Fistulising skin metastases in Crohn's disease: a case report and review of the literature.

BACKGROUND: Metastatic Crohn's disease is a rare disorder characterized by various granulomatous skin lesions that occur independently of gastrointestinal tract involvement. However, currently there is no standardized care or specific treatment. Therapeutic approaches include immunosuppressive agents, such as corticosteroids, azathioprine, and monoclonal antibodies targeting inflammatory cytokines like tumor necrosis factor (TNF). CASE PRESENTATION: We present a case of a 29-year-old western European woman with significant blind ending abdominal subcutaneous fistulas and abscesses, who sought evaluation in the dermatology department. Histological examination revealed multiple epithelioid cell granulomas. There was no evidence of infectious or rheumatologic diseases such as sarcoidosis. The tentative diagnosis was metastatic Crohn's disease, which was not related to an intestinal manifestation of the disease. The patient responded to infliximab but had to discontinue it due to an allergic reaction. Subsequent adalimumab treatment failed to induce clinical remission; thus, therapy was switched to ustekinumab, resulting in a positive response. Written informed consent for publication of their clinical details and clinical images was obtained from the patient. For our study more than 1600 publications were screened for cases of metastatic Crohn's disease on PubMed database. 59 case reports with 171 patients were included in the analysis and evaluated for localization, diagnostic and therapeutic approaches, and complications and were summarized in this review. CONCLUSION: The successful ustekinumab treatment of a patient with metastatic Crohn's disease underscores the potential of this minimally investigated therapeutic option, highlighting the need for future treatment guidelines given the increasing prevalence of such cases.

Botulinum Neurotoxin A in the Treatment of Pharyngocutaneous Fistula after Salvage Surgery in Head and Neck Cancer Patients: Our Preliminary Results.

Objective: To analyze the effect of intraparotid injection of botulinum neurotoxin A (BoNT-A) on salivary production and the course of pharyngocutaneous fistula (PCF) in post-radiation therapy salvage surgery. Methods: A total of 13 patients who had undergone total laryngectomy or pharyngolaryngectomy were treated with BoNT-A to both parotid glands, within three days from PCF onset. The salivary flow was evaluated using a subjective rating scale as the percentage of normal function from 0% (no saliva) to 100% (normal saliva flow), before injection, every day for 2 weeks, and once a week for three months. PCFs were monitored daily. Results: Spontaneous closure of PCF occurred in 7/13 (53.84%) cases 13.6 days (range: 7-18) after treatment; 6/13 (46.16%) patients needed revision surgery. Salivary flow significantly decreased in all patients seven days after injection (from 67.2% to 36.4%; p < 0.05). Patients who had undergone either conservative or surgical treatment did not differ in salivary flow before injection, whereas the mean percentages of salivary flow calculated at each time point after injection were different (p < 0.05). Conclusions: BoNT-A contributed to the closure of the fistula in most of our cases. The subjective perception of salivary flow predicted the closure of PCF. The mean time to closure may contribute to establishing the timing of PCF surgical treatment.

Relationship between clinical remission of perianal fistulas in Crohn's disease and serum adalimumab concentrations: A multi-center cross-sectional study.

BACKGROUND: Crohn's disease (CD) is complicated by perianal fistulas in approximately 20% of patients. Achieving permanent fistula closure remains a challenge for physicians. An association between serum anti-tumor necrosis factor-α concentrations and clinical outcomes in patients with CD has been demonstrated; however, little information is available on serum adalimumab (ADA) concentrations and remission of perianal fistulas in such patients. AIM: To study the relationship between serum ADA concentrations and clinical remission of CD-associated perianal fistulas. METHODS: This cross-sectional study of patients with CD-associated perianal fistulas treated with ADA was performed at four French hospitals between December 2013 and March 2018. At the time of each serum ADA concentration measurement, we collected information about the patients and their fistulas. The primary study endpoint was clinical remission of fistulas defined as the absence of drainage (in accordance with Present's criteria), with a PDAI ≤ 4, absence of a seton and assessment of the overall evaluation as favorable by the proctologist at the relevant center. We also assessed fistula healing [defined as being in clinical and radiological (magnetic resonance imaging, MRI) remission] and adverse events. RESULTS: The study cohort comprised 34 patients who underwent 56 evaluations (patients had between one and four evaluations). Fifteen patients had clinical remissions (44%), four of whom had healed fistulas on MRI. Serum ADA concentrations were significantly higher at evaluations in which clinical remission was identified than at evaluations in which it was not [14 (10-16) vs 10 (2-15) µg/mL, P = 0.01]. Serum ADA concentrations were comparable at the times of evaluation of patients with and without healed fistulas [11 (7-14) vs 10 (4-16) µg/mL, P = 0.69]. The adverse event rate did not differ between different serum ADA concentrations. CONCLUSION: We found a significant association between high serum ADA concentrations and clinical remission of CD-associated perianal fistulas.

Staged surgical treatment for infection of total disc arthroplasty: three cases and a narrative review of the literature.

Although total disc arthroplasty (TDA) is a common procedure for selected cases of degenerative disc disease, until today there are only two cases of TDA infections reported in the literature. We report three cases of postoperative TDA infections, two developed cutaneous fistulas. To eradicate the infectious site, a staged removal of the device, resolute debridement, and stabilization plus fusion is proposed. Surgeons are challenged by (1) major retroperitoneal vessels adherent to the device, (2) surrounding scar tissue, (3) accompanying retroperitoneal abscess, and (4) technical issues when removing and replacing the implant. A staged multidisciplinary team approach involving vascular and plastic surgery as well as spine specialists is mandatory to achieve good results.

A Functional Approach to Posttraumatic Salivary Fistula Treatment: The Use of Botulinum Toxin.

This manuscript highlights key aspects regarding the practical use of botulinum toxin for the conservative nonsurgical treatment of a rarely encountered, but significant posttraumatic complication-the parotid salivary fistula. It adds information to the scarce existing literature on the subject. The authors outline the main differences between postoperative and trauma-related parotid injury regarding salivary fistula treatment. A total of 6 patients with trauma-related salivary fistulas have been treated by Abobotulinum toxin A injections over the course of 5 years. The technique is detailed, describing the doses used in the presence of parenchyma and duct injuries, the location and number of injection points in relation to the wound pattern. The results were favorable, leading to the healing of the salivary fistulas in all patients, with 1 injection session, without additional conservative treatment. In our experience, the use of botulinum toxin is of great benefit for treating salivary fistulas in a traumatic context.

[Congenital cysts and fistulas on the neck in adults]. / Kystes et fistules congenitaux du cou chez l'adulte.

Cervical congenital malformations are relatively common in children. They can also be found in adults. The embryological development of the cervical region is closely related to the branchial clefts. This must be a diagnosis made by elimination; a cervical tumor must evoke the diagnosis of cancer. A cutaneous fistula or a cervical tumor, chronic or recent appearance in an inflammatory context, are the clinical signs. The thyroglossal duct cysts and the second branchial clefts cysts are the most common causes of median and lateral cervical cysts, respectively. Imaging contributes greatly to the orientation and diagnostic evaluation of the extent of the lesions. Treatment is initially based on antibiotic therapy and then on complete surgical excision, away from an infectious episode, the sole guarantee for the absence of local recurrence.

Linezolid-induced black hairy tongue.

Black hairy tongue (BHT) is a self-limiting disorder characterized by abnormal hypertrophy and elongation of filiform papillae on the surface of the tongue. The exact mechanism of drug-induced BHT is unknown. Several factors have been implicated and included smoking or chewing tobacco, drinking alcohol, poor oral hygiene and antibiotics such as tetracyclines and penicillins. We report a quite uncommon case of Linezolid-induced BHT in a patient with a long-lasting history of chest wall infection.

Tubercular esophagocutaneous fistula: Rare case report.

Tubercular esophagocutaneous fistula is a rare entity with only about five cases reported so far. It can be as a result of primary involvement of esophagus by tuberculosis or due to spread of infection from adjacent structures like lungs or mediastinal lymph nodes. The fistula usually heals with initiation of antitubercular therapy and surgery is rarely required. Here we report a case of 65-year-old diabetic male who developed esophagocutaneous fistula secondary to caseation of mediastinal lymph nodes and was successfully treated with antitubercular treatment.

Cutaneous fistulization of the hydatid disease: A PRISMA-compliant systematic review.

AIM: To provide an overview of the medical literature on cutaneous fistulization in patients with hydatid disease (HD). METHODS: According to PRISMA guidelines a literature search was made in PubMed, Medline, Google Scholar, and Google databases were searched using keywords to identify articles related to cutaneous fistulization of the HD. Keywords used were hydatid disease, hydatid cyst, cutaneous fistulization, cysto-cutaneous fistulization, external rupture, and external fistulization. The literature search included case reports, review articles, original articles, and meeting presentations published until July 2016 without restrictions on language, journal, or country. Articles and abstracts containing adequate information, such as age, sex, cyst size, cyst location, clinical presentation, fistula opening location, and management, were included in the study, whereas articles with insufficient clinical and demographic data were excluded. We also present a new case of cysto-cutaneous fistulization of a liver hydatid cyst. RESULTS: The literature review included 38 articles (32 full text, 2 abstracts, and 4 unavailable) on cutaneous fistulization in patients with HD. Among the 38 articles included in the study, 22 were written in English, 13 in French, 1 in German, 1 in Italian, and 1 in Spanish. Forty patients (21 males and 19 females; mean age ±â€Šstandard deviation, 54.0 ±â€Š21.5 years; range, 7-93 years) were involved in the study. Twenty-four patients had cysto-cutaneous fistulization (Echinococcus granulosus); 10 had cutaneous fistulization (E multilocularis), 3 had cysto-cutaneo-bronchio-biliary fistulization, 2 had cysto-cutaneo-bronchial fistulization; and 1 had cutaneo-bronchial fistulization (E multilocularis). Twenty-nine patients were diagnosed with E granulosis and 11 had E multilocularis detected by clinical, radiological, and/or histopathological examinations. CONCLUSION: Cutaneous fistulization is a rare complication of HD. Complicated HD should be considered in the differential diagnosis of cases presenting with cutaneous fistulization, particularly in regions where HD is endemic.

Ustekinumab for the Treatment of Refractory Crohn's Disease: The Spanish Experience in a Large Multicentre Open-label Cohort.

BACKGROUND: Ustekinumab is a fully human monoclonal antibody against IL-12/23. Ustekinumab induced clinical response and maintained higher rate of response than placebo in patients with Crohn's disease (CD). This study aims to assess the effectiveness and safety of ustekinumab in refractory patients with CD in real-life practice. METHODS: Consecutive patients with CD who were treated with subcutaneous ustekinumab between March 2010 and December 2014 were retrospectively included in a multicenter open-label study. Clinical response was defined by Harvey-Bradshaw index score and assessed after the loading doses, 6, 12 months, and last follow-up. RESULTS: One hundred sixteen patients were included, with a median follow-up of 10 months (interquartile range: 5-21). Clinical response after loading ustekinumab was achieved in 97/116 (84%) patients. The clinical benefit at 6, 12 months, and at the end of the follow-up was 76%, 64%, and 58%, respectively. Dose escalation was effective in 8 of 11 (73%) patients. Perianal disease also improved in 11 of 18 (61%) patients with active perianal fistulae. The initial response to ustekinumab and previous use of more than 2 immunosuppressant drugs were associated with a clinical response to ustekinumab maintenance therapy. In contrast, previous bowel resection predicted a long-term failure with ustekinumab. Adverse events were reported in 11 (9.5%) patients, but none required ustekinumab withdrawal. CONCLUSIONS: Subcutaneous ustekinumab is effective and safe in a high proportion of patients with CD that were resistant to conventional immunosuppressant and antitumor necrosis factor drugs.

Therapeutic approach to Crohn disease: possible parallels with hidradenitis suppurativa. / Abordaje terapéutico de la enfermedad de Crohn: posibles paralelismos con la hidradenitis supurativa.

The current controversy in the setting of dermatology surrounding the treatment of inflammatory diseases, and specifically hidradenitis suppurativa, bears strong similarities with the debate concerning inflammatory bowel disease (IBD) that took place several years ago. That debate led to new perspectives on this disease and, in particular, its treatment after the development of biological agents.

Clinical experience of the use of adalimumab in the management of hidradenitis suppurativa. Comparison of response rates with Crohn disease. / Experiencia clínica del uso de adalimumab en el manejo de la hidradenitis supurativa. Comparando las tasas de respuesta con la enfermedad de Crohn.

The recent approval of adalimumab as the first treatment to be approved for the management of hidradenitis suppurativa has represented a before and after in the control of this chronic inflammatory disease. Given the inflammatory burden of this cutaneous disease, in the last few years hidradenitis suppurativa has been compared with inflammatory bowel disease, particularly with Crohn disease, to the point of considering hidradenitis suppurativa as "Crohn disease of the skin". These two chronic inflammatory diseases show sufficient similarities to consider whether treatment response based on the inflammatory load could also be similar. The present article aims to analyse the efficacy of adalimumab in hidradenitis suppurativa in comparison with a truly comparable disease, Crohn disease, with a view to evaluating therapeutic response rates and to drawing conclusions on the therapeutic success obtained in this disabling cutaneous disease.

Effect of postoperative use of diclofenac on pharyngocutaneous fistula development after primary total laryngopharyngectomy: Results of a single-center retrospective study.

BACKGROUND: Primary total laryngopharyngectomy is the treatment of choice in many cases of locally advanced hypopharyngeal and laryngeal cancer. Development of pharyngocutaneous fistulae is the most common postoperative complication. A recent Danish study showed significantly increased rates of anastomosal leakage after colorectal resection in patients receiving diclofenac treatment. METHODS: We retrospectively analyzed 67 patients after primary total laryngopharyngectomy to determine whether diclofenac increases the risk for development of pharyngocutaneous fistula analogously to leakage in the colorectal area. RESULTS: The fistula rate in the total study population (n = 67) was 19.4%. In the group receiving diclofenac postoperatively (n = 31), the fistula rate was 25.8%. In the patient group not receiving diclofenac (n = 36), the fistula rate was 13.9% (p = .219). CONCLUSION: Our results suggest that cyclooxygenase-2 (COX-2) selective nonsteroidal anti-inflammatory drugs (NSAIDs) should be administered with caution after laryngopharyngectomy. Additional studies on larger cohorts are required to further evaluate our findings. © 2015 Wiley Periodicals, Inc. Head Neck 38: E1515-E1520, 2016.

[Multiple sites extrapodal actinomycetoma: Favorable outcome to treatment with a combination of cotrimoxazole and NSAI]. / Mycétome actinomycosique extrapodal plurifocal : bonne réponse au traitement par l'association cotrimoxazole et AINS.

Mycetoma is a bacteriological or fungal infectious disease affecting the skin and/or soft tissues, which can be complicated by bone involvement. The most common feature is a tumor of the foot, but extrapodal localizations have been described. We report one case of a 47-year-old man who presented with tumefaction of a leg with multiple skin fistulae. Histopathological examination permitted to confirm the diagnosis of actinomycetoma and TDM showed the degree of bone and soft tissues involvement. Our case was characterized by the very inflammatory aspect of the tumor, its localization to the leg without foot involvement, the modest functional signs compared to the importance of radiological bone involvements, the deep destruction of the fibula while the tibia was apparently intact and the good response to treatment. In spite of its characteristic features, diagnosis of mycetoma is still late in our country, often with bone and/or articular spread. Priority may be given to measures for reduction of mycetoma diagnosis lateness.

[Acetazolamide in the resolution of cerebrospinal fluid cutaneous fistula after peridural analgesia: case report]. / Acetazolamida en el manejo de la fístula de líquido cefalorraquídeo posterior a analgesia peridural: reporte de caso.

BACKGROUND: Cerebrospinal fluid cutaneous fistula following spinal anesthesia is a serious and rare complication which mandates prompt diagnosis, although the treatment modalities are not well codified. CLINICAL CASE: Female aged 50 with a stage IIB cervical carcinoma; a peridural catheter was passed at lumbar level; three days after surgery, refers severe headache and to corroborate leakage cerebrospinal fluid through the puncture. The prescription was antibiotics and acetazolamide 250mg every 8hours for five days with favorable evolution. CONCLUSION: In this case, management with acetazolamide and suture of the fistula inhibits cerebrospinal fluid leakage without blood patch.

The Role of Medication in Spontaneous Gastrocutaneous Fistula Closure.

PURPOSE: Factors precipitating persistence of gastrocutaneous fistulas (GCFs) are not clearly understood. The role of proton pump inhibitors (PPIs) or histamine receptor antagonists in GCF closure is not yet studied. We aimed to identify whether these medications influence spontaneous GCF closure. METHODS: Retrospective review was performed on children who underwent gastrostomy tube insertion and removal from January 2010 to February 2013. Spontaneous GCF closure rates and medication use during gastrostomy tube removal were investigated. RESULTS: Of the 97 patients included, 48 had spontaneous GCF closure, whereas 49 required operative closure. When comparing these two groups, no significant difference existed in spontaneous GCF closure rates among patients who were on ranitidine, PPIs, or both (p = 0.09, p = 0.83, p = 0.06 respectively). Spontaneous closure occurred more in older patients (2.7 ± 4.1 vs. 0.9 ± 1.6 years, p < 0.01) and in patients without fundoplication at time of tube insertion (12.5 vs. 30.6%, p = 0.05). There were more laparoscopic placements in the group that closed spontaneously (83 vs. 61%, p = 0.02). Mean gastrostomy tube presence was longer in patients who required surgery than those with spontaneous closures (18.7 ± 10.3 vs. 35.5 ± 36.6 months, p < 0.01). CONCLUSION: Ranitidine or PPI use upon removal of gastrostomy tubes does not seem to facilitate spontaneous GCF closure in children.