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Revista Mexicana de Ortopedia Pediátrica

ISSN 2007-087X (Print)
Órgano Oficial de la Sociedad Mexicana de Ortopedia Pediátrica
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2024, Number 1-3

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Rev Mex Ortop Ped 2024; 26 (1-3)

Mild respiratory distress associated with massive chylothorax and massive bone destruction. Gorham Stout syndrome. Study of a case

Vera-Nava A, Flores-Fernández M, Vargas-Gonzalez R, Castelán-Estrada ER, Marrufo SE
Full text How to cite this article 10.35366/118244

DOI

DOI: 10.35366/118244
URL: https://dx.doi.org/10.35366/118244

Language: Spanish
References: 8
Page: 42-47
PDF size: 296.91 Kb.


Key words:

Gorham-Stout, skeletal muscle failure, respiratory distress, chylothorax, invasive osteolysis, bisphosphonates.

ABSTRACT

Gorham's syndrome, evanescent or phantom bone or idiopathic massive osteolysis is a little known pathologic entity, whose etiopathogenic cursor is still unknown. However, histologically it has typically been demonstrated bone resorption and alteration at the level of vascular or lymphatic tissues which clinically is accompanied by respiratory distress of insidious onset associated with massive chylothorax and radiographic data of massive osteolysis in different anatomical bone regions. In this article we present a clinical case of a schoolboy who, due to the lack of knowledge of the disease, is treated as a septic arthritis, and later, due to the torpid evolution, it is treated in a multidisciplinary approach in order to reach the diagnosis.


REFERENCES

  1. Moreno-Regidor A, Borrego-Ratero D, Cabo-Rodríguez A. Síndrome de Gorham-Stout. Rev Ortop Traumatol. 2004; 48(4): 295-297.

  2. Malpica Ramírez LM, Fernández de Lara CLG, Cuautli TLA, Cuéllar MY, Solís SD, Malpica RJD. Enfermedad de Gorham en la columna lumbar. Rev Esp Méd Quir. 2012; 17(4): 330-333.

  3. Domínguez Samaniego SV, Guzmán Vega O. Síndrome de gorham stout. Caso clínico. Pediatría (Asunción). 2020; 47(3): 175-179.

  4. Hong PY, Zhang XB, Zeng HQ, Zhao YL, Huang MH. Gorham-Stout syndrome: a chylothorax disease with bony destruction: a case report. Medicine (Baltimore). 2022; 101(50): e32105.

  5. Páez CFM, Morillo DMC, Dorado GA. Una causa infrecuente de quilotórax. El síndrome de Gorham-Stout. Arch Bronconeumol. 2017; 53(11): 640.

  6. Yang T, Liu L. Gorham-Stout syndrome: a case of a 7-year-old girl with massive osteolysis and chylothorax. Am J Respir Crit Care Med. 2024; 209(1): 101-103.

  7. Hurtado GR, Nortes CE, Ruiz MJ, Paredes MM. Síndrome de Gorham. Rev Osteoporos Metab Min. 2018; 10(2): 96-97.

  8. Momanu A, Caba L, Gorduza NC, Arhire OE, Popa AD, Ianole V, Gorduza EV. Gorham-Stout disease with multiple bone involvement-challenging diagnosis of a rare disease and literature review. Medicina. 2021; 57(7): 681. Available in: https://doi.org/10.3390/medicina57070681



EVIDENCE LEVEL

IV. Estudio de caso, restrospectivo, revisión sistemática.




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Rev Mex Ortop Ped. 2024;26