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Abstract Background Oral Submucous Fibrosis (OFMF) is an oral potentially malignant disorder (OPMDs), strongly linked to betel quid chewing. It exhibits a significantly higher rate of malignant transformation compared to other OPMDs. The use of Intralesional Triamcinolone Acetonide Injection has emerged as a highly effective treatment option and has become the cornerstone of managing this condition. Case Presentation A 44-year-old female and a 40-year-old male presented with burning sensation and limited mouth opening, leading to diagnosis of OSMF. Both patients were treated with Triamcinolone Acetonide (TAC) Intralesional injections. Following a few months of treatment, a significant improvement in mouth opening was observed. However, both patients began experiencing symptoms such as facial rounding (mooning of the face), a buffalo hump, uneven hair growth, and swelling in the lower extremities. Upon recognizing these symptoms as indicative of Cushing’s Syndrome, the administration of TAC injection was discontinued. Both patients were referred to a higher-level medical facility for confirmatory tests, which revealed elevated cortisol levels in both morning (Cortisol A.M) and evening (Cortisol P.M). Conclusion TAC injection has been established as an effective treatment for OSMF. However, it is crucial to closely monitor patients for any adverse effects resulting from the treatment, which may arise from high dosage or increased frequency.

Background: Oral Submucous Fibrosis (OFMF) is an oral potentially malignant disorder (OPMDs), strongly linked to betel quid chewing. It exhibits a significantly higher rate of malignant transformation compared to other OPMDs. The use of Intralesional Triamcinolone Acetonide Injection has emerged as a highly effective treatment option and has become the cornerstone of managing this condition. Case Presentation: A 44-year-old female and a 40-year-old male presented with burning sensation and limited mouth opening, leading to diagnosis of OSMF. Both patients were treated with Triamcinolone Acetonide (TAC) Intralesional injections. Following a few months of treatment, a significant improvement in mouth opening was observed. However, both patients began experiencing symptoms such as facial rounding (mooning of the face), a buffalo hump, uneven hair growth, and swelling in the lower extremities. Upon recognizing these symptoms as indicative of Cushing's Syndrome, the administration of TAC injection was discontinued. Both patients were referred to a higher-level medical facility for confirmatory tests, which revealed elevated cortisol levels in both morning (Cortisol A.M) and evening (Cortisol P.M). Conclusion: TAC injection has been established as an effective treatment for OSMF. However, it is crucial to closely monitor patients for any adverse effects resulting from the treatment, which may arise from high dosage or increased frequency. [ABSTRACT FROM AUTHOR]

Abstract Background Glucocorticoids, among the most widely utilized drugs in veterinary medicine, are employed to treat a wide variety of diseases; however, their use often induces adverse events in dogs. The efficacy of glucocorticoids usually depends on dosage, although differences in sensitivity to glucocorticoids in individual animals have been reported. Glucocorticoids bind to the cytoplasmic glucocorticoid receptor (GR), which is expressed in almost all cells. These receptors are key factors in determining individual sensitivity to glucocorticoids. This study examined individual differences in glucocorticoid sensitivity in dogs, focusing on reactivity of the GR to prednisolone. Results We first molecularly cloned the GR gene from a healthy dog. We discovered a mutant GR in a dog suspected to have iatrogenic Cushing syndrome. The mutant GR had extra nucleotides between exons 6 and 7, resulting in a truncated form of GR that was 98 amino acids shorter than the wild-type dog GR. The truncated GR exhibited very low reactivity to prednisolone, irrespective of concentration. Conclusions We have identified the truncated form of canine GR in a dog with iatrogenic Cushing syndrome. This truncated form showed the very less sensitivity to glucocorticoid in vitro, unfortunately, we could not elucidate its clinical significance. However, our data is a first report about the function of canine GR, and will facilitate the analysis of canine glucocorticoid sensitivity.

Background: Glucocorticoids, among the most widely utilized drugs in veterinary medicine, are employed to treat a wide variety of diseases; however, their use often induces adverse events in dogs. The efficacy of glucocorticoids usually depends on dosage, although differences in sensitivity to glucocorticoids in individual animals have been reported. Glucocorticoids bind to the cytoplasmic glucocorticoid receptor (GR), which is expressed in almost all cells. These receptors are key factors in determining individual sensitivity to glucocorticoids. This study examined individual differences in glucocorticoid sensitivity in dogs, focusing on reactivity of the GR to prednisolone. Results: We first molecularly cloned the GR gene from a healthy dog. We discovered a mutant GR in a dog suspected to have iatrogenic Cushing syndrome. The mutant GR had extra nucleotides between exons 6 and 7, resulting in a truncated form of GR that was 98 amino acids shorter than the wild-type dog GR. The truncated GR exhibited very low reactivity to prednisolone, irrespective of concentration. Conclusions: We have identified the truncated form of canine GR in a dog with iatrogenic Cushing syndrome. This truncated form showed the very less sensitivity to glucocorticoid in vitro, unfortunately, we could not elucidate its clinical significance. However, our data is a first report about the function of canine GR, and will facilitate the analysis of canine glucocorticoid sensitivity. [ABSTRACT FROM AUTHOR]

Cystic fibrosis (CF) is not known to directly affect the adrenal gland, but commonly used CF therapies do impact the function of the hypothalamic-pituitary-adrenal (HPA) axis. By binding to the glucocorticoid receptor, medications such as inhaled and oral corticosteroids can enhance the systemic effects of cortisol and result in iatrogenic Cushing syndrome. Prolonged use suppresses the body's ability to make cortisol, resulting in iatrogenic adrenal insufficiency upon medication discontinuation. Chronic use of inhaled and oral corticosteroids can negatively affect bone health, growth, and glucose metabolism. This chapter provides practical guidelines regarding the screening, diagnosis, and treatment of iatrogenic adrenal insufficiency. As the guidelines are mainly derived from the asthma literature, this chapter also highlights the need for studies to evaluate the impact of CF therapies on adrenal function and other CF-endocrinopathies. • Common CF therapies are known to affect adrenal function by binding to the glucocorticoid receptors. • Patients can present with clinical features consistent with adrenal hyper- or hypo-function. • Long-term use of oral and inhaled corticosteroids can negatively affect bone health, growth, and glucose metabolism. • Patients at risk of adrenal insufficiency should be screened, diagnosed, and treated with hydrocortisone. • CF studies are needed to determine the impact of CFTR modulator therapy on adrenal insufficiency. [ABSTRACT FROM AUTHOR]

Prolonged use of topical corticosteroids, particularly in infants, albeit rare, may lead to Cushing syndrome. Central nervous system abnormalities including brain atrophy and delayed myelination on cranial magnetic resonance imaging has been reported in patients with corticosteroid treatment. We herein report a 5-month-old female infant referred to Department of Pediatric Endocrinology, Edirne, Turkey with brain atrophy and myelination delay that might be due to iatrogenic Cushing syndrome caused by topical corticosteroid use. [ABSTRACT FROM AUTHOR]

Prolonged potent topical glucocorticoid therapy in infants can cause iatrogenic Cushing’s syndrome. This case highlights the rarity of poor weight gain in iatrogenic Cushing’s syndrome. A 17-month-old boy was referred to outpatients pediatric endocrine clinic for evaluation of growth failure. On presentation his weight was 9.7kg (5th percentile) and height was 72cm (-3.6 SD below mean for age and sex). Systemic examination revealed grossly moon-like face, hypertrichosis and thin skin in the genital area. His mother reported using local clobetasol for the previous seven months for his diaper dermatitis. Baseline plasma cortisol was low (0.3ng/ml, normal range: 60 to 280ng/ml). During standard dose of synthetic adrenocorticotropic hormone test, the peak cortisol level was 0.4ng/ml (N>180ng/ml) and was consistent with hypothalamic–pituitary–adrenal axis suppression. The patient’s clinical presentation and laboratory investigations confirmed the diagnosis of secondary adrenal insufficiency and iatrogenic Cushing’s syndrome. He was treated successfully by discontinuing use of clobetasol. His appearance and growth returned to normal within two months. Morning cortisol was 101.2ng/ml after stopping the oral physiologic dose of hydrocortisone. Our case differed from other reports of iatrogenic Cushing’s syndrome by presenting in poor weight gain rather than obesity.

Key Clinical Message Our case report discusses the usefulness of administering romiplostim as a second-line treatment before splenectomy in a cirrhotic patient with immune thrombocytopenia who developed corticosteroid-induced Cushing's syndrome. Corticosteroids were tapered and consequently withdrawn. The patient made a full recovery postsplenectomy. [ABSTRACT FROM AUTHOR]

Hematopoietic stem cell transplantation (HSCT) outcomes in X-linked severe combined immune deficiency are most effective when performed with patients <3 months of age and without coexisting morbidity, and with donor cells from a matched sibling. Even under such favorable circumstances, outcomes can be suboptimal, and full cellular engraftment may not be complete, which results in poor B or natural killer cell function. Protein losing enteropathies can accompany persistent immune deficiency disorders with resultant low serum globulins (immunoglobulin A [IgA], IgG, IgM) and lymphopenia. Patients with immune disorders acquire infections that can be predicted by their immune dysfunction. Fungal infections are typically noted in neutropenic (congenital or acquired) and T-cell deficient individuals. Coexisting fungal infections are rare, even in hosts who are immunocompromised, and they require careful evaluation. Antifungal treatment may result in drug-drug interactions with significant complications. [ABSTRACT FROM AUTHOR]

Prolonged potent topical glucocorticoid therapy in infants can cause iatrogenic Cushing's syndrome. This case highlights the rarity of poor weight gain in iatrogenic Cushing's syndrome. A 17-month-old boy was referred to outpatients pediatric endocrine clinic for evaluation of growth failure. On presentation his weight was 9.7kg (5th percentile) and height was 72cm (-3.6 SD below mean for age and sex). Systemic examination revealed grossly moon-like face, hypertrichosis and thin skin in the genital area. His mother reported using local clobetasol for the previous seven months for his diaper dermatitis. Baseline plasma cortisol was low (0.3ng/ml, normal range: 60 to 280ng/ml). During standard dose of synthetic adrenocorticotropic hormone test, the peak cortisol level was 0.4ng/ml (N>180ng/ml) and was consistent with hypothalamic-pituitary-adrenal axis suppression. The patient's clinical presentation and laboratory investigations confirmed the diagnosis of secondary adrenal insufficiency and iatrogenic Cushing's syndrome. He was treated successfully by discontinuing use of clobetasol. His appearance and growth returned to normal within two months. Morning cortisol was 101.2ng/ml after stopping the oral physiologic dose of hydrocortisone. Our case differed from other reports of iatrogenic Cushing's syndrome by presenting in poor weight gain rather than obesity. [ABSTRACT FROM AUTHOR]

Topical steroids are widely used by dermatologists and other physicians for the treatment of skin diseases such as diaper dermatitis. Here, we report a 4 month-old boy with Cushing syndrome that was developed secondary to potent topical corticosteroid (diflucortolon valerate) use for only 3 days. We tried to warn the physicians about the warning signs of Cushing syndrome in patients who suffer from regional obesity, and emphasized the importance of explanation of the usage, and duration of treatment while prescribing topical steroid creams especially during infancy. [ABSTRACT FROM AUTHOR]

Iatrogenic Cushing syndrome induced by oral and parenteral corticosteroid administration is a well-known complication, and necessary precautions have to be taken. Cushing syndrome, however, following treatment with glucocorticoid-containing eye drops is a very rare complication. To the best of our knowledge, there have been only four reported cases in the literature. Herein, we present an infant boy who developed Cushing syndrome after receiving dexamethasone-containing eye drops after bilateral cataract extraction to prevent postoperative inflammatory complications. At the age of 5 months, after approx. 3 months of dexamethasone therapy, the patient presented with cushingoid facies, nephrocalcinosis and failure to grow. Iatrogenic Cushing syndrome was diagnosed and dexamethasone-containing eye drops were reduced and eventually stopped. Follow-up examinations revealed catch-up growth. Conclusion: Ocularly administered corticosteroids may have substantial systemic side effects in infants. [ABSTRACT FROM AUTHOR]

We report a case of subcutaneous emphysema and pneumomediastinum secondary to pyomyositis and necrotizing fasciitis over the right arm of a woman with underlying diabetes mellitus and iatrogenic Cushing syndrome. Gas produced by the culprit pathogen extensively dissected the subcutaneous fat and fascia of the patient's right arm and distantly spread to her face, neck, back, and thoracic wall and penetrated the soft tissue cephalically bordering her sternum, resulting in pneumomediastinum. The patient improved with antimicrobial therapy and localized debridement and fasciotomy over her right arm. [ABSTRACT FROM AUTHOR]