Indian Journal of Case Reports
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Chief-Editor: Dr. Amit Agarwal
ISSN:(Print) 2454-129X (Online) 2454-1303
Frequency: Quarterly
Language: English
Open Access Peer-reviewed journal
Web site: https://atharvapub.net/index.php/IJCR/index
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Item A very rare case of adenomatoid hyperplasia of minor salivary gland in a 55 year old male patient(Mansa STM Publishers, 2024-10) Johannas, RE; Kadiri, S; Sameera, PRSS; Sri, Vidhya TM; Mohan, KVM.Adenomatoid hyperplasia of minor salivary glands (AHMSG) is an extremely rare non-neoplastic enlargement of minor salivary glands with uncertain pathogenesis. It is most commonly seen in males around 40–50 years of age. The most common sites are hard or soft palate. Clinically, the lesion presents as a sessile tumor-like nodule which mimics neoplasm with histological findings of benign hyperplasia and hypertrophy of mucous glands. Herein, we report a very rare case of AHMSG in a 55-year-old male patient who presented with asymptomatic swelling over the floor of the mouth since 8 months.Item Pathological complete response after neoadjuvant cyclin-dependent kinase 4/6 inhibitor and neoadjuvant endocrine therapy in locally advanced hormone receptor-positive breast cancer(Mansa STM Publishers, 2024-10) Mahajan, A; Majumdar, SK; Barik, SK; Das, DK; Parida, DK; Patnayak, R; Baisakh, MR.Breast cancer (BC) comprises different molecular subtypes, each having its own individual prognostic factors and thus having a distinct treatment algorithm. With the advent of cyclin-dependent kinase 4/6 (CDK 4/6) inhibitors, hormone receptor-positive (HR+) BC now has an additional targeted therapeutic option apart from endocrine therapy. As of now, CDK 4/6 inhibitors are Food and Drug Administration (FDA) approved in the metastatic and adjuvant settings. However, for the neoadjuvant setting, systemic chemotherapy is the standard of care. Systemic chemotherapy may not be a suitable option for elderly and frail patients in terms of associated side effects. Thus, due to tailored oncological management, many phase II and III trials of neoadjuvant CDK 4/6 inhibitors are ongoing. CDK 4/6 inhibitors are better tolerated than systemic chemotherapy and have less reported adverse events. Here, we report a case of locally advanced BC (HR+) achieving pathological complete response from neoadjuvant CDK 4/6 inhibitor with minimal toxicity. Thus, CDK 4/6 inhibitors appear to have a promising and upcoming role in the neoadjuvant setting for hormone-positive BC.Item Unveiling the Unconventional: Exploring Atypical Manifestations of Scrub Typhus(Mansa STM Publishers, 2024-10) Singh, K; Singh, I.Scrub typhus is a rickettsial illness caused by Orientia tsutsugamushi. It is a common cause of undifferentiated febrile illness, especially in the Indian subcontinent, usually with multiorgan involvement, though central nervous involvement is rare. Here, we demonstrate a case of a young female who presented with fever, headache, vomiting, and seizures. Her examination was remarkable for generalized blanchable macular rash, opsoclonus–myoclonus along with neck rigidity and mild epigastric tenderness. Contrast-enhanced magnetic resonance imaging brain was suggestive of leptomeningitis. She also had mild pancreatitis, transaminitis, thrombocytopenia, and transaminitis. Given the ongoing tropical fever burden in Northern India, further workup was done and enzyme-linked immunosorbent assay anti-scrub typhus immunoglobulin M came as positive. Doxycycline along with supportive treatment was started, and all signs and symptoms improved gradually.Item Erupting mandibular second molar concomitant with complex odontome and supernumerary tooth in a 13-year-old boy: A case report(Mansa STM Publishers, 2024-10) Krishnan, MA; Vidhya Vijayan, MC; Sreejith, VP; Puthalath, U; Tom, JJ.Odontomas are the most frequent hamartomatous developmental abnormality. The condition frequently correlates with one or more unerupted teeth and is often detected through the failure of teeth to erupt at the expected time. Although most cases were discovered that were impacted within the jaw, there are scenarios where odontomas have erupted into the oral cavity. The majority of odontomas are asymptomatic; however, these anomalies can cause delayed eruption, impaction, or even result in the retention of primary teeth. Odontomas are atypical calcified aggregations of dental tissues such as enamel, dentin, pulp, and cementum and originate from odontogenic epithelium thereby, classifying them as mixed odontogenic tumors. Literature suggests that their etiology can be local trauma, infection, or genetic mutations, yet the precise cause remains unknown. This case report presents a complex odontome and a supernumerary tooth associated with a partially erupted second molar in a 13-year-old patient with radiological manifestations and surgical management.Item A full thickness and partial rotator cuff tear with retraction of fibers and reconstruction with palmaris longus graft(Mansa STM Publishers, 2024-11) Basha, MS; Dharani, NV.The report mainly appraises full-thickness tears at myotendinous junctions which are rare and only described in the supraspinatus and infraspinatus muscles. There is very little literature describing both tears occurring concurrently. After suffering mechanical damage to her shoulder, a 64-year-old woman was examined for pain in the shoulder on movement; the patient underwent radiographic and clinical evaluation which revealed full-thickness rotator cuff tears with retraction of fibers. Hence, the plan of care is to offer the best surgical techniques to guarantee a good result and a quick recovery. A full-thickness tear at the myotendinous junctions of the supraspinatus tear and partial tear at the subscapularis tendon and infraspinatus tendon at humeral attachment was confirmed radiographically in this case. To make a diagnosis, a high level of clinical suspicion must be supported by specific magnetic resonance imaging characteristics.Item Isolated dextrocardia and rheumatoid arthritis with mitral stenosis with pulmonary hypertension with atrial fibrillation: A rare association(Mansa STM Publishers, 2024-01) Shashidhara, KC; Chukkapalli, SS; Duggirala, AC; Madala, SS; Prabhu, PP.This case presents a rare occurrence of rheumatoid arthritis (RA) in a 50-year-old woman with concurrent mitral valvular disease and dextrocardia. Diagnosed with seropositive RA 5 years earlier, the patient exhibited fever, cough, and progressive dyspnea. Clinical examination revealed irregular tachycardia, elevated jugular venous pressure, and signs of heart failure. Laboratory results confirmed seropositive RA, anemia, and elevated inflammatory markers. Electrocardiogram and echocardiogram indicated dextrocardia, atrial fibrillation, and moderate mitral stenosis. This case highlights the unusual association of RA with valvular heart disease and the added complexity of dextrocardia. Recognizing diverse cardiac manifestations in RA is crucial, contributing to the growing evidence linking autoimmune disorders to cardiovascular complications. Further research is essential to understand the intricate relationship between RA and unique cardiac abnormalities.Item Inverted and impacted mandibular third molar: A rare case report(Mansa STM Publishers, 2024-10) Sreejith, VP; Puthalath, U; Tom, JJ; Krishnan, MA; Vidhya, Vijayan MC.A tooth is considered to be impacted when it fails to erupt fully or emerge partially. Impaction is most common with wisdom teeth because they are the last tooth to erupt in the oral cavity. An impacted tooth is embedded within soft tissue or bone for various reasons. Tooth may also become twisted, tilted, or displaced as they try to emerge in closed space resulting in impaction. A tooth is said to be inversed when it is placed reversed and seated upside down. In the mandible, ascending ramus is the most common site for third molars. Inversion of the impacted tooth is a rare condition. In this literature, a very rare case of inverted and impacted mandibular third molar has been reported in a 28-year-old female patient with swelling and pus discharge, considering the associated symptoms, surgical removal was performed.Item Primary Ewing Sarcoma of Kidney in an Adult: An Oddball(Mansa STM Publishers, 2024-10) Mahajan, N; Mahanta, N; Ray, A; Phukan, M; Saini, N.Ewing sarcoma (ES) is a malignant bone and soft-tissue tumor derived from neuroectoderm. It occurs, mostly, in pediatric adolescents with the histopathological features of invasiveness. The occurrence of ES of the kidney is rare, comprising only 1% of all renal tumors which are limited to case reports and small case series. Herein, we report a case of a 44-year-old male patient, presenting with the right flank pain and hematuria. Computed tomography scan demonstrated a right midpolar exophytic renal mass with intraluminal right renal vein and inferior vena cava (IVC) thrombus without visceral metastasis suggesting the diagnosis of renal cell carcinoma (T3BN0M0). The patient underwent the right radical nephrectomy with IVC thrombectomy. In view of the aggressiveness of the tumor, the patient received adjuvant chemotherapy (i.e., four cycles of vincristine, adriamycin, and cyclophosphamide alternating every 3 weeks with four cycles of ifosphamide and etoposide). The patient had near-complete resolution of the tumor with no thrombus seen in the follow-up scan. We could not complete six cycles of chemotherapy which were planned initially due to recurrent cytopenias. Six months postchemotherapy, the patient is clinically and radiologically disease-free and is in close follow-up. We are reporting this case to highlight the rarity of this entity and its challenging clinicopathological diagnosis when presenting as a renal tumor.Item Carcinoma buccal mucosa with cutaneous metastasis: An unusual presentation(Mansa STM Publishers, 2024-10) Singhal, L; Gupta, RK; Bhatnagar, M; Paul, B; Agrawal, D.Head-and-neck cancer is a common cancer in India. Oral cavity cancer accounts for 50% of all head and neck sites. The most common sites of distant metastasis are the lung, liver, and bone. The incidence of cutaneous metastasis is a very rare site. We report a case of early-stage carcinoma buccal mucosa post-surgery and adjuvant radiotherapy developed skin metastasis shortly after completion of treatment. A 37-year-old female non-smoker, non-alcoholic diagnosed with early-stage carcinoma right buccal mucosa cT1N0M0. She underwent surgery of wide local excision with marginal mandibulectomy and extended supra-omohyoid neck dissection. In post-surgery, the histopathological report was moderately differentiated squamous cell carcinoma, pT2N1Mx. She completed her adjuvant radiotherapy 59.4Gy/33 fractions with six cycles of concurrent chemotherapy cisplatin 40 mg/m2. She developed difficulty in swallowing, increased oral secretions, and thickening over right-sided scar marks shortly after 10 weeks of completion of treatment. Rapidly, she developed multiple cutaneous nodules over both sides of the entire face and neck. Dermal biopsy reveals metastatic squamous cell carcinoma. Skin metastasis from head-and-neck malignancy is an uncommon entity and in the early stage, it is very rare. Initially, it is difficult to diagnose as it looks like some disseminated bacterial or fungal infection but we should always keep in mind this entity as cancer can metastasize and present in any atypical forms.Item A case report of arthrocentesis in temporomandibular joint internal derangement(Mansa STM Publishers, 2024-10) Thanseeh, M; Rohith, R; Sreejith, VP; Puthalath, U; Tom, JJ.In this case report, a 56-year-old male presented to our department of oral and maxillofacial surgery with a chief complaint of pain during mouth opening on the left joint region. The patient also complained of limited mouth opening and temporomandibular joint (TMJ) pain. Arthrocentesis was performed under aseptic conditions. Clinical evaluation of the patients was done before the procedure, 1 week, 1 month, and 4 months postoperatively. Intensity of TMJ pain and maximal mouth opening were recorded at each follow-up visit. There was a significant improvement in mouth opening and a reduction in pain scores in the postoperative period. Arthrocentesis is a simple and safe procedure for patients with internal derangement with a closed lock for improving mouth opening and decreasing pain.Item Primary pancreatic hydatid cyst; typical radiological signs: A case report(Mansa STM Publishers, 2024-10) Mohsen, S; Mohammed, N; Dakika, MA; Gawargios, FA.Pancreatic hydatid cysts are rare, especially in pediatric populations, and their diagnosis presents a remarkable challenge because of the non-specific clinical presentation and the limitations of available diagnostic tools, particularly in resource-limited settings. Our case highlights the importance of maintaining a high index of suspicion for hydatid disease, even in pediatric patients presenting with non-specific abdominal symptoms, particularly in rural regions, where diagnostic resources may be limited. Early recognition and accurate diagnosis are crucial for guiding appropriate therapeutic interventions and preventing potential complications associated with this rare but clinically significant entity.Item Multidisciplinary management of a patient with eyelid ecthyma gangrenosum due to Pseudomonas aeruginosa infection(Mansa STM Publishers, 2024-09) Gallego-Amorós, M; Barba, IT; Suárez, AH; Rodríguez, ER; Cortés, SP.Pseudomonas aeruginosa is a Gram-negative bacillus, responsible for frequently serious infections that cause high morbidity and mortality, especially in immunosuppressed hosts or with underlying malignant processes. Ecthyma gangrenosum is a highly suggestive, though not pathognomonic, manifestation of disseminated P. aeruginosa infection. The palpebral presentation of the latter is estimated to be <6%, so initial clinical suspicion and early multidisciplinary management are of great importance. This article aims to show the diagnostic and therapeutic challenge of a patient who abruptly presents septicemia by Pseudomonas, accompanied by severe periocular skin symptoms in a tertiary hospital.Item Management of a Case of Recurrent Cutaneous Adenoid Cystic Carcinoma of the Scalp with Radiation Therapy(Mansa STM Publishers, 2024-09) Gupte, A; Jaiswal, P; Kakade, A; Dandekar, PR.Primary cutaneous adenoid cystic carcinoma (PCACC) is a rare tumor originating within the skin, typically presenting as a solid, gradually enlarging nodule or mass. We report a unique case of recurrent PCACC of the scalp in a 90-year-old male, initially diagnosed in 2010 and managed with surgical excision. Despite initial treatment, the lesion recurred twice, necessitating re-irradiation due to ineligibility for surgical resection at the second recurrence. We discuss the treatment modalities and outcomes of this case, highlighting the importance of a multidisciplinary approach in managing rare malignancies. Our case underscores the feasibility and efficacy of radiotherapy as a palliative treatment option for recurrent PCACC, particularly in patients unsuitable for surgical intervention.Item Silent giant: A case report of retroperitoneal schwannoma(Mansa STM Publishers, 2024-09) Saini, R; Singh, P; Yadav, R; Kodela, J.Schwann cells of the peripheral nerve sheath give birth to soft-tissue tumors known as schwannomas (neurilemmomas). Schwannomas of the retroperitoneum are uncommon. Diagnosis is frequently delayed in view of unspecific clinical symptoms and non-typical radiologic findings. The preferred course of therapy is total surgical excision. The prognosis is favorable; however, further monitoring is required due to the possibility of recurrence and malignant change. In this case report, we describe a case of retroperitoneal schwannoma in a 44-year-old male and examine the pertinent research.Item Complex presentation of atypical hemolytic uremic syndrome: A case study(Mansa STM Publishers, 2024-09) Gangireddy, VR; Varshitha, BV; Annapureddy, SR; Chadarajupalli, B; Yalamuri, RR.This case study discusses atypical hemolytic uremic syndrome (aHUS) in a 67-year-old female who presented with hypertension and hyperlipidemia 8 days after a suspected foodborne illness. Her initial presentation included acute respiratory and renal failure in which her condition required extensive re-warming, intubation, pressor or vasopressor therapy, continuous renal replacement therapy or hemodialysis, broad-spectrum antibiotic therapy, and blood and culture workups. Kidney function also showed signs of renal failure, metabolic acidosis, thrombocytopenia, and an elevated level of lactate dehydrogenase in the laboratory results. Radiological investigations demonstrated enteritis, colitis, and an ischemic infarct without hemorrhage. Multidisciplinary consultations achieved the importance and urgency of early diagnosis and treatment of aHUS to prevent irreversible organ damage. This case highlights the role of a timely diagnosis and interprofessional patient care in the treatment of severe aHUS.Item A rare case of drug-induced agranulocytosis: A challenging diagnosis andmanagement approach(Mansa STM Publishers, 2024-09) Ali, M; Viqar, U.Agranulocytosis is a severe adverse drug reaction characterized by a significant decrease in the number of neutrophils, making patients highly susceptible to infections. I present a rare case of drug-induced agranulocytosis in a 45-year-old male patient who presented with a sudden onset of fever, sore throat, and severe fatigue. Infectious and autoimmune etiologies were ruled out by thorough examinations. A thorough medication history indicated that a new drug for the treatment of hypertension had just been started (chlorthalidone). The patient was immediately placed on broad-spectrum antibiotics and granulocyte colony-stimulating factor after the medication was rapidly stopped. The patient’s neutrophil count gradually increased with prompt management, and he displayed clinical improvement. This instance emphasizes the value of prompt diagnosis and treatment of drug-induced agranulocytosis to avert potentially fatal consequences.Item Laparoscopic low anterior resection of rectum: A case report with review ofliterature(Mansa STM Publishers, 2024-09) Shandilaya, U; Joshi, A.Low anterior resection (LAR) is the standard operative treatment for rectal cancer. Laparoscopic anterior resections were first attempted in the 1990s. With the passage of time, laparoscopic instrumentation, energy sources, and laparoscopic techniques underwent a significant improvement. With all the above advancements, at the turn of the 20th century, there emerged strong evidence in the literature in favor of laparoscopic therapy for rectal cancer. Large-volume multicenter studies, various randomized controlled trials, and many review analyses of large databases, for example, Cochrane; conclusively showed that laparoscopy was not inferior to open surgery in oncological completeness of the resection; in colorectal cancer. Given its cosmetic superiority, lesser post-operative pain, and earlier resumption of routine day-to-day activity, laparoscopic radical colorectal cancer surgery has become standard practice in advanced laparoscopic health-care setups all over the world. Herein, we present the case of a 77-year-old gentleman who was diagnosed with low rectal cancer, underwent a laparoscopic LAR, and is disease-free until date, on serial surveillance investigations; over a post-operative follow-up period of just over 9 years.Item A rare case of adult rhabdomyosarcoma with adnexal metastasis responding tonon-conventional chemotherapy(Mansa STM Publishers, 2024-09) Sinha, N; Porwal, J; Sehrawat, A.Rhabdomyosarcoma (RMS) is a pediatric soft tissue sarcoma having a rare incidence among adults. It is a highly chemo-radiosensitive tumor among the pediatric population but has poor biology in adults with dismal outcomes. Due to the rarity of adult RMS, there are no well-defined treatment guidelines for adult RMS and are mostly managed with pediatric-defined protocols but lack similar response. Here, we present the case of an adult female with extremely atypical metastatic site RMS which showed an extraordinary response to non-pediatric protocol, rather than adult sarcoma-based protocol. This might give an idea to have more research and trials in the future to implement adult sarcoma-based therapy among adult RMS cases.Item Hypokalemic paralysis and proximal renal tubular acidosis secondary to tenofovir-induced Fanconi syndrome: A contributing cause of death(Mansa STM Publishers, 2024-09) Karve, M; Suryawanshi, VR; Attarde, G; Asad, AH; Purandare, B.Nucleotide reverse transcriptase inhibitors (NRTIs, primarily cidofovir and adefovir, less likely tenofovir) pose a well-known risk of nephrotoxicity. Acute renal failure, nephrogenic diabetes insipidus (NDI), and Fanconi syndrome (FS) are the primarily reported renal adverse effects associated with NRTIs. In this report, we describe a case of a 52-year-old female who presented to the outpatient department of a tertiary-care facility with polyuria, polydipsia, convulsions, myalgias, and hypokalemic paralysis (quadriparesis) after 10 months exposure to tenofovir disoproxil fumarate (TDF), which she was taking for HIV and Hepatitis-B virus coinfection. The patient developed hypokalemic paralysis and proximal renal tubular acidosis (pRTA) with normal anion gap hyperchloremic metabolic acidosis. On evaluation, renal tubulopathy was evident, which was resolved post-TDF discontinuation and therapeutic corrections. A diagnosis of TDF-induced FS, pRTA, and NDI were considered, as there were no clear alternative explanations. This report also highlights the characteristics of TDF-induced FS from the published case reports in the Indian context.Item Diagnosis on dilemma: Arteriovenous malformation with scar pregnancy with old retained product of conception(Mansa STM Publishers, 2024-09) Chandanan, A; Swaroop, N; Saxena, A; Prasad, A; Varshney, A; Chaudhari, JJ; Garhwal, P.Cesarean scar pregnancy (CSP) is described as the placement of a gestational sac within the scar from a previous cesarean operation and is considered an ectopic pregnancy. Ectopic pregnancy situated in a cesarean section scar is a rare but potentially life-threatening event. Because of its rarity, there are no universal guidelines to manage this condition. CSP has no pathognomonic signs or symptoms and its presentation varies considerably. Here, we report a case of scar pregnancy containing an old retained product of conceptus.