McBride and the midfacial granuloma syndrome

Friedmann, I.

doi:10.1258/00222150260369381

Cited by 4 publications

(2 citation statements)

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“…6 Midfacial granulomatosis, described as enigmatic by some authors, has several names: Robert Woods in 1921, "malignant granuloma of the nose", Stewart in 1933, "progressive lethal granulomatous ulceration of the nose", Tsokos in 1980, "idiopathic destructive disease of the midline. 7,8 The lesions in our patient were identical to those described by Stewart and repeated by Valerio 4 and other authors. 2,[9][10][11] These authors described the course of the disease by the appearance of a nonspecific rhinosinusitis, followed by the development of an ulcerous lesion with purulent discharge.…”

Section: Discussionsupporting

confidence: 89%

“…Therapeutic success with systemic corticosteroids, dapsone, clofazimine, antimalarials and PUVA therapy (photochemotherapy) have also been reported. 4,7,18 Friedmann 12 presented a case of granulomatosis that responded well to corticosteroid therapy and in addition had benefited from methotrexate treatment and Cald-Well-Luc; death occurred following extension of the lesions into the middle cranial fossa 6 years later.…”

Section: Discussionmentioning

confidence: 99%

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Mediofacial malignant granuloma: a case report and review of the literature

Aliou,

Ibrahima,

Sayon

et al. 2023

JOENTR

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We report a case of malignant midfacial granuloma or idiopathic midfacial granulomatosis in a 50-year-old male farmer with no previous history. He was admitted to the department in an emergency context. The history was reportedly 18 months old. He presented a symptomatology made of nasal obstruction, fever, headache and rhinorrhea with an altered general state. He consulted several health facilities. The evolution was marked by the aggravation of the symptomatology. The examination at admission noted an alteration of the general state. Examination of the ENT sphere revealed ulcerative-necrotic lesions with a destructive nasal origin, with bone sequestration, midface edema, and extension to the sinuses and hard palate. The CT scan revealed bony and cartilaginous lysis, irregular thickening of the nasal pyramid, continuity of the nasal septum, and isodensity of the maxillary, ethmoidal, and left sphenoidal sinus walls without total filling. Deep biopsies were performed under general anesthesia. Histology was consistent with a granulomatous reaction with progressive tissue destruction. Mycobacterium tuberculosis was absent. Medical treatment was initiated. The evolution was marked by the extension of the lesions, malnutrition and the appearance of generalized oedema. The patient died on the 45th day of his hospitalization in a septic shock situation. Conclusion: Mediofacial granulomatosis is a difficult lesion to diagnose and manage. It requires careful histological exploration in order to differentiate it from other mediofacial destructive lesions. The evolution is most often towards extension and lethal complications.

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Section: Discussionsupporting

confidence: 89%

Section: Discussionmentioning

confidence: 99%

Mediofacial malignant granuloma: a case report and review of the literature

Aliou,

Ibrahima,

Sayon

et al. 2023

JOENTR

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Mucor irregularis Infection and Lethal Midline Granuloma: A Case Report and Review of Published Literature

Lun

2012

Mycopathologia

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Mucor irregularis (Rhizomucor variabilis) infection and lethal midline granuloma (LMG) are characterized by progressive swelling, ulceration, and destruction of the central face that is usually fatal. Pathological features are inflammation, necrosis, and granulation. LMG has been called by various names, and in recent years, it has been known as NK/T cell lymphoma. However, diagnosis still relies on the progressive necrosis course rather than malignancy in histology. The disease has long challenged physicians, particularly when it worsens with radiotherapy or chemotherapy but sometimes achieves total remission without anti-malignancy therapies. We describe a 35-year-old man who had typical clinical-pathological symptoms of LMG, which turned out to be primary M. irregularis infection; that was diagnosed by positive tissue culture and fungal elements in histology. The patient was successfully treated with antifungal therapy (liposomal amphotericin B, total 4,600 mg and amphotericin B total 277 mg, over a duration of 70 days). We hereby review current knowledge about the epidemiology, clinical manifestations, radiographic characteristics, and pathologic features of LMG with those of M. irregularis infection and their associations. We conclude that primary M. irregulars infection can mimic the clinico-pathological symptoms of LMG and the condition responds favorably to aggressive antifungal therapy.

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