Displaying publications 21 - 40 of 45 in total

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  1. Siow SL, Tee SC, Wong CM
    J Med Case Rep, 2015;9:49.
    PMID: 25890166 DOI: 10.1186/s13256-015-0519-6
    Paraesophageal hernia with intrathoracic mesentericoaxial type of gastric volvulus is a rare clinical entity. The rotation occurs because of the idiopathic relaxation of the gastric ligaments and ascent of the stomach adjacent to the oesophagus through the hiatus defect, while the gastroesophageal junction remains in the abdomen. The open approach remains the gold standard therapy for most patients. Here we report the case of a patient with such a condition who underwent a successful laparoscopic surgery. A literature search revealed that this is the first case report from Southeast Asia.
  2. Kasinathan G, Kori AN, Azmie NM
    J Med Case Rep, 2019 Jul 22;13(1):223.
    PMID: 31327318 DOI: 10.1186/s13256-019-2164-y
    INTRODUCTION: Post-transplant lymphoproliferative disorder is a serious disorder which occurs post hematopoietic stem cell transplant or solid organ transplantation. T-prolymphocytic leukemia is a T cell type monomorphic post-transplant lymphoproliferative disorder which accounts for only 2% of all mature lymphocytic leukemias in adults over the age of 30.

    CASE PRESENTATION: A 59-year-old man of Chinese ethnicity presented to our hematology unit with headache, lethargy, and exertional dyspnea for the past 1 month. He underwent an uneventful cadaveric renal transplant 20 years ago for chronic glomerulonephritis-induced end-stage renal disease. He had been on long-term immunosuppressants since then consisting of orally administered prednisolone 10 mg daily and orally administered cyclosporine A 50 mg twice daily. On examination, he was pale with a palpable liver and spleen. He had a functioning renal graft. Marrow flow cytometry confirmed T-prolymphocytic leukemia with lymphocytes expressing CD2, CD3, CD7, CD52, and TCL-1. His human T-cell lymphotropic virus and Epstein-Barr virus serology and deoxyribonucleic acid (DNA) were negative. He was treated with one cycle of cyclophosphamide, doxorubicin, vincristine, and prednisone chemotherapy to which he failed to respond. In view of his renal allograft, he was not suitable for alemtuzumab due to the risk of nephrotoxicity. He was given orally administered venetoclax but he died on day 17 due to severe auto tumor lysis syndrome.

    CONCLUSION: The place of immunophenotyping in the diagnosis and treatment of this disorder is of significant importance. More research needs to be carried out to further comprehend the pathophysiology and treatment modalities for this disorder.

  3. Wan SA, Teh CL, Jobli AT, Cheong YK, Chin WV, Tan BB
    J Med Case Rep, 2019 Jan 08;13(1):8.
    PMID: 30626451 DOI: 10.1186/s13256-018-1940-4
    BACKGROUND: Gout is a monosodium urate deposition disease which is prevalent worldwide. The usual manifestations are crystal arthropathy and tophi deposition in the soft tissues. Spinal tophi may also occur and are rarely reported, resulting in various clinical manifestations such as back pain, spinal cord compression, radiculopathy, and even mimicking epidural abscess and spondylodiscitis.

    CASE PRESENTATION: We report a case of a 42-year-old Chinese man with underlying gout who presented with back pain and radiculopathy. The diagnosis of spinal tophi was unsuspected and he was initially treated for epidural abscess and spondylodiscitis. He underwent a laminectomy and posterolateral fusion during which tophus material was discovered. He recovered and medications for gout were started.

    CONCLUSION: Spinal tophi are rare. The diagnosis is difficult and spinal tophi may be mistaken for epidural abscess, spondylodiscitis, or neoplasm.
  4. Sachdev Manjit Singh B, Wan SA, Cheong YK, Chuah SL, Teh CL, Jobli AT
    J Med Case Rep, 2021 Feb 23;15(1):94.
    PMID: 33618728 DOI: 10.1186/s13256-020-02642-z
    BACKGROUND: Arthritis is rarely reported as a paraneoplastic manifestation of occult malignancy. We report herein two cases of paraneoplastic arthritis due to occult malignancy. CASE 1: The patient was a 65-year-old woman of asian descent who was a former smoker with a history of spine surgery performed for L4/L5 degenerative disc disease. She presented with a 1-month history of oligoarthritis affecting both ankle joints and early morning stiffness of about 3 hours. Laboratory tests were positive for antinuclear antibody at a titer of 1:320 (speckled) but negative for rheumatoid factor. She was treated for seronegative spondyloarthritis and started on prednisolone without much improvement. A routine chest radiograph incidentally revealed a right lung mass which was found to be adenocarcinoma of the lung. She was treated with gefitinib and her arthritis resolved. CASE 2: The patient was a 64-year-old woman of asian descent, nonsmoker, who presented with a chief complaint of asymmetrical polyarthritis involving her right wrist, second and third metacarpophalangeal joints, and first to fifth proximal interphalangeal joints. She was treated for seronegative rheumatoid arthritis (RA) and started on sulfasalazine, with poor clinical response. Six months later, she developed abdominal pain which was diagnosed as ovarian carcinoma by laparotomy. Her arthritis resolved following treatment of her malignancy with chemotherapy.

    CONCLUSION: In summary, paraneoplastic arthritis usually presents in an atypical manner and responds poorly to disease-modifying antirheumatic drugs. Accordingly, we recommend screening for occult malignancy in patients presenting with atypical arthritis.

  5. Yap JF, Wai YZ, Ng QX, Lim LT
    J Med Case Rep, 2019 May 06;13(1):131.
    PMID: 31056080 DOI: 10.1186/s13256-019-2064-1
    BACKGROUND: This is a case report of an iatrogenic intralenticular broken steroid (Ozurdex™) implant in a patient with uveitis. There are only a few case reports on broken Ozurdex™ implants in the vitreous cavity, with none of them involving the crystalline lens. A few authors have described the accidental injection of an Ozurdex™ implant into the crystalline lens, but all of the implants remained in one piece in the lens and none of them were broken. We report an unusual case of an Ozurdex™ implant which was injected inadvertently into the crystalline lens, resulting in a broken Ozurdex™ implant with an entry and exit wound through the posterior capsule of the lens.

    CASE PRESENTATION: An ophthalmic trainee performed an Ozurdex™ intravitreal injection into a 48-year-old Asian man's right eye under aseptic conditions. This patient was then followed up for further management. On day 7 post-procedure, a slit lamp examination revealed that the Ozurdex™ implant was injected into the intralenticular structure of his right eye and had fractured into two pieces. The posterior capsule of the right lens was breached, with one half of the Ozurdex™ implant stuck at the entry and the other stuck at the exit wound of the posterior capsule. This patient underwent right eye cataract extraction and repositioning of the fractured implant; he made an uneventful recovery.

    CONCLUSIONS: Ophthalmologists should be aware of the potential risk of injecting an Ozurdex™ implant into an anatomical structure other than the vitreous cavity. Adequate training and careful administration of the Ozurdex™ implant are necessary to avoid such a complication, which fortunately is rare.

  6. Lim TH, Wai YZ, Chong JC
    J Med Case Rep, 2021 May 12;15(1):267.
    PMID: 33980269 DOI: 10.1186/s13256-021-02826-1
    BACKGROUND: Frosted branch angiitis (FBA) is an uncommon ocular sign with multiple causes. With the recent outbreak of coronavirus disease 2019 (COVID-19), many cases of ocular manifestation in association with this disease have been reported. However, as yet we have no complete understanding of this condition. We report here the first case of FBA in a human immunodeficiency virus-infected patient with coexisting cytomegalovirus (CMV) and COVID-19 infection.

    CASE PRESENTATION: A 33-year-old Malay man with underlying acquired immunodeficiency syndrome receiving highly active antiretroviral therapy was referred to the Opthalmology Department with complaints of blurry vision for the past 2 months. He had tested positive for and been diagnosed with COVID-19 1 month previously. Clinical examination of the fundus revealed extensive perivascular sheathing of both the artery and vein suggestive of FBA in the right eye. Laboratory testing of nasal swabs for COVID-19 polymerase chain reaction (PCR) and serum CMV antibody were positive. The patient was then admitted to the COVID-19 ward and treated with intravenous ganciclovir.

    CONCLUSION: Clinicians should be aware of and take the necessary standard precautions for possible coexistence of COVID-19 in an immunocompromised patient presenting with blurred vision, eye redness, dry eye and foreign body sensation despite the absence of clinical features suggestive of COVID-19. Whether FBA is one of the ocular signs of co-infection of COVID-19 and CMV remains unknown. Further studies are needed to provide more information on ocular signs presented in patients with concurrent COVID-19 and CMV infections.

  7. Engku-Husna EI, Nik-Ahmad-Zuky NL, Muhammad-Nashriq K
    J Med Case Rep, 2020 Nov 02;14(1):209.
    PMID: 33131504 DOI: 10.1186/s13256-020-02506-6
    BACKGROUND: Müllerian duct anomaly is a rare condition. Many cases remain unidentified, especially if asymptomatic. Thus, it is difficult to determine the actual incidence. Müllerian duct anomaly is associated with a wide range of gynecological and obstetric complications, namely infertility, endometriosis, urinary tract anomalies, and preterm delivery. Furthermore, congenital anomalies in pregnant mothers have a high risk of being genetically transmitted to their offspring.

    CASE PRESENTATION: We report a case of a patient with unsuspected müllerian duct anomaly in a term pregnancy. A 33-year-old Malay woman with previously uninvestigated involuntary primary infertility for 4 years presented with acute right pyelonephritis in labor at 38 weeks of gestation. She has had multiple congenital anomalies since birth and had undergone numerous surgeries during childhood. Her range of congenital defects included hydrocephalus, for which she was put on a ventriculoperitoneal shunt; imperforated anus; and tracheoesophageal fistula with a history of multiples surgeries. In addition, she had a shorter right lower limb length with limping gait. Her physical examination revealed a transverse scar at the right hypochondrium and multiple scars at the posterior thoracic region, levels T10-T12. Abdominal palpation revealed a term size uterus that was deviated to the left, with a singleton fetus in a nonengaged cephalic presentation. The cervical os was closed, but stricture bands were present on the vagina from the upper third until the fornices posteriorly. She also had multiple rectal prolapses and strictures over the rectum due to previous anorectoplasty. An emergency cesarean delivery was performed in view of the history of anorectoplasty, vaginal stricture, and infertility. Intraoperative findings showed a left unicornuate uterus with a communicating right rudimentary horn.

    CONCLUSION: Most cases of müllerian duct anomaly remain undiagnosed due to the lack of clinical suspicion and the absence of pathognomonic clinical and radiological characteristics. Because it is associated with a wide range of gynecological and obstetric complications, it is vital for healthcare providers to be aware of its existence and the role of antenatal radiological investigations in its diagnosis. The presence of multiple congenital abnormalities and a history of infertility in a pregnant woman should warrant the exclusion of müllerian duct anomalies from the beginning. Early detection of müllerian duct anomalies can facilitate an appropriate delivery plan and improve the general obstetric outcome.

  8. Zakaria NF, Ahmad NI, Mokhtar E, Zukiman WZHHW, Shah AM
    J Med Case Rep, 2021 Jul 30;15(1):420.
    PMID: 34325745 DOI: 10.1186/s13256-021-02895-2
    BACKGROUND: Patients who undergo peritoneal dialysis (PD) are at risk of gut bacteria translocation leading to peritonitis when there is chronic diarrhea. Chronic diarrhea is defined as any course of diarrhea that lasts at least 4 weeks, which can be continuous or intermittent. Chronic diarrhea of any duration may cause dehydration, electrolyte imbalance, and life-threatening hypovolemic shock. In PD patients, excessive ultrafiltration from the exchanges, combined with severe gastrointestinal loss, may cause hypovolemic shock, electrolyte imbalance, and metabolic acidosis. There are multiple causes of chronic diarrhea in PD patients including infective causes, mitotic lesions, and rarely the regular and excessive use of laxatives, which is a diagnosis of exclusion.

    CASE PRESENTATION: We report a case of Melanau lady with chronic diarrhea secondary to laxative usage in a patient being treated with automated peritoneal dialysis (APD). The patient went into hypovolemic shock, but luckily did not contract peritonitis. A colonoscopy revealed brown to black discoloration of the colon, a feature suggestive of melanosis coli. A biopsy of the intestine further confirmed the diagnosis by histopathological examination. Withdrawal of laxatives and the introduction of probiotics improved the symptoms tremendously.

    CONCLUSIONS: The chronic use of laxatives in PD patients can potentially lead to a devastating problem; thus, the management team must monitor treatment commencement appropriately.

  9. Nik-Ahmad-Zuky NL, Seoparjoo A, Husna EIE
    J Med Case Rep, 2021 Sep 08;15(1):448.
    PMID: 34493340 DOI: 10.1186/s13256-021-03030-x
    BACKGROUND: Placenta accreta is known to be associated with significant maternal morbidity and mortality-primarily due to intractable bleeding during abortion or delivery at any level of gestation. The complications could be reduced if placenta accreta is suspected in a patient with a history of previous cesarean delivery and the gestational sac/placenta is located at the lower part of the uterus. Then, a proper management plan can be instituted, and complications can be reduced. The diagnosis of placenta accreta in the first trimester of pregnancy is considered uncommon.

    CASE PRESENTATION: A 34-year-old Malay, gravida 4, para 3, rhesus-negative woman was referred from a private hospital at 13 weeks owing to accreta suspicion for further management. She has a history of three previous lower-segment cesarean sections. She also had per vaginal bleeding in the early first trimester, which is considered to indicate threatened miscarriage. Transabdominal ultrasound revealed features consistent with placenta accreta spectrum. She was counseled for open laparotomy and hysterectomy because of potential major complication if she continued with the pregnancy. Histopathological examination revealed placenta increta.

    CONCLUSION: A high index of suspicion of placenta previa accreta must be in practice in a patient with a history of previous cesarean deliveries and low-lying placenta upon ultrasound examination during early gestation.

  10. Baharin MF, Dhaliwal JS, Sarachandran SV, Idris SZ, Yeoh SL
    J Med Case Rep, 2016;10(1):188.
    PMID: 27356510 DOI: 10.1186/s13256-016-0944-1
    Wiskott-Aldrich syndrome is a rare X-linked disorder characterized by microthrombocytopenia, eczema, and recurrent infections. It is caused by mutations of the WAS gene. Microthrombocytopenia has been regarded as the key criteria in diagnosing this rare condition. However, in this case report, we describe a case of Wiskott-Aldrich syndrome with normal platelet size.
  11. Lu HT, Nordin R, Othman N, Choy CN, Kam JY, Leo BC, et al.
    J Med Case Rep, 2016 Aug 10;10(1):221.
    PMID: 27510438 DOI: 10.1186/s13256-016-1018-0
    BACKGROUND: Many cases of cardiac masses have been reported in the literature, but in this case report we described a rare case of biatrial cardiac mass that represented a challenge for diagnosis and therapy. The differentiation between cardiac masses such as thrombi, vegetations, myxomas and other tumors is not always straightforward and an exact diagnosis is important because of its distinct treatment strategy. Transthoracic/esophageal echocardiography and cardiac magnetic resonance play an important role in establishing the diagnosis of cardiac masses. However, no current noninvasive diagnostic tool has the ability to absolutely diagnose cardiac masses; obtaining a pathological specimen by surgical resection of cardiac masses is the only reliable method to diagnose cardiac masses accurately. Our case report is an exception in that the final diagnosis was affirmed by empirical anticoagulation therapy based on clinical judgment and noninvasive characterization of biatrial mass.

    CASE PRESENTATION: We described a 54-year-old Malay man with severe mitral stenosis and atrial fibrillation who presented with a biatrial mass. Transthoracic/esophageal echocardiography and cardiac magnetic resonance detected a large, homogeneous right atrial mass typical of a thrombus, and a left atrial mass adhering to interatrial septum that mimicked atrial myxoma. The risk factors, morphology, location, and characteristics of the biatrial cardiac mass indicated a diagnosis of thrombi. However, our patient declined surgery. As a result, the nature of his cardiac masses was not specified by histology. Of note, his left atrial mass was completely regressed by long-term warfarin, leaving a residual right atrial mass. Thus, we affirmed the most probable diagnosis of cardiac thrombi. During the course of treatment, he had an episode of non-fatal ischemic stroke most probably because of a thromboembolism.

    CONCLUSIONS: Noninvasive characterization of cardiac mass is essential in clarifying the diagnosis and directing treatment strategy. Anticoagulation is a feasible treatment when the clinical assessment, risk factors, and imaging findings indicate a diagnosis of thrombi. After prolonged anticoagulation therapy, complete resolution of biatrial thrombi was achievable in our case.

  12. Atuk FA, Basuni JBM
    J Med Case Rep, 2018 May 21;12(1):140.
    PMID: 29779493 DOI: 10.1186/s13256-018-1689-9
    BACKGROUND: While gestational trophoblastic disease is not rare, hydatidiform mole with a coexistent live fetus is a very rare condition occurring in 0.005 to 0.01% of all pregnancies. As a result of the rarity of this condition, diagnosis, management, and monitoring will remain challenging especially in places with limited resources and expertise. The case we report is an interesting rare case which presented with well-described complications; only a few similar cases have been described to date.

    CASE PRESENTATION: We report a case of a 21-year-old local Sarawakian woman with partial molar pregnancy who presented with severe pre-eclampsia in which the baby was morphologically normal, delivered prematurely, and there was a single large placenta showing molar changes.

    CONCLUSION: Even though the incidence of this condition is very rare, recognizing and diagnosing it is very important for patient care and it should be considered and looked for in patients presenting with pre-eclampsia.

  13. Tan JH, Mohamad Y, Tan CLH, Kassim M, Warkentin TE
    J Med Case Rep, 2018 May 19;12(1):131.
    PMID: 29776439 DOI: 10.1186/s13256-018-1684-1
    BACKGROUND: Symmetrical peripheral gangrene is characterized as acral (distal extremity) ischemic limb injury affecting two or more extremities, without large vessel obstruction, typically in a symmetrical fashion. Risk factors include hypotension, disseminated intravascular coagulation, and acute ischemic hepatitis ("shock liver"). In contrast, venous limb gangrene is characterized by acral ischemic injury occurring in a limb with deep vein thrombosis. Both symmetrical peripheral gangrene and venous limb gangrene present as acral limb ischemic necrosis despite presence of arterial pulses. The coexistence of symmetrical peripheral gangrene and venous limb gangrene is rare, with potential to provide pathophysiological insights.

    CASE PRESENTATION: A 42-year-old Chinese man presented with polytrauma (severe head injury, lung contusions, and right femur fracture). Emergency craniotomy and debridement of right thigh wound were performed on presentation. Intraoperative hypotension secondary to bleeding was complicated by transient need for vasopressors and acute liver enzyme elevation indicating shock liver. Beginning on postoperative day 5, he developed an acute platelet count fall (from 559 to 250 × 109/L over 3 days) associated with left iliofemoral deep vein thrombosis that evolved to bilateral lower limb ischemic necrosis; ultimately, the extent of limb ischemic injury was greater in the left (requiring below-knee amputation) versus the right (transmetatarsal amputation). As the presence of deep vein thrombosis is a key feature known to localize microthrombosis and hence ischemic injury in venous limb gangrene, the concurrence of unilateral lower limb deep vein thrombosis in a typical clinical setting of symmetrical peripheral gangrene (hypotension, proximate shock liver, platelet count fall consistent with disseminated intravascular coagulation) helps to explain asymmetric limb injury - manifesting as a greater degree of ischemic necrosis and extent of amputation in the limb affected by deep vein thrombosis - in a patient whose clinical picture otherwise resembled symmetrical peripheral gangrene.

    CONCLUSIONS: Concurrence of unilateral lower limb deep vein thrombosis in a typical clinical setting of symmetrical peripheral gangrene is a potential explanation for greater extent of acral ischemic injury in the limb affected by deep vein thrombosis.

  14. Phang ZH, Miskon MFB, Ibrahim SB
    J Med Case Rep, 2018 Jul 18;12(1):211.
    PMID: 30016981 DOI: 10.1186/s13256-018-1751-7
    BACKGROUND: Blunt trauma causing brachial artery injury in a young patient is very rare. Cases of brachial artery injury may be associated with closed elbow dislocation or instability. Elbow dislocation may not be evident clinically and radiologically on initial presentation.

    CASE PRESENTATION: This is a case of a 37-year-old, right-hand dominant, Malay man who fell approximately 6 meters from a rambutan tree and his left arm hit the tree trunk on his way down. He was an active tobacco smoker with a 20 pack year smoking history. On clinical examination, Doppler signals over his radial and ulnar arteries were poor. He proceeded with emergency computed tomography angiogram of his left upper limb which showed non-opacification of contrast at the distal left brachial artery just before the bifurcation of the left brachial artery at his left elbow joint. Radiographs and computed tomography scan also showed undisplaced fracture of left lateral epicondyle and radial head with no evidence of elbow dislocation. He subsequently underwent left brachial to brachial artery bypass which was done using reversed saphenous vein graft and recovered well. His fractures were treated using 90 degree long posterior splint for 2 weeks and he was then allowed early range of motion of the left elbow. This patient developed left elbow dislocation 6 weeks postoperatively. Closed manipulative reduction of his left elbow resulted in incomplete reduction. The functional outcome of his left elbow was limited with a range of motion of left elbow of 0-45 degrees. However, he was not keen for surgery to stabilize his elbow joint during his last follow-up 6 months post injury.

    CONCLUSIONS: This is an uncommon case of brachial artery injury in a civilian caused by blunt trauma associated with occult elbow instability/dislocation and minor fractures around the elbow joint. The treatment of brachial artery injury with clinical evidence of distal ischemia is surgical revascularization. The possibility of elbow instability and dislocation need to be considered in all cases of brachial artery injury because early radiographs and computed tomography scans may be normal. Short-term posterior splint immobilization is not sufficient to prevent recurrent dislocations.

  15. Alias H, Yong WL, Muttlib FAA, Koo HW, Loh CK, Lau SCD, et al.
    J Med Case Rep, 2018 Sep 17;12(1):276.
    PMID: 30223886 DOI: 10.1186/s13256-018-1806-9
    BACKGROUND: Acquired thrombotic thrombocytopenia purpura is very rarely encountered in children. It is often misdiagnosed initially when the condition is not inherited.

    CASE PRESENTATION: We describe a 3-year-old Malay boy who presented with simple febrile seizure and had no neurological deficit, however, he was found to have microangiopathic hemolytic anemia, thrombocytopenia, and elevated serum lactate dehydrogenase. An ADAMTS13 assay results showed zero activities (0%), and markedly high level of ADAMTS13 inhibitor (93.15 U/mL) confirming the diagnosis of secondary thrombotic thrombocytopenia purpura. He received fresh frozen plasma infusions for 3 days and subsequently his platelet levels normalized. Serial ADAMTS13 assay results showed improvement. He was also given a short course of prednisolone after which the ADAMTS13 activity normalized (> 114%) at the end of prednisolone course.

    CONCLUSIONS: At presentation, acquired thrombotic thrombocytopenia purpura in a very young child is commonly misdiagnosed as other conditions like idiopathic thrombocytopenic purpura, Evans syndrome, atypical hemolytic-uremic syndrome, or malignancy. ADAMTS13 assay should be performed early when thrombotic thrombocytopenia purpura is suspected as this condition is associated with dire consequences.

  16. Nyanti L, Samsudin A, Tiong IK
    J Med Case Rep, 2019 Jun 21;13(1):188.
    PMID: 31221202 DOI: 10.1186/s13256-019-2122-8
    BACKGROUND: Leser-Trélat syndrome, which manifests as eruptive multiple seborrheic keratoses, is a rare paraneoplastic sign. Hyponatremia in the elderly population is an often overlooked but potentially sinister biochemical abnormality. Cancer-related causes of hyponatremia include syndrome of inappropriate antidiuretic hormone secretion, cerebral or renal salt wasting, and adrenal dysfunction. We report a case of an elderly man who presented with both syndrome of inappropriate antidiuretic hormone secretion and Leser-Trélat syndrome, and was eventually found to have renal malignancy.

    CASE PRESENTATION: A 74-year-old indigenous Malaysian man with underlying chronic kidney disease presented with recurrent admissions for hyponatremia with parameters indicative of syndrome of inappropriate antidiuretic hormone secretion, constitutional symptoms, and diffuse skin lesions suggestive of multiple seborrheic keratoses. A radiological workup revealed metastatic renal cell carcinoma with evidence of metastasis to the brain, adrenal glands, bone, and lungs.

    CONCLUSIONS: To the best of our knowledge, renal malignancy presenting as syndrome of inappropriate antidiuretic hormone secretion and Leser-Trélat concurrently is rare. The causes of hyponatremia in the elderly, approach to investigation, and value as a poor prognostic marker in malignancy are highlighted. We also discuss Leser-Trélat syndrome, its pathophysiology, and its possible implications on clinical practice.

  17. Chuah SL, Jobli AT, Wan SA, Teh CL
    J Med Case Rep, 2021 Oct 19;15(1):526.
    PMID: 34663471 DOI: 10.1186/s13256-021-03103-x
    BACKGROUND: Cerebellar degeneration is a rare and severe presentation of primary Sjögren syndrome. There are few case reports of cerebellar degeneration associated with different autoimmune diseases, especially with systemic lupus erythematosus and neuro-Behcet's disease. There are only six patients reported worldwide to be affected by cerebellar atrophy associated with primary Sjögren syndrome. In this report, we describe a patient with primary Sjögren syndrome who presented with ataxia due to cerebellar degeneration.

    CASE PRESENTATION: We report the case of a 37-year-old Chinese woman with primary Sjögren syndrome who presented with ataxia over 3 months associated with tremor of the limbs. Magnetic resonance imaging of the brain revealed bilateral cerebellar atrophy. Based on the presence of cerebellar signs with magnetic resonance imaging brain findings, she was diagnosed as cerebellar degeneration secondary to primary Sjögren syndrome. She was treated with methylprednisolone, hydroxychloroquine, and two cycles of monthly intravenous cyclophosphamide. Subsequently, she refused further treatment, and her neurological symptoms remained the same upon the last clinic review. Primary cerebellar degeneration is rarely associated with primary Sjögren syndrome. The pathogenesis of the neurological manifestations in primary Sjögren syndrome is unclear. Treatment involves corticosteroids and immunosuppressive agents with no consensus of a specific therapy for the management of primary Sjögren syndrome with central nervous system involvement.

    CONCLUSIONS: Cerebellar degeneration is a rare presentation of primary Sjögren syndrome. Early diagnosis and treatment of this condition is needed to ensure a good outcome.

  18. Gouda W, Alsaqabi F, Moshrif A, Abbas AS, Abdel-Aziz TM, Islam MA
    J Med Case Rep, 2021 Oct 07;15(1):497.
    PMID: 34620236 DOI: 10.1186/s13256-021-03072-1
    BACKGROUND: Macrophage activation syndrome is classified as a secondary form of hemophagocytic lymphohistiocytosis. It is a hyperinflammatory complication observed to be comorbid with a variety of autoimmune diseases, including adult-onset Still's disease and systemic juvenile idiopathic arthritis. Macrophage activation syndrome is less commonly detected in adult patients with systemic lupus erythematosus, which, if untreated, can be fatal, though determining the optimum treatment strategy is still a challenge.

    CASE PRESENTATION: Herein, we report a case of macrophage activation syndrome in a 33-year-old Egyptian female as an unusual complication of a systemic lupus erythematosus flare in adult patients. Our patient was initially treated with a combination of intravenous methylprednisolone pulse therapy and intravenous immunoglobulin therapy, which was followed by a course of oral prednisolone and oral cyclosporine with little response. Switching from oral prednisone to intravenous dexamethasone sodium phosphate showed a more favorable clinical and biochemical response.

    CONCLUSION: Macrophage activation syndrome is less commonly detected in adult patients with systemic lupus erythematosus. Our case demonstrates that dexamethasone sodium phosphate can be a successful alternative treatment for patients with systemic lupus erythematosus complicated by macrophage activation syndrome in whom the response to pulse methylprednisolone was inadequate to manage their illness, proving to be remarkably effective in a relatively short time frame.

  19. Yii ESS, Azli AW, Sitaram PN
    J Med Case Rep, 2022 Jan 04;16(1):17.
    PMID: 34983625 DOI: 10.1186/s13256-021-03232-3
    BACKGROUND: Sodium-glucose cotransporter 2 inhibitors are among the new-generation oral antihyperglycemic agents that have been used in the treatment of type 2 diabetes mellitus. With the recent coronavirus disease 2019 pandemic and rise of cases in the third wave, diagnosis of life-threatening euglycemic diabetic ketoacidosis may easily be overlooked or missed.

    CASE PRESENTATION: We present the case of a 37-year-old Malay gentleman with underlying type 2 diabetes mellitus on empagliflozin, who presented to our hospital with symptomatic coronavirus disease 2019 infection and diabetic ketoacidosis. He developed severe rebound euglycemic diabetic ketoacidosis due to the continuous usage of empagliflozin for glycemic control alongside intravenous insulin.

    CONCLUSIONS: Physicians should have a high index of suspicion in diagnosing and managing euglycemic diabetic ketoacidosis, including withholding treatment of sodium-glucose cotransporter 2 inhibitors during the acute management of diabetic ketoacidosis.

  20. Awang MH, Hatta SFWM, Mohamad AF, Ghani RA
    J Med Case Rep, 2023 Mar 02;17(1):73.
    PMID: 36859300 DOI: 10.1186/s13256-023-03764-w
    BACKGROUND: Denosumab is known to cause abnormalities in calcium homeostasis. Most of such cases have been described in patients with underlying chronic kidney disease or severe vitamin D deficiency. Previous bariatric surgery could also contribute to hypocalcemia and deterioration in bone health.

    CASE PRESENTATION: We present a case of a 61-year-old Malay female with worsening bilateral limb weakness, paresthesia, and severe carpopedal spasm a week after receiving subcutaneous denosumab for osteoporosis. She had a history of gastric bypass surgery 20 years ago. Post gastric bypass surgery, she was advised and initiated on lifelong calcium, vitamin D, and iron supplementations that she unfortunately stopped taking 5 years after surgery. Her last serum blood tests, prior to initiation on denosumab, were conducted in a different center, and she was told that she had a low calcium level; hence, she was advised to restart her vitamin and mineral supplements. Laboratory workup revealed severe hypocalcemia (adjusted serum calcium of 1.33 mmol/L) and mild hypophosphatemia (0.65 mmol/L), with normal magnesium and renal function. Electrocardiogram showed a prolonged QTc interval. She required four bolus courses of intravenous calcium gluconate, and three courses of continuous infusions due to retractable severe hypocalcemia (total of 29 vials of 10 mL of 10% calcium gluconate intravenously). In view of her low vitamin D level of 33 nmol/L, she was initiated on a loading dose of cholecalciferol of 50,000 IU per week for 8 weeks. However, despite a loading dose of cholecalciferol, multiple bolus courses, and infusions of calcium gluconate, her serum calcium hovered around only 1.8 mmol/L. After 8 days of continuous intravenous infusions of calcium gluconate, high doses of calcitriol 1.5 μg twice daily, and 1 g calcium carbonate three times daily, her serum calcium stabilized at approximately 2.0 mmol/L. She remained on these high doses for over 2 months, before they were gradually titrated down to ensure sustainability of a safe calcium level.

    CONCLUSION: This case report highlights the importance of screening for risk factors for iatrogenic hypocalcemia and ensuring normal levels before initiating denosumab. The patient history of bariatric surgery could have worsened the hypocalcemia, resulting in a more severe presentation and protracted response to oral calcium and vitamin D supplementation.

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