[A rare cause of hypercorticism: ACTH-secreting pheochromocytoma (a case report)]. / Une cause rare d'hypercorticisme: un phéochromocytome sécrétant l'ACTH (à propos d'un cas).

Ectopic ACTH-secreting pheochromocytoma is a very rare cause of Cushing´s syndrome, posing diagnostic and therapeutic challenges. We here report the case of a female patient with suspected severe Cushing´s syndrome associated with melanoderma, arterial hypertension resistant to triple therapy and unbalanced diabetes treated with insulin therapy. Biologically, urinary ethoxylated, 24-hour urinary free cortisol and ACTH were very high. Imaging showed a 3.5 cm left adrenal mass. The patient underwent left adrenalectomy after medical preparation, with good clinico-biological outcome. Anatomopathological examination confirmed the diagnosis of pheochromocytoma. This case study highlights the importance of measuring methoxylated derivatives in any patient with ACTH-dependent Cushing´s syndrome associated with an adrenal mass. The aim is to ensure early treatment and avoid life-threatening complications.

Uncommon Thigh Mass in Neurofibromatosis Type 1: Unveiling Aggressive Epithelioid Sarcoma.

Background: Patients with neurofibromatosis type I (NF1) have an increased risk of developing soft-tissue sarcomas, particularly those related to the nervous system. Epithelioid sarcoma (ES) is an exceptionally rare subtype of soft-tissue sarcoma, with limited knowledge about its clinical presentation and optimal management in NF1. This report aims to provide insights into the characteristics and outcomes of ES in NF1 patients. Case description: A 37-year-old man with a history of NF1 presented with a progressively worsening mass on his right inner thigh. An MRI scan revealed a well-defined tissue mass originating from the adductor magnus muscle, later confirmed as ES through histopathology and immunohistochemistry. Considering poor local and general prognosis, the multidisciplinary team recommended salvage hip disarticulation, however the patient refused and opted for palliative marginal resection to reduce the tumour size. The patient's condition declined rapidly, and he succumbed six days after the surgery. Conclusion: This case highlights the rarity of ES in NF1 patients and underscores the potential for malignant tumour development in this population. Further research is needed to improve our understanding and management of sarcomas in the context of NF1. LEARNING POINTS: Patients with neurofibromatosis type 1 or von Recklinghausen's disease have a higher risk than those with other types of neurofibromatosis of developing benign or malignant soft-tissue tumours especially related to the nervous system.Epithelioid sarcoma is an extremely rare subtype of soft-tissue sarcoma and is exceptionally associated with neurofibromatosis type 1.A multidisciplinary approach remains essential in the diagnosis, management, and treatment of soft-tissue sarcomas in patients with neurofibromatosis type 1.

First report of Sarcocystis falcatula in naturally infected Razorbill auks (Alca torda) collected in Tunisian Mediterranean Sea shores.

Sarcocystis spp. are apicomplexan cyst-forming parasites that can infect numerous vertebrates, including birds. Sarcosporidiosis infection was investigated in three muscles (breast, right and left thigh muscle) and one organ (heart) of four Razorbill auks (Alca torda) stranded between November and December 2022 on the shores of the Mediterranean Sea in Nabeul and Bizerte governorates, Northern Tunisia. Two of the four tested A. torda were PCR positive for 18S rRNA Sarcocystis spp. gene. Among the examined 16 muscles/organs, only one breast and one right thigh were Sarcocystis spp. PCR-positive (12.5% ± 8.3, 2/16). Our results showed a relatively high molecular prevalence of Sarcocystis spp. in Razorbill auks (A. torda). Sarcocystis spp. sequence described in the present study (GenBank number: OR516818) showed 99.56-100% identity to Sarcocystis falcatula. In conclusion, our results confirmed the infection of Razorbill auks (A. torda) by S. falcatula. Further research is needed on different migratory seabirds' species in order to identify other Sarcocystis species.

Primary extrahepatic hydatid cyst of the thigh, an unusual presentation of sciatica: A case report of a diagnostic challenge.

INTRODUCTION: Sciatica, a condition characterized by pain along the sciatic nerve distribution, is commonly associated with nerve compression or irritation. However, its etiology can vary, including rare non-spinal causes such as hydatid cysts. We present a case of hydatid cyst in the thigh causing sciatica-like symptoms, highlighting the diagnostic challenges and management approach. CASE PRESENTATION: A 40-year-old patient with a history of pulmonary tuberculosis presented with persistent lumbosciatic pain despite conservative treatment. Physical examination revealed left sciatica without spinal abnormalities. MRI revealed a hydatid cyst in the thigh, causing nerve irritation. Surgical resection of the cyst was performed, achieving symptom resolution. DISCUSSION: Hydatid cysts in skeletal muscles are rare, with atypical presentations complicating diagnosis. Localization in the thigh, particularly the biceps femoris muscle, is uncommon. Diagnostic modalities include imaging and serological tests, while treatment involves surgical excision and postoperative albendazole therapy. CONCLUSION: Recognition of rare presentations like thigh hydatid cysts causing sciatica-like symptoms is crucial for timely diagnosis and management. This case emphasizes the importance of considering unusual etiologies in refractory sciatica cases and underscores the complexity of medical diagnosis. Increased awareness among healthcare providers can lead to improved patient outcomes and prevent diagnostic delays.

An unusual cause of inguinal swelling: Primary synovial osteochondromatosis.

Inguinal swelling should raise suspicion for hip joint osteochondromatosis. Early recognition, accurate diagnosis, and prompt surgical intervention are essential for optimizing patient outcomes.

One-stage combined approach en bloc vertebrectomy for primary Ewing's sarcoma of mobile spine in an adult patient: 3 years following aggressive surgery for a rare entity.

Key Clinical Message: Currently, there is no consensus on the optimal management of nonmetastatic Ewing's sarcoma of the mobile spine. However, associated to chemotherapy, aggressive surgery with en bloc wide resection seems to improve local control and survival. Abstract: Primary Ewing's sarcoma (EWS) of non-sacral spine is extremely rare, especially in middle-age. Therapeutic strategy aims: to large tumor resection, to provide spine stability and to avoid recurrence through chemo and radiotherapy. We report a case of thoracic spine EWS in an adult treated by combined approach en bloc vertebrectomy.

Conservative surgical management of surface osteosarcoma using 3D printing technology: An unusual case report and literature review.

INTRODUCTION AND IMPORTANCE: Surface osteosarcoma, a rare variant of osteosarcoma, poses unique challenges in diagnosis and treatment. This report discusses the application of 3D printing technology in the surgical management of a complex case involving a 27-year-old female patient with surface osteosarcoma in the proximal tibia. CASE PRESENTATION: A 27-year-old female patient presented with a progressively growing mass on her right knee, initially misdiagnosed as a benign bone tumor. Over five years, the lesion expanded from a well-corticated metaphyseal-epiphyseal outgrowth on the proximal tibia to involve the anterior tibial tuberosity. Radiological and histological evaluations confirmed well-differentiated paraosteal surface osteosarcoma. A multidisciplinary team opted for a conservative surgical approach, including resection of the anterior tibial tuberosity and patellar tendon. Precision was enhanced through 3D printing technology, which provided custom cutting guides. The reconstruction involved non-vascularized peroneus and iliac crest bone grafts. CLINICAL DISCUSSION: Accurate differentiation from benign lesions presents challenges. Achieving surgical resection with clear margins is pivotal for favourable outcomes, particularly in young patients. Chemotherapy yields limited benefits in low-grade surface osteosarcomas. Functional prognosis hinges on effective post-resection reconstruction. 3D printing technology facilitates meticulous surgical planning and guidance, enhancing the success of conservative surgical interventions. CONCLUSION: This case underscores the significance of a multidisciplinary approach, accurate diagnosis, and the integration of 3D printing technology in managing surface osteosarcomas. Conservative surgical resection, guided by precise planning and reconstruction, is critical for preserving functionality. Continued research and the adoption of innovative techniques hold promise for improving the quality of life and functional outcomes of individuals grappling with musculoskeletal tumors.

Unusual case of lateral subtalar joint dislocation associated with calcaneal fracture and lateral malleolus fracture.

Introduction: Subtalar dislocation is defined by a simultaneous total separation of the talocalcaneal and talonavicular joints. Lateral variety is the rarest. It is the result of a high energy trauma. It occurs following forced eversion, with the foot locked in a rut with pressure on the lateral aspect of the leg. Articular fractures of the calcaneus occur as a result of axial impact on the greater tuberosity.We report the case of a lateral subtalar dislocation associated with a calcaneal fracture. Case presentation: A 62-year-old man who presented to the emergency room following a 9 m fall. Initial examination revealed a swelling of the right foot and ankle with a deformity giving the appearance of a "flat foot" without skin opening. The neuro-vascular examination was normal.A radiological assessment including an antero-posterior and lateral X-ray of the right ankle and a axial view if the calcaneus, showed a calcaneal articular fracture and a lateral dislocation of the subtalar joint. CT scan confirmed the diagnosis. Closed reduction by external maneuvers failed. Through a lateral mini-approach centered on the subtalar joint, open reduction of the dislocation was achieved and controlled fluoroscopically. Osteosynthesis of the calcaneal fracture was performed using a percutaneous pinning. Consolidation was obtained at 3 months post-operatively.At the last follow-up, the patient reported occasional pain. The radiological assessment showed early arthritic changes of the subtalar joint. Conclusion: In rare cases of subtalar dislocation, the direction of displacement, associated bone injuries, and open reduction would be responsible for a poor functional outcome. At present, there is no certain way to avoid subtalar osteoarthritis.

Giant cell tumor of the tendon sheath of the toe: case report of an unusual localization.

INTRODUCTION AND IMPORTANCE: The purpose of this study is to highlight a rare entity of unusual location and to report our therapeutic attitude in this regard, while detailing the diagnostic criteria and therapeutic management. CASE PRESENTATION: We describe the case of a 32 years old woman with a swelling of the big toe initially neglected by the patient, the evolution is marked by a discomfort when wearing shoes, it was a firm mass of 2 cm slightly painful without local inflammatory signs with bone lysis on radiography, The MRI showed an invasion of the flexor and extensor tendons of the hallux, our approach was a carcinological tumor exeresis and arthrodesis while preserving the toe since the pedicle was not invaded, the histological examination confirmed the diagnosis and the evolution is good without tumor recurrence. CLINICAL DISCUSSION: A giant cell tumor of the tendon sheaths (GCTTC) usually presents as a firm slow growing mass, the radiological assessment shows bone erosions, an MRI for extension assessment and histological confirmation are mandatory, malignant degenerations are unusual but should always be investigated, treatment is based on the most complete surgical resection to avoid the risk of recurrence, adjuvant radiotherapy can be done in case of incomplete resection but it is a controversial subject. CONCLUSION: GCTTC are benign tumors that may be locally malignant because of the invasion of noble structures, which makes their complete exeresis difficult. The diagnosis is based on clinical and radiological criteria and requires histological confirmation.

Bone metastases from renal cell carcinoma: 4 years after aggressive surgeries and anti-angiogenic therapy.

Management of bone metastases from renal cell carcinoma (RCC) has significantly changed after the era of targeted therapy that improved the overall survival (OS). Surgical decision-making remains a subject of controversy. We report a case of pelvic bone metastasis from RCC, 2 months after nephrectomy and surgery of a revealing clavicular metastasis.

First molecular detection of Neospora caninum from naturally infected slaughtered camels in Tunisia.

BACKGROUND: Neospora caninum has been documented to infect most domestic wildlife but is known to primarily infect dogs and cattle and is considered an important cause of abortion in camels. OBJECTIVE: The aim of this study was to estimate the molecular detection of Neospora caninum in tissues of naturally infected camelids. METHODS: Brain, tongue (bottom and tip) and masseter muscles from 35 slaughtered camelids from Tataouine and Médenine regions were collected (n = 140 samples). PCR was used to amplify and detect N. caninum DNA in tissues samples followed by sequencing of some PCR products. A phylogenetic tree was then constructed to compare the partial sequences of the ITS1 gene with GenBank sequences. Histopathology examination was used to detect Neospora spp. cysts, but no lesions were observed. RESULTS: The overall molecular detection of N. caninum in camelids was 34.3% (12/35). The highest molecular detection of N. caninum was recorded in animals of more than 3 years old (6/9) and in animals aged between 1 and 3 years old (4/12). Whilst, the lowest molecular detection (2/14) was observed in animals 1 year or younger (p = 0.035). There were no significant differences in molecular detection of N. caninum according to both locality and gender (p > 0.05). Similarly, there was no difference of prevalence between different anatomical locations. Comparison of the partial sequences of the ITS1 gene revealed 100-95.5% similarity among our N. caninum amplicon (MW551566) and those deposited in GenBank. CONCLUSION: These results highlight the presence of a risk infection by N. caninum in camels. For preventing N. caninum infection further studies are needed to improve our knowledge about the epidemiology of neosporosis in North Africa.

Infestation rate of lice on donkeys in Northeast Tunisia.

The present survey was carried out during autumn 2013 (September, October and November) in five localities of Jendouba district. A total number of 300 donkeys were examined and all present ectoparasites found on animals were collected and examined under stereomicroscope. The infestation rate by lice was estimated to 12.7% (38/300) there was no difference between infestation rates in males (11.5%; 22/192) and females (14.8%; 16/108). The highest infestation rate was observed in donkeys aged between 10 and 14 years, they represented 19.6% of the infested donkeys. Two lice species were collected with no statistically difference between the two infestation rates: Haematopinus asini (4%; 12/300) and Bovicola ocellatus (5.3%; 16/300). The lice were collected from donkeys of the five regions with no statistically difference between the infestation rates. A specific programme to control these parasites is recommended to improve the welfare of the working donkeys in Tunisia and in other North African countries.

Infestation of small ruminants by the metacestode stage of Taenia hydatigena in slaughterhouse, North East Tunisia.

BACKGROUND: Cysticercus tenuicollis (larvae of Taenia hydatigena) is a frequent cosmopolitan endoparasite of ruminants. The infestation by this parasite is underestimated since it is neither zoonotic nor inducing high economic losses in the sheep sector. METHODS: This study aimed at estimating different parasitological indicators, Cysticercus tenuicollis infestation in small ruminants using a slaughterhouse-based survey in Northeast Tunisia. A total number of 3,692 sheep and 78 goats were examined in the slaughterhouse of Menzel Temime. RESULTS: The overall prevalence was estimated to be 2.8 (106/3692) and 8.9% (7/78) in sheep and goats, respectively. The abundance in these two species was 0.24 and 0.05 and the intensity 1.97 and 2.85, respectively. In goats, all the cysts were found in the mesentery, whilst, in sheep, the majority were in mesentery (96%) but also on the liver in 2% of the cases and in both organs (2%). There were only cysts with aqueous liquid with predominantly middle-sized cysts (1 to 3 cm) corresponding to 63.15 and 70.34% in sheep and goats, respectively. CONCLUSION: Since, in small ruminants, the infestation by C. tenuicollis indicators are not high, low cost control measures should be implemented to eliminate this parasite in Northeast Tunisia.

Correlation of polyphenolic content with radical-scavenging capacity and anthelmintic effects of Rubus ulmifolius (Rosaceae) against Haemonchus contortus.

Phenolic content, antioxidant and anthelmintic activities of herbal extracts are of particular interest to drug industry; plant extracts with significant anthelmintic activity have the potential to be used as alternatives to conventional chemical drugs. In the present study, Rubus ulmifolius fruit extracts obtained using solvents of increasing polarity (water, methanol, chloroform and hexane) were examined for their antioxidant and anthelmintic activities in correlation with their polyphenolic content. In vitro antioxidant activity of all extracts was carried out using free radical-scavenging activity by 2,2-diphenyl-1-picrylhydrazyl (DPPH) and 2,2'-azinobis-(3-ethilenebenzotiazolin)-6-sulfonic acid (ABTS) radical cation. In vitro anthelmintic activities were investigated on the egg and adult worms of Haemonchus contortus from sheep in comparison to albendazole. Total polyphenol content of R. ulmifolius was higher in more polar extract, ranging from 64.5 in aqueous extract to 1.57 mg gallic acid equivalents per gram of dry weight (GAE/g DW) in hexanic extract. Likewise, highest amounts of flavonoids and condensed tannins were found in aqueous extract (28.06 mg QE/g and 7.42 mg CE/g DW, respectively) compared to hexanic extract (0.71 mg QE/g and 0.29 mg CE/g DW, respectively) (p<0.05). Both DPPH and ABTS antioxidant assays showed that all tested extracts possess free radical scavenging activity, while the inhibitory concentration 50% (IC50) range values were similar for both assays (2.13-45.54 µg/mL and 1.2-43.82 µg/mL, respectively). All plant extracts showed ovicidal activity at all tested concentrations. Fruit methanolic (IC50=2.76mg/mL) and aqueous (IC50=2.08 mg/mL) extracts showed higher inhibitory effects than chloroformic (IC50=7.62 mg/mL) and hexanic (IC50=12.93 mg/mL) extracts on egg hatching (p<0.05). There was a significant correlation of total polyphenol, flavonoids and tannins content with scavenging of either DPPH (r=0.722, 0.764 and 0.752, p<0.01, respectively) or ABTS radicals (r=0.893, 0.765 and 0.722, p<0.01, respectively) and with inhibition of egg hatching (r=0.874, 0.883 and 0.862, p<0.01, respectively). Highest inhibition of motility (100%) of worms was observed 8h post-exposure in aqueous and methanolic extract at 8 mg/mL. To our knowledge, these results depict for the first time that R. ulmifolius possesses in vitro anthelmintic properties.

Non-cytotoxic Thymus capitata extracts prevent Bovine herpesvirus-1 infection in cell cultures.

BACKGROUND: Bovine herpes virus type 1 (BHV-1) still causes great economic loss to the livestock industry and trade because there aren't any available drugs that proved to be fully effective against it. In this study, the cytotoxicity and the antiviral activities of the Thymus capitata extracts were evaluated for the development of new, non toxic and specific anti-herpesvirus drug. Aqueous extracts (AE), ethanolic extracts (EE) and essential oil (EO) of the aerial parts of Thymus capitata were analyzed to determine their chemical compositions by gas chromatography, and high performance liquid chromatography combined with mass spectrometry. Their cytotoxicity and antiviral activities against Bovine Herpesvirus type 1 (BHV-1) were evaluated by quantifying the reduction of the viral cytopathic effect using Madin-Darby Bovine Kidney cell line with colorimetric assay. T. capitata extracts were added at different stages of the viral infection to investigate and better quantify their potential inhibitory effects. RESULTS: Polyphenols and flavonoids were the major compounds found in T. capitata EO, EE and AE. The cytotoxic concentrations at 50% were 48.70, 189 and 289 µg ml(-1) for EO, EE and AE, respectively. The inhibitor concentrations at 50% for the EO, EE and AE, were 3.36, 47.80 and 164 µg ml(-1), respectively. The selectivity index anti-BHV-1 values were 14.49, 3.95 and 1.81 for EO, EE and AE, respectively. Thus, the EO extracts were the most efficient antiviral compounds. T. capitata extracts affect mainly the adsorption of BHV-1 virus to host cells. CONCLUSION: T. capitata extracts inhibit the viral replication by interfering with the early stages of viral adsorption and replication. Thus, T. capitata is a potential candidate for anti-herpesvirus treatment.

Thrombotic thrombocytopenic purpura and systemic lupus erythematosus: a rare and life-threatening association.

INTRODUCTION: The association between thrombotic thrombocytopenic purpura (TTP) and systemic lupus erythematosus (SLE) is uncommon. Diagnosis is often difficult because of their clinical and biological similarities. The presence of TTP in SLE worsens the prognosis and causes high mortality in the absence of early therapeutic interventions. CASE REPORT: We report the case of a 20 year-old man, admitted with nephrotic range proteinuria, hematuria and rapidly progressive renal failure. He also had anemia, thrombocytopenia and pericardial effusion. The diagnosis of SLE was made based on these clinical findings along with positive antinuclear and anti dsDNA antibodies. Renal biopsy revealed class IV/ V lupus nephritis (LN) with active lesions of thrombotic microangiopathy. The evolution of neurological deficit, persistent thrombocytopenia and active microangiopathic changes suggested the diagnosis of associated TTP. The patient was treated initially with corticosteroids and cyclophosphamide. Plasmapheresis could only be started 16 days later. Mycophenolate mofetil and rituximab were successively tried in the absence of improvement in renal function and persistent thrombocytopenia. The patient's neurological condition deteriorated necessitating transfer to the intensive care unit and mechanical ventilation. There he developed pneumonia and died of septic shock two months after presentation. CONCLUSION: The coexistence of TTP and SLE needs to be considered early in SLE patients with complicated course. It may not respond to the conventional immunosuppressive treatment of SLE.

Prime-boost immunisation against tropical theileriosis with two parasite surface antigens: evidence for protection and antigen synergy.

Current methods for control of tropical theileriosis in cattle suffer from several disadvantages that could be circumvented by development of an effective sub-unit vaccine. Previous work has utilised two major surface antigens (SPAG-1 and Tams1) and conventional adjuvants to provide partial protection against parasite challenge. In this study we have delivered these antigens using the prime-boost system and analysed whether a combination regime can enhance protection against lethal challenge. Delivery of the boost as recombinant protein or expressed from a recombinant MVA vector was also assessed. The results confirmed that immunisation with Tams1 alone could reduce the severity of several disease parameters compared to non-immunised controls and these effects were more marked when recombinant protein was used for boosting compared to MVA delivery. A similar outcome was obtained by immunisation with SPAG-1 alone. Significantly, delivery of SPAG-1 and Tams1 as a cocktail showed enhanced protection. This was manifest by significant improvement in a large range of clinical and parasitological parameters and, most dramatically, by the survival and recovery of 50% of the immunised animals compared to 0% of the controls. Analysis of the antibody response post-challenge showed that while there was a strong response to Tams1, no response to SPAG-1 was detected. In contrast, lymphoproliferation assays showed a significant enhancement of response at day 7 post-challenge in calves of the SPAG-1 group but a dramatic decrease of the proliferation activity in all three groups receiving Tams1. We conclude that immunisation with a cocktail of SPAG-1 and Tams1 generates a synergistic protective response that significantly improves the efficacy of recombinant vaccination against tropical theileriosis. Potential effector mechanisms that could mediate this response are discussed.