Ocular Mycobacterium haemophilum infection originating in the cornea: a case report.

BACKGROUND: Mycobacterium haemophilum is a slow-growing non-chromogenic nontuberculous Mycobacterium species that can cause skin infection or arthritis in an immunocompromised population or in children. Primary infection of the healthy adult cornea is rare. The special requirements for culture make this pathogen difficult to diagnose. The study aims to report the clinical manifestation and treatment process of corneal infection and notify the awareness of M. Haemophilus keratitis among clinicians. This is the first case report of primary M. haemophilum infection in the cornea of healthy adults reported in the literature. CASE PRESENTATION: A 53-year-old healthy goldminer presented with left eye redness and a history of vision loss for four months. The patient was misdiagnosed with herpes simplex keratitis until M. haemophilum was detected using high-throughput sequencing. Penetrating keratoplasty was performed, and a large number of mycobacteria were detected by Ziehl-Neelsen staining of the infected tissue. Three months later, the patient developed conjunctival and eyelid skin infections that manifested as caseous necrosis of the conjunctiva and skin nodules. After excision and debridement of the conjunctival lesions and systemic antituberculosis drug treatment for 10 months, the patient was cured. CONCLUSION: M. haemophilum could cause primary corneal infection in healthy adults, which is an infrequent or rare infection. Owing to the need for special bacterial culture conditions, conventional culture methods do not provide positive results. High-throughput sequencing can rapidly identify the presence of bacteria, which aids in early diagnosis and timely treatment. Prompt surgical intervention is an effective treatment option for severe keratitis. Long-term systemic antimicrobial therapy is crucial.

[Two severe cases of disseminated cutaneous nontuberculous mycobacteriosis due to Mycobacterium haemophilum]. / Zwei schwere Fälle von disseminiert-kutanen, nichttuberkulösen Mykobakteriosen durch Mycobacterium haemophilum.

Mycobacterium haemophilum is a rare pathogen belonging to the group of slowly growing nontuberculous mycobacteria (NTM) that can cause infections, especially in immunocompromised patients. Detection by culturing is difficult because M. haemophilum only grows under special cultivation conditions. Therefore, it is believed that the pathogen is too rarely identified as a cause of disease overall. In addition to patients with severe immunodeficiency, e.g. due to acquired immunodeficiency syndrome (AIDS), chemotherapy or immunosuppression after transplantation, patients with underlying rheumatic diseases are increasingly described in the literature, who are at risk due to the immunosuppressive treatment regimen. Clinically, ulcerative skin alterations, lymphadenopathy and arthropathy are in the foreground. In immunosuppressed patients with unclear skin lesions, infections due to M. haemophilum should be considered and specific microbiological diagnostics should be initiated.

Mycobacterium haemophilum infection with cutaneous involvement: two case reports and an updated literature review: Mycobacterium haemophilum skin infection.

Mycobacterium haemophilum (MH) is a slow-growing, non-tuberculous Mycobacterium that most commonly causes infections in immunocompromised patients. The skin is the most prevalent site of infection and can be an isolated presentation or part of a disseminated disease. Herein, we reported a case of isolated MH infection of the hand and a case of disseminated MH infection with multiple skin lesions. In addition, other MH cases with cutaneous involvement over the last 10 years, from 2011-2022, were reviewed and analyzed. Among the 79 included cases, the common skin findings in MH infections included nodules, ulcers, abscesses, swelling, and pustules. Middle-aged patients with iatrogenic immunosuppression from glucocorticoids, mycophenolate mofetil, cyclosporine, and cyclophosphamide are the most susceptible to MH infection, with a higher risk of dissemination to internal organs. Disseminated MH infections commonly present as tenosynovitis, arthritis/arthralgia, or osteomyelitis. There is a lack of strong evidence for treatment; however, triple therapy of quinolone, macrolides, and rifampicin is most often used in clinical practice. The overall prognosis is good. The presence of iatrogenic immunocompromised diseases, lesions involving the proximal limbs, and dissemination of MH infections are associated with worse clinical outcomes.

Cryptococcal fungemia and Mycobacterium haemophilum cellulitis in a patient receiving ruxolitinib: a case report and literature review.

BACKGROUND: Ruxolitinib is a novel oral Janus kinase inhibitor that is used for treatment of myeloproliferative diseases. It exhibits potent anti-inflammatory and immunosuppressive effects, and may increase the risk of opportunistic infections. Here, we report a rare case of Cryptococcus neoformans and Mycobacterium haemophilum coinfection in a myelofibrosis patient who was receiving ruxolitinib. CASE PRESENTATION: A 70-year-old Thai man who was diagnosed with JAK2V617F-mutation-positive primary myelofibrosis had been treated with ruxolitinib for 4 years. He presented with cellulitis at his left leg for 1 week. Physical examination revealed fever, dyspnea, desaturation, and sign of inflammation on the left leg and ulcers on the right foot. Blood cultures showed positive for C. neoformans. He was prescribed intravenous amphotericin B deoxycholate with a subsequent switch to liposomal amphotericin B due to the development of acute kidney injury. He developed new onset of fever after 1 month of antifungal treatment, and the lesion on his left leg had worsened. Biopsy of that skin lesion was sent for mycobacterial culture, and the result showed M. haemophilum. He was treated with levofloxacin, ethambutol, and rifampicin; however, the patient eventually developed septic shock and expired. CONCLUSIONS: This is the first case of C. neoformans and M. haemophilum coinfection in a patient receiving ruxolitinib treatment. Although uncommon, clinicians should be aware of the potential for multiple opportunistic infections that may be caused by atypical pathogens in patients receiving ruxolitinib.

Mycobacterium haemophilum skin and soft tissue infection in a kidney transplant recipient: A case report and summary of the literature.

Non-tuberculous mycobacteria are ubiquitous pathogens causing infections in immunocompromised patients. Here, we describe a kidney transplant recipient who developed skin and soft tissue infection by Mycobacterium haemophilum, complicated by tenosynovitis and fluid collection, following an injury sustained to her right foot. Her immunosuppressant dose was reduced, and she underwent prolonged antimicrobial therapy followed by surgical debridement with a favorable outcome. Non-tuberculous mycobacteria should be considered as a potential etiology of subacute skin and soft tissue infections.

Mycobacterium haemophilum scleritis: two case reports and review of literature.

BACKGROUND: Mycobacterium haemophilum is a rare and emerging nontuberculous mycobacteria (NTM). It normally causes localized or disseminated systemic diseases, particularly skin infections and arthritis in severely immunocompromised patients. There have been 5 cases of M. haemophilum ocular infections reported in the literature. Only 1 case presented with scleritis with keratitis. Here, we reported 2 cases of M. haemophilum scleritis. One of them was immunocompetent host and had keratitis with radial keratoneuritis as a presenting sign. CASE PRESENTATION: Case 1: A 52-year-old Thai female with rheumatoid arthritis presented with scleritis. Conjunctival scraping was carried out and the culture result was positive for M. haemophilum. Despite receiving systemic and topical antibiotics, her clinical symptoms and signs worsened. Surgical debridement was performed. After surgery, the lesion was significantly improved and finally turned to conjunctival scarring. Case 2: A 32-year old healthy Thai male without underlying disease presented with nodular scleritis and keratouveitis with multiple radial keratoneuritis. Surgical debridement of the scleral nodule was performed. Initial microbiological investigations were negative. Herpes ocular infections was suspected. Topical antibiotics, oral acyclovir, low-dose topical steroids and systemic steroids were started. The scleral inflammation subsided but later the keratitis relapsed, requiring corneal biopsy. Histopathology of the specimen revealed acid-fast bacteria and M. haemophilum was identified by polymerase chain reaction (PCR) and sequencing. The diagnosis of Mycobacterial keratitis was made. Although using the combination of systemic and topical antibiotics, his clinical status progressively deteriorated. Multiple therapeutic penetrating keratoplasties were required to eradicate the infection. No recurrence was found during the 1-year follow-up in both cases. CONCLUSIONS: M. haemophilum can cause scleritis and keratitis, even in immunocompenent host. Radial keraoneuritis is first described in M. haemophilum keratitis. NTM keratitis should be considered in the differential diagnosis of patients with radial keratoneuritis. Increased awareness and early diagnosis using appropriate culture conditions and molecular techniques are important for the proper treatment of this infection. Prompt surgical intervention appears to be vital for successful management of M. haemophilum scleritis and keratitis.

Clinical Characteristics and Treatment Outcomes for Patients Infected with Mycobacterium haemophilum.

Mycobacterium haemophilum is a nontuberculous mycobacterium that can infect immunocompromised patients. Because of special conditions required for its culture, this bacterium is rarely reported and there are scarce data for long-term outcomes. We conducted a retrospective study at Siriraj Hospital, Bangkok, Thailand, during January 2012-September 2017. We studied 21 patients for which HIV infection was the most common concurrent condition. The most common organ involvement was skin and soft tissue (60%). Combination therapy with macrolides and fluoroquinolones resulted in a 60% cure rate for cutaneous infection; adding rifampin as a third drug for more severe cases resulted in modest (66%) cure rate. Efficacy of medical therapy in cutaneous, musculoskeletal, and ocular diseases was 80%, 50%, and 50%, respectively. All patients with central nervous system involvement showed treatment failures. Infections with M. haemophilum in HIV-infected patients were more likely to have central nervous system involvement and tended to have disseminated infections and less favorable outcomes.

Cluster of Lymphadenitis due to Nontuberculous Mycobacterium in Children and Adolescents 8-15 Years of Age.

BACKGROUND: Nontuberculous mycobacteria (NTM) lymphadenitis is an under-recognized entity, and data of the true burden in children are limited. Without a high index of suspicion, diagnosis may be delayed and microbiological detection is challenging. Here, we report a cluster of NTM lymphadenitis experienced in Korean children. METHODS: Subjects under 19 years of age diagnosed with NTM lymphadenitis during November 2016-April 2017 and April 2018 were included. Electronic medical records were reviewed for clinical, laboratory and pathological findings. Information regarding underlying health conditions and environmental exposure factors was obtained through interview and questionnaires. RESULTS: A total of ten subjects were diagnosed during 18 months. All subjects were 8-15 years of age, previously healthy, male and had unilateral, nontender, cervicofacial lymphadenitis for more than 3 weeks with no significant systemic symptoms and no response to empirical antibiotics. Lymph nodes involved were submandibular (n = 8), preauricular (n = 6) and submental (n = 1). Five patients had two infected nodes and violaceous discoloration was seen in seven subjects. Biopsy specimens revealed chronic granulomatous inflammation and acid-fast bacteria culture identified Mycobacterium haemophilum in two cases and NTM polymerase chain reaction was positive in two cases. Survey revealed various common exposure sources. CONCLUSION: NTM lymphadenitis is rare but increasing in detection and it may occur in children and adolescents. Diagnosis requires high index of suspicion and communication between clinicians and the laboratory is essential for identification of NTM.

Late-onset postoperative Mycobacterium haemophilum endophthalmitis masquerading as inflammatory uveitis: a case report.

BACKGROUND: Although atypical mycobacteria had been increasingly found in various ocular infections in the past decades, a slow-growing Mycobacterium haemophilum (M. haemophilum) was scarcely reported. Similar to tuberculous infection, the presentation can masquerade as low-grade granulomatous intraocular inflammation with partial response to corticosteroids. Besides, the special requirements for culture make this pathogen difficult to diagnose. The study aims to report the clinical presentation and notify the awareness of NTM endophthalmitis among clinicians. This is the first case report of late-onset, postoperative M. haemophilum endophthalmitis in the literature. CASE PRESENTATION: A 66-year-old man with non-insulin-dependent diabetes mellitus (NIDDM) manifested chronic granulomatous inflammation in the left eye after multiple glaucoma surgeries. With a diagnosis of noninfectious panuveitis, he was treated with systemic corticosteroids. The inflammation initially responded to therapy although it subsequently worsened and became purulent endophthalmitis. The vitreous cultures grew M. haemophilum. Intraocular and systemic antimicrobial treatments were administered early, but the patient eventually turned blind. CONCLUSIONS: M. haemophilum endophthalmitis is a rare but serious intraocular complication leading to loss of vision or eyeball. Awareness of atypical mycobacterial infections is necessary especially in patients with impaired immune function, previous intraocular surgery, and corticosteroid resistance. Proper laboratory investigations and treatments should be performed. However, due to the rarity of the disease, the development of guidelines for its investigation and therapy is still challenging.

Successful management of Mycobacterium haemophilum lower extremity cutaneous infection in a matched-unrelated donor stem cell transplant recipient.

Nontuberculous mycobacterial infections can often occur in individuals with adequate immune function. Such infections typically have cutaneous involvement and are caused by rapidly growing mycobacterium. Other nontuberculous mycobacteria species, like Mycobacterium haemophilum, almost always present as opportunistic infections occurring in severely immunocompromised hosts. Here, we present a complicated and protracted course of diagnosing M. haemophilum lower extremity cutaneous infection in a matched-unrelated donor stem cell transplant recipient.

Mycobacterium haemophilum infection in a renal transplant patient with inflammatory bowel disease.

A 61-year-old immunosuppressed renal transplant patient with inflammatory bowel disease presented with tender pink nodules on the trunk and extremities. An initial biopsy was suggestive of metastatic Crohn disease, but after disease persistence, a second biopsy revealed disseminated Mycobacterium haemophilum. Atypical mycobacterial infections should be considered in immunosuppressed patients. This case highlights the complexities of diagnosing such infections in patients with an underlying granulomatous condition and the particular growth requirements of M. haemophilum.

Facial Skin Lesions in Children Caused by Nontuberculous Mycobacteria.

BACKGROUND: Nontuberculous mycobacteria rarely cause facial skin lesions in immunocompetent children. AIM: I describe the clinical features and treatment of nontuberculous mycobacteria facial lesions. MATERIALS AND METHODS: The diagnosis of a facial nontuberculous mycobacteria infection was established using polymerase chain reaction. RESULTS: Of 286 children with confirmed nontuberculous mycobacteria infection, 14 (4.9%; median age 50 mos, range 9-156 mos; 5 [36%] male, 9 [64%] female) had nontuberculous mycobacteria facial skin lesions. Six (43%) had lesions on the cheek and five (36%) in the medial eye corner. Polymerase chain reaction results confirmed the presence of Mycobacterium haemophilum in eight patients (57%) and Mycobacterium avium in six patients (43%). The facial lesions were treated using a combination of clarithromycin and rifabutin for 12 weeks, with a median healing time of 4 months. CONCLUSION: Nontuberculous mycobacteria facial lesions are rare in immunocompetent children. The diagnosis requires a high index of suspicion. Nonsurgical treatment is preferable, because surgical excision of the cutaneous lesions might lead to undesirable visible facial scars.

Disseminated Mycobacterium haemophilum skeletal disease in a patient with interferon-gamma deficiency.

Disseminated non-tuberculous mycobacterial (NTM) infection is rare in immunocompetent adults. Anti-interferon-gamma (IFN-γ) autoantibodies have recently been associated with NTM infections, particularly in patients of Asian ethnicity. We describe a case of disseminated Mycobacterium haemophilum skeletal infection due to anti IFN-γ autoantibodies in a 71-year-old Cambodian man. He responded to a combination of anti-mycobacterial antibiotics without requirement for immunomodulator therapy. Testing for acquired IFN-γ deficiency due to IFN-γ autoantibodies should be considered when standard tests for immunodeficiency are negative in patients with unusual or severe opportunistic infections, including NTM.

Recurrent Mycobacterium haemophilum in a renal transplant recipient.

Mycobacterium haemophilum is a rare isolate of non-tuberculous Mycobacterium which has been reported to affect immunocompromised patients. We report a case of a 32-year-old renal transplant patient with M. haemophilum infection initially involving his left sinus which was treated with appropriate antimicrobial therapy for thirteen months. Two weeks after cessation of antibiotics the infection rapidly recurred in his skin and soft tissues of his hands and feet. This case highlights the difficult diagnostic and therapeutic implications of atypical infections in transplant patients. To our knowledge this is the first reported case of relapsed M. haemophilum infection in a renal transplant recipient.