Syndrome d’activation macrophagique secondaire à une fièvre boutonneuse méditerranéenne

Publication date: Available online 10 July 2018

Source: Annales de Dermatologie et de Vénéréologie

Author(s): A.-S. Bertrand, M. Fondain, P. Rullier, C. Fontaine, B. Guillot

Résumé

Introduction

Le syndrome d'activation macrophagique (SAM) est une pathologie rare de pronostic sévère définie par des critères cliniques, biologiques et cyto-histologiques. Les infections représentent une cause classique de SAM. Les SAM secondaires à une fièvre boutonneuse méditerranéenne sont rares ; seuls quelques cas sont rapportés dans la littérature.

Observations

Nous rapportons les cas de deux hommes de 77 et 63 ans qui avaient une éruption maculo-purpurique fébrile avec escarre d'inoculation associée à des anomalies biologiques comprenant cytopénie, hyperferritinémie et hypertriglycéridémie. Une hémophagocytose était présente dans les deux cas. La sérologie et la PCR Rickettsia conorii étaient positives en faveur d'une infection récente, confirmant le diagnostic de fièvre boutonneuse méditerranéenne (FBM). Ces éléments permettaient dans les deux cas de poser le diagnostic de SAM secondaire à une FBM.

Discussion

Le premier cas de SAM a été décrit en 1979. Seize cas de SAM secondaires à une FBM sont décrits dans la littérature. La présence d'une cytopénie associée à une hyperferritinémie et une hypertriglycéridémie au cours d'une FBM est fortement évocatrice du diagnostic de SAM. Le pronostic dépend du délai diagnostique et des facteurs propres à l'hôte. L'association d'immunosuppresseurs à l'antibiothérapie peut être nécessaire pour obtenir la guérison.

Conclusion

Les rickettsioses peuvent induire un syndrome d'hémophagocytose ; la survenue possible de cette complication de mauvais pronostic mérite d'être connue.

Summary

Introduction

Haemophagocytic syndrome (HS) is a rare disease with a severe prognosis that is defined by clinical, laboratory and histopathological criteria. Infections represent the classical cause of HS. HS secondary to Mediterranean spotted fever (MSF) is rare with only a few cases being reported in the literature.

Observations

We report two cases of HS secondary to MSF in 2 men aged 77 and 63 years presenting a febrile maculo-purpuric eruption with inoculation ulcer associated with laboratory abnormalities (cytopenia, elevated ferritin, hypertriglyceridaemia). Haemophagocytosis was present in 2 cases. Serology and PCR for Rickettsia conorii were positive and militated in favour of recent infection responsible for the diagnosis of MSF.

Discussion

The first case of HS was described in 1979. Sixteen cases of HS secondary to MSF are described in the literature. Cytopenia associated with hyperferritinaemia and hypertriglyceridaemia strongly suggests MSF complicated by HS. The prognosis depends on the time elapsed since diagnosis and host-specific factors. Immunosuppressants and antibiotics may be necessary to ensure healing.

Conclusion

Rickettsioses can induce HS, and this potential complication with a severe prognosis must be known.

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Increasing nerve autograft length increases senescence and reduces regeneration

Background: Nerve grafting with an autograft is considered the gold standard. However, the functional outcomes of long (>3cm) nerve autografting are often poor. We hypothesized that a factor contributing to these outcomes is the graft microenvironment, where long compared to short autografts support axon regeneration to different extents. Methods: A rat sciatic nerve defect model was used to compare regeneration within short (2cm) and long (6cm) isografts. Axon regeneration and cell populations within grafts were assessed using histology, retrograde labeling of neurons regenerating axons, immunohistochemistry, qRT-PCR, and electron microscopy at 4 and/or 8 weeks. Results: At 8 weeks for distances of both 1 and 2cm from the proximal coaptation (equivalent regenerative distance), long isografts had reduced numbers of regenerated fibers compared to short isografts. Similarly, the number of motoneurons regenerating axons was reduced in the presence of long isografts compared to short isografts. Considering the regenerative microenvironments between short and long isografts, cell densities and general populations within both short and long isografts were similar. However, long isografts had significantly greater expression of senescence markers, which included senescence associated β-galactosidase, p21, and p16, as well as distinct chromatin changes within Schwann cells. Conclusions: This study shows that axon regeneration is reduced within long compared to short isografts, where long isografts contained an environment with an increased accumulation of senescent markers. While autografts are considered the "gold standard" for grafting, these results demonstrate that we must continue to strive for improvements in the autograft regenerative environment. Financial Disclosure Statement: The authors have the following to disclose: None Presented at: American Society for Surgery of the Hand 2016 meeting in Austin, Tx and the Plastic Surgery Research Council 2017 meeting in Durham, NC. Disclosures: None of the authors has a financial interest in any of the products, devices, or drugs mentioned in this manuscript. Acknowledgements: This work was supported in part by the National Institutes of Neurological Disorders and Stroke of the National Institutes of Health (NIH) under award number R01 NS086773 to Washington University and in part by the Plastic Surgery Foundation under award ID 346670 to Wood through Washington University. The content is solely the responsibility of the authors and does not represent the views of the NIH, the Plastic Surgery Foundation, or Washington University. Corresponding author: Matthew D. Wood, PhD, Division of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine, Campus Box 8238, 660 South Euclid Avenue, St. Louis, MO 63110, USA. woodm@wudosis.wustl.edu ©2018American Society of Plastic Surgeons

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Outcomes of Corticosteroid Treatment for Trigger Finger by Stage.

Background: While steroid injection remains a common first-line treatment of trigger fingers, clinical experience suggests that not all trigger fingers respond the same. The purpose of this study was to utilize a classification system for trigger finger that is simple, reproducible, and produces clearly definable, clinically relevant cutoff points to determine if responsiveness to steroid injection correlates to clinical staging. Methods: This was a prospectively-collected longitudinal study of trigger finger patients separated into four stages of severity. Each subject received a single injection of 6 mg dexamethasone acetate. One-month outcomes were analyzed to evaluate efficacy of steroid injection. These outcomes were further stratified based on baseline characteristics and stage of triggering Results: A total of 99 digits and 69 subjects were included. Two variables were found to be significant in predicting response to initial injection: 1. multiple affected digits, and 2. stage severity. Patients with multiple involved fingers were 5.8 times more likely to have no resolution of symptoms compared with those with a single affected finger. For every level of stage increase, the odds doubled for having no resolution of symptoms. Conclusions: Steroid injection remains a viable first-line option for patients presenting with mild triggering (Stage 1 and 2). For more severe triggering (Stage 3 and 4) or multiple affected digits, success of steroid injection is significantly lower at one month. For the latter patients, surgery may be a more reasonable initial treatment. Presented at: The American Association of Plastic Surgeons Annual Meeting, Austin, TX, 2017 Financial Disclosure Statement: Funded by a 2012 PSF/AAHS research grant Products Mentioned: N/A Corresponding Author: Robert A. Weber, MD, 2401 S. 31st St. Temple, TX, USA. E-mail address: Robert.Weber@BSWHealth.org ©2018American Society of Plastic Surgeons

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Treatment of Apert hand syndrome: Strategies for achieving a 5-digit hand

Background: Apert hand reconstruction requires complex surgical planning. The purpose of this study is to describe our 8 year surgical experience with Apert syndrome hand reconstruction, and provide specific surgical strategies for achieving a 5-digit hand in Upton type I and II hands. Methods: A retrospective analysis of consecutive Apert patients who underwent web space releases between 2007 and 2015 was performed. Demographic, surgical, and outcome data were verified through medical records, clinical photographs, radiographic images, and patient interviews. Results: A total of 41 Apert patients (23 [56.1%] male and 18 [43.9%] female) were treated at our Hospital since 2007. A 5-digit hand was achieved in all patients (100%) with Upton type I and II hands, and in 8 patients (72.7%) with Upton type III hands. A 4-digit hand was obtained in 3 of 11 patients (27.3%) with Upton type III hands. Four (25%) of 20 patients with Upton type I hands, 3 (30%) of 10 patients with Upton type II hands, and 6 (54.5%) of 11 patients with Upton type III hands, required subsequent revision for aesthetic reasons. Conclusion: Upton type III hands have demonstrated higher revision rates than type I and II hands, regardless of whether a 4 or 5-digit hand is obtained. Treatment strategies for Apert hands based on hand type are offered to guide 4-web-space release in all patients with Upton type I and II hands. Funding: None Conflict of interest: The authors have no personal financial or institutional interest in any of the drugs, materials, or devices described in this article. Corresponding author: Cassio Eduardo Raposo-Amaral, MD, PhD, Institute of Plastic and Craniofacial Surgery, SOBRAPAR Hospital, Av. Adolpho Lutz, 100, Caixa Postal: 6028, Campinas, São Paulo 13084-880, Brazil. E-mail: cassioraposo@hotmail.com ©2018American Society of Plastic Surgeons

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Aesthetic Comparison of Two Different Types of Webspace Reconstruction for Finger Syndactyly

Background: Syndactyly reconstruction incorporates techniques of applying skin grafts or dorsal advancement flaps without the use of skin grafts. Comparative outcome studies of these two approaches are lacking. Our study compares the long-term aesthetic outcomes of these two flap techniques. Methods: Forty-five patients were included in our study. The methods of reconstruction used were a dorsally-based rectangular flap with skin graft from the groin and a dorsal pentagonal advancement flap without skin grafting. Eighteen independent raters completed a Visual Analogue Scale (VAS) as well as a unique classification scale to subjectively assess aesthetic outcomes. We used univariate analyses to determine which variables significantly influenced the outcome score. We then used multivariable regression models to compare the two flap types. Results: Dorsally-based rectangular flaps with skin graft had statistically significantly better VAS scores and greater odds of receiving an 'Excellent' rating when compared to dorsal pentagonal advancement flaps. Conclusions: Despite the use of skin graft and their associated donor-site morbidity, dorsal rectangular flaps may offer better overall aesthetic outcomes for patients. Future comparative studies should incorporate functional and patient-reported outcomes to better assess the optimal reconstruction type. Financial Disclosure Statement: This publication was supported in part by the National Institute of Arthritis and Musculoskeletal and Skin Diseases (2 K24-AR053120-06). The consent is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. Corresponding Author: Kevin C. Chung MD, MS, Section of Plastic Surgery, University of Michigan Health System, 2130 Taubman Center, SPC 5340, 1500 E. Medical Center Drive, Ann Arbor, MI, 48109-5340, Kecchung@med.umich.edu, Phone 734-936-5885, Fax 734-763-5354 ©2018American Society of Plastic Surgeons

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Re-establishment of Lymphatic Drainage after Vascularized Lymph Node Transfer in a Rat Model

Background: Vascularized lymph node transfer (VLNT) has recently received attention as a potential surgical treatment for lymphedema. Despite good results in some series, the mechanism and benefits of VLNT have yet to be fully understood. This study aimed to investigate the re-establishment of drainage into transferred lymph nodes following VLNT in a rat model. Methods: Seven rats underwent VLNT. The operation performed on each rat consisted of two parts. First, the left groin lymph node basin with superficial epigastric vessels was harvested as a free flap. Second, the flap was re-attached in the left groin of the rat via end-to-end microvascular anastomoses. Anastomosis patency was assessed immediately post-op and at the time of animal sacrifice. The rats were evaluated for re-establishment of lymphatic flow into the transplanted nodes at 1 month intervals for at least 6 months post-op. This was accomplished non-invasively by injecting the rats in their flanks with fluorescent indocyanine green (ICG) which was detected using a PDE infrared camera. Results: Anastomoses were patent in all 7 rats immediately postop. No ICG uptake was seen in the transplanted lymph node basins in the first 2 months post-op in any of the rats. In 5 of 7 rats, however, ICG uptake was demonstrated in the transplanted lymph node basin by 6 months (average 13 weeks). Conclusions: We report uptake of ICG in 5 of 7 rats at an average of 13 weeks following lymph node transplantation, consistent with the re-establishment of lymphatic drainage into the transplanted nodes. Financial Disclosure Statement: None of the authors have any conflicts of interest nor disclosures. No funding was received for this article. Portions presented at: American Society for Reconstructive Microsurgery (ASRM) 2017 in Waikoloa, Hawaii Northeastern Society of Plastic Surgeons (NESPS) 2016 in Baltimore, MD Acknowledgments: We would like to thank Yanping Sun, PhD and Christopher Damoci from the Small Animal Imaging Shared Resource (SAISR) division of the Herbert Irving Comprehensive Cancer Center at Columbia University for their assistance in obtaining our MRI studies. Corresponding author: Jeffrey A. Ascherman, MD, Herbert Irving Pavilion, 161 Fort Washington Avenue Suite: 511, New York, NY 10032, jaa7@cumc.columbia.edu ©2018American Society of Plastic Surgeons

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Facial implants; controversies an d criticism, a comprehensive review of the current literature.

BACKGROUND: Polyethylene (Medpor) and Silicone are two of the most popular materials used today for facial skeleton implantation. Previous studies have identified common complications with the use of these implants, but patient follow up has been short. This review of literature examines complications and patient follow up in cases using Medpor and Silicone implants for reconstructive and aesthetic surgeries of the mid and lower face over the last 20 years. METHOD: A literature search was conducted through the PUBMED database. Key words used were ("Mandible Implants" or "Malar Implants" or "Chin Implants") AND ("Reconstruction" or "Augmentation") AND ("Medpor" or "Silicone"). RESULT: There were 9 studies with 626 patients in the Medpor group and 5 studies with 365 patients for Silicone. Silicone had a higher incidence of infections and displacements. Medpor showed a higher incidence of prominence problems. Exposure/extrusion rates were low for both implants. Chin and mandibular implants were the safest, while malar implants had a high incidence of prominence problems. The average follow up for Medpor was 36.6 months and 24 months for Silicone. There were wide ranges of follow up times, from 2 weeks up to 15 years. A limited number of articles included an averaged time within their ranges. Reported follow ups were not linked to specific complications. CONCLUSION: Medpor implantation is more common than Silicone. Complications rates are low with the use of both materials. Patient follow up is deficient and hasn't improve in the last 20 years, raising questions on the reliability of complications rates. FD - None of the authors have financial disclosures. Corresponding author: Yoel Rojas, MD1, Department of Plastic Surgery, Nassau University Medical Center, East Meadow, NY 11554 ©2018American Society of Plastic Surgeons

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