻胃管を抜去するなどの対応が遅れ，治癒までの期間が延長しうる。とくに救命・集中治療部門において経鼻胃管は留置されていることの多いデバイスであり，経鼻胃管を留置する際は本合併症に留意して管理を行う必要がある。 椎体前面の外傷性変化が一因になったと考えられた経鼻胃管症候群の1例を経験した。経鼻胃管はよく留置されるデバイスであり，両側声帯麻痺を発症した場合，緊急気道確保を行うとともに，経鼻胃管症候群を鑑別に挙げて治療を行う必要がある。 本論文の要旨は第122回近畿救急医学研究会（2021年，大阪）で発表した。 本稿のすべての著者に規定された利益相反はない。

Peripheral T-Cell Lymphoma Manifesting as Multifocal Central Nervous System Disease with Prominent Spinal Cord Involvement (P2-13.001)

<h3>Objective:</h3> To emphasize the need for timely and wide-reaching diagnostic testing in the evaluation of acute-onset multifocal CNS disease with prominent spinal cord involvement. <h3>Background:</h3> Metastatic CNS neoplasms, although rare, should be considered early in rapidly declining patients with multifocal disease in whom immune-mediated or infectious etiologies have been excluded. <h3>Design/Methods:</h3> N/A <h3>Results:</h3> A 53-year-old male with Celiac disease on azathioprine presented with one-week of vertigo, diplopia, and dysarthria upon one-year of malaise and cachexia. The patient’s examination revealed right eyelid ptosis, bilateral abducens palsies, restricted up-gaze, dysarthria, and diffuse dysmetria and hyporeflexia. MRI showed a right T2 hyperintense contrast-enhancing cerebellar lesion and a small left cerebellar diffusion-restricting lesion. CSF was inflammatory (nucleated cell 6 cells/mcL, protein 181 mg/dL, glucose 30 mg/dL). Workup for autoimmune encephalitis, myelin-oligodendrocyte-glycoprotein-IgG, aquaporin-4-IgG, and infections etiologies were negative. He was empirically treated with intravenous methylprednisolone and immunoglobulin without benefit. He continued to decline and repeat CSF was again inflammatory (28 cells/mcL, protein 494 mg/dL), without clonal cells. Repeated MRI showed new left frontal leptomeningeal and cauda equina enhancement extending into the adjacent musculature. Electromyography showed proximal myopathy and subsequent biopsy was unrevealing. A bone marrow and brain biopsy were declined due to development of pulmonary embolism and multiorgan failure. He continued to decline and developed multifocal strokes, expiring 6 weeks after presentation. Autopsy demonstrated peripheral T cell lymphoma (PTLC). <h3>Conclusions:</h3> Intramedullary non-CNS spinal metastases represent less than 8.5% of all metastatic tumors and 3% of all spinal metastases. PTCL represents 15% of non-Hodgkin lymphomas and can rarely involve the CNS parenchyma or leptomeninges. Persistent enhancement, elevated protein, and minimal response to steroids or subsequent rebound should raise suspicion for neoplasm, especially considering these patients’ rapid decline can make CNS biopsy inadvisable later in the disease course. <b>Disclosure:</b> Dr. Singh has nothing to disclose. Dr. Carozza has nothing to disclose. Dr. Misulis has received publishing royalties from a publication relating to health care. Dr. Shah has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Horizon. Dr. Shah has received publishing royalties from a publication relating to health care.

E025 A complex case of giant cell arteritis presenting with visual loss, tongue and scalp infarction and multiple cranial neuropathies

Abstract Background/Aims Giant cell arteritis (GCA) is the most common of the vasculitides. Visual loss due to vascular occlusion is a well recognised complication of GCA. It is important to remember that other ischaemic complications can occur, leading to atypical clinical features. We present a case of a patient with GCA who did not complain of headaches, but presented with at least five cranial neuropathies, including a right optic neuropathy, ischaemic ulceration of her tongue and a large area of scalp necrosis. Methods We present the results of our patient case review and literature search. Results An 82-year-old retired landlady who was an ex-smoker presented with haemoptysis and was diagnosed with non-small-cell lung cancer. She was treated with chemotherapy and radiotherapy in 2020, then had possible disease progression on CT thorax in 2021. She developed thigh pain, swallowing difficulties and decreased oral intake in 2021, and subsequently developed severe oral ulceration, dehydration and delirium and was admitted to the frailty unit. On examination she had a very large scalp lesion, oral lesions, and multiple cranial neuropathies (right optic neuropathy, right partial third nerve, right seventh nerve, recurrent laryngeal, and bilateral twelfth nerve palsies). Her anterior ischemic optic neuropathy meant she could only see hand movements with the right eye. Blood tests revealed anaemia of chronic disease and a CRP of over 200 mg/L. Ultrasound scan of the temporal arteries showed a halo sign confirming the diagnosis of GCA. CT angiography showed vertebral artery wall thickening and stenosis. She was treated with intravenous methylprednisolone followed by high dose oral prednisolone, and started on methotrexate. She improved symptomatically. The cranial neuropathies resolved and the area of scalp necrosis and ischemic tongue ulceration healed, but vision was not regained. Her inflammatory markers never normalised and she was subsequently started on tocilizumab, with the approval of her oncologist. Her ESR and CRP quickly fell to normal. It remains to be seen how biologic treatment may impact her malignancy. Conclusion This case highlights multiple atypical presenting features of GCA and the need for a high index of suspicion to treat early and avoid irreversible visual loss. A review of the literature reveals that cranial neuropathies secondary to ischemia can be the presenting feature in GCA and multiple cranial neuropathies can occur. This is the first case in the literature, that we are aware of, which involves this number of cranial neuropathies alongside extensive scalp and tongue infarction. These features have all resolved with treatment, with the exception of unilateral permanent visual loss. Disclosure J. Jade: None. V. Barradell: None. A. Coote: None.

Teaching NeuroImages: Rupture and spontaneous resolution of a P1 perforator pseudoaneurysm

A 47-year-old man with a history of smoking and hypertension had a severe, sudden headache. Head CT showed subarachnoid hemorrhage (SAH) (figure, A). Progressive ophthalmoplegia developed without alteration of consciousness. Catheter angiography was normal (figure, B). Brain MRI documented bilateral thalamic infarcts (figure, C). Repeat catheter angiography after 4 days showed a 2-mm dissecting pseudoaneurysm of a thalamoperforator (figure, D and E). Serial noninvasive vascular studies documented no growth. Six weeks later, a catheter angiogram demonstrated complete resolution of the pseudoaneurysm (figure, F).Bilateral third nerve palsy persisted at 1 year follow-up. Dissecting pseudoaneurysms of perforating posterior circulation vessels can present with a pattern suggestive of perimesencephalic SAH. Presence of associated neurologic deficits should increase the level of suspicion. Spontaneous resolution can occur without invasive treatment.1

Patient with neck swelling.

An approximately 70-year-old female with a history of anaplastic thyroid cancer with metastasis to the lungs and cervical spine presented to the emergency department with a new fluctuant neck swelling of approximately 1 week's duration (Figure 1, Video 1). The mass was non-tender but oscillated paradoxically with respirations. The patient also noted a mildly hoarse voice. One month previously, she underwent total radical thyroidectomy with extensive soft tissue resection, including cricotracheal resection with primary re-anastomosis. The patient underwent computed tomography with contrast of the neck. (Figure 2). The computed tomography revealed a large, predominantly air-filled collection in the anterior neck measuring 7 × 6 × 6 cm and communicating with a contained tracheal perforation. The patient was taken to the operating room the following day, confirming breakdown of the cricotracheal repair. The patient underwent debridement and revision tracheostomy. Tracheal dehiscence is the complete disruption of the tracheal wall. This is in contrast to tracheal diverticulum, which represents an outpouching of the tracheal wall.1, 2 Anastomotic dehiscence occurs in 2.5%–11% of postoperative patients.3, 4 Known risk factors for dehiscence include diabetes and laryngotracheal resections, as well as long resections, pediatric age group, preoperative tracheostomy, and tension on the anastomosis. The current case is unusual in the timing of the diagnosis (1 month after surgery) and the large size of the perforation.5, 6 Surgical complications of tracheal repair may occur in up to 27% of patients. Bilateral recurrent nerve palsy is the most common complication. Other potential complications include anastomotic dehiscence, hypoparathyroidism, glottic edema, anastomotic stenosis, bleeding, respiratory insufficiency, and ventilator dependency.7, 8

Перший досвід імплантації стимулятора діафрагмального нерва в немовляти з білатеральним парезом діафрагми

Phrenic nerve injury is not frequent, but well recognized complication of birth trauma caused by brachial plexus injury. Diaphragmatic paresis is usually unilateral, and cases of bilateral damage are rare. Diaphragmatic paralysis led to development of severe respiratory disorders, which required the prolonged intensive care, often with the applying of mechanical ventilation. Although is it possible the spontaneous restoration the function of the diaphragm, this requires the long-term of respiratory support. The general accepted guidelines of the treatment of infants with paresis/paralysis of diaphragm is absent. Diaphragmatic plication considered as the main surgical method of treatment. By that, diaphragmatic plication not always had a positive effect. The aim of the study was to present new possibility in the treatment of infants with bilateral diaphragmatic paralysis. Clinical case. We presented the first experience of transthoracic implantation of diaphragm pacemaker in infant with bilateral diaphragmatic paralysis. The newborn was hospitalized with the respiratory disorders’ syndrome. The paresis of both hemidiaphragms was reviled on chest X-ray. For 7 months child required the mechanical ventilation, due to unsuccessful attempts to transfer the child to spontaneous breathing caused the decrease of saturation. With the aim to restore spontaneous breathing, the implantation of diaphragm pacemaker, under the thoracoscopic control, was performed initially at the right side and next at the left side. The effectiveness of the treatment was confirmed by the electroneuromyography and ultrasonography. Conclusions. Implantation of the diaphragmatic pacemaker my be the method of treatment in infants with bilateral diaphragmatic paralysis. The research was carried out in accordance with the principles of the Helsinki Declaration. The informed consent of the patient was obtained for conducting the studies. No conflict of interests was declared by the authors.

JAPANESE ENCEPHALITIS VIRUS TRIGGERED OVERLAPPING MILLER FISHER SYNDROME WITH BICKERSTAFF ENCEPHALITIS IN A YOUNG FILIPINO CRUISE LINE WORKER.

Insidious onset of bilateral paralysis preceded by fever is most likely encephalitis.Japanese encephalitis virus led to the development of variant forms of Guillain-Barré syndrome in our patient.Supportive care resulted in significant recovery despite the severity of the condition.

Percutaneous suture lateralization for bilateral vocal fold paralysis - How we do

Bilateral abductor cord paralysis is rare. But it requires urgent surgical intervention due to upper airway obstruction at glottis level. Percutaneous suture lateralization seems to be a safe and effective treatment, which enable patients to be devoid of tracheostomy without significant influence on phonation or swallowing function. Here we explain the technique of suture lateralization of vocal cord with the available resources.