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Fulltext Giant epidermal cyst with intramuscular extension: a rare occurrence

Low SF, Sridharan R, Ngiu CS

BMJ Case Rep, 2015 Feb 06;2015.
PMID: 25661748 DOI: 10.1136/bcr-2013-202534

An epidermal cyst is the most common type of cyst to occur in subcutaneous tissue. When its size is greater than 5 cm, it is recognised as a giant epidermal cyst. A subcutaneous giant epidermal cyst with intramuscular extension is extremely rare. The authors report a case of a 74-year-old man who presented with a painless, slow-growing left gluteal mass of 6-month duration. Examination revealed a large left gluteal mass that was fixed to the underlying structures. A small epidermal cyst with visible punctum was noted at the medial aspect of the mass. MRI demonstrated a large, lobulated left gluteal lesion measuring 20 cm×16 cm×10 cm. The lesion was partly within the gluteal maximus muscle and partly within the subcutaneous tissue. MRI and ultrasound features of the lesion were consistent with a giant epidermal cyst with intramuscular extension. The lesion was excised and histology confirmed the diagnosis.
Fulltext Camurati-engelmann disease association with hypogonadism and primary hypothyroidism

Low SF, Abu Bakar N, Ngiu CS

Iran Red Crescent Med J, 2014 Aug;16(8):e9481.
PMID: 25389497 DOI: 10.5812/ircmj.9481

INTRODUCTION: Camurati-Engelmann disease (CED) is a rare autosomal dominant disease with various phenotypic expressions. The symptoms usually develop during childhood. The hallmark of the disease is bilateral symmetric diaphyseal hyperostosis of the long bones with progressive involvement of the metaphysis. The epiphysis is strictly spared. The common clinical symptoms are pain of the extremities, muscle wasting, waddling gait, and lethargy. CED is rarely seen in conjunction with hypogonadism. CED-associated hypothyroidism has not been reported yet. Clinical assessment and skeletal survey are important to make the diagnosis.
CASE PRESENTATION: Hereby we reported a case of CED with concomitant hypogonadism and hypothyroidism. Serial plain radiographs of the patient showed classic and progressive diaphyseal cortical hyperostosis of the long bone.
CONCLUSIONS: Hyperostosis of the skull was observed in the present case. The characteristic osseous changes of CED were highlighted and the differential diagnoses were discussed.
Fulltext Osseous haemophilic pseudotumour and concurrent primary hyperparathyroidism: a diagnostic conundrum

Low SF, Sridharan R, Ngiu CS, Haflah NH

BMJ Case Rep, 2014;2014.
PMID: 24729114 DOI: 10.1136/bcr-2013-203282

Pseudotumours are rare, occurring in 1-2% of severe haemophiliacs. Osseous locations are far less frequent than soft tissue location. We report a case of a 43-year-old man with haemophilia A, who presented with a gradually enlarging left thigh mass for 8 months. There were no constitutional symptoms. Plain radiograph showed an expansile lytic lesion with 'soap-bubble' appearance arising from the left femur diaphysis. On MRI, it appeared as a non-enhancing, multilobulated lesion expanding the medullary and subperiosteal spaces. The mass exhibited concentric ring sign with heterogeneous intermediate signal intensity in the core lesion, reflective of chronic haematoma with blood degradation products of different stages. A diagnosis of haemophilic pseudotumour was made. Hypercalcaemia, however, raised a diagnostic dilemma as bone malignancy needed to be considered. An open excisional biopsy and subsequent amputation confirmed the diagnosis of osseous haemophilic pseudotumour. Nuclear medicine study later revealed a concurrent parathyroid adenoma.
Fulltext Midgut malrotation with congenital peritoneal band: a rare cause of small bowel obstruction in adulthood

Low SF, Ngiu CS, Sridharan R, Lee YL

BMJ Case Rep, 2014;2014.
PMID: 24717859 DOI: 10.1136/bcr-2013-202690

Midgut malrotation commonly presents in the neonatal period, and rarely manifests its symptoms in adulthood with an estimated incidence of 0.2-0.5%. Nevertheless, the symptoms are non-specific with no strong pointers towards the clinical diagnosis. Consequently, the diagnosis is usually disclosed with imaging or surgery. We report a case of small bowel obstruction secondary to a congenital peritoneal band with underlying midgut malrotation in a 48-year-old man.
Fulltext Probable hepatic tuberculosis masquerading as Klatskin tumour in an immunocompetent patient

Hanafiah M, Alhabshi SM, Bag T, Low SF

BMJ Case Rep, 2013;2013.
PMID: 24343799 DOI: 10.1136/bcr-2013-010262

A 55-year-old immunocompetent woman was presented with features of obstructive jaundice and a clinicoradiological picture suggestive of a hilar cholangiocarcinoma (Klatskin tumour). However, caseating granulomatous lesion associated with miliary nodules were revealed intraoperatively. The lesion responded to standard antituberculous therapy. This unusual presentation highlights the considerable diagnostic challenge in such case.
Fulltext Severe thrombocytopenia due to hypersplenism treated with partial splenic embolisation

Hanafiah M, Shahizon AM, Low SF, Shahrina MH

BMJ Case Rep, 2013 Jul 05;2013.
PMID: 23833091 DOI: 10.1136/bcr-2013-010163

A 35-year-old woman with background of liver cirrhosis and portal hypertension secondary to chronic hepatitis C presented with complication of hypersplenism and thrombocytopenia. She developed severe menorrhagia requiring multiple blood transfusions. In addition, her interferon therapy was withheld owing to the underlying thrombocytopenia. Partial splenic embolisation was performed, which improved her platelet counts. Subsequently, the menorrhagia was resolved and her interferon therapy was restarted.
Fulltext Spinal neurocytoma with extensive syringohydromyelia

Hanafiah M, Low SF, Sridharan R, Young B

BMJ Case Rep, 2013;2013.
PMID: 24132448 DOI: 10.1136/bcr-2013-201285
Fulltext Challenges in imaging and histopathological assessment of a giant cell tumour with secondary aneurysmal cyst in the patella

Low SF, Hanafiah M, Nurismah MI, Suraya A

BMJ Case Rep, 2013;2013.
PMID: 24057334 DOI: 10.1136/bcr-2013-200790

The patella is an uncommon site for all primary and metastatic bone tumours and primary intra-osseous tumours of the patella are very rare. A majority of the patella tumours are benign. We report a patient with a sudden onset swelling and pain of the right knee following a staircase fall. The plain radiograph showed an expansile multiseptated patella lesion and it was further assessed with an MRI. The radiological findings and the initial histopathological features from a limited sample were suggestive of a primary aneurysmal bone cyst. However, the final histopathological diagnosis from a more adequate specimen was a giant cell tumour with a secondary aneurysmal bone cyst.
Fulltext Nocardiosis in a patient with rheumatoid arthritis treated with rituximab and a summary of reported cases

Ngiu CS, Said MS, Periyasamy P, Low SF

BMJ Case Rep, 2010;2010.
PMID: 22778377 DOI: 10.1136/bcr.11.2009.2421

Rituximab is a B-cell-depleting monoclonal anti-CD20 antibody. It is widely used in haematology and rheumatology. However, usage of rituximab in immunosupressed patient has been associated with various opportunistic infections. The authors reported a case of refractory rheumatoid arthritis treated with rituximab, which later presented with non-resolving pneumonia with pulmonary nodule. Percutaneous computer tomogram guided lung biopsy was arranged to confirm the suspicion of tuberculosis, but did not yield conclusive results. Later, she presented left-chest abscess and underwent incision and drainage. The pus culture and sensitivity confirmed pulmonary nocardiosis with chest wall dissemination. She was treated with 2-week course of trimethoprim sulfamethoxazole and responded. The authors also reviewed published cases of nocardiosis post-rituximab.
Fulltext A CASE OF INFILTRATING LOBULAR CARCINOMA OF THE BREAST WITH GASTRIC METASTASIS 22 YEARS AFTER INITIAL SURGERY

Hanafiah M, Sidek S, Low SF, Ngiu CS

Acta Clin Croat, 2021 Mar;60(1):136-140.
PMID: 34588734 DOI: 10.20471/acc.2021.60.01.20

Gastric metastasis from breast carcinoma is uncommon and rarely encountered in our daily practice. We report a case of late gastric metastasis from previous infiltrating lobular carcinoma of the breast. The patient had extended disease-free interval of 22 years prior to metastasis. Gastroscopy and fluoroscopy showed changes appearing like linitis plastica of the body of the stomach. Computed tomography of the thorax and abdomen demonstrated pulmonary metastasis and mediastinal lymphadenopathy. Biopsy of the mediastinal lymph node and stomach lesion confirmed metastatic adenocarcinoma from breast carcinoma based on immunohistochemistry staining. Immunohistochemistry staining of both specimens revealed strong positivity for cytokeratin 7 and negative for cytokeratin 20.
Fulltext Osteochondrogenesis with Autologous Peripheral Blood Stem Cells for Osteochondral Lesions of the Talus: Report of Five Cases

Saw KY, Jee C, Ramlan A, Dawam A, Saw YC, Low SF

Malays Orthop J, 2022 Nov;16(3):128-131.
PMID: 36589370 DOI: 10.5704/MOJ.2211.019

Osteochondral lesions of the talus (OLTs) may progress to ankle arthritis needing ankle arthroplasty or arthrodesis. We report five cases of OLTs treated along the principles developed for chondrogenesis of the knee joint with autologous peripheral blood stem cells (PBSCs), resulting in repair and regeneration of the bone and cartilage components. Improvement in Ankle Osteoarthritis Scale (AOS) scores with minimum two years follow-up showed statistical significance (p < 0.05).
Fulltext Intracavernous internal carotid artery pseudoaneurysm

Sridharan R, Low SF, Mohd MR, Kew TY

Singapore Med J, 2014 Oct;55(10):e165-8.
PMID: 25631906

Epistaxis is commonly encountered in otorhinolaryngologic practice. However, severe and recurrent epistaxis is rarely seen, especially that originating from a pseudoaneurysm of the intracavernous internal carotid artery (ICA). We herein present the case of a 32-year-old man who was involved in a motor vehicle accident and subsequently developed recurrent episodes of profuse epistaxis for the next three months, which required blood transfusion and nasal packing to control the bleeding. Computed tomography angiography revealed a large intracavernous ICA pseudoaneurysm measuring 1.7 cm × 1.2 cm × 1.0 cm. The patient underwent emergent four-vessel angiography and coil embolisation and was discharged one week later without any episode of bleeding. He remained asymptomatic after three-month and one‑year intervals. This case report highlights a large intracavernous ICA pseudoaneurysm as a rare cause of epistaxis, which requires a high index of suspicion in the right clinical setting and emergent endovascular treatment to prevent mortality.
Dendritic fibromyxolipoma: a variant of spindle cell lipoma with extensive myxoid change, with cytogenetic evidence

Wong YP, Chia WK, Low SF, Mohamed-Haflah NH, Sharifah NA

Pathol. Int., 2014 Jul;64(7):346-51.
PMID: 25047505 DOI: 10.1111/pin.12176

Dendritic fibromyxolipoma (DFML), a rare, recently described distinct benign soft tissue tumor, has many clinicopathological features reminiscent of spindle cell lipoma and solitary fibrous tumor with myxoid change. It is distinguished histologically from both entities by the presence of spindle and stellate cells with dendritic cytoplasmic prolongations, prominent myxoid stroma with abundant keloidal collagen and occasional small plexiform vascular proliferation. We describe a case of histologically confirmed DFML of the left shoulder in a 67-year-old male, in which subsequent cytogenetic analysis revealed deletion involving 13q14.3 region in all the tumor cells, typically detected in spindle cell lipoma. In the presence of many clinicopathological similarities between DFML and spindle cell lipoma including chromosomal abnormalities, we postulate that DFML is merely a rare variant of spindle cell lipoma with extensive myxoid degeneration, and may not be considered as a separate entity. The possible differential diagnosis and their distinguishing features are briefly discussed.
Fulltext Interleukin-23 and its correlation with disease activity, joint damage, and functional disability in rheumatoid arthritis

Dalila AS, Mohd Said MS, Shaharir SS, Asrul AW, Low SF, Shamsul AS, et al.

Kaohsiung J. Med. Sci., 2014 Jul;30(7):337-42.
PMID: 24924839 DOI: 10.1016/j.kjms.2014.02.010

The purpose of this study was to compare the serum interleukin (IL)-23 levels between rheumatoid arthritis (RA) patients and healthy controls and to determine the correlation of IL-23 levels with disease activity, joint damage and functional disability in RA. Serum samples were obtained from 45 patients with RA and 45 healthy controls. The enzyme-linked immunosorbent assay method was used for quantitative analysis of IL-23. All the RA patients were assessed for disease activity based on the 28-joint disease activity score, joint damage based on modified Sharp score, and functional ability using the Health Assessment Questionnaire-Disability Index. The mean serum IL-23 level was much higher among the RA patients (24.50 ± 13.98 pg/mL) compared to the controls (5.98 ± 3.40 pg/mL; p < 0.01). There was a significant positive relationship between IL-23 levels and disease activity and questionnaire scores (p = 0.003 and 0.020, respectively). On logistic regression analysis, IL-23 levels were significantly higher in patients with moderate to high disease activity (p = 0.008, odds ratio = 1.073, 95% confidence interval = 1.019-1.130) and patients with significant functional disability (p = 0.008, odds ratio = 1.085, 95% confidence interval = 1.021-1.153). RA patients have significantly higher levels of serum IL-23. The IL-23 levels correlate well with disease activity and functional disability but not with radiographic joint damage.

Study site: Rheumatology clinic, Pusat Perubatan Universiti Kebangsaan Malaysia (PPUKM)
Fulltext Non Secretory Multiple Myeloma With Extensive Extramedullary Plasmacytoma: A Diagnostic Dilemma

Low SF, Mohd Tap NH, Kew TY, Ngiu CS, Sridharan R

Iran J Radiol, 2015 Jul;12(3):e11760.
PMID: 26528383 DOI: 10.5812/iranjradiol.11760v2

Multiple myeloma (MM) is characterized by progressive proliferation of malignant plasma cells, usually initiating in the bone marrow. MM can affect any organ; a total of 7 - 18% of patients with MM demonstrate extramedullary involvement at diagnosis. Non-secretory multiple myeloma (NSMM) is a rare variant that accounts for 1 - 5% of all cases of multiple myeloma. The disease is characterized by the absence of monoclonal gammopathy in serum and urine electrophoresis. Our case report highlights the diagnostic challenge of a case of NSMM with extensive extramedullary involvement in a young female patient who initially presented with right shoulder pain and bilateral breasts lumps. Skeletal survey showed multiple lytic bony lesions. The initial diagnosis was primary breast carcinoma with osseous metastases. No monoclonal gammopathy was found in the serum or urine electrophoresis. Bone marrow and breast biopsies revealed marked plasmacytosis. The diagnosis was delayed for a month in view of the lack of clinical suspicion of multiple myeloma in a young patient and scant biochemical expression of non-secretory type of multiple myeloma.
Fulltext Osteochondral Regeneration in the Knee Joint with Autologous Peripheral Blood Stem Cells plus Hyaluronic Acid after Arthroscopic Subchondral Drilling: Report of Five Cases

Saw KY, Anz AW, Jee CS, Low SF, Dawam A, Ramlan A

Orthop Surg, 2024 Feb;16(2):506-513.
PMID: 38087402 DOI: 10.1111/os.13949

BACKGROUND: Treatment of osteochondral defects (OCDs) of the knee joint remains challenging. The purpose of this study was to evaluate the clinical and radiological results of osteochondral regeneration following intra-articular injections of autologous peripheral blood stem cells (PBSC) plus hyaluronic acid (HA) after arthroscopic subchondral drilling into OCDs of the knee joint.
CASE PRESENTATION: Five patients with OCDs of the knee joint are presented. The etiology includes osteochondritis dissecans, traumatic knee injuries, previously failed cartilage repair procedures involving microfractures and OATS (osteochondral allograft transfer systems). PBSC were harvested 1 week after surgery. Patients received intra-articular injections at week 1, 2, 3, 4, and 5 after surgery. Then at 6 months after surgery, intra-articular injections were administered at a weekly interval for 3 consecutive weeks. These 3 weekly injections were repeated at 12, 18 and 24 months after surgery. Each patient received a total of 17 injections. Subjective International Knee Documentation Committee (IKDC) scores and MRI scans were obtained preoperatively and postoperatively at serial visits. At follow-ups of >5 years, the mean preoperative and postoperative IKDC scores were 47.2 and 80.7 respectively (p = 0.005). IKDC scores for all patients exceeded the minimal clinically important difference values of 8.3, indicating clinical significance. Serial MRI scans charted the repair and regeneration of the OCDs with evidence of bone growth filling-in the base of the defects, followed by reformation of the subchondral bone plate and regeneration of the overlying articular cartilage.
CONCLUSION: These case studies showed that this treatment is able to repair and regenerate both the osseous and articular cartilage components of knee OCDs.
Role of high resolution ultrasound in ulnar nerve neuropathy

Radhika S, Lee YL, Low SF, Fazalina MF, Sharifah Majedah IA, Suraya A, et al.

Med J Malaysia, 2015 Jun;70(3):158-61.
PMID: 26248778 MyJurnal

AIM: This study was conducted to measure the cross sectional area (CSA) of the ulnar nerve (UN) in the cubital tunnel and to evaluate the role of high-resolution ultrasonography in the diagnosis of ulnar nerve neuropathy (UNN).
MATERIALS AND METHODS: This was a cross sectional study with 64 arms from 32 patients (34 neuropathic, 30 nonneuropathic). Diagnosis was confirmed by nerve conduction study and electromyography. The ulnar nerves were evaluated with 15MHz small footprint linear array transducer. The ulnar nerve CSA was measured at three levels with arm extended: at medial epicondyle (ME), 5cm proximal and 5cm distal to ME. Results from the neuropathic and nonneuropathic arms were compared. Independent T-tests and Pearson correlation tests were used. P value of less than 0.05 was considered significant.
RESULTS: Mean CSA values for the UN at levels 5cm proximal to ME, ME and 5cm distal to ME were 0.055, 0.109, 0.045 cm(2) respectively in the neuropathic group and 0.049, 0.075, 0.042 cm2 respectively in the non-neuropathic group. The CSA of the UN at the ME level was significantly larger in the neuropathic group, with p value of 0.005. However, there was no statistical difference between the groups at 5cm proximal and distal to the ME, with p values of 0.10 and 0.35 respectively.
CONCLUSION: There is significant difference in CSA values of the UN at ME between the neuropathic and non-neuropathic groups with mean CSA value above the predetermined 0.10cm(2) cut-off point. High-resolution ultrasonography is therefore useful to diagnose and follow up cases of elbow UNN.
Arthroscopic Subchondral Drilling Followed by Injection of Peripheral Blood Stem Cells and Hyaluronic Acid Showed Improved Outcome Compared to Hyaluronic Acid and Physiotherapy for Massive Knee Chondral Defects: A Randomized Controlled Trial

Saw KY, Anz AW, Ng RC, Jee CS, Low SF, Dorvault C, et al.

Arthroscopy, 2021 08;37(8):2502-2517.
PMID: 34265388 DOI: 10.1016/j.arthro.2021.01.067

PURPOSE: The purpose of this study was to evaluate the safety and efficacy of intra-articular injections of autologous peripheral blood stem cells (PBSCs) plus hyaluronic acid (HA) after arthroscopic subchondral drilling into massive chondral defects of the knee joint and to determine whether PBSC therapy can improve functional outcome and reduce pain of the knee joint better than HA plus physiotherapy.
METHODS: This is a dual-center randomized controlled trial (RCT). Sixty-nine patients aged 18 to 55 years with International Cartilage Repair Society grade 3 and 4 chondral lesions (size ≥3 cm2) of the knee joint were randomized equally into (1) a control group receiving intra-articular injections of HA plus physiotherapy and (2) an intervention group receiving arthroscopic subchondral drilling into chondral defects and postoperative intra-articular injections of PBSCs plus HA. The coprimary efficacy endpoints were subjective International Knee Documentation Committee (IKDC) and Knee Injury and Osteoarthritis Outcome Score (KOOS)-pain subdomain measured at month 24. The secondary efficacy endpoints included all other KOOS subdomains, Numeric Rating Scale (NRS), and Magnetic Resonance Observation of Cartilage Repair Tissue (MOCART) scores.
RESULTS: At 24 months, the mean IKDC scores for the control and intervention groups were 48.1 and 65.6, respectively (P < .0001). The mean for KOOS-pain subdomain scores were 59.0 (control) and 86.0 (intervention) with P < .0001. All other KOOS subdomain, NRS, and MOCART scores were statistically significant (P < .0001) at month 24. Moreover, for the intervention group, 70.8% of patients had IKDC and KOOS-pain subdomain scores exceeding the minimal clinically important difference values, indicating clinical significance. There were no notable adverse events that were unexpected and related to the study drug or procedures.
CONCLUSIONS: Arthroscopic marrow stimulation with subchondral drilling into massive chondral defects of the knee joint followed by postoperative intra-articular injections of autologous PBSCs plus HA is safe and showed a significant improvement of clinical and radiologic scores compared with HA plus physiotherapy.
LEVEL OF EVIDENCE: Level I, RCT.

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