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1.
Int J Surg Case Rep ; 111: 108831, 2023 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-37738831

RESUMO

INTRODUCTION: Sheehan's syndrome (SS) is a rare cause of hypopituitarism resulting from postpartum haemorrhage and pituitary necrosis. It remains an underdiagnosed condition, especially in developing countries due to poor obstetric care and home deliveries. This case report highlights the significance of recognizing atypical presentations of SS, such as pancytopenia, to aid in early diagnosis and management. CASE PRESENTATION: A 40-year-old female presented with acute abdomen symptoms and was initially diagnosed with acalculous cholecystitis. However, a detailed history revealed a history of postpartum haemorrhage 18 years prior, leading to a provisional diagnosis of SS. Further investigations confirmed panhypopituitarism, including hypothyroidism, hypocortisolism, and hypogonadism. Notably, the patient also exhibited pancytopenia, a rarely reported haematological manifestation of SS. DISCUSSION: SS often presents with nonspecific symptoms, leading to delayed or missed diagnoses. In this case, the patient's initial presentation of acute abdomen symptoms was attributed to secondary adrenal insufficiency due to panhypopituitarism. The presence of pancytopenia, along with hyponatremia, further complicated the clinical picture. Hormone replacement therapy led to a remarkable improvement in the patient's condition, emphasizing the importance of early diagnosis and intervention. CONCLUSION: SS is a common cause of panhypopituitarism in developing countries, but its atypical presentations, such as pancytopenia, are rare and often overlooked. This case highlights the need for increased awareness among clinicians to consider SS in patients with unexplained haematological abnormalities, particularly in regions with high rates of postpartum haemorrhage. Early recognition and appropriate hormone replacement therapy can significantly improve patients' outcomes and prevent long-term complications associated with this underdiagnosed syndrome.

2.
Int J Surg Case Rep ; 107: 108350, 2023 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-37271024

RESUMO

INTRODUCTION: Spigelian hernia is an uncommon hernia presenting as a protrusion of abdominal contents through the spigelian fascia, lateral to the rectus abdominis. In some rare cases, Spigelian hernia can occur alongside cryptorchidism, which forms a recognized syndrome found in male infants with Spigelian hernia. This is a relatively unreported syndrome with very limited literature available regarding it, none of which is reported in Pakistan in adults. PRESENTATION OF CASE: We report a case of a 65-year-old male with right sided obstructed spigelian hernia along with the rare finding of testis in the hernial sac. The patient was successfully managed by transperitoneal primary repair (herniotomy) with orchiectomy. The patient recovered uneventfully and was discharged 5 days after the surgery. DISCUSSION: The exact pathophysiology of this syndrome remains unclear. Three theories have been proposed to explain this syndrome, including the primary defect being Spigelian hernia leading to undescended testes (Al-Salem), testicular maldescent preceding the formation of the hernia (Raveenthiran), or the absence of the inguinal canal leading to the development of a rescue canal due to the undescended testes (Rushfeldt et al.). In this case, the absence of gubernaculum was confirmed suggesting the findings to be consistent with Rushfeldt's theory. The surgical team proceeded with hernial repair and orchiectomy. CONCLUSION: In conclusion, Spigelian-Cryptorchidism syndrome is a rare syndrome in adult male, with an unclear pathophysiology. Management of this condition involves repair of the hernia along with either orchiopexy or orchiectomy, depending upon the risk factors involved.

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