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Fulltext Recurrent Submandibular Sialolithiasis in a Child

Thong HK, Mohamad Mahbob H, Sabir Husin Athar PP, Tengku Kamalden TMI

Cureus, 2020 Dec 19;12(12):e12163.
PMID: 33489575 DOI: 10.7759/cureus.12163

Sialolithiasis is a commonly encountered disease of the salivary glands, reported to represent up to 30% of all salivary gland diseases. However, the condition is rarely encountered in the pediatric population. The formation of a salivary stone is believed to be secondary to the deposition of calcium salts around a nidus. The formation of a nidus is commonly associated with desquamated epithelial or sloughing from a recent bacterial infection. Patients with submandibular sialolithiasis usually present with acute swelling over the neck associated with pain, fever, and purulent intraoral discharge. Neglected and poorly treated acute infection may progress to life-threatening abscess formation. Here we are describing our encounter with a 10-year-old boy with recurrent submandibular sialolithiasis. He was initially treated with conservative measures and antibiotics regimen. Failure of medical treatment and recurring symptoms led to submandibular gland excision followed by a full recovery.
Fulltext A Rare Case of an Undiagnosed Middle Ear Tumor Due to Late Referral

Ng JJ, Ong HY, Nasseri Z, Azmi MI, Abdullah A

Cureus, 2021 Jan 08;13(1):e12584.
PMID: 33457146 DOI: 10.7759/cureus.12584

Facial nerve tumors constitute about 5% of all facial nerve paralysis. As it is relatively uncommon, it could be misdiagnosed. We encountered an 18-year-old girl who had right facial weakness since the age of four, referred to otorhinolaryngology clinic for further evaluation only when her hearing deteriorated and the facial weakness worsened. Further investigation revealed facial nerve schwannoma. Facial nerve paralysis in the pediatric age group is uncommon and should be examined in detail to rule out other possible etiologies besides Bell's palsy.
Pain Perception During the Phases of Manual Reduction of Distal End Radius Fracture With a Periosteal Block

Wan Ali WAA, Soh EZF, Abdullah S, Narin Singh PSG, Ahmad AA, Sapuan J

Cureus, 2021 Jan 13;13(1):e12691.
PMID: 33604222 DOI: 10.7759/cureus.12691

Introduction Closed reduction is an effective method of treatment for distal end radius fractures. We present a case series of patients with distal end radius fractures who underwent closed manipulative reduction using the periosteal block. We describe the technique in detail and examine its efficacy in pain lowering effect during closed reduction. Methods Nineteen patients with distal end radial and ulnar fractures were included and grouped based on the Frykman classification. The reduction was performed using a periosteal block of 10 ml of 2% lignocaine injection. The severity of pain was recorded by utilizing the visual analog scale (VAS) in five phases: 1) before injection, 2) after 15 minutes of analgesia in a resting position, 3) during minimal motion, 4) during full manipulation and reduction, and 5) post-procedure. The VAS scoring was classified as painless (VAS score of 0), mild pain (VAS score between 1-3), and painful (VAS score of 4 and above). Results The study included 19 patients [median age of 53 years (range: 18-88 years)]; there were 11 (58%) males and eight (42%) females. The mechanism of injury was a fall (n=12, 63%) or a motor vehicle accident (n=7, 37%). There was a statistically significant reduction of pain between phase one and all the other phases. Between the different fracture configurations, there was no significant difference in pain reduction. The most painful phase was expected to be phase four, ie, during full manipulation, in which four (21%) patients had a VAS score of 0, 12 (63%) patients had a VAS score between 1-3, and three (16%) patients had a VAS score of 4. Thus, 16 out of 19 patients (84%) had no or minimal pain during the most painful phase. There were no complications from the periosteal blocks. Conclusions The periosteal nerve block is an effective procedure providing satisfactory analgesia during the reduction of distal radial and ulnar fractures. It has no side effects and is free from complications associated with conventional sedation.
Fulltext Non-Traumatic Snapping Tendon on the Dorsal Aspect of the Thumb: A Diagnostic Challenge

Kesu Belani L, Abdullah S, Soh EZF, Abd Jabar F, Nasseri Z

Cureus, 2021 Apr 11;13(4):e14417.
PMID: 33987066 DOI: 10.7759/cureus.14417

A snapping tendon on the dorsal aspect of the thumb is a rare condition as opposed to the common triggering on the volar aspect of the thumb. This condition is known as triggering of the extensor pollicis longus (EPL). A 21-year-old female presented with a clicking or snapping sensation that was felt on the dorsum of her thumb when it is extended. There was no history of trauma. She worked in an ice-cream parlor with repetitive scooping ice-cream motions. Her triggering immediately resolved on releasing the EPL fascia ulnar to Lister's tubercle. Upon wake-up surgery, we could immediately confirm this. We recommend dynamic ultrasound as an investigation and do not recommend MRI. The surgical method of choice is either wake-up surgery or wide-awake local anesthesia no tourniquet (WALANT) surgery.
Fulltext Myelin Oligodendrocyte Glycoprotein (MOG) Optic Neuritis: A Case Series

Nurul-Ain M, Khairul Kamal ZN, Wan Hitam WH, Abd Munaaim M, Mohd Zaki F

Cureus, 2021 Apr 13;13(4):e14452.
PMID: 33996312 DOI: 10.7759/cureus.14452

Myelin oligodendrocyte glycoprotein (MOG) antibody disease has been recognised as a distinct demyelinating disorder. Optic neuritis has been reported as the most common presentation and manifestation of this spectrum disorder. This is a case series of three MOG optic neuritis patients. Patients involved are female with disease onset ranging between 7- and 37-year-old. Most of these patients experienced symptoms of profound reduced visual acuity with eye pain. All three patients had optic disc swelling upon first presentation and they experienced at least one episode of bilateral simultaneous optic neuritis. Only one patient had demonstrable optic nerve enhancement on magnetic resonance imaging (MRI). Disease was confirmed through positive MOG antibody. Patients typically responded well to intravenous methylprednisolone (IVMP) during acute attack of optic neuritis. However, one patient had suboptimal response to IVMP after multiple relapses. We noted multiple relapses of optic neuritis are common in MOG patients. MOG optic neuritis is a devastating, but treatable condition. Aggressive treatment during acute optic neuritis attack and relapse prevention may favour a good visual prognosis in MOG antibody disease.
Fulltext Orbital Tumor As First Manifestation of Metastatic Renal Cell Carcinoma

Tan SY, Bastion MC, Mohd Khialdin S

Cureus, 2021 Jul;13(7):e16275.
PMID: 34373823 DOI: 10.7759/cureus.16275

Orbital metastasis from renal cell carcinoma (RCC) is uncommon. Orbital tumor, as the first presentation of RCC, is rare as the majority of orbital metastases occur after a confirmed diagnosis of primary cancer. We report a case of the metastatic orbital tumor as the first manifestation of RCC, which presented with painless left eye proptosis for two months' duration, associated with blurring of vision and diplopia. Otherwise, the systemic review was unremarkable. Examination showed left eye non-axial proptosis with a pulsatile, multilobulated mass over the left supraorbital area extending to the left frontal region, limited ocular motility, and impaired optic nerve functions. CT of the orbit showed a mass arising from the left frontal and greater wing of the left sphenoid bone, with infiltration to the left lateral rectus, left superior oblique, and lacrimal gland. Further systemic investigation with CT thorax, abdomen, and pelvis revealed left RCC with para-aortic nodes, lungs, and bone metastases. The patient was planned for palliative care.
Fulltext Neck of Femur Fracture in Young Patients With End-Stage Renal Disease and Hyperparathyroidism: A Report of Three Cases and Proposed Treatment Algorithm

Raj JJ, Kow RY, Ramalingam S, Low CL

Cureus, 2021 Jul;13(7):e16155.
PMID: 34367767 DOI: 10.7759/cureus.16155

Secondary hyperparathyroidism is a complication arising from untreated end-stage renal disease (ESRD). It can invariably lead to osteoporosis and subsequently cause pathological neck of femur (NOF) fracture. Despite being young, osteosynthesis in neck of femur fractures of these patients often leads to nonunion and implant failure due to severely osteoporotic bone. We present our experience in managing three young patients with ESRD and secondary hyperthyroidism who sustained NOF fractures. All three patients were successfully treated and showed no complication at one year post-operation. Based on our experience and literature review, we propose a simple algorithm to guide the management of these patients.
Fulltext Metastatic Vascular Pleomorphic Leiomyosarcoma in a Previously Treated Cervical Carcinoma Patient: A Diagnostic Dilemma

Ghani UHM, Leong JF, Sani MH, Rosli N, Mohd-Haflah NH

Cureus, 2021 Jul;13(7):e16182.
PMID: 34367789 DOI: 10.7759/cureus.16182

Vascular leiomyosarcoma is a rare malignant tumour of the smooth muscle connective tissue. Patients are usually asymptomatic in the early stages and only present when the lesion causes compressive or obstructive effects or has metastasized. We report a case of vascular pleomorphic leiomyosarcoma in a 70-year-old lady with a background history of squamous cell carcinoma of the cervix. She presented with a three-month history of low back pain, which radiated to the anterior bilateral thigh. Initial radiological findings revealed metastatic lesions involving the spine and lungs. Two spinal biopsies done were inconclusive. Increasing severity of pain over the right thigh prompted further imaging, which revealed bilateral femoral lesions. The patient underwent surgery which involved excision of the tumour in the right proximal femur with skeletal reconstruction using megaprosthesis. Proximal femoral nail was performed for the left femur. Intra-operatively, tumour was noted at the anteromedial aspect of the proximal right thigh surrounding the superficial femoral vein. Histopathological report of the right thigh mass finally confirmed a diagnosis of vascular pleomorphic leiomyosarcoma. The patient presented four months later with bilateral pulmonary embolism with deep vein thrombosis in addition to progression of the disease.
Fulltext Retinal Nerve Fiber Layer and Macular Thickness in Parkinson's Disease Patients

Abd Hamid MR, Wan Hitam WH, Abd Halim S

Cureus, 2021 Jul;13(7):e16224.
PMID: 34367824 DOI: 10.7759/cureus.16224

OBJECTIVE: This study aimed to evaluate the retinal nerve fiber layer (RNFL) thickness and macular thickness in Parkinson's disease (PD) patients.
METHODS: The present study is a comparative cross-sectional, hospital-based study. A total number of 64 PD patients and 64 controls were recruited. Candidates that fulfilled the criteria with normal ocular examinations were undergone optical coherent tomography (OCT) examinations of the right eye. RNFL and macular thickness were evaluated.
RESULTS: There was a statistically significant reduction in RNFL thickness in average (adjusted mean 84.32 vs 95.93, p ≤ 0.001), superior (adjusted mean 105.15 vs 118.13, p ≤ 0.010), and inferior (adjusted mean 104.95 vs 126.55, p ≤ 0.001) PD group compared to the control group. The macula thickness also was significantly reduced in average (adjusted mean 266.51 vs 281.34, p = 0.015), central (adjusted mean 236.37 vs 255.55, p = 0.001), outer superior (adjusted mean 269.16 vs 278.19, p = 0.014), outer inferior (adjusted mean 256.34 vs 272.24, p ≤ 0.001), and outer nasal (adjusted mean 287.64 vs 302.84, p = 0.001) PD group compared to the control group. There was a significant positive correlation between RNFL thickness and visual acuity among PD patients in the inferior segment with p = 0.020 and nasal segment with p ≤ 0.001. There was also a significant positive correlation between macular thickness and visual acuity among PD patients in the inner temporal segment with p = 0.006, outer superior segment with p = 0.003, and outer temporal segment with p ≤ 0.001.
CONCLUSION: The mean of the average, superior, and inferior RNFL thickness was significantly lower in the PD group compared to the control. The mean of the average, central, outer superior, outer inferior, and outer nasal macular thickness was significantly lower in the PD group compared to the control.
Fulltext Good Visual Outcome in Post Traumatic Exogenous Endophthalmitis Caused by Trichophyton Species

Supahiah P, Bin Ahmad NA, Teh WM, Bt Abd Jalil NF, Md Din N

Cureus, 2021 Jun;13(6):e16071.
PMID: 34367742 DOI: 10.7759/cureus.16071

Trichophyton species is a dermatophytic fungus commonly found in the skin, nails, hair, and other organic matters such as palm trees and soil. We report a rare case of a 23-year-old man who had a penetrating injury to the eye from the leaves of a palm tree and subsequently developed exogenous endophthalmitis. Culture from the vitreous tap revealed Trichophyton sp as the causative organism. Early vitrectomy and adequate intravitreal injection of amphotericin B resulted in good visual outcomes in an otherwise blinding condition. This is the first reported case of exogenous endophthalmitis secondary to Trichophyton species. Early diagnosis and prompt treatment may help improve visual outcomes.
Fulltext Integra: An Alternative Option for Reconstruction of Extensive Finger Defects With Exposed Bones

Hamdan AM, Al-Chalabi MMM, Wan Sulaiman WA

Cureus, 2021 Jul;13(7):e16223.
PMID: 34367823 DOI: 10.7759/cureus.16223

Extensive soft tissue injuries with exposed joints, bones, and tendons due to trauma of the hand -particularly fingers- necessitate soft tissue reconstruction and coverage. However, these injuries are challenging; therefore, various management options for traumatic skin loss over fingers are widely performed. These options comprise wound care permitting wound contracture without surgical intervention, full or split-thickness skin grafting, skeletal shortening and primary closure, and various types of flaps. We present a case of successfully placed Integra over the exposed phalangeal bones followed by split-thickness skin grafting a few weeks later, with a good outcome. We conclude that Integra is an alternative, safe and effective method for reconstructing severely traumatized fingers with exposed bone, tendon, or joint without sacrificing outcome.
Fulltext Scurvy: A Neglected Cause of Muscle Pain and Weakness in a Child With Picky Eating Behavior

Kow RY, Nik Abdul Adel NA, Abdul Razak AH, Low CL, Awang MS

Cureus, 2021 Jul;13(7):e16289.
PMID: 34405060 DOI: 10.7759/cureus.16289

Ascorbic acid (vitamin C) is an essential micronutrient that the human's body cannot synthesize endogenously. Scurvy, a disease of ascorbic acid deficiency, can manifest in a myriad of presentations. Due to its rarity in the modern world, scurvy is considered as a disease of the past. We present a paediatric case of scurvy with musculoskeletal manifestations as a result of picky eating behavior. We report a previously healthy nine-year-old boy who presented with unexplained progressive bilateral lower limb generalized weakness and pain for two months. All initial biochemical and radiological investigations were unremarkable. Upon further history taking, he had severe picky eating behavior which raised the suspicion of scurvy. The diagnosis was confirmed with a serum ascorbic acid test. After ascorbic acid supplementation, his symptoms resolved immediately. Further food behavioral modification counselling to his family members helped to change his diet in a lasting way. As a result, he had no recurrence of symptoms. This case highlights the importance of having a high index of suspicion for an uncommon disease and emphasizes the need for a detailed dietary history upon patient's presentation.
Antibiotic Irrigation: A Promising Unconventional Method for Facial Carbuncle

Ng JJ, Gendeh H, Ong HY, Gopalan S, Abdul Karaf JH

Cureus, 2021 Apr 27;13(4):e14710.
PMID: 34079672 DOI: 10.7759/cureus.14710

Carbuncle is conventionally treated with combinations of intravenous antibiotics and surgical intervention; be it saucerization or incision and drainage. Cosmesis outcome might be unfavorable following surgical intervention, especially when the facial region is involved. Skin grafting surgery may even be needed as a second-stage procedure for a larger wound. We reported a series of three facial carbuncles treated successfully with a new improvised method. Our method includes performing a stab incision prior to draining of pus, coupled with minimal wound debridement, followed by regular irrigation of the wound with antibiotics containing solution. Based on the three cases reported in this article, we conclude that this method is more superior as there is more skin preservation, better patient tolerance, shorter hospital stays, and favorable cosmesis outcome.
Fulltext Orbital Cellulitis Secondary to Dental Abscess in Children

Abdul Satar H, Yaakub A, Md Shukri N, Ahmad Tajudin LS

Cureus, 2021 Apr 09;13(4):e14392.
PMID: 33981511 DOI: 10.7759/cureus.14392

Paediatric orbital cellulitis is a potential sight- and life-threatening condition. It is a serious infection in children that can result in significant complications, including blindness, cavernous sinus thrombosis, cerebral venous sinus thrombosis, meningitis, subdural empyema, and brain abscess. Of the patients with orbital cellulitis, 17% died from meningitis, and 20% of the survivors had permanent loss of vision. Therefore, the potential for sight- and life-threatening complications makes prompt diagnosis and early treatment very important. We report here a case of a two-year-old girl who presented with a three-day history of left periorbital swelling, preceded by left upper perioral swelling that extended upward to the left cheek and left lower lid and was associated with low-grade fever. The patient had been admitted and was treated as having left preseptal with facial cellulitis; the patient was started on intravenous amoxicillin/clavulanic acid (200 mg three times per day dose), and chloramphenicol ointment was applied to the periorbital area. On day 3, the condition worsened, and dental examination showed multiple dental caries, upper gum swelling and abscess, and mobility of teeth 61 and 62 (two baby teeth). Contrast-enhanced computed tomography (CECT) of the orbit, paranasal, and brain showed a left periosteal abscess collection extending to the inferomedial region of the orbit. Examination and tooth extraction were performed under general anesthesia. The intraoperative results showed the presence of a left upper gum abscess, which was possibly the primary source of infection. Clinical improvement was observed postoperatively. Orbital cellulitis can be a complication of a dental abscess. This case emphasizes the importance of primary tooth care in children. A lack of care can result in fatal complications.
Fulltext Acute Proptosis in a Diabetic Patient: Diagnostic and Therapeutic Dilemmas

Khairul-Anwar I, Tai E, Hussein A, Zunaina E

Cureus, 2021 Jun;13(6):e15580.
PMID: 34277202 DOI: 10.7759/cureus.15580

An elderly diabetic lady presented with a painful swollen right eye and blurred vision for one week, preceded by right eye redness for six months. Her right eye best-corrected visual acuity was finger counting at 1 m. There was right eye proptosis, limited extraocular muscle movements, corkscrew vessels, chemosis and elevated intraocular pressure, but no bruit. Fever was absent. Computed tomography of the brain and orbit showed thickened extraocular muscles and intraconal fat streakiness, with normal superior ophthalmic vein and concavity of the cavernous sinus. Intravenous antibiotics resulted in limited clinical improvement. The subsequent response to oral prednisolone was dramatic, with the improvement of visual acuity to 20/60 after three doses. In cases of atypical orbital cellulitis where antibiotics fail, one should consider differential diagnoses such as orbital inflammatory disease, vascular anomalies and masqueraders. We discuss the approach to the diagnosis of acute proptosis in a diabetic patient and highlight the role of corticosteroids in idiopathic orbital inflammatory disease.
Fulltext Case Series on Endogenous Klebsiella pneumoniae Endophthalmitis: More Than Meets the Eye

Danapal P, Mustapha M, Abdul Malek NS, Yeak J, A Qamarruddin F

Cureus, 2021 Jun;13(6):e15929.
PMID: 34336431 DOI: 10.7759/cureus.15929

Endogenous endophthalmitis (EE) is a rare but potentially sight-threatening disease with an appreciable mortality rate. Diabetes mellitus remains the most frequently associated condition especially in the Asian population, which potentiates Klebsiella pneumoniae involvement. Endogenous Klebsiella pneumoniae endophthalmitis (EKE) usually has a poor final visual outcome despite treatment with intravitreal and systemic antibiotics. We report three cases of EKE with systemic involvement Klebsiella pneumoniae invasive syndrome (KPIS). KPIS was diagnosed in three patients with multiple comorbidities who presented with a blurring of vision and eye redness. Patient 1 was a 63-year-old Malay man diagnosed with left eye panophthalmitis with multifocal liver and prostate abscesses. He underwent drainage of the liver abscess and eventually evisceration of the left eye due to scleral perforation. Patient 2 was a 66-year-old Malay woman diagnosed with left eye endophthalmitis. Due to hemodynamic instability, vitrectomy was delayed and eventually sustained corneal perforation and eviscerated. The patient eventually succumbed to infection. Patient 3 was a 42-year-old Malay woman diagnosed with KPIS, renal abscess, lung abscess, and left endogenous endophthalmitis. She underwent a vitrectomy but her postoperative vision remained poor. All patients received multiple intravitreal antibiotics and systemic antibiotics. KPIS is frequently associated with catastrophic disabilities. Our cases highlight the importance of an early suspicion of systemic involvement in patients presenting with EKE. Prompt diagnosis, emergent radiographic evaluation, early adequate drainage, and appropriate treatment with antibiotics potentially improve survival and visual prognosis.
The Epidemiological Profile of Open Globe Injuries and Prognostic Factors in a Tertiary Care Centre

Ng HR, Chee SF, Chai KS, Chong MF, Mustapha M

Cureus, 2021 Jun;13(6):e15846.
PMID: 34327079 DOI: 10.7759/cureus.15846

Purpose To describe the epidemiology of open globe injuries and its prognostic factors from the perspectives of a tertiary care centre in northern Malaysia. Methods A retrospective study of open globe injuries in a period of three years between June 2017 and May 2020. Patients presenting with open globe injuries were identified and recruited from hospital census. Case records were retrieved and analysed after recruitment. Results A total of 114 patients with 118 open globe injuries were included in the study. Four patients had bilateral eye involvement. Men were found to have seven and a half times higher rate of ocular injuries than women. The predominant age group of ocular trauma presentation was in younger adults between the age of 21 and 30 years old. Work-related injuries account for almost half of the globe injuries (48.7%) followed by motor vehicle accident-related, domestic accident-related, and others. The significant factors related to the visual outcome are presenting visual acuity (VA), presence of relative afferent pupillary defect (RAPD) and vitreous loss. Conclusion The factors related to visual outcomes in this study may aid the clinician in determining the visual prognosis of ocular injuries. Young working males were the most susceptible group to sustain penetrating ocular injuries due to their job nature. Health education and safety at workplace are essential to reduce the occurrence of ocular injuries.
Fulltext MRI Evaluation of Anterolateral Ligament of the Knee: A Cross-Sectional Study in Malaysia

Kow RY, Low CL, Siron Baharom KN, Sheikh Said SNB

Cureus, 2021 Jun;13(6):e15758.
PMID: 34290935 DOI: 10.7759/cureus.15758

Introduction After detailed anatomical delineation of the anterolateral ligament (ALL) of the knee, there is a surge in research on this anatomical structure. Owing to the anatomical variation and lack of experience in the identification of this structure, magnetic resonance (MR) evaluation of the ALL produces mixed results. It was aimed to evaluate the ALL using the routinely performed MR imaging of the knee and to determine any associated factors with ALL injuries. Materials and methods Thirty-six MR images of the knee from 31 patients from January 1, 2017, to June 30, 2017, are evaluated. MR sequences performed include T1-weighted, T2-weighted, proton density (PD), and PD fat saturation (FS). All MR images were double-read by two authors and approved by a consultant radiologist with more than 20 years of radiological experience. The ALL was divided into three portions: femoral, meniscal, and tibial, and the ALL was considered fully visualized when all three portions were seen on MR images. Results At least a portion of the ALL was visualized in 27 scans (75%), and it was fully visualized in 20 scans (55.6%). The femoral portion was the most commonly identified (75%), followed by the meniscal portion (69.4%) and the tibial portion (58.3%). ALLs were best visualized on coronal view in PD FS with the lateral inferior genicular artery as a guide to locate the bifurcation of the meniscal and tibial components. Conclusion The ALL can be visualized in routine 1.5-T MR imaging, either full delineation (55.6%) or partially visualized (75%). It is best characterized via a PD-weighted sequence with fat saturation on the coronal plane. The ALL injury was associated with an anterior cruciate ligament (ACL) injury.
Fulltext Thoracolumbar Fracture in Disseminated Idiopathic Skeletal Hyperostosis

Chia Hua L, Mokhtar SA

Cureus, 2021 May 25;13(5):e15222.
PMID: 34188976 DOI: 10.7759/cureus.15222

Disseminated idiopathic skeletal hyperostosis (DISH) is a form of ankylosing spinal disorders, which is at high risk of fracture because of the rigidity of the spinal column and reduced bone quality. The patients with DISH are at higher risk of fall because of the poor muscle tone, rigid spine column, and positive sagittal balance. The management of spinal fractures in these patients proves to be challenging because of the altered biomechanics and alignment of the spine. Furthermore, most patients have multiple comorbidities with high intraoperative burden, and osteoporosis itself will impair any implant purchase of the bone. Here, we report a case of thoracolumbar fracture in DISH where both conservative and surgical approaches were utilized, with unfortunate results in both, and a brief review of the literature on its management.
Fulltext Clinical Puzzles and Decision-Making in Antisynthetase Syndrome

Yehudina Y, Trypilka S, Isayeva A

Cureus, 2021 Jun 25;13(6):e15931.
PMID: 34221776 DOI: 10.7759/cureus.15931

We present a challenging clinical case of an antisynthetase syndrome (ASS) with a four-year follow-up. The disease debuted with skin manifestations and interstitial lung disease (ILD), then the severe Raynaud's phenomenon came to the fore with the development of occlusive vasculopathy and critical digital ischemia. After the relief of vascular lesions, the severity of the condition was determined by ILD. The use of combined pulse therapy with cyclophosphamide and methylprednisolone, treatment with intravenous immunoglobulin made it possible to reduce the activity of ASS: lung lesion and the progression of vasculopathy. However, after the termination of an unplanned pregnancy, the patient again experienced an exacerbation with ILD progression. It was decided to use rituximab, against which the patient's condition was stabilized. Clinical and laboratory remission was achieved, which was maintained for a year and a half. However, the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) pandemic triggered a re-exacerbation of the pulmonary domain of the disease, which forced us to use a nintedanib with a positive clinical and instrumental effect.

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